Evolution of the biopsychosocial profile in multiple sclerosis: Influence of medical variables associated with the disease
Abstract
En la presente tesis doctoral se ha llevado a cabo una revisión sistemática y cinco trabajos empíricos y pioneros que abordan distintitos perfiles biopsicosociales en la EM asociados a variables médicas y sociodemográficas, así como psicológicas. Además, se explora su evolución en el tiempo. En concreto, el primer trabajo pretende proporcionar una amplia revisión sobre factores clínicos, sociodemográficos y psicosociales de riesgo y protección para la calidad de vida de adultos con EM. También tiene como objetivo analizar intervenciones psicológicas destinadas a mejorar la calidad de vida en la EM. Para el análisis cualitativo 106 artículos cumplieron con los criterios de inclusión y exclusión. La discapacidad, fatiga, depresión, daños cognitivos y desempleo se identificaron como factores de riesgos para la calidad de vida, mientras que una alta autoestima, autoeficacia, resiliencia y apoyo social mostraron ser factores de protección. La revisión analizó un amplio espectro de enfoques de intervenciones psicológicas destinadas a mejorar la calidad de vida, como la atención plena, la terapia cognitivo-conductual, los grupos de autoayuda y la autogestión. La gran mayoría de las intervenciones mejoró la calidad de vida en alguno de sus aspectos. El objetivo del segundo trabajo fue explorar la evolución de salud mental y calidad de vida en pacientes con EM durante un periodo de 18 meses de seguimiento, e identificar factores de predicción. A una muestra de 314 pacientes se le aplicó en dos ocasiones durante un periodo de 18 meses: Cuestionario de Salud Breve SF-12 y el Cuestionario de Salud General (GHQ-28). Mediante los resultados obtenido en la prueba T para muestras dependientes y análisis de regresión binomial, se comprobó que existe posibilidad de una mejora de calidad de vida significativa de gran tamaño en el transcurso de 18 meses, incluso 12 años después del diagnóstico de EM. El estudio demostró la importancia de las variables sociodemográficas y clínicas para predecir la calidad de vida y la salud mental. El tercer estudio pretendió investigar cómo las características sociodemográficas y clínicas se relacionan con el uso de estrategias de afrontamiento y el apoyo social percibido. Además, exploró el impacto de estos factores sobre la calidad de vida. Se evaluó a 314 pacientes dos veces durante un periodo de seguimiento de 18 meses mediante: el Cuestionario de Afrontamiento COPE-28, la Escala Multidimensional de Apoyo Social Percibido (MSPSS) y Cuestionario de Salud Breve (SF-12). Se aplicaron las pruebas T dependientes y regresiones múltiple lineales. Este estudio pudo identificar variables sociodemográficas y clínicas asociadas a estrategias de afrontamiento disfuncionales, como la autoculpabilización y la negación, que predicen una peor calidad de vida mental, al contrario que la reinterpretación positiva. El objetivo del cuarto estudio fue explorar el PGT en pacientes con EM e identificar posibles determinantes de este. También comparó el crecimiento postraumático de 209 pacientes y sus cuidadores. 260 pacientes rellenaron el Inventario de Crecimiento Post-traumático PGTI-21 en tres momentos diferentes durante un periodo de 36 meses. Se ejecutaron análisis ANOVA de un factor, regresiones lineales múltiples y pruebas T para muestras independientes. Los resultados obtenidos muestran un incremento positivo de PGT durante el periodo de 36 meses. La influencia ejercida de variables clínicas, demográficas y de salud mental señala la necesidad de adoptar un enfoque más personalizado para poder entender el proceso de PGT en la EM. Un PGT similar en pacientes y cuidadores a nivel basal, sugiere que la elaboración del PGT es un proceso cognitivo interrelacionado en la diada paciente-cuidador. El quinto artículo pretendió analizar la influencia de PGT basal y tiempo de evaluación sobre el uso de estrategias de afrontamiento. Igualmente, exploró el papel predictor de las estrategias de afrontamiento sobre el PGT. Para ello 260 pacientes con EM contestaron el Inventario de Crecimiento Post-traumático PGTI-21 en tres momentos diferentes en un periodo de 36 meses de seguimiento. Trás realizar modeles análisis factoriales mixtos ANOVA 3X3 y regresiones lineales múltiples, se llegó a la conclusión de que para potenciar el desarrollo de PGT en la EM es necesario intervenciones tempranas que promuevan estrategias de afrontamiento adaptativas como: la reinterpretación positiva, el apoyo social, el afrontamiento activo, la planificación, el uso de religión y la autodistracción. El objetivo del sexto artículo fue investigar la influencia del PGT y salud mental en el uso de estrategias de afrontamiento en cuidadores de EM e identificar predictores biopsicosociales del afrontamiento reactivo y proactivo. Para evaluar a los 209 pacientes se utilizó: Cuestionario de Salud Breve (SF-12), Cuestionario de Salud General (GHQ-28), Inventario de Crecimiento Post-traumático PGTI-21, Cuestionario de Afrontamiento COPE-28, la Escala Multidimensional de Apoyo Social Percibido (MSPSS). Seguidamente se calculó un modelo factorial ANOVA 3X3 y modelos de regresión múltiple. Según los resultados obtenidos, puede concluirse de manera general que una mejora salud mental está asociada con un afrontamiento proactivo, mientras que el PGT se relaciona con una amplia gama de estrategias de afrontamiento tanto proactivas como reactivas en cuidadores con pacientes con EM.
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1 Evolution of the biopsychosocial profile in multiple sclerosis: Influence of medical variables associated with the disease Tesis Doctoral Departamento de Personalidad, Evaluación y Tratamiento Psicológicos Universidad de Sevilla Irene Gil González
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3 Tesis presentada por Dña. Irene Gil González y realizada bajo la dirección de la Dra. Mª Ángeles Pérez San Gregorio y el Dr. Agustín Martín Rodríguez en el Departamento de Personalidad, Evaluación y Tratamiento Psicológicos de la Universidad de Sevilla, para la obtención del Grado de Doctor y la Mención Internacional en el Título de Doctor. En Sevilla, a 9 de junio de 2023.
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5 AGRADECIMIENTOS A todos/as los/as participantes de esta esta investigación, que han hecho posible esta tesis. A la Dra. Mª Ángeles Pérez San Gregorio y al Dr. Agustín Martín Rodríguez, por involucrarse en cada paso de este camino y depositar la confianza imprescindible para desarrollar autonomía. Gracias por todas las correcciones y revisiones. Gracias por todos los “llámame que estoy disponible”, “lo puedes organizar como tú quieras” y “lo vemos con detenimiento”. Gracias por la disponibilidad y atención como directores, que han dado resultados tan productivos. A la Unidad de Esclerosis Múltiple del Hospital Virgen Macarena, por una acogida tan cálida y generosa. Gracias a cada uno/a de los/as voluntarios/as por participar. Espero seguir recordando muchos años las lecciones de vida que me regalasteis en cada entrevista. Habéis puesto en mis manos el arte de vivir con lo que se tiene. A la Universidad de Sevilla, a la Facultad de Psicología y al Departamento de Personalidad, Evaluación y Tratamiento Psicológicos por darme la oportunidad de hacer esta tesis e impartir docencia. Gracias a todos/as compañeros/as de las asignaturas compartidas. Especialmente gracias a María Luisa Avargues, por ser tan cercana y ponérmelo todo tan fácil, tengo un recuerdo precioso de Ética. A mi madre y padre, que son tan únicos. Gracias por haber aplaudido cada uno de mis logros y haberme recordado que mis esfuerzos académicos no son suficientes, son más que sobresalientes con independencia del resultado. Gracias por haberme inculcado que es más feliz el que viene a jugar que el que gana una competición. A mis abuelos, por mimarme tanto desde una mirada con la baba caída. Por todas las horas jugando a la escuela y a la esteticien, los huevitos pasados por agua, las dobles raciones de helados de Juan Ramón y los disfraces de Doña Rogelia. A mi abuela Araceli por encontrar siempre la ocasión para presumir de nieta. A mi abuela Mariquita, por ser infinita y arrolladora, tiene más de siete vidas en su voz. Frasquito, sé que te hubiera encantado verme trabajar tanto como a mí que me vieras. Te tengo presente a diario. A mi tía Juana, por aumentar cada año la colección de momentos esperpénticos. Ser tu sobrina es muy divertido. Gracias por verme especial. A Isa, por aprender juntas a amarnos sin complejos y con mucho orgullo. Por ponerle tanto empeño es ser mejores compañeras cada día. Gracias por hacerte una experta en JCR y todo lo que le rodea a esta tesis. Contigo ha sido más fácil.
6 A mis amigas, por seguir creciendo juntas después de tantas primaveras. Gracias por formar una familia elegida tan bonita, tan grande, tan nuestra. Sois mi clan. A mi amiga Elena, la niña de Palomares. Podría ser mi abogada, porque es una fiel defensora de mis derechos. Gracias por vivir mis avances como triunfos. Es muy bonito todo lo que recorrimos. A mis compañeros/as de tesis, por darnos calor y apoyo en los momentos complicados. Gracias por las dudas resueltas y documentos intercambiados. Especialmente gracias por el primer año de tesis que disfrutamos como niños chicos. Nos debemos todos los cafés y cervezas que el COVID se llevó. A mis amigas de Limerick y Boston, por hacer de mis estancias un campamento Santillana. Gracias por darle color a lo mejor del doctorado. A mi grupo de Gestalt, que es hogar. Gracias por haber sido entorno de descubrimiento en un camino que ha concurrido con esta tesis. Os llego conmigo. The last but not least, a mi mujer científica favorita, mi hermana. No solo ibas a ser la mentora de planes perversos como cortarme las pestañas o el flequillo con cuchilla. Gracias por ser mi número de emergencias y cargar con el bicho desde 1994. Gracias por aparecer con grandes proyectos que no pensaba que fuera capaz de hacer, y por supuesto, ayudarme a conseguirlos. Ojalá nunca se gaste el brillo de tus ideas. Y sin lugar a duda, gracias a mí, por aprender tanto y hacer lo mucho poquito a poco.
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8 ÍNDICE 1. MARCO LEGISLATIVO DE LA TESIS DOCTORAL ........................................... 11 2.INTRODUCCIÓN .......................................................................................................... 13 3. OBJECTIVES ................................................................................................................ 23 4.TRABAJOS QUE CONFORMAN LA TESIS DOCTORAL .................................... 24 4.1. Primer trabajo titulado “Quality of life in adults with multiple sclerosis: A systematic review” ........................................................................................................... 24 Abstract ............................................................................................................................. 25 Introduction ...................................................................................................................... 25 Methods ............................................................................................................................. 26 Results ............................................................................................................................... 28 Discussion.......................................................................................................................... 48 References ......................................................................................................................... 52 4.2. Segundo trabajo titulado “Predicting improvement of quality of life and mental health over 18-months in multiple sclerosis patients” ..................................... 67 4.3. Tercer trabajo titulado “Coping with multiple sclerosis: Reconciling significant aspects of health-related quality of life”...................................................... 68 Abstract ............................................................................................................................. 69 Introduction ...................................................................................................................... 69 Methods ............................................................................................................................. 70 Results ............................................................................................................................... 72 Discussion.......................................................................................................................... 77 References ......................................................................................................................... 81 4.4. Cuarto trabajo titulado “Beyond the boundaries of disease - Significant post-traumatic growth in multiple sclerosis patients and caregivers” ......................... 86 Abstract ............................................................................................................................. 87 Introduction ...................................................................................................................... 87 Methods ............................................................................................................................. 90 Results ............................................................................................................................... 94 Discussion.......................................................................................................................... 99 References ....................................................................................................................... 104 4.5. Quinto trabajo titulado “Coping strategies furthering post-traumatic growth in multiple sclerosis: A longitudinal study” ..................................................... 110 Abstract ........................................................................................................................... 111 Introduction .................................................................................................................... 111 Method ............................................................................................................................ 112
9 Results ............................................................................................................................. 115 Discussion........................................................................................................................ 122 References ....................................................................................................................... 127 4.6. Sexto trabajo titulado “Significance of post-traumatic growth and mental health for coping in multiple sclerosis caregivers” ....................................................... 132 Abstract ........................................................................................................................... 133 Introduction .................................................................................................................... 133 Methods ........................................................................................................................... 135 Results ............................................................................................................................. 138 Discussion........................................................................................................................ 144 References ....................................................................................................................... 149 5. OVERVIEW OF RESULTS ....................................................................................... 154 6. DISCUSIÓN ................................................................................................................. 159 6.1 Primer trabajo: identificar los factores de riesgo y prevención de la calidad de vida en adultos con esclerosis múltiple ...................................................... 159 6.2 Tercer, cuarto y quinto trabajo: identificar la influencia de las variables clínicas y sociodemográficas sobre el perfil biopsicosocial de pacientes con esclerosis múltiple .......................................................................................................... 159 6.3 Segundo, tercer, cuarto y quinto trabajo: analizar la evolución del perfil biopsicosocial en función de ciertas variables médicas en tres momentos diferentes durante un periodo de seguimiento de 36 meses. ...................................... 162 6.4 Cuarto y sexto trabajo: explorar el perfil biopsicosocial y comparar los diferentes niveles de crecimiento post-traumático entre las personas diagnosticadas de EM y sus cuidadores. ...................................................................... 167 6.5 Limitaciones y procedimientos de minimización de las mismas en los seis trabajos ........................................................................................................................... 169 6.6 Aplicabilidad y utilidad de los resultados en el área de salud ............................. 170 7. CONCLUSIONS .......................................................................................................... 173 8. RESUMEN / SUMMARY ........................................................................................... 176 9. REFERENCIAS .......................................................................................................... 180 10. ANEXOS ......................................................................................................................... i 10.1. Separata del trabajo titulado: “Quality of life in adults with multiple sclerosis: A systematic review” ......................................................................................... i 10.2. Separata del trabajo titulado: “Predicting improvement of quality of life and mental health over 18 months in multiple sclerosis” ........................................... xxii 10.3. Separata del trabajo titulado: “Coping with multiple sclerosis: Reconciling significant aspects of health related quality of life” .............................. xxiii
16 estrategias de afrontamiento designadas como desadaptativas y calidad de vida. Carver (1997) dividió las estrategias en tres categorías: centradas en el problema, centradas en la emoción y disfuncionales. Cerea et al. (2021) descubrió una asociación positiva entre calidad de vida mental y un afrontamiento centrado en la resolución de problemas, y una relación negativa con la descarga emocional y el afrontamiento pasivo. La bibliografía científica revela que los pacientes con EM utilizan menos estrategias activas y más estrategias de evitación y emocionales que la población general (Keramat Kar et al., 2019). La EM no sólo tiene un impacto en las personas que la padecen, también en la vida de los más allegados. Esta tesis además explora el perfil biopsicosocial de familiares, así como sus factores de riesgos y prevención. La calidad de vida en familiares se ve dañada, especialmente cuando ejercen el rol de cuidador informal ante la presencia de un curso inestable y síntomas moderados o graves (Di Cara et al., 2020; Hlabangana & Hearn, 2020; Merali et al., 2020). El estrés emocional y la reducción del bienestar físico y mental de cuidadores ha sido extensamente reportados en los últimos años. Hlabangana & Hearn (2020) informaron que la mitad de su muestra cumplía con criterios clínicos de depresión, así como su puntuación en calidad de vida era más baja comparado con la población general. Gafari et al. (2017) reportaron que todos los participantes sufrieron en algún momento síntomas de ansiedad, depresión y estrés, identificando periodos extensos de cuidado como el origen de su malestar. Sin embargo, aquellos familiares que perciben un mayor apoyo social de su entorno presentan menos síntomas depresivos, con independencia de la gravedad de la EM (Bambara et al., 2014). Diversos estudios señalan el efecto beneficioso del apoyo social para preservar el bienestar físico y la salud mental de los cuidadores (Dȩbska et al., 2020; Maguire & Maguire, 2020; Treder-Rochna, 2020a; van der Hiele et al., 2019). Las estrategias utilizadas para preservar el bienestar del cuidador durante la adaptación a la EM son de gran importancia. Según el Modelo Transaccional del Estrés y Afrontamiento de Lazarus & Folkman (1984), la tarea de cuidar consiste en mantener el equilibrio entre las demandas funcionales y los recursos emocionales propios del cuidador (Bassi et al., 2020). Se ha demostrado que el afrontamiento del estrés, los comportamientos que promueven la salud, el apoyo al compromiso y el replanteamiento positivo son estrategias útiles para los cuidadores, al contrario que la evitación (Maguire & Maguire, 2020; Rajachandrakumar & Finlayson, 2021). Las consecuencias perjudiciales de la EM también pueden coexistir con cambios favorables. Tanto pacientes como cuidadores han comunicado encontrar un significado positivo o beneficios a su experiencia con la EM, como reconocer su propia fortaleza personal
17 y su apoyo social (Maguire & Maguire, 2020; Rajachandrakumar & Finlayson, 2021). Este fenómeno recibe el nombre de CPT, refiere a un crecimiento interior como resultado de lidiar con un acontecimiento de vida amenazante. El CPT es un constructo multivariado que comprende una serie de beneficios como el crecimiento personal, el fortalecimiento de las relaciones personales y cambios en prioridades y objetivos vitales (Pakenham, 2007). Cuidadores y pacientes de EM normalmente reportan un mayor aprecio de la vida, sensación de liberación, un mejor cuidado de sí mismos y sus relaciones personales (Baka et al., 2021; Kivi et al., 2019; Pakenham, 2005a, 2007). Debido a su carácter beneficioso, esta tesis también plantea como un objetivo relevante el estudio de perfiles biopsicosociales asociados al CPT, así como la identificación de factores de riesgo y protección de este, tanto en pacientes como en familiares. Como el crecimiento experimentado por pacientes y cuidadores parece mantener una relación positiva y recíproca (Ackroyd et al., 2011), se propone la comparación del CPT en ambos grupos. Los resultados sobre la relación entre CPT y variables médicas son controvertidos. Hay estudios que no encuentran relación entre CPT y variables como el curso, duración de la enfermedad o gravedad (Ackroyd et al., 2011), mientras que otros encuentran una relación positiva entre la duración y daño neurológico sufrido con el grado de crecimiento experimentado (Pakenham & Cox, 2008, 2009). Asimismo, el curso remitente-recidivante parece presentar mayor grado de CPT (Barskova & Oesterreich, 2009). En salud mental, el CPT ha mostrado una relación positiva con la ansiedad y negativa con los síntomas depresivos (Barskova & Oesterreich, 2009). La falta de energía de la depresión puede obstaculizar el desarrollo del crecimiento (Kivi et al., 2019; Hart et al., 2008). Otros estudios no han encontrado ninguna asociación significativa entre salud mental y CPT en EM (Ackroyd et al., 2011). En el desarrollo de CPT, las estrategias de afrontamiento han demostrado ser un factor fundamental. Estrategias adaptativas como la reinterpretación positiva y la búsqueda de apoyo social son las que mejor predicen el CPT (Aflakseir & Manafi, 2018; Mohr et al., 1999; Pakenham, 2005, 2007; Prati & Pietrantoni, 2009). Por otro lado, estrategias consideradas menos adaptativas como la evitación, predicen un menor grado de CPT (Lorefice et al., 2018). Varios estudios coinciden en el efecto beneficioso del apoyo social para preservar el bienestar y la salud mental de los cuidadores y pacientes de EM lo que influye favorablemente al desarrollo de CPT. Tener un ambiente seguro donde poder compartir y expresarse emocionalmente parece favorecer a la elaboración y procesamiento del trauma (Dȩbska, 2020; Maguire & Maguire, 2020; Treder-Rochna, 2020b; van der Hiele 2019).
18 En resumen, en la presente Tesis Doctoral se proponen seis investigaciones centradas en el estudio del perfil biopsicosocial en la EM, ya publicadas en revistas indexadas en la Journal Citation Reports (JCR) de ISI Web of Knowledge. El primer trabajo es una revisión sistemática, enfocada en recopilar los factores de riesgo y protección de la calidad de vida en EM y analizar posibles intervenciones psicológicas para mejorarla. Los cinco trabajos restantes son empíricos centrados en: a) identificar la influencia de las variables clínicas y sociodemográficas al perfil biopsicosocial (calidad de vida, la salud mental, estrategias de afrontamiento, el apoyo social y CPT) en pacientes, b) analizar la evolución del perfil biopsicosocial en pacientes y c) explorar y comparar los diferentes niveles de CPT y sus perfiles biopsicosociales asociados en pacientes con EM y sus cuidadores. En la Tabla 1 se resumen los objetivos (generales y específicos), así como los aspectos que conciernen al método (diseño, tamaño muestral, análisis estadísticos) de cada uno de los seis trabajos que conforman esta Tesis Doctoral.
19 Tabla 1. Objetivos y método de los trabajos que conforman la presente Tesis Doctoral Objetivo general Identificar los factores de riesgo y prevención de la calidad de vida en adultos con EM Título del trabajo Quality of life in adults with multiple sclerosis: A systematic review Objetivo específico -Identificar factores clínicos, sociodemográficos y psicosociales de protección y riesgo para la calidad de vida en adultos con EM -Estudiar la calidad de vida en pacientes con EM en diferentes momentos durante la trayectoria de la enfermedad -Analizar posibles intervenciones psicológicas centradas en promover la calidad de vida en la EM Método -Revisión sistemática Objetivo general Identificar la influencia de las variables clínicas y sociodemográficas sobre el perfil biopsicosocial de pacientes con EM Título del trabajo Coping with multiple sclerosis: Reconciling significant aspect of health-related quality of life Beyond the boundaries of disease - Significant posttraumatic growth in multiple sclerosis patients and caregivers Coping strategies furthering post-traumatic growth in multiple sclerosis: A longitudinal study Objetivo específico -Estudiar la relación entre variables sociodemográficas y clínicas y el uso de estrategias de afrontamiento, así como apoyo social en la EM -Identificar predictores clínicos del CPT de pacientes con EM -Identificar estrategias de afrontamiento predictoras del CPT en tres tiempos diferentes Método -Diseño longitudinal (T1 = nivel basal, T2 = 18 meses después) -n = 314 pacientes -Correlaciones de Pearson y Spearman -Regresión lineal múltiple -Diseño longitudinal (T1 = nivel basal, T2 = 18 meses y T3 = 18 meses después) -n = 260 pacientes -ANOVA de un factor -Prueba T para muestras independientes -Regresión lineal múltiple -Diseño longitudinal (T1 = nivel basal, T2 = 18 meses y T3 = 18 meses después) -n = 260 pacientes -ANOVA factorial mixto 3x3 -Modelo de regresión lineal múltiple Objetivo general Analizar la evolución del perfil biopsicosocial en función de ciertas variables médicas en tres momentos diferentes durante un periodo de seguimiento de 36 meses Título del trabajo Predicting improvement of quality Coping with multiple sclerosis: Reconciling significant aspect of Beyond the boundaries of disease - Significant post-traumatic growth Coping strategies furthering posttraumatic growth in
20 of life and mental health over 18-months in multiple sclerosis patients health-related quality of life in multiple sclerosis patients and caregivers multiple sclerosis: A longitudinal study Objetivo específico -Investigar los cambios en la calidad de vida y la salud mental de pacientes con EM durante un periodo de 18 meses -Comparar la calidad de vida de pacientes con EM con la población general española -Identificar predictores clínicos y sociodemográficos del componente físico y mental de la calidad de vida y la salud mental -Analizar el papel de las estrategias de afrontamiento y el apoyo social, así como los factores sociodemográficos y clínicos, como predictores de la calidad de vida en la EM durante 18 meses de seguimiento -Explorar los posibles cambios de CPT en adultos con EM en tres tiempos diferentes durante 36 meses -Investigar la asociación entre CPT y estrategias de afrontamiento en un periodo de 36 meses Método -Diseño longitudinal (T1 = nivel basal, T2 = 18 meses después) -n = 314 pacientes -Prueba T para muestras relacionadas -Prueba T para muestras independientes -Regresión logística binaria -Diseño longitudinal (T1 = nivel basal, T2 = 18 meses después) -n = 314 pacientes -Correlaciones de Pearson y Spearman -Regresión lineal múltiple -Diseño longitudinal (T1 = nivel basal, T2 = 18 meses y T3= 18 meses después) -n = 260 pacientes -ANOVA de un factor -Prueba T para muestras independientes -Regresión lineal múltiple -Diseño longitudinal (T1 = nivel basal, T2 = 18 meses y T3 = 18 meses después) -n = 260 pacientes -ANOVA factorial mixto 3x3 -Modelo de regresión lineal múltiple Objetivo general Explorar el perfil biopsicosocial y comparar los diferentes niveles de crecimiento post-traumático entre las personas diagnosticadas de EM y sus cuidadores
21 Título del trabajo Beyond the boundaries of disease - Significant post-traumatic growth in multiple sclerosis patients and caregivers Significance of post-traumatic growth and mental health for coping in multiple sclerosis caregivers Objetivo específico -Comparar el CPT en pacientes y familiares con EM -Explorar el uso de diferentes estrategias de afrontamiento en cuidadores con EM en función de los niveles de crecimiento y salud mental -Identificar potenciales predictores biopsicosociales de un afrontamiento proactivo y reactivo Método -Diseño longitudinal (T1 = nivel basal, T2 = 18 meses y T3 = 18 meses después) -n = 260 pacientes - n = 209 familiares -Prueba T para muestras independientes -Regresión lineal múltiple -Diseño transversal -n = 209 familiares -ANOVA factorial 3x3 -Regresión lineal múltiple
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23 3. OBJECTIVES 1. Discuss quality of life (QOL) risk and protective factors in adults with MS: 1.1 To identify clinical, sociodemographic, and psychosocial risk and protective factors for QOL in adults with MS. 1.2 To study QOL in MS patients at different times during their disease history. 1.3 To analyze psychological interventions aimed at improving QOL in MS. 2. To identify the influence of clinical and sociodemographic variables on the biopsychosocial profile of MS patients: 2.1 To investigate the association between sociodemographic and clinical characteristics in MS and the use of coping strategies as well as social support. 2.2 To determine possible MS clinical determinants of post-traumatic growth. 2.3 To identify coping strategies predicting post-traumatic growth in MS. 3. To analyze the evolution of the biopsychosocial profile according to certain medical variables at three different points in time during a 36-month follow-up period: 3.1 To investigate changes in HQOL and mental health in a sample of MS patient over an 18-months follow-up period. 3.2 To identify clinical and sociodemographic basal predictors of QOL and mental health in an 18-months follow-up period. 3.3 To compere MS patients QOL with the general Spanish population. 3.4 To study the role of coping strategies and social support as well as sociodemographic and clinical factors as mental and physical QOL predictors in MS over an 18-months follow-up period. 3.5 To examine possible changes in post-traumatic growth in MS at three different times in a 36-month follow up period. 3.6 To investigate the association between post-traumatic growth and coping strategies over the course of 36 months. 4. To explore the biopsychosocial profile and compare the different levels of post-traumatic growth (PGT) among people diagnosed with MS and their caregivers: 4.1 Compare the PGT level of patients and caregivers. 4.2 To explore the use of different coping strategies in caregivers with MS according to PGT levels and mental health. 4.3 To determine potential biopsychosocial predictors of proactive and reactive coping in MS caregivers.
24 4.TRABAJOS QUE CONFORMAN LA TESIS DOCTORAL 4.1. Primer trabajo titulado “Quality of life in adults with multiple sclerosis: A systematic review” Este trabajo corresponde al artículo publicado que se referencia a continuación: Gil-González, I., Martín-Rodríguez, A., Conrad, R., & Pérez-San-Gregorio, M. Á. (2020). Quality of life in adults with multiple sclerosis: A systematic review. BMJ Open, 10, e041249. https://doi.org/10.1136/bmjopen- 2020-041249
25 Abstract Objective: In recent years, quality of life (QoL) in multiple sclerosis (MS) has been gaining considerable importance in clinical research and practice. Against this backdrop, this systematic review aimed to provide a broad overview of clinical, sociodemographic and psychosocial risk and protective factors for QoL in adults with MS and analyze psychological interventions for improving QoL. Method: The literature search was conducted in the Scopus, Web of Science and ProQuest electronic databases. Document type was limited to articles written in English, published from January 1, 2014 to January 31, 2019. Information from the selected articles was extracted using a coding sheet and then qualitatively synthesized. Results: The search identified 4886 records. After duplicate removal and screening, 106 articles met the inclusion and exclusion criteria for qualitative synthesis and were assessed for study quality. Disability, fatigue, depression, cognitive impairment, and unemployment were consistently identified as QoL risk factors, whereas higher self-esteem, self-efficacy, resilience and social support proved to be protective. The review analyzed a wide spectrum of approaches for QoL psychological intervention, such as mindfulness, cognitive-behavioral therapy, self-help groups and self-management. The majority of interventions were successful in improving various aspects of QoL. Conclusion: Adequate biopsychosocial assessment is of vital importance to treat risk and promote protective factors to improve QoL in patients with MS in general care practice. Key words: Multiple sclerosis, quality of life, protective and risk factors, mental and physical quality of life. Introduction The Constitution of the World Health Organization (WHO) (2019) declares health to be “…a state of complete physical, mental and social well-being and not merely the absence of disease or infirmity.” Quality of life (QoL) is a multidimensional concept that encompasses the domains included in this definition of health (Bernstein, 1995; Patti & Pappalardo, 2010). Its introduction in medical literature dates back to 1960 (Post, 2014), with its importance continuously growing to date (Gellert, 1993). Multiple Sclerosis (MS) is a chronic neurodegenerative condition, characterized by a wide range of symptoms and a highly unpredictable prognosis, which can severely affect patient QoL (Benito-León et al., 2003; Hyarat et al., 2019; Yalachkov et al., 2019). MS patients tend to report lower QoL than the general population (Amtmann et al., 2018: McCabe & McKern, 2002; Pittock et al., 2004; Schmidt et al., 2012). This diminished QoL may be due to their impaired functioning in daily living, more so if the help of caregivers is required, impeding
32 (Farran et al., 2016; Ledesma et al., 2018) suppression (Grech et al., 2018), and selfcontrolling coping (Phillips et al., 2014) were associated with lower QoL. Coping strategies were also identified as relevant mediator variables. Problem-focused, emotion-focused, and stopping unpleasant emotion coping strategies were partial mediators between fatigue (Mikula et al., 2015b) or type D personality (Mikula et al., 2016) and QoL as measured by the mental composite score (MCS). Other psychological factors According to Van Damme et al. (2016) acceptance of the illness is a protective factor for QoL. The role of flexible adjustment and tenacious goal pursuit in achieving personally blocked goals was not as clear, although their findings showed a trend towards a positive relationship. Resilience was confirmed as a protective factor of QoL in MS (Nakazawa et al., 2018; Nery-hurwit et al., 2018). Moreover, Koelmel et al. (2017) highlighted its role as a mediator variable in the relationship between social support and MCS. High levels of self-efficacy (Strober, 2018; Wilski & Tasiemski, 2016), self-esteem (Wilski & Tasiemski, 2016), illness identity (Wilski & Tasiemski, 2016) and sense of coherence (Calandri et al., 2018) correlated with higher QoL, and self-esteem mediated in the relationship of social support with MCS (Mikula et al., 2017). Ultimately, cognitive fusion, the extent to which people feel fused with or attached to their thoughts, mediated the relationship between stigma and QoL in MS (Valvano et al., 2016).
33 Table 2. Characteristics of included articles. Authors, Publication year Study design Quality of life measurement Sample size (N) Age (mean) Sex (Female %) Main results Risk factors Protective factors Clinical variables Gupta et al. (2014) Crosssectional Short Form Health Survey 12 (SF-12) N = 74451 47.9 years 51.3 % EDSS (PCS) Gross et al. (2017) Crosssectional Short Form Health Survey 36 (SF-36) N = 810 RRMS 48.9years SPMS 55.7 years RRMS 71.6 % SPMS 56.2 % Progressive MS type (PCS) Zhang et al. (2019) Crosssectional EuroQol 5- Dimensions (EQ-5D) N = 1958 55.3 years 78.1% Progressive MS type onset Rezapour et al. (2017) Crosssectional Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 171 35.7 years 76.6% Relapses in the last 3 months Mild EDSS RRMS Type Marck et al. (2017) Crosssectional Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 2296 45.5 years 82.2% Pain Milinis et al. (2016) Crosssectional Leeds MS Quality of Life Scale (MSQoL) N = 701 48.8 years 72% Spasticity Zettl et al. (2014) Crosssectional EuroQol 5- Dimensions (EQ-5D) Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 414 48.6 years 64.3 % Spasticity Leonavicius et al. (2016) Crosssectional Short Form Health Survey 36 (SF-36) N = 137 44.7 years 72.3% Fatigue (MCS) Garg et al. (2016) Crosssectional Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 89 54.26 years 66% Fatigue Fernández-Muñoz et al. (2015) Crosssectional Short Form Health Survey 36 (SF-36) N = 108 44 years 55% Fatigue Weiland et al. (2015) Crosssectional Multiple Sclerosis Quality of Life- N = 2738 45.5 years 82.3% Fatigue
34 54 (MSQoL- 54) Aygünoğlu et al. (2015) Crosssectional Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 120 34.24 years 70 % Fatigue Vister et al. (2015) Crosssectional World Health Organization Disability Assessment Schedule (WHODAS) 2.0 N = 210 50.8 years 72.4 % Fatigue Tabrizi et al. (2015) Crosssectional Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 217 36.2 years 79 % Fatigue Poor sleep quality Low MCS (PCS) White et al. (2019) Crosssectional EuroQol 5- Dimensiones (EQ-5D) N = 531 51.60 years 70.1 % Sleep disorder Barin et al. (2018) Crosssectional EuroQol 5- Dimensions (EQ-5D) EuroQol Visual Analogue Scale (EQ-VAS) N = 855 48 years 72.7 % Fatigue Balance Spasticity Paralysis Walking difficulties Kratz et al. (2016) Crosssectional Short-Form Health Survey 36 (SF-36) N = 180 50.5 years 78 % Fatigue (MCS) Pain (MCS) Memory loss (MCS) Colbeck et al. (2018) Crosssectional RAND-36 Health Item Survey (RAND-36) N = 30 - 73.33% Cognitive fatigue Low sensory sensitivity Sensation avoiding Grech et al. (2015) Crosssectional Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 107 48.8 years 77.6 % Cognitive inflexibility Sgaramella et al. (2014) Crosssectional Quality of life questionnaire (QoL) N = 39 42.2 years 71.8 % Executive function Khalaf et al. (2016) Crosssectional Short Form Health Survey 36 (SF-36) N = 1048 47.8 years 81% Lower urinary tract symptoms Vitkova et al. (2014) Crosssectional Short Form Health Survey 36 (SF-36) N = 223 38.4 years 67.3 % Bladder dysfunction (PCS) Sexual dysfunction (MCS)
35 Qaderi et al. (2014) Crosssectional Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 132 36.9 years 100 % Sexual problems (PCS and MCS) Schairer et al. (2014) Crosssectional Short-Form Health Survey 12 (SF-12) N = 6138 50.6 years 74.7 % Sexual dysfunction Ma et al. (2017) Crosssectional Multiple Sclerosis Impact Scale (MSIS-29) N = 231 40.2 years 58.4 % Sleep disorders Psychosocial variables Ledesma et al. (2018) Crosssectional World Health Organization Quality of Life Questionnaire (WHOQoL- BREF) N = 26 39.2 years 57.5% Problem avoidance Social withdrawal Wishful thinking Self-criticism Anxiety Depression Problem resolution Cognitive restructuring Emotional social and instrumental support Emotional expression Grech et al. (2018) Crosssectional Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 107 48.8 years 77.57% Behavioral disengagement Suppression and self-control Emotional venting Acceptance Growth Restrain Zengin et al. (2017) Crosssectional World Health Organization Quality of Life Questionnaire (WHOQoL- BREF) N = 214 36-46 years 53.2% Self-distraction Denial Substance use Planning Active coping Acceptance Positive reinterpretation Social support Farran et al. (2016) Crosssectional Multiple Sclerosis International Quality of Life Questionnaire (MusiQoL) N = 34 36 years 56% Self-criticism Escape avoidance Distancing Self-controlling Emotional social support Instrumental social support Planful problem solving Positive reappraisal Mikula et al. (2014) Crosssectional Short Form Health Survey 36 (SF-36) N = 113 40.8 years 77 % Problem focused coping Stopping unpleasant emotion Getting support Van Damme et al. (2016) Crosssectional Short-Form Health Survey 36 (SF-36) N = 117 41 years 70.2 % Acceptance (PCS and MCS) Tenacious goal pursuit (PCS) Flexible goal adjustment (MCS)
36 Wilski et al. (2016) Crosssectional Multiple Sclerosis Impact Scale (MSIS-29) N = 257 47.9 years 69.93% Self-efficacy Self-esteem Illness identity Nery-Hurwit et al. (2018) Crosssectional Function Neutral Health- Related Quality of Life Short Form (FuNHRQOL- SF) N = 259 48.6 years 84.23% Resilience Self-compassion Calandri et al. (2018) Crosssectional Short Form Health Survey 12 (SF-12) N = 90 37 years 61.1 % Sense of Coherence Fernández-Muñoz et al. (2018) Crosssectional Short Form Health Survey 36 (SF-36) N = 108 44 years 55 % Depression Pham et al. (2018) Crosssectional Short Form Health Survey 12 (SF-12) N = 310 49 years 73.6 % Anxiety Prisnie et al. (2018) Longitudina l (T1 = basal level/ T2 = 2 weeks later) Short Form Health Survey 12 (SF-12) N = 139 40 years 70.5% Anxiety Depression Alsaadi et al. (2018) Crosssectional World Health Organization Quality of Life Questionnaire (WHOQoL- BREF) N = 80 35.1 years 65 % Anxiety Depression Labiano-Fontcuberta et al. (2015) Crosssectional Functional Assessment of Multiple Sclerosis (FAMS) N = 157 41.7 years 66.9% Depression Anxiety Anger expression-in Paziuc et al. (2018) Crosssectional Short-Form Health Survey 36 (SF-36) N = 60 46 years 85 % Trait anxiety State anxiety Depression Extraversion Emotional Stability Phillips et al. (2014) Crossseccional World Health Organization Quality of Life questionnaire( WHOQoL- BREF) N = 32 44.0 years 75 % Emotional problems Salhofer-Polanyi et al. (2018) Crosssectional Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 139 40.0 years 70.5% Depressive temperament Cyclothymic temperament Hyperthymic temperament Demirci et al. (2017) Crosssectional Short Form Health Survey 36 (SF-36) N = 74 35.3 years 65.51% Type D personality
37 Mikula et al. (2015) Crosssectional Short Form Health Survey 36 (SF-36) N = 116 40.4 years 72.4% Social participation (MCS y PCS) Costa et al. (2017) Crosssectional Short Form Health Survey 36 (SF-36) N = 150 41.7 years 70.7% Social support Clinical, psychosocial, and demographic variables Nakazawa et al. (2018) Crosssectional Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 63 41.7 years 66.7 % EDSS level Resilience Ciampi et al. (2018) Crosssectional Multiple Sclerosis Impact Scale (MSIS-29) N = 43 57.2 years 65.1 % EDSS level Fatigue Depression Fernández-Jiménez et al. (2015) Crosssectional Functional Assessment of Multiple Sclerosis (FAMS) N = 97 47.3 years 82.5 % EDSS level Depression Klevan et al. (2014) Crosssectional Short Form Health Survey 36 (SF-36) N = 93 41.8 years 69 % EDSS (PCS) Fatigue Depression Apathy Williams et al. (2014) Crosssectional Short-Form Health Survey 36 (SF-36) Short-Form Health Survey 12 (SF-12) N = 447 49.3 years 70.02 % Pain (PCS) Muscle spasms (PCS) Stiffness (PCS) Depression (MCS) Hyncicova et al. (2018) Crosssectional Short Form Health Survey 36 (SF-36) N = 67 32.3 years 53.7% Number and severity of symptoms Fatigue Stress Depression Anxiety Shahrbanian et al. (2015) Crosssectional Person Generated Index (PGI) N = 188 43 years 74% Pain Fatigue Irritability Anxiety Depression Sleep disorder Cognitive deficit Strober et al. (2018) Crosssectional Short Form Health Survey 36 (SF-36) N = 69 40.4 years 89.5% Pain Fatigue Behavioral disengagement Denial Depression Anxiety High neuroticism Acceptance Growth Emotional social and instrumental support Planning Active coping
38 Low extroversion Low selfefficacy Positive reinterpretation Humor Dymecka et al. (2018) Crosssectional Multiple Sclerosis Impact Scale (MSIS-29) N = 137 46.5 years 53.3 % Fatigue Upper-limb disability Lower-limb disability Cognitive disorders Emotional problems Samartzis et al. (2014) Crosssectional Short Form Health Survey 36 (SF-36) N = 100 40.5 years 64 % Perceived planning/organiz ation dysfunction Perceived retrospective memory dysfunction Depression Brola et al. (2016) Crosssectional EuroQol 5- Dimensions (EQ-5D) EuroQol Visual Analogue Scale (EQ-VAS) Multiple Sclerosis Impact Scale (MSIS-29) N = 2385 37.8 years 69.7% EDSS level MS duration Lack of DMD treatment Age Brola et al. (2017) Crosssectional EuroQol 5- Dimensions (EQ-5D) EuroQol Visual Analogue Scale (EQ-VAS) Multiple Sclerosis Impact Scale (MSIS-29) N = 765 44.9 years 67.7 % EDSS MS duration Be unemployed Age No immunomodulat ory therapy Abdullah et al. (2018) Crosssectional Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 200 35.1 years 68% Motor symptoms Low resistance Sensory symptoms Low income Be unemployed Nickel et al. (2018) Crosssectional Multiple Sclerosis International Quality of Life (MusiQoL) N = 1220 47.8 years 76 % EDSS Comorbidity High educational level High employment status Campbell et al. (2017) Crosssectional Functional assessment of N = 62 49.4 years Cognitive deficit Be unemployed
39 multiple sclerosis (FAMS) EuroQol 5- Dimensions (EQ-5D) 69.35% Chiu et al. (2015) Crosssectional Short Form Health Survey 12 (SF-12) N = 157 43.8 years 86% Be unemployed Disability adjusted employment Boogar et al. (2018) Crosssectional Multiple Sclerosis Quality of Life- 54 (MSQoL- 54) N = 193 38.1 years 64.8 % High disability Depression Low socioeconomic status Positive story treatment Bishop et al. (2015) Crosssectional Quality of Life Scale (QOLS) N = 1839 54 years 78.1 % Number and severity of symptoms Perceived stress High educational level High employment status Job satisfaction Job match Cioncoloni et al. (2014) Crosssectional Short Form Health Survey 36 (SF-36) N = 57 41.7 years 68.42% EDSS level Fatigue Pain Bladder dysfunction Bowel dysfunction Depressive manifestations Sleeping problems Introverted personality Be unemployed Cichy et al. (2016) Crosssectional Quality of Life Scale (QOLS) N = 703 63 years 76 % Progressive MS Progressive diagnosis Number and severity of symptoms Perceived stress Be male Not married/not living with significant other Unable to meet living expenses Mediational variables Mediator variable Mediated relation Mikula et al. (2016) Crosssectional Short Form Health Survey 36 (SF-36) N = 156 40 years 75 % Coping strategies Problem focused Emotional focused Stopping Personality type D and MCS
40 Social factors Social support (Costa et al., 2017) and participation (Mikula et al., 2015a) were positively related with QoL. Several mediators in this relationship were mentioned above. Demographic factors Employment was found to be the leading sociodemographic factor influencing QoL. Several studies displayed an association between unemployment and lower QoL (Abdullah & Badr, 2018; Blora et al, 2017; Campbell et al., 2017; Cioncoloni et al., 2014; Chiu et al., 2015). Others showed a positive correlation between jobs adapted to disability (Chiu et al., 2015), job match and job satisfaction (Bishop et al., 2015), high employment status (Bishop et al., 2015; Nickel et al., 2018), and QoL in MS. Low socioeconomic status (Boogar et al., 2018) and financial straits (Cichy et al., 2016) were also risk factors for lower QoL. Brola et al. (2016, 2017) noted that not having access to an adequate pharmacological treatment put QoL in danger. Congruent with this finding, Boogar et al. (2018) found a positive treatment experience to be a protective factor. Other sociodemographic variables related to poorer QoL in MS were male sex (Cichy et al., 2016), old age (Brola et al. 2016, 2017), unmarried or living with significant others (Cichy et al., 2016), whereas a higher education was a protective factor (Nickel et al., 2018). Mikula et al. (2015) Crosssectional Short Form Health Survey 36 (SF-36) N = 154 40.05 years 76% Coping strategies Fatigue and MCS and PCS Mikula et al. (2017) Crosssectional Short Form Health Survey 36 (SF-36) N = 74 35.3 years 65.51% Self-esteem Social participation and MCS Koelmel et al. (2017) Longitudina l (T1 = basal level/ T2 = 10 weeks later/ T3 = 26 weeks later/ T4 = 52 weeks later) Short Form Health Survey 8 (SF-8) N = 163 52.2 years 87.1% Resilience Social support and MCS Valvano et al. (2016) Crosssectional Leeds MS Quality of Life Scale (MSQoL) N = 128 45.5 years 85% Cognitive fusion Stigma and QoL EDSS, expanded disability status scale; PCS, physical composite; RRMS, remittent remitting; SPMS, secondary progressive; MS, multiple sclerosis; MCS, mental composite score; DMD, disease modifying drug; QoL, quality of life.
41 Disease history Table 3 summarizes the characteristics of studies focusing on QoL at different ages and times in the disease history. Some of the selected studies examined QoL in MS in its early years. According to Possa et al. (2017), QoL decreased in the first year of diagnosis, as assessed by the MCS and physical composite score (PCS). Stern et al. (2018) found the worst QoL in the youngest group of MS patients. Calandri et al. (2017) found that during the first three years from diagnosis, problem-solving and avoidance coping strategies had a positive effect on QoL. Nourbakhsh et al. (2016) also studied factors influencing the development of QoL in the first three years. Their results showed that higher baseline levels of fatigue and depression predicted worse QoL as assessed by the PCS, whereas lower cognitive functioning and higher fatigue predicted a worse MCS. Another study on QoL in MS by Buhse et al. (2014) focused on old age. These authors identified neurological impairment, physical disability, depression, and comorbidity with thyroid disease as risk factors for worse QoL as assessed by the PCS in a sample of elderly MS patients. On the contrary, being widowed and employed were identified as protective PCS factors. In a longitudinal study, Kinkel et al. (2015) showed that a second clinical event consistent with clinically defined MS, higher EDSS at the time of diagnosis and an earlier MS onset predicted a decrease in PCS 10 years after diagnosis. Bueno et al. (2015) also showed that progression from benign MS to non-benign MS predicted a decrease in PCS 25-30 years after diagnosis. Some longitudinal predictors of QoL identified have been: longer MS duration predicted worse QoL two years later (Baumstarck et al., 2015), and worse EDSS predicted worse QoL two (Baumstarck et al., 2015), six (Tepavcevic et al., 2014), and ten (Chruzander et al., 2014) years later. Depression predicted worse QoL six (Tepavcevic et al., 2014) and ten (Chruzander et al., 2014) years later, and stronger pain (Young et al., 2017) and cognitive impairment (Chruzander et al., 2014) predicted worse QoL ten years later.
48 questionnaire (WHOQoL- BREF) 42.2 years 59% relationships (p < 0.001) in the SH group. Based on symptoms and self-management Mulligan et al. (2016) Fatigue selfmanagement program “Minimize Fatigue, Maximize Life: Creating Balance with Multiple Sclerosis (MFML)” Longitudinal (T1 = 1 month pre-treatment/ T2 = pre-treatment/ T3 = posttreatment). Short Form Health Survey 12 (SF-12) N = 24 49.3 years 100% Positive but not significant changes in SF-12 (p > 0.05). Thomas et al. (2014) Group-based fatigue management (FACETS) Longitudinal (T1 = 1 week before treatment/ T2 = 1 month post-treatment/ T3 = 4 month post-treatment/ T4 = 12 month post-treatment) Multiple Sclerosis Impact Scale (MSIS-29) Short Form Health Survey 36 (SF-36) N intervention/ control = 84/ 80 48 years 73% Changes in physical health MSIS-29 (p = 0.046) and vitality SF-36 (p = 0.03) at T4. Ehde et al. (2015) Telephone- Delivered Self- Management (SM) Longitudinal (T1 = before group randomization/ T2 = posttreatment/ T3 = 6 month posttreatment/ T4 = 12 month posttreatment) Short Form Health Survey 8 (SF-8) N intervention/ control = 75/ 88 51 years 89.3% MCS and PCS increase at T2, T3 and T4 (p < 0.05). Feicke et al. (2014) Education program for selfmanagement competencies (S.MS) Longitudinal (T1 = 1 basal level/T2 = posttreatment /T3 = 6 month posttreatment) Hamburg quality of life questionnaire in multiple sclerosis (HAQUAMS) N intervention/ control = 31/ 33 41.9 years 87.1% Stable positive changes in QoL (p = 0.007). Other psychological intervention Leclaire et al. (2018) Group Positive Psychology Longitudinal (T1 = basal level /T2 = posttreatment) Short Form Health Survey 36 (SF-36) N = 11 53.5 years 100% Increase in SF-36 vitality subscale score (p = 0.016). Increase in mental health SF-36 subscale (p = 0.098) that did not reach statistical significance. FAMS, functional assessment of multiple sclerosis; QoL, quality of life; PCS, physical component score; MCS, mental component score; MSQoL-54, multiple sclerosis quality of life instrument; CBT, cognitive behavioral therapy; SF-36, short form health survey 36; MSIS-29, multiple sclerosis impact scale; EQ-5D, euroqol 5-dimensions; HBT, hope-based group therapy; RR, relapsing-remitting; SH, self-help group; SF-12, short-form health survey Discussion Firstly, the present systematic review was intended to identify risk and QoL protective factors in MS. The results showed that the EDSS was most employed for assessment of functional
49 impairment (Blora et al, 2016, 2017; Boogar et al., 2018; Ciampi et al., 2018; Cioncoloni et al., 2014; Fernández-Jiménez et al., 2015; Gupta et al., 2014; Klevan et al., 2014; Nakazawa et al., 2018; Nickel et al., 2018; Rezapour et al., 2017). As expected, the number and severity of symptoms and associated impairment appeared to play a crucial role in QoL. Fatigue, (Aygünoğlu et al., 2015; Barin et al., 2018; Ciampi et al., 2018; Dymecka & Bidzan, 2018; Fernández-Muñoz et al., 2015; Garg et al., 2016; Hyncicova et al., 2018; Klevan et al., 2014; Kratz et al., 2016; Leonavicius, 2016; Radfar, 2015; Shahrbanian et al., 2015; Strober, 2018; Tabrizi &; Vister et al., 2017; Weiland et al., 2015) cognitive impairment (Campbell et al., 2017; Colbeck, 2018; Dymecka & Bidzan, 2018; Kratz et al., 2016; Ma et al., 2017; Samartzis et al., 2014; Shahrbanian et al., 2015) , and pain (Boogar et al., 2018; Kratz et al., 2016; Marck et al., 2017; Shahrbanian et al., 2015; Strober, 2018; Williams et al., 2014), in particular, were the focus of a large number of studies, and were confirmed as important risk factors. Longitudinal studies suggested that greater fatigue (Nourbakhsh et al., 2016), pain (Young et al., 2017), and cognitive impairment (Chruzander et al., 2014; Nourbakhsh et al., 2016) also predicted worse QoL up to 10 years later. This has important clinical implications, as treatment of the abovementioned symptoms should be prioritized. In general, functional impairment (Baumstarck et al., 2015; Chruzander et al., 2014; Tepavcevic et al., 2014), as well as longer duration of illness (Baumstarck et al., 2015), were predictors of QoL two to 10 years later, whereas disease progression (Bueno et al., 2015) from benign to non-benign MS predicted QoL as measured by the PCS up to 30 years later. Among the emotional symptoms, there was convincing evidence that depression (Alsaadi et al., 2017; Boogar et al., 2018; Ciampi et al., 2018; Cioncoloni et al., 2014; Fernández- Jiménez et al., 2015; Fernández-Muñoz et al., 2018; Hyncicova et al., 2018; Klevan et al., 2014; Labiano-Fontcuberta et al., 2015; Ledesma et al., 2018; Paziuc & Radu, 2018; Prisnie et al., 2018; Samartzis et al., 2014; Shahrbanian et al., 2015; Strober, 2018; Williams et al., 2014), along with depressive temperament (Salhofer-Polanyi et al., 2018) and anxiety (Alsaadi et al., 2017; Hyncicova et al., 2018; Labiano-Fontcuberta et al., 2015; Ledesma et al., 2018; Paziuc & Radu, 2018; Pham et al., 2018; Prisnie et al., 2018; Strober, 2018; Zhang et al., 2019), were associated with lower QoL, and that depression also predicted QoL up to 10 years later (Chruzander et al., 2014). The coping strategies applied obviously influenced QoL in MS, however their effect depended on the specific circumstances of the disease history. For example, problem-solving and avoidance coping, normally classified as opposite strategies, both seemed to have a positive effect on the MCS in the first three years of diagnosis (Calandri et al., 2017).
50 However, in general, strategies associated with denial (Strober, 2018; Zengin et al., 2017) and avoidance of the challenges of the disease, such as problem avoidance (Farran et al., 2016; Ledesma et al., 2018), behavioral disengagement (Grech et al., 2018; Strober, 2018), distancing (Farran et al., 2016), self-distraction (Zengin et al., 2017), social withdrawal (Ledesma et al., 2018), wishful thinking (Ledesma et al., 2018),were associated with a lower QoL. On the other hand, strategies based on acceptance and active commitment, such as active coping, humor, problem resolution, cognitive positive restructuring, and emotional expression, led to higher QoL in MS (Farran et al., 2016; Grech et al., 2018; Ledesma et al., 2018; Mikula et al., 2014; Strober, 2018; Zengin et al., 2017). Obviously, there is a close connection between the active confrontation of the challenges of illness and specific personality-based convictions, such as a high self-efficacy. Thus, higher self-efficacy (Strober, 2018; Wilski & Tasiemski, 2016), selfesteem (Wilski & Tasiemski, 2016), and sense of coherence (Calandri et al., 2018) improved QoL in MS. Regarding sociodemographic influences on QoL, not surprisingly, unemployment, a low socioeconomic status (Boogar et al., 2018) and financial difficulties (Cichy et al., 2016) proved to be major risk factors (Abdullah & Badr, 2018; Blora et al, 2017; Campbell et al., 2017; Chiu et al., 2015; Cioncoloni et al., 2014). In keeping with the negative influence of the scarcity of resources, lack of access to therapy was also identified as a risk factor (Blora et al, 2016, 2017). The second aim of this systematic review was to study QoL in MS patients at different times during their disease history. Two studies showed diminishing QoL in MS patients in its early stage (Possa et al., 2017; Stern et al., 2018). This might have to do with the fact that patients being diagnosed with a severe chronic disease need a certain time to come to terms with this emotional shock. Oscillation between avoidance and problem-solving, which both have a positive influence in the first three years after diagnosis (Calandri et al., 2017), may be behind this inner struggle. In older patients, neurological impairment and physical disability (Calandri et al., 2017), which represent the age-associated increase in physical impairment, were identified as risk factors for QoL in MS. Finally, the third aim of this review was to analyze psychological interventions for the improvement of QoL in MS. Symptomatic improvement of psychopathology usually at the center of psychotherapy outcome studies, was not the primary focus of our review (Geiser et al., 131). Eight of the intervention studies specifically treated depressive symptomatology (Carletto et al., 2017; Case et al., 2018; Chruzander et al., 2016; Ghodspour et al., 2018; Graziano et al., 2014; Kikuchi et al., 2019; Spitzer & Pakenham, 2018) either with
51 mindfulness-based or cognitive-behavioral approaches, both of which proved to be successful. Three studies were specifically directed towards the treatment of fatigue (Case et al., 2018; Mulligan et al., 2016; Thomas et al., 2014) by light guided imagery or self-management programs. Both the imagery and self-management group intervention approaches were successful, whereas the individual self-management program did not show significant improvement. A variety of mindfulness-based approaches (Besharat et al., 2017; Blankespoor et al., 2017; Simpson et al., 2017) and a Community-based intervention were directed at stress reduction (Spitzer & Pakenham, 2018). Three of the four studies showed some kind of improvement in QoL, including the only study with a control group. Several of the interventions were designed to reinforce protective factors in MS patients. Graziano et al. (2014) focused on identity redefinition, sense of coherence and self-efficacy. Pakenham et al. (2018) implemented a program based on resilience training, and the program by Blair et al. (2017) focused on the improvement of emotion regulation. All of them were successful in improving QoL, confirming the alternative focus on protective factors instead of risk factors. A wide spectrum of interventions based on social support concentrated on reinforcement of the social network of MS patients, for example, self-help groups (Eliášová et al., 2015), hope-based group therapy (Liu et al., 2017), supportive-expressive therapy (Abolghasemi et al., 2016), and social cognitive training with support partners (Jongen et al., 2014, 2016). All interventions aimed at helping people overcome MS barriers in daily living by strengthening their social support, improving some aspects of QoL. This is consistent with the studies mentioned above (Costa et al., 2017; Mikula et al., 2015a) and emphasizes the importance of social support and participation as a protective factor for QoL. The main limitation of this study was the impossibility of carrying out a quantitative synthesis of the results, due to the heterogeneity of methodologies and designs in the articles included. Due to the vast number of topics and limited resources our search was restricted to a five-year period through January 2019. This review was intended to give a broad overview of QoL in MS. The findings show the importance of clinical, psychosocial and demographic variables as QoL risk and protective factors. A variety of psychological interventions ranging from mindfulness-based and cognitive-behavioral approaches to self-help groups addressing these factors were identified as promising options for improving QoL. These findings have important clinical implications.
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66 Young, J., Amatya, B., Galea, M. P., & Khan, F. (2017). Chronic pain in multiple sclerosis: A 10-year longitudinal study. Scandinavian Journal of Pain, 16, 198-203. https://doi.org/10.1016/j.sjpain.2017.04.070 Ysrraelit, M. C., Fiol, M. P., Gaitán, M. I., & Correale, J. (2018). Quality of life assessment in multiple sclerosis: Different perception between patients and neurologists. Frontiers in Neurology, 8. https://doi.org/10.3389/fneur.2017.00729 Zengin, O., Erbay, E., Yıldırım, B., & Altındağ, Ö. (2017). Quality of life, coping, and social support in patients with multiple sclerosis: A pilot study. Turk Noroloji Dergisi, 23(4), 211-218. https://doi.org/10.4274/tnd.37074 Zettl, U. K., Henze, T., Essner, U., & Flachenecker, P. (2014). Burden of disease in multiple sclerosis patients with spasticity in Germany: Mobility improvement study (Move I). European Journal of Health Economics, 15(9), 953-966. https://doi.org/10.1007/s10198- 013-0537-5 Zhang, Y., Taylor, B. V., Simpson, S., Blizzard, L., & van der Mei, I. (2019). Patient-reported outcomes are worse for progressive-onset multiple sclerosis than relapse-onset multiple sclerosis, particularly early in the disease process. European Journal of Neurology, 26(1), 155-161. https://doi.org/10.1111/ene.13786
67 4.2. Segundo trabajo titulado “Predicting improvement of quality of life and mental health over 18-months in multiple sclerosis patients” Este trabajo corresponde al artículo publicado que se referencia a continuación: Gil-González, I., Pérez-San-Gregorio, M. Á., Conrad, R., & Martín-Rodríguez, A. (2021). Predicting improvement of quality of life and mental health over 18-months in multiple sclerosis patients. Multiple Sclerosis and Related Disorders, 53, e041249. http://doi.org/ 10.1016/j.msard.2021.103093 Este artículo no está disponible por embargo en Multiple Sclerosis and Related Disorders. Puede consultarlo en el siguiente enlace: http://doi.org/ 10.1016/j.msard.2021.103093
68 4.3. Tercer trabajo titulado “Coping with multiple sclerosis: Reconciling significant aspects of health-related quality of life” Este trabajo corresponde al artículo publicado que se referencia a continuación: Gil-González, I., Martín-Rodríguez, A., Conrad, R., & Pérez-San-Gregorio, M. Á. (2023). Coping with multiple sclerosis: Reconciling significant aspects of health-related quality of life. Psychology, Health and Medicine, 28(5), 1167-1180. http://doi.org/ 10.1016/j.msard.2021.103093
69 Abstract Multiple sclerosis (MS) symptoms and unpredictability can damage patient well-being. This study is aimed to investigate the relation between sociodemographic and clinical characteristics and the use of coping strategies as well as social support on health-related quality of life (HRQOL). We evaluated 314 MS outpatients of Virgen Macarena University Hospital in Sevilla/Spain (mean age 45 years, 67.8% women) twice over an 18-months follow up period by Brief COPE Questionnaire (COPE-28), Multidimensional Scale of Perceived Social Support (MSPSS) and 12-Item Short Form Health Survey (SF-12). Female gender was significantly related to religion (r = 0.175, p < 0.001), self-distraction (r = 0.160, p < 0.001) and self-blame (r = 0.131, p < 0.05). Age correlated positively with religion (r = 0.240, p < 0.001), and self-blame (r = 0.123, p < 0.05). Progressive MS as well as functional impairment (EDSS) showed a positive relation with denial (r = 0.125, p < 0.05; r = 0.150, p < 0.001). Longer duration since diagnosis was related to lower perceived support from family (r = - 0.123, p < 0.05). EDSS (β = -0.452, p < 0.001) was the strongest negative predictor of physical HRQOL followed by age (β = -0.123, p < 0.001), whereas family support was a protective factor (β = 0.096, p < 0.001). Denial (β = -0.132, p < 0.05), self-blame (β = -0.156, p <0.05), female gender (β = -0.115, p < 0.05) and EDSS (β = -0.108, p < 0.05) negatively impacted on mental HRQOL 18 months later, whereas positive reframing (β = 0.142, p < 0.05) was a protective factor. Our study could identify sociodemographic and clinical variables associated with dysfunctional coping strategies, such as self-blame and denial, which specifically predict worse mental HRQOL as opposed to positive reframing. Diminishing dysfunctional coping and supporting cognitive reframing may contribute to improve HRQOL in MS. key words: multiple sclerosis; coping strategies; social support; health related quality of life; risk and protective factors. Introduction Coping strategies play an essential role in adaptation to multiple sclerosis (MS) and Carnero Contentti et al. (2021) point out a negative relationship between maladaptive coping and HRQOL. Carver (1997) divided coping strategies into three categories: problem-focused, emotion-focused, and dysfunctional coping. Generally, active coping, problem solving, planning, positive reframing, acceptance, emotional and instrumental social support were related to a higher HRQOL in MS. Whilst, avoidance, behavioural disengagement, selfdistraction, denial, emotion-focused, self-criticism and venting were associated with lower HRQOL (Gil-Gonzalez et al., 2020).
70 Particularly, Bassi et al. (2021) found a negative association between avoidance coping and physical HRQOL. In addition, Cerea et al. (2021) discovered a positive association between mental HRQOL and problem solving and a negative with emotional discharge and passive coping (Cerea et al., 2021; Krstić et al., 2021). The scientific literature revealed that MS patients use less active and more avoidance and emotional coping than the general population (Keramat Kar et al., 2019). In dealing with MS, intrapsychic and interpersonal mechanisms are closely intertwined. A study by Homayuni et al. (2021) found that MS patients described coping strategies and social support as HRQOL facilitators. In fact, social support has been related to improvements regarding fatigue (Mikula et al., 2020), pain (Alphonsus & D’Arcy, 2021), depression and anxiety (Hanna & Strober, 2020; Mikula et al., 2020; Ratajska et al., 2020), thereby also protecting employment (Iwanaga et al., 2018). Social support also influences patients’ attitudes on medication selection as they consider significant others’ opinions (Visser et al, 2020). In summary, there is evidence that directly and/or indirectly higher social support is related to better HRQOL (Bassi et al., 2021; Dȩbska et al., 2020; Gil-González et al., 2020; Kever et al., 2021; Ratajska et al., 2020), while lower social support is related to worse HRQOL (Costa et al., 2017; Strober, 2018). The present study aimed at investigating (1) sociodemographic and clinical factors underlying coping strategies and social support in adults with multiple sclerosis (MS), and (2) the role of coping strategies and social support as well as sociodemographic and clinical factors as predictors for quality of life in MS over an 18 months’ follow-up period. Methods Participants and procedures The sample was recruited between June 2017 and May 2018 (T1), and December 2018 and December 2019 (T2) at Virgen de la Macarena University Hospital in Sevilla/Spain. Inclusion criteria were: (1) confirmed MS diagnosis; (2) age over 18, and (3) mental, physical and cognitive capability to participate and sign informed consent. The study was approved by the responsible Ethics Committee (0846-N-18). Instruments Clinical and sociodemographic information were collected from the medical data base and a questionnaire.
71 Coping strategies The Spanish version of Brief COPE Questionnaire (COPE-28) was applied to study the patients use of different actions in dealing with stressful situations (Morán et al., 2010). COPE-28 has 28 items grouped into the following 14 dimensions: (1) acceptance; (2) emotional support; (3) humor; (4) positive reframing; (5) religion; (6) active coping; (7) instrumental support; (8) planning; (9) behavioral disengagement; (10) denial; (11) selfdistraction; (l2) self-blaming; (13) substance use; (14) venting. Items are scored on a fourpoint Likert scale (from 0 to 3). Higher scores indicate greater use (Carver, 1997). Cronbach’s alpha ranged from 0.60 to 0.88 for the 14 subscales. Social Support Participants perception of social support was measured by the Multidimensional Scale of Perceived Social Support (MSPSS). The MSPSS comprises 12-items scored on a 7-point Likert scale ranging from 1 to 7. The total score varies from 12 to 84 (Arechabala and Miranda, 2002; Zinnet, 1988). Cronbach’s alpha in our sample ranged from 0.91 to 0.96 for the subscales. Health related Quality of life The 12-Item Short Form Health Survey (SF-12) consists of 12 items scored on a 3 or 5-point Likert scale. The SF-12 consists of eight domains: physical functioning, role-physical, bodily pain, general health, vitality, social functioning, role-emotional and mental health. Subscales scores range from 0 (worst) to 100 (best). These subscales are combined to form the Physical Component Summary Score (PCS) and the Mental Component Summary Score (MCS) (Vilgaut et al., 2008; Ware et al., 2002). In our sample, dimensions Cronbach’s alpha ranged from 0.70 to 0.96 at T1 and from 0.64 to 0.96 at T2. Cronbach’s alpha for the PCS and MCS was 0.92 and 0.88, respectively (Maruish, 2012). Statistics Pearson and Spearman correlations are presented for coping strategies/social support and clinical/demographic variables. Stepwise regression analyses identify quality of life predictors and determine their relative contribution. Two multivariate models were built with MCS and PCS scores at T2 as dependent variables. Sociodemographic (gender, age, partnership, educational level and occupation), clinical variables (EDSS, MS subtype, months since diagnosis and months since the outbreak), coping strategies and social support at T1 were considered as predictors.
72 All tests were computed using SPSS-v26. Significance level was set to p < 0.05. Effect size coefficient were calculated using G*Power Software. Coefficients were interpreted according to Cohen (1988) guidelines; for correlations: p ≥ 0.10 small, ≥ 0.30 medium, and ≥ 0.50 large effect and in multiple regression: f2 ≥ 0.02 small, ≥ 0.15 medium, and ≥ 0.35 large effect. Results The final sample comprised 314 MS patients (dropout rate 19.69%; see figure 1). Figure 1. Flow chart As can be seen in Table 1, the sample was composed of 213 (67.8%) females and 101 (32.2%) males. Mean age was 45.31 years (± 10.77), range from 19 to 78 years. The predominant MS type was remittent 272 (86.6) and mean EDSS score was 3.17 (± 1.92). Table 1. Clinical and sociodemographic characteristics T1 Sample N = 391 T2 Sample N = 314 Gender n (%) Male 123 (31.5) 101 (32.2) Female 268 (68.5) 213 (67.8) Age (M ± SD) 45.66 ± 11.13 45.31 ± 10.77 Partnership n (%) No partner 108 (27.6) 85 (27.1) Partner 283 (72.4) 229 (72.9) Occupation n (%) Employed/In education 135 (34.5) 116 (36.9) Unemployed 256 (65.5) 198 (63.1) Educational level n (%) Primary education 65 (16.6) 44 (14) Secondary education 128 (32.7) 102 (32.5) University or higher 198 (50.6) 168 (53.5) EDSS (M ± SD) 3.38 ± 2.06 3.17 ± 1.92 MS subtype n (%)
73 Remittent 326 (83.4) 272 (86.6) Progressive 65 (16.6) 42 (13.4) Months since diagnosis (M ±S D) 145.31 ± 89.49 145.68 ± 89.56 Months since outbreak (M ± SD) 184.90 ± 108.47 186.11 ± 111.18 Sociodemographic/clinical variables and coping strategies Female gender correlated with higher use of self-distraction (r = 0.160, p < 0.001), religion (r = 0.175, p < 0.001), and self-blame (r = 0.131, p < 0.05). Age also correlated positively with religion (r = 0.240, p < 0.001), and self-blame (r = 0.123, p < 0.05). Higher educational level was related to a higher use of planning (r = 0.167, p < 0.001), seeking emotional support (r = 0.119, p < 0.05), and venting (r = 0.151, p < 0.001). Being unemployed was related to a lower use of venting (r = -0.121, p < 0.05) and higher use of denial (r = 0.133, p < 0.05) as well as religion (r = 0.112, p < 0.05). Progressive MS subtype showed a negative relation with venting (r = -0.134, p < 0.05) and a positive relation with denial (r = 0.125, p < 0.05). Months since diagnosis positively correlated with self-blame (r = 0.147, p < 0.001), as well as months since the outbreak (r = 0.143, p <0.05), which also correlated negatively with active coping (r = -0.115, p < 0.05). EDSS was related to a higher use of behavioral disengagement (r = 0.112, p < 0.05), denial (r = 0.150, p < 0.001), substance use (r = 0.124, p < 0.05), and humor (r = 0.120, p < 0.05). There were no significant correlations in regard to partnership status in the use of coping strategies. Effect sizes coefficients (p) of significant correlations ranged from 0.33 to 0.48, medium effects (Table 2). Sociodemographic/clinical variables and perceived social support Age (r = -0.130, p <0.05) and progressive MS subtype (r = -0.114, p < 0.05) were negatively related with social support from friends (see Table 3). Being without a partner showed a negative relation with social support from significant others (r = -0.128, p < 0.05). Higher educational level (r = -0.119, p < 0.05) and longer duration since diagnosis (r = -0.123, p < 0.05) was related to lower perceived support from family. With regard to gender, occupation, months since diagnosis outbreak and EDSS no significant associations were found with social support. For significant results correlation effect sizes (p) were medium (from 0.34 to 0.38).
80 (Freud, 1925). Denial in MS may impact on every aspect of health behaviour, e.g. medication adherence or regular physician appointments (Chandra et al., 2007). The second dysfunctional coping strategy negatively impacting mental HRQOL was self-blame, as found in previous studies (Koltuniuk et al., 2021). Self-blame often is closely associated with a feeling of helplessness and a lack of self-efficacy as well as depression (Zahn et al., 2015). Regarding sociodemographic predictors, female gender was a risk factor for mental HRQOL. Accordingly, Khader et al. (2019) showed that female MS patients suffer greater mental health impairments. As expected, EDSS was a predictor of poorer mental HRQOL, but its contribution to the mental HRQOL is lower than to the physical. Physical disabilities can impede social activities and endanger social relationships (Costa et al., 2017). Consequently, patients with higher level of disability are at higher risk to suffer from mental distress in the long run. In accordance with our previous research, positive reframing was identified as the only protective factor for mental HRQOL (Gil-González et al., 2020; Ukueberuwa and Arnett, 2019; Wilski et al., 2019). This finding has important clinical implications as positive reframing is also used as a fundamental psychological technique known as cognitive restructuring in cognitive-behavioural therapy to change the way a situation or experience is viewed. A previous study could show the positive impact of cognitive restructuring on pain symptoms in multiple sclerosis patients (Jensen et al., 2011). Clinical and practice implications Based on these findings, health care professionals should be sensitized to promote psychosocial interventions to facilitate MS adaptation and HRQOL at the onset of disease. Social skills training is recommended to improve communication and strengthen relationships with significant others. Cognitive behavioral therapy and acceptance and commitment therapy might be helpful to promote protective coping strategies such as positive reframing and acceptance, and prevent from avoidance coping strategies, such as denial and self-blame. Limitations and strengths The external validity of the study is limited due to the non-random selection of participants. Moreover, only self-report instruments were used. Nevertheless, our sample is very heterogeneous in terms of sample characteristics. As primary strengths the longitudinal design, the large sample size and the rather low dropout rate can be remarked.
81 Conclusions The present findings reveal the importance of patients clinical and demographic features when studying coping strategies and social support in MS. The identification of longitudinal risk and protective factors emphasize the necessity to integrate intrapsychic and interpersonal processes to provide a richer insight into the dynamics of HRQOL in MS and to identify potential therapeutic targets. Recommendations for future research Future research is required to further disentangle the complexity of these connections by analysing direct and indirect pathways in longitudinal studies. This could improve the efficacy of interventions through a more individualized therapeutic approach in MS. References Alphonsus, K. B., & D’Arcy, C. (2021). Is there an association between social support and pain among individuals living with multiple sclerosis? Journal of Evidence-Based Integrative Medicine, 26, 1-6. https://doi.org/10.1177/2515690X21991995 Amtmann, D., Bamer, A. M., Nery-Hurwit, M. B., Liljenquist, K. S., & Yorkston, K. (2019). Factors associated with disease self-efficacy in individuals aging with a disability. Psychology, Health and Medicine, 24(10), 1171-1181. https://doi.org/10.1080/13548506.2019.1612082 Arechabala Mantuliz, M. C., & Miranda Castillo, C. (2002). Validation of a scale of perceived social support in a group of elders under control in a hypertension program in the metropolitan region. Ciencia y Enfermeria, 8(1), 49-55. Assirelli, G., & Tosi, M. (2013). Education and family ties in Italy, France and Sweden. Journal of Educational and Social Research, 3(7), 379. Bassi, M., Grobberio, M., Negri, L., Cilia, S., Minacapelli, E., Niccolai, C., Pattini, M., Pietrolongo, E., Quartuccio, M. E., Viterbo, R. G., Allegri, B., Amato, M. P., Benin, M., De Luca G., Falautano, M., Gasperini, C., Patti, F., Trojano, M., & Delle Fave, A. (2021). The contribution of illness beliefs, coping strategies, and social support to perceived physical health and fatigue in multiple sclerosis. Journal of Clinical Psychology in Medical Settings, 28(1), 149-160. https://doi.org/10.1007/s10880-019-09692-6 Carnero Contentti, E., López, P. A., Alonso, R., Eizaguirre, B., Pettinicchi, J. P., Tizio, S., Tkachuk, V. & Caride, A. (2021). Coping strategies used by patients with relapsing
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86 4.4. Cuarto trabajo titulado “Beyond the boundaries of disease - Significant posttraumatic growth in multiple sclerosis patients and caregivers” Este trabajo corresponde al artículo publicado que se referencia a continuación: Gil-González, I., Pérez-San-Gregorio, M. Á., Conrad, R., & Martín-Rodríguez, A. (2022). Beyond the boundaries of disease - Significant post-traumatic growth in multiple sclerosis patients and caregivers. Frontiers in Psychology, 13, 903508. http://doi.org/ 10.3389/fpsyg.2022.903508
87 Abstract Despite the negative repercussions of a chronic disease, multiple sclerosis (MS) might also lead to positive consequences. This longitudinal study explored post-traumatic growth in MS patients and attempted to identify possible determinants. Post-traumatic growth of 260 patients and their caregivers was compared. A subset of 209 patients and caregivers were evaluated at baseline. Patients filled in the Posttraumatic Growth Inventory and General Health Questionnaire at three different times over a 36-month follow-up period. Patient posttraumatic growth significantly increased over the follow-up period (p < 0.001) with large effect sizes on almost every subscale. Higher score on the Expanded Disability Status Scale, higher pain severity, female gender, and higher anxiety were positive predictors of posttraumatic growth, while more interference of pain, higher level of education, and more social dysfunction were negative predictors. Post-traumatic growth did not differ significantly between patients and caregivers. Our results showed significant positive intrapsychic changes of MS patients over a 36-month follow-up period up to 12 years from diagnosis. The potential influence of clinical, demographic, and mental health variables underlines the need for a personalized approach to be able to understand and sustain these processes. Comparable posttraumatic growth levels in patient-caregiver dyads at baseline suggest interdependently driven cognitive processes stabilizing well-being. Future research is recommended for further insight into the underlying cognitive processes. Keywords: multiple sclerosis; post-traumatic growth; clinical variables; longitudinal; patients; caregivers. Introduction Multiple sclerosis (MS) is a chronic disease affecting physical, mental, and social well-being (Gil-González et al., 2020; Faraclas et al., 2022). While struggling with the unpredictability and variability of symptoms, MS patients may also undergo intrapsychic processes leading to positive mental and physical changes that impact on all areas of life. MS patients usually report greater appreciation of life, sense of liberation, taking better care of themselves and strengthening of close relationships (Baka et al., 2021; Kivi et al., 2019; Pakenham, 2005a, 2007). The term post-traumatic growth coined by Tedeschi & Calhoun (2004) is defined as “an experience of positive change that occurs as a result of the struggle with highly challenging life crises.” “Post-traumatic” highlights that growth is experienced after a critical life event, not just minor stressful episodes. “Growth” refers to inner development of capabilities and functioning due to modifications in mental and emotional awareness following adverse experience (Kivi et al., 2019). Post-traumatic growth must be differentiated
88 from other, perhaps overlapping, similar concepts. For instance, resilience is the ability to adapt to change and uncertainty after an adverse and stressful situation. This flexibility prevents people from experiencing the full blow of a critical life event, consequently experiencing less trauma, and thereby, limiting post-traumatic growth. The main difference between post-traumatic growth and resilience is that while resilience permits the person to survive and adapt to the adverse situation without significant difficulties, in post-traumatic growth, a stress-induced positive change leads the person to function better than before the traumatic experience. Thus, it has been demonstrated that MS patients with a higher level of resilience experience less post-traumatic growth (Younesi et al., 2020). Post-traumatic growth and psychological adjustment to MS are closely related, as both concepts imply regaining functionality and quality of life. However, post-traumatic growth means a level of functioning surpassing the level previous to the critical life event. Irvine et al. (2009) identified the following topics related to potentially traumatic events and highly stressful situations for MS patients: reaction to and impact of the diagnosis; limitations in social activities due to impairments; role in society and self-worth; changes in relationship dynamics and dependency, particularly when careers are involved; attitudes and reactions of others to MS patients who feel that unaffected people cannot understand it. Thus, the extent to which the diagnosis itself or its impact on different areas of life is a major traumatic experience is highly individual. It is evident that those mechanisms leading to significant post-traumatic growth have not been fully understood. We try to understand these mechanisms by looking at specific factors, which may contribute to this development. The identification of all relevant factors, their specific contribution as well as interdependence enable us to fully understand the underlying mechanisms of post-traumatic growth. Post-traumatic growth has been widely explored in various medical conditions, for example, acquired brain damage (Rogan et al., 2013), cardiac outpatient (Leung et al., 2010), myocardial infarction (Hosseini Golafshani et al., 2021; Rahimi et al., 2016), cancer (Heidarzadeh et al., 2014), and liver transplantation (Pérez-San-Gregorio et al., 2017a). There is evidence that most MS patients undergo some degree of post-traumatic growth (Aflakseir & Manafi, 2018; Younesi et al., 2020), even though the underlying mechanisms are poorly understood. Pakenham & Cox (2009) discovered a significant connection between duration of MS and personal growth, but only in patients who had been diagnosed over 15 years before. The majority of studies showed controversial associations between sociodemographic or clinical variables and post-traumatic growth (Ackroyd et al., 2011; Hart et al., 2008;
89 Heidarzadeh et al., 2014). Both younger and older age have been found to be positively related to post-traumatic growth (Heidarzadeh et al., 2014; Henson et al., 2021; Leung et al., 2010). Analysis of level of education has also shown inconsistent results. Some studies have found a positive correlation between post-traumatic growth and education, probably because it provides access to more resources for such development. However, others have found an association with a lower level of education, where intense search for social support is a key factor in post-traumatic growth (Henson et al., 2021). In mental health, post-traumatic growth has shown a positive relationship with anxiety and negative with depressive symptoms (Barskova & Oesterreich, 2009). It might be argued that in depression, the ability for inner growth is hampered by a lack of energy and inner drive. Therefore, some psychological treatments have increased post-traumatic growth by reducing demoralization and depressive symptoms (Kivi et al., 2019) as well as amplifying positive affects (Hart et al., 2008). Other studies have not found any significant association between mental health and post-traumatic growth in MS (Ackroyd et al., 2011). As MS is a degenerative and uncurable neurological disorder with onset most often in young adulthood, it has a strong impact on patients’ families and relatives, particularly on primary caregivers. Many studies have focused on caregiver distress in coming to terms with the repercussions of the disease (Bassi et al., 2016; Pakenham et al., 2012; Tramonti et al., 2019). However, there is also qualitative and quantitative research indicating that care of MS patients may also lead to positive experiences (Rajachandrakumar & Finlayson, 2021). In adapting to their new role, caregivers might become aware of personal strengths and family resources, or make healthy modifications in their lifestyle (Maguire & Maguire, 2020). In this context the importance of the patient-caregiver dyad, especially coping strategies, has been emphasized. Dyads using avoidance coping have shown less growth than those dealing with MS in an active and mutually supportive manner (Wawrziczny et al., 2021). Couples in which one partner is an MS patient who attended therapeutic programs where they had the opportunity to share and reevaluate their experience, showed greater post-traumatic growth (Neate et al., 2019). Thus, benefits in patients and caregivers are strongly related. Nonetheless, care recipients typically report higher levels of post-traumatic growth. It has been suggested that the direct personal experience of a traumatic event has a particularly high psychological impact challenging one’s whole personality. This might lead to new self-definition and deeper meaning (Ackroyd et al., 2011; Pakenham, 2005b; Treder-Rochna, 2020b).
96 Table 3. T1 Post-traumatic growth (PGI-21) Multiple linear regression model. Dependent variable T1 post-traumatic growth (PGI-21) F (14,245) R2 B p SE.B β f2 Model 1 1.956, p = 0.032 0.096 67.413 11.01 0.106 Gender 3.134 0.330 3.213 0.062 Age -0.319 0.069 0.174 -0.143 Educational Level -3.881 0.068 2.115 -0.118 EDSS level 1.858 0.042 0.911 0.152 MS type 0.018 0.997 5.093 0.000 DMD type -1.042 0.439 1.343 -0.051 Months since diagnosis 0.013 0.662 0.029 0.048 Months since outbreak -0.005 0.856 0.025 -0.021 Pain severity 0.914 0.387 1.056 0.101 Pain interference -0.919 0.319 0.920 -0.122 Somatic symptoms 0.107 0.823 0.477 0.021 Anxiety and insomnia -0.925 0.028 0.419 -0.209 Social dysfunction -1.370 0.010 0.530 -0.214 Severe depression -0.529 0.226 0.436 -0.102 Dependent variable T2 post-traumatic growth (PGI-21) F (14,245) R2 B p SE.B β f2 Model 2 2.693, p = 0.001 0.133 74.745 11.259 0.153 Gender 6.849 0.022 2.971 0.146 Age -0.217 0.166 0.156 -0.106 Educational Level -1.197 0.530 1.902 -0.629 EDSS level 2.007 0.025 0.888 0.018 MS type -1.707 0.716 4.687 -0.028 DMD type -0.486 0.699 1.254 -0.025 Months since diagnosis -0.007 0.789 0.026 -0.268 Months since outbreak -0.002 0.934 0.022 -0.009 Pain severity -0.516 0.608 1.005 -0.65 Pain interference 1.190 0.253 1.034 0.148 Somatic symptoms 0.022 0.960 0.447 0.005 Anxiety and insomnia 0.424 0.253 0.370 0.100 Social dysfunction -2.085 <0.000 1 0.515 -0.367 Severe depression -0.386 0.378 0.437 -0.065 Dependent variable T2 post-traumatic growth (PGI-21) F (14,245) R2 B p SE.B β f2 Model 3 2.054, p = 0.015 0.054 85.295 10.387 0.057 Gender 4.578 0.085 2.649 0.109 Age -0.107 0.441 0.139 -0.058 Educational Level -4.640 0.007 1.172 -0.071 EDSS level 1.309 0.096 0.783 0.135 MS type -4.578 0.240 3.886 -0.087
97 EDSS, Expanded Disability Status Scale; MS, multiple sclerosis; DMD, Disease modifying Drug. S, small effect size. Model 2 [F (14,245) = 2.693, R2 = 0.133, p = 0.001] showed that female gender (β = 0.146, p = 0.022), and high EDSS (β = 0.018, p = 0.025) predicted PGI-21 total score at T2 positively and social dysfunction (β = -0.367, p < 0.001) predicted it negatively. The variables in Model 2 accounted for 13.3% of total PGI-21 score variance at T2 with a medium effect size (f 2 = 0.153). In Model 3 [F (14,245) = 2.054, R2 = 0.054, p = 0.015] education (β = -0.071, p = 0.007) and pain interference (β = -0.271, p = 0.030) were the negative predictors of PGI-21 total score at T3 and pain severity (β = 0.330, p = 0.005) was a positive predictor. Model 3 variables explained 5.4% of PGI-21 total score variance at T3 with a small effect size (f 2 = 0.054). The comparison of patient and caregiver PTG1-21 subscale scores at T1 did not show statistically significant differences: Relating to others (t = 0.271, p = 0.79), New Possibilities (t = -0.925, p = 0.356), Personal Strength (t = -1.028, p = 0.305), Spiritual Change (t = 0.378, p = 0.378), Appreciation of Life (t = -0.884, p = 0.706) and Total Post-Traumatic Growth (t = -0.450, p = 0.653). Effect sizes were null (d from 0.015 to 0.081). Mean scores, standard deviation and paired t-test results are reported in Table 4 and Figure 3. Table 4. Post-traumatic growth (PGI-21) comparison between MS patients and family members (paired t-test). Patients Family members M ± SD M ± SD t (p) Cohens´d Relating to others 2.25 ± 1.31 2.22 ± 1.28 0.271 (0.79) 0.017 (N) New Possibilities 2.04 ± 1.28 2.14 ± 1.21 -0.925 (0.356) 0.080 (N) Personal Strength 2.61 ± 1.34 2.72 ± 1.38 -1.028 (0.305) 0.082 (N) Spiritual Change 1.31 ± 1.47 1.39 ± 1.50 0.378 (0.378) 0.054 (N) Appreciation of life 2.89 ± 1.47 2.84 ± 1.38 -0.884 (0.706) 0.028 (N) Total post-traumatic growth 47.68 ± 23.04 48.51 ± 23.38 -0.450 (0.653) 0.036 (N) N, null effect size. DMD type -0.819 0.488 1.118 -0.045 Months since diagnosis -0.015 0.434 0.020 -0.71 Months since outbreak 0.018 0.208 0.014 0.114 Pain severity 2.412 0.005 0.861 0.330 Pain interference -2.031 0.030 0.931 -0.271 Somatic symptoms -0.717 0.184 0.516 -0.132 Anxiety and insomnia 0.386 0.568 0.290 0.100 Social dysfunction 0.316 0.568 0.551 0.056 Severe depression -0.331 0.390 0.384 -0.070
98 Figure 3. Mean score (standard deviation) of patient and caregiver post-traumatic growth. At T3 patients affected by COVID-19 (n = 123) compared to patients unaffected by COVID-19 (n = 137) did show significantly higher scores on the following scales: Relating to Others (t = 2.020, p = 0.044), New Possibilities (t = 2.868, p = 0.004), Spiritual Change (t = 3.699, p < 0.001), Appreciation of Life (t = 3.010, p = 0.002) and total Post-traumatic Growth (t = 3.188, p = 0.002). Effect sizes were small (d from 0.207 to 0.458). No significant differences were found between the COVID-19-affected group and the total sample (d from 0.110 to 0.303, null and small effect sizes), except for Spiritual Change (t = 2.131, p = 0.034, d = 0.303, small effect size). Results were similar when the unaffected group was compared with the total sample (d from 0.097 to 0.220, null and small effect sizes), the only significant difference was for Spiritual Change (t = 2.066, p = 0.039, d = 0.220, small effect size; Table 5). Table 5. COVID impact at T3, mean score and comparisons. Mean score ± SD Comparisons p (Cohens d) A. Covid affected (n = 123) B. No Covid affected (n = 137) C. Total sample (n = 260) A-B A-C B-C Relating to others 4.05 ± 0.92 3.81 ± 0.99 3.92 ± 0.97 0.044 (0.251) 0.197 (0.137) 0.197 (0.112) New possibilities 3.97 ± 1.08 3.59 ± 1.05 3.77 ± 1.08 0.004 (0.357) 0.080 (0.182) 0.080 (0.169) Personal strength 3.98 ± 1.12 3.76 ± 1.00 3.86 ±1.06 0.086 (0.207) 0.293 (0.110) 0.363 (0.097) Spiritual change 2.28 ± 1.95 1.42 ± 1.80 1.83 ± 1.92 < 0.0001 (0.458) 0.034 (0.303) 0.039 (0.220) Appreciation of life 4.09 ± 1.08 3.63 ± 1.38 3.85 ± 1.24 0.002 (0.371) 0.066 (0.206) 0.107 (0.160) Total PTG 80.95 ± 18.85 73.37 ± 19.42 76.95 ± 19.49 0.002 (0.396) 0.058 (0.208) 0.082 (0.184) S, small effect size; N, Null effect size.
99 Discussion In recent decades, there has been growing interest in positive changes derived from diagnosis and adaptation to MS. The first objective of this study was to analyze post-traumatic growth and its development in a large sample of MS patients. Over the course of 36 months, there were significant increases in the total score and all the subscales except for spiritual change with large effect sizes. This confirms our first hypothesis. Less change in the Spiritual change subscale might be explained partly by methodological limitations of the questionnaire, as it is the only subscale consisting of just two items, and this limits its sensitivity to change. In addition, spiritual processes in particular might take longer to develop, exceeding the follow up period of 36 months. This is corroborated by the fact that even though the total increase on the spiritual subscale is less than on the others, the increase between T2 and T3 is more than threefold higher than the increase from T1 to T2. Spiritual Change is the most complex concept in the framework of post-traumatic growth, as it involves the definition of oneself in relation to the universe, which involves self, significant others, society, and any powers which may transcend and influence these concepts. In the two relevant items, the post-traumatic growth inventory specifically asks about “better understanding of spiritual matters” and “stronger religious faith.” A new orientation in this framework is highly challenging and requires time. Against this backdrop it is understandable that spirituality is usually stronger in older people (Malone & Dadswell, 2018). When comparing post-traumatic growth levels in the current study with previous observations in MS patients, it is important to consider the assessment time. Compared to previously reported levels, total post-traumatic scores were lower at T1 and T2, but higher at T3 (Aflakseir & Manafi, 2018; Kivi et al., 2019). At T2, total scores were higher than in patients with acquired brain damage (Rogan et al., 2013) or cardiac outpatients (Leung et al., 2010), at T3 higher than in cancer and myocardial infarction patients (Ackroyd et al., 2011; Barskova and Oesterreich, 2009). These findings are in keeping with previous results showing a higher level of post-traumatic growth in MS than other chronic diseases (Aflakseir and Manafi, 2018). Obviously, study design and the duration of follow up strongly influence the degree of post-traumatic growth reported. Regarding the specific pattern of post-traumatic growth in our study, the Appreciation of life and Personal strength dimensions were highest at T1. Contrary to this pattern, related research in MS (Kivi et al., 2019), cancer (Heidarzadeh et al., 2014), and myocardial infarction (Rahimi et al., 2016) has reported Spiritual change and Relating to others to be the most prominent post-traumatic growth dimensions. These
100 differences might be explained by specific sociocultural circumstances of the patients in the samples favoring more individualistic approaches, such as in Western industrial societies or particularistic approaches as in Eastern societies. In the conceptual foundation of post-traumatic growth by Tedeschi & Calhoun (2004), the successful management of distressing emotions is a prerequisite for cognitive processing and schema changes that might lead to post-traumatic growth. In this line of reasoning, persistent distress could be essential to developing the maximum level of post-traumatic growth. In MS patients, the need to deal with the variability of MS symptoms, chronicity, and unpredictable prognosis causes persistent distress. This continual coping could fuel cognitive processing and contribute to post-traumatic growth. This study also analyzed possible factors influencing post-traumatic growth. Clinical variables, EDSS, pain interference and severity were predictors of post-traumatic growth at T2 and T3, respectively. Even though the vast majority of studies found no significant linear associations between disease severity and post-traumatic growth in either MS or other chronic conditions like acquired brain damage (Rogan et al., 2013) or cancer (Heidarzadeh et al., 2014), there is evidence that the subjectively felt magnitude of the trauma is an important variable, and in serious medical conditions, level of disability can determine its impact. Our result is in agreement with the study by Leung et al. (2010) relating post-traumatic growth to functional status in cardiac outpatients. Our study is the first to show a significant relationship between pain and post-traumatic growth in MS. Previous studies did not reveal these significant associations (Barskova & Oesterreich, 2009). We found pain severity to predict more post-traumatic growth, whereas interference predicted less. How can this seemingly contradictory finding be explained? Pain intensity mirrors the subjective perception of this negative sensation, which may induce coping strategies to manage or mitigate it. The deep psychological impact of a traumatic event is often described by affected individuals as painful, and therefore, pain intensity could even be considered a reminder of the traumatic stressor, and the ongoing traumatic pain fuels the process of adaptation and growth. As opposed to pain intensity, pain interference means the degree to which an individual is unable to manage the pain, possibly feeling overwhelmed and helpless. This reflects inability to cope with pain or actively deal with it and use it for personal growth. As neither time since diagnosis nor onset predicted post-traumatic growth, our second hypothesis could not be confirmed. This association was only found in one previous study of a sample of MS patients who had been diagnosed over 15 years before. In most other studies, no clinical or sociodemographic predictors of post-traumatic growth could be identified (Ackroyd et al., 2011; Hart et al., 2008;
101 Pakenham, 2005b; Pakenham & Cox, 2009; Prati & Pietrantoni, 2009). Interestingly, significant prediction of post-traumatic growth by time since diagnosis or transplantation could not be confirmed in either cancer (Heidarzadeh et al., 2014) or liver transplant patients (Pérez- San-Gregorio et al., 2017b). In other words, in none of these empirical studies did the exact time since diagnosis predict the inner process of post-traumatic growth. This might indicate that the psychological process is driven by highly complex cognitions and does not necessarily develop in a continuously ongoing stream, but rather by long unforeseeable phases of rumination and sudden life-changing insights. Therefore, these idiosyncratic processes reflect a highly individual inner time flow. Tedeschi & Calhoun (2004) themselves emphasized the nonlinearity of post-traumatic growth and proposed, for example, a complex association curve between trauma strength and post-traumatic growth. Accordingly, the underlying mechanisms of post-traumatic growth are closely related to cognitive factors, which lead to the restructuring of personal narratives. In this context the presence of deliberate versus intrusive cognitions as well as frequency, timing, valence and content of cognitions need to be taken into account (Tedeschi & Calhoun, 2004). By demographic factors, female gender and lower education were predictors of higher post-traumatic growth at T2. Previous studies have consistently shown a close association between female gender and post-traumatic growth (Henson et al., 2021). In this context, the tendency of women to deliberately ruminate on constructive issues, such as personal strengths or the importance of social connections, has been suggested as a relevant mechanism (Vishnevsky et al., 2010). This cognitive process is facilitated by the stronger tendency of women to share their experiences with others (Henson et al., 2021). This finding is supported by previous research in cancer and HIV survivors (Barskova & Oesterreich, 2009). Moreover, lower education was a positive predictor of post-traumatic growth. As mentioned above, there are several studies demonstrating that individuals with less education tend to rely more on others and use the resources of social relationships, which may be a crucial resource in fostering growth from adversity (Heidarzadeh et al., 2014; Henson et al., 2021). Of the mental health variables, greater social dysfunction predicted lower levels of posttraumatic growth at T1 and T2, and greater anxiety and insomnia higher post-traumatic growth at T1. It has consistently been shown that sharing emotions as well as disclosure of the traumatic event in a safe social environment is helpful for post-traumatic growth development. Additionally, talking to others indirectly promotes post-traumatic growth by reducing depressive symptoms (Henson et al., 2021; Leung et al., 2010; Prati & Pietrantoni, 2009). On
102 the contrary, difficulties in relating to others and sharing, as in social dysfunction, can hamper progress of post-traumatic growth. Anxiety and insomnia positively predicted post-traumatic growth at T1. Anxiety, as opposed to depression, is associated with more of a sympathetic tone, which may induce active coping mechanisms to handle the stressor and reduce the stress reaction. A similar association between post-traumatic growth and anxiety has previously been reported in MS patients as well as in acquired brain injury survivors (Barskova & Oesterreich, 2009). In an attempt to find external/circumstantial variables that could explain post-traumatic growth in MS, we studied the differences between patients who had experienced that the COVID pandemic had impacted on their lives and those who had not. The COVID-19- affected group showed higher post-traumatic growth levels than the unaffected group in the final evaluation (T3). The onset of the pandemic might be a further significant life event, experienced as a significant stressor and adding to the impact of highly personal life events like MS, thereby increasing post-traumatic growth. However, as the pandemic began near the end of the study, its impact on overall changes in post-traumatic growth should not be overestimated. Apparently, objective clinical parameters, such as EDSS, MS type, disease duration, and symptomatology do not directly influence post-traumatic growth, but rather, their subjectively felt impact and intrapsychic coping strategies in dealing with them. Active coping, such as positive reframing, acceptance, and seeking emotional and social support could lead to higher post-traumatic growth than passive strategies such as avoidance and denial. Therefore, further research on post-traumatic growth in MS coping strategies should be considered essential. Based on our findings the underlying mechanisms in post-traumatic growth involve the ongoing intrapsychic impact of a traumatic event, which manifests in pain, anxiety, or disability, which, however, does not substantially hamper social interactions but rather strengthens social relationships. Therefore, social dysfunction, pain interference and male gender are factors contributing to significantly less growth. Our third hypothesis could not be confirmed, as there were no significant differences at T1 between patient and caregiver total post-traumatic scores or subscales. In previous studies where time since diagnosis was comparable to our study (about 10 years vs. 12 years in our study) significantly higher post-traumatic growth was reported in MS patients than in caregivers (Ackroyd et al., 2011; Pakenham, 2005b). A similar outcome was found after liver transplantation (Pérez-San-Gregorio et al., 2017b).
103 A possible explanation for these differences could be that in our sample disability was lower with an EDSS score of 3.7, indicating that patients were still able to walk without issues, compared to 5.1 in the study by Ackroyd et al. (2011), meaning the disability was severe enough to impair full daily activities and ability to work a full day without special provisions and patients were only able to walk without aid or rest for 200 m. The other study (Pakenham, 2005b) did not provide an EDSS score. Nevertheless, our study corroborated empirical findings insofar as MS led to posttraumatic growth not only in patients but also in caregivers. Recent research has highlighted the importance of open communication about personal experiences, creating a “communal sense of the illness” which fosters growth and personal gain in the MS patient-caregiver dyad (Neate et al., 2018, 2019; Treder-Rochna, 2020). By means of empathy and charitable understanding, caregivers delve into the patient’s inner world, fueling cognitive changes in their own. This might be easier when the caregiver is the partner, which was the case in 65% of our sample. The fruitful connection of otherwise distinct inner worlds may be a good example of “shared pain is of half the pain.” Future research could find out whether patientcaregiver post-traumatic growth patterns still match over a 36-month longitudinal study. Implication for clinical practices Our study confirmed significant post-traumatic growth in MS even an average 12 years from diagnosis even without any psychotherapeutic help to support or enhance this development. Nevertheless, as stronger pain interference and social dysfunction, as well as higher education and male gender were associated with less growth, it would be important to optimize posttraumatic growth in patients with these characteristics. In clinical practice, cognitive behavioral therapy has been shown to support post-traumatic growth (Knaevelsrud et al., 2010). Cognitive behavioral therapy is the most empirically validated form of psychotherapy and has undergone remarkable development categorized in three phases or waves. The evolution started with behavioral therapy, in the second wave, cognitive influences on behavior were addressed in cognitive therapy, and in the third wave, acceptance-based therapies focused on mindfulness and compassion. Acceptance and Commitment Therapy (ACT), Dialectic Behavioral Therapy (DBT) and Mindfulness-Based Stress Reduction (MBSR) as well as mindfulness-based cognitive therapy (MBCT) belong to the so-called third wave. The essence of Acceptance and Commitment Therapy (ACT) is that it helps the individual find important values and goals for directing behavioral change. Dialectic Behavioral Therapy (CBT) aims at emotion regulation, distress tolerance, self-acceptance,
104 and validation. It can be helpful for patients whose personality structure is less well-integrated and for comorbid personality disorder. Finally, Mindfulness-Based Stress Reduction (MBSR), and Mindfulness-Based Cognitive Therapy (MBCT), including components of CBT, focus on awareness of the present moment and acceptance (Zarotti et al., 2022). All these therapies help patients to stay focused on the present moment and accept thoughts and feelings without judgment, which might be particularly helpful in patients suffering from pain interference or social dysfunction, as by accepting their situation, patients can acquire more control and feel less helpless, similar to what has been demonstrated for similarly traumatic diseases (Eccles et al., 2021). Acceptance and creating meaning from a stressful situation, reframing the life-impacting event, validating the personal experience, appreciating life and redefining life goals are psychological mechanisms involved in an MS patients’ posttraumatic growth that can be supported by third-wave CBT intervention. Limitations and strength The main weakness of the study was non-random sampling at a single tertiary care center, which limits its external validity. In addition, the use of self-report questionnaires increases the risk of bias due to social desirability. Evaluation of mental health with a semi-structured interview, for instance, could be more reliable. Nonetheless, the large sample size and low dropout rate, its heterogeneity and longitudinal design over 36 months are major strengths of this trial. Conclusions Our results suggest significant positive consequences for MS patients over a 36-month followup, even 12 years from diagnosis. Higher EDSS, more severe pain, less pain interference, female gender, lower education, higher anxiety, and lower social dysfunction showed positive linear associations with post-traumatic growth. These results highlight the importance of a thorough assessment of a wide spectrum of sociodemographic and clinical variables leading to further knowledge of the underlying mechanisms of post-traumatic growth in MS. Comparable levels of post-traumatic growth in MS patients and caregivers provide insight into possibilities for personal growth driven by care and empathy, which can be seen as an inspiring and hopeful message. References Ackroyd, K., Fortune, D. G., Price, S., Howell, S., Sharrack, B., & Isaac, C. L. (2011). Adversarial growth in patients with multiple sclerosis and their partners: relationships with
105 illness perceptions, disability, and distress. Journal of Clinical Psychology in Medical Settings, 18, 372-379. https://doi.org/10.1007/s10880-011-9265-0 Aflakseir, A., & Manafi, F. (2018). Posttraumatic growth and its relationship with cognitive emotion regulation strategies in patients with multiple sclerosis in shiraz, Iran. Practice in Clinical Psychology, 6, 57-62. https://doi.org/10.29252/ nirp.jpcp.6.1.57 Baka, C., Chatira, K., Karademas, E. C., & Kafetsios, K. G. (2021). Patients’ perspective on the psychological impact of multiple sclerosis on their life. Illness, Crisis & Loss, 1-15. https://doi.org/10.1177/10541373211006883 Barskova, T., & Oesterreich, R. (2009). Post-traumatic growth in people living with a serious medical condition and its relations to physical and mental health: A systematic review. Disability and Rehabilitation, 31, 1709-1733. https://doi.org/10.1080/ 09638280902738441 Bassi, M., Falautano, M., Cilia, S., Goretti, B., Grobberio, M., Pattini, M., Pietrolongo, E., Viterbo, R.G., Amato, M.P., Benin, M., Lugaresi, A., Minacapelli, E., Montanari, E., Patti, F., Trojano, M. & Delle Fave, A (2016). Illness perception and well-being among persons with multiple sclerosis and their caregivers. Journal of Clinical Psychology in Medical Settings, 23, 33-52. https://doi.org/10.1007/s10880-015-9425-8 Cohen, J. (1988). Statistical power analysis for the behavioral sciences. Routledge Academic. Eccles, F. J. R., Craufurd, D., Smith, A., Davies, R., Glenny, K., Homberger, M., Rose, L., Theed, R., Peeren, S., Rogers, D., Skitt, Z., Zarotti, N. & Simpson, J. (2021). Experiences of mindfulness-based cognitive therapy for premanifest huntington's disease. Journal of Huntington's Disease, 10(2), 277-291. https://doi.org/10.3233/ JHD-210471 Faraclas, E., Lynn, J., Lau, J. D., & Merlo, A. (2022). Health-related quality of life in people with multiple sclerosis: how does this population compare to population-based norms in different health domains? Journal of Patient-Reported Outcomes, 6, 12. https://doi.org/10.1186/s41687-022-00415-4 Gil-González, I., Martín-Rodríguez, A., Conrad, R., & Pérez- San-Gregorio, M. A. (2020). Quality of life in adults with multiple sclerosis: a systematic review. BMJ Open, 10, e041249. https://doi.org/10.1136/bmjopen-2020- 041249 Goldberg, D., Gater, R., Sartorius, N., Ustun, T., Piccinelli, M., Gureje, O., Gureje, O., & Rutter, C. (1997). The validity of two versions of the GHQ in the WHO study of mental illness in general health care. Psychological Medicine, 27(1), 191-197 https://doi.org/10.1017/S0033291796004242
112 promoting personal growth among people with physical disability (Kim et al., 2020). The following coping strategies were identified as contributing factors of PTG in specific health conditions: problem focused active coping, which involves direct actions to deal with the stressor, in cervical dystonia (O’Connor et al., 2022); acceptance and putting into perspective in hemodialysis (Arjeini et al., 2020); emotional engaging in hematopoietic cell transplantation (Schwartz et al., 2022); instrumental and emotional social support in liver transplant recipients (Pérez-San-Gregorio et al., 2017) and hematopoietic cell transplant recipients (Schwartz et al., 2022). In people with acquired brain damage self-distraction and humor were associated with higher PTG in addition to active coping, emotional and instrumental support, positive reframing, planning, acceptance, and use of religion/spirituality (Rogan et al., 2013). With regard to MS there is growing evidence for the relevance of coping and PTG for wellbeing and mental health. Thus, in a recent study using semi-structured interviews reevaluation of life has been described as a beneficial consequence of MS experience (Baka et al., 2022). In quantitative studies, reappraisal and positive reframing were found to predict PTG (Aflakseir & Manafi, 2018) and personal benefit finding (Pakenham, 2005). However, there is a lack of studies exploring these associations over a longer period of time. Longitudinal data indicate an increasing progression of PTG in MS and corroborate the influence of clinical, demographic, and mental health variables, with small to medium effect sizes (Gil-González et al., 2022). When studying personal benefit finding an improvement tendency over time was also observed and suggests that substantial improvement of personal growth appears later in MS adaptation (Aflakseir & Manafi, 2018). An ideographic study exploring people with MS disease experience highlights patients expression of gratitude for family support enhancement, religious faith promotion and making a new identity meaning in a long run (Kassie et al., 2021). Against this backdrop the present study aims to investigate the association between posttraumatic growth and coping strategies over the course of 36 months with special emphasis on strategies positively predicting PTG. Method Participants and procedure Outpatients of Virgen Macarena University Hospital were asked to participate in three different periods: June 2017-May 2018 (T1), December 2018-December 2019 (T2), and May 2020-April 2021 (T3).
113 Eligibility criteria included: a) confirmed MS diagnosis according to McDonald criteria verified by their primary care neurologist; b) aged over 18; and c) mental, physical, and cognitive capability to fulfill the assessment protocol. Figure 1 represents the sample selection process. The research was authorized by the responsible Ethics Committee (0846-N-18). All participants were provided with oral and written instructions and gave their in-formed consent to take part in the study. Instruments Participants completed a standardized form on sociodemographic features. We collected relevant clinical information from a medical data base. Post-traumatic growth The Spanish version of the Post-traumatic Growth Inventory (PGI-21) assesses patient perception of personal benefit after their experience with MS (Tedeschi & Calhoun, 1996; Weiss & Berger, 2006). PGI-21 consists of 21 items, scored on a Likert-type scale from 0 (“no change”) to 5 (“very great degree of change”). Items are grouped into the following five dimensions: relating to others, new possibilities, personal strength, spiritual change and appreciation of life. In the present sample, Cronbach´s alpha was 0.92, 0.91, and 0.93 for the total score scale and 0.74-0.80, 0.77-0.83, and 0.72-0.88 for the five subscales dimensions, at T1, T2 and T3 respectively.
114 Figure 1. Study flow chart. CIS, clinically isolated syndrome. Coping strategies Brief COPE Questionnaire (COPE-28) evaluates different actions in dealing with stressful situations (Carver, 1997). COPE-28 contains 28 items scored on a Likert scale from 0 (“I have not been doing this at all”) to 3 (“I have been doing this a lot”). Test results pro-vide information about 14 subscales: (1) acceptance; (2) emotional support; (3) humor; (4) positive reframing; (5) religion; (6) active coping; (7) instrumental support; (8) planning; (9) behavioral disengagement; (10) denial; (11) self-distraction; (12) self-blaming; (13) substance use; and (14) venting. The Spanish version was applied to our sample (Morán et al., 2010). Cronbach’s alpha ranged from 0.61 to 0.84 at T1; 0.70 to 0.96 at T2; and 0.63 to 0.95 at T3, for the above mentioned subscales. Expanded Disability Status Scale (EDSS) Expanded Disability Status Scale (EDSS) is the predominantly used scale to measure disability in MS. Its efficacy and reliability have been proven at every disease stage. EDSS scores range from 0, indicating regular neurological functioning, to 10. The scales assess different Functional Systems (FS): Visual Functions, Brainstem Functions, Pyramidal Functions, Cerebellar functions, Sensory Functions, Bowel and Bladder Functions, Cerebral Functions, and Ambulation. A 5-points EDSS score means no ambulatory problem. In EDSS scores over 5-points, the ambulation status is the main factor determining the degree of disability (Haber & LaRocca, 1985; Kurtzke, 1983; Şen, 2018).
115 Statistical Analysis Descriptive analyses were calculated to report sample characteristics. One way analysis of variance (ANOVA) was used for quantitative variables (age, EDSS, months since diagnosis and months since the outbreak) and Chi-squared test for qualitative variables (gender, partnership, occupation, educational level and MS subtype) to detect differences in sociodemographic and clinical variables between three subgroups defined by PTG level (low, medium, and high). A 3x3 mixed factorial ANOVA with Bonferroni post-hoc tests was calculated to study the influence of initial post-traumatic growth level (low: ≥ 36, medium: 37-60 and high: 61- 97) on the use of coping strategies in follow up assessments (T1, T2 and T3). To identify potential biopsychosocial predictors of post-traumatic growth level, three different stepwise multivariate regression models were calculated. Total post-traumatic growth level at T1, T2, and T3 were considered dependent variables. Coping strategies at T1, T2, and T3 were introduced as predictors. Statistics were conducted using SPSS v26. Significance level was set at p < 0.05. Effect size coefficients were calculated using G*Power Software and interpreted according to Cohen´s (1988) recommendations; for w (0.10 = small, 0.30 = medium, and 0.50 = large), for f (0.10 = small, 0.25 = medium, and 0.40 = large), f2 (0.02 = small, 0.15 = medium, and 0.35 = large effects) and d (0.20 = small, 0.50 = medium, and 0.80 = large effects) (Cohen, 1988). Results The final sample was composed of 260 adults with MS. The sample comprised 179 (68.83%) women and 81 (31.27%) men. Mean age was 45 (SD = 10.60). Mean EDSS at T1 was 3.20 (SD = 1.93) and the most reported MS type was relapsing remittent. Participants demographic and clinical characteristics are reported in Table 1.
116 Table 1. Comparison of sociodemographic and clinical characteristic of the three different posttraumatic growth level groups at T1. Posttraumatic growth level Intergroup comparison Effect size Low (n = 85) Medium (n = 84) High (n = 91) χ2 p Cohen´s w Gender n (%) 0.222 0.895 0.029 (N) Male 28 (32.9) 26 (31) 27 (29.7) Female 57 (67.1) 58 (69) 64 (70.3) Partnership n (%) 1.871 0.392 0.084 (N) No partner 59 (69.4) 66 (78.6) 68 (74.7) Partner 26 (30.6) 18 (21.4) 23 (25.3) Occupation n (%) 1.247 0.536 0.068 (N) Employed/In education 34 (40) 27 (32.1) 31 (34.1) Unemployed 51 (60) 57 (67.9) 60 (65.9) Educational level n (%) 8.039 0.090 0.175 (S) Primary education 8 (9.4) 10 (11.9) 17 (18.7) Secondary education 26 (30.6) 34 (40.5) 22 (24.2) University or higher 51 (60) 40 (47.6) 52 (57.1) MS subtype n (%) 0.541 0.763 0.046 (N) Remittent 76 (89.4) 72 (85.7) 80 (87.9) Progressive 9 (10.6) 12 (14.3) 11 (12.1) F (2,257) p Cohen´s d Age (M ± SD) 45.7 ± 11.1 45.4 ± 10.9 44.1±9.9 0.581 0.560 0.015 (N) EDSS (M ± SD) 3.1 ± 1.8 3.1 ± 1.9 3.4 ± 2.1 0.704 0.495 0.040 (N) Months since diagnosis (M ± SD) 140.3 ± 98.4 147.6 ± 92.3 146.2 ± 77.8 0.159 0.853 0.021 (N) Months since outbreak (M ± SD) 181.4 ± 112.9 190.8 ± 112.4 184.4 ± 108.4 0.084 0.919 0.020 (N) L, large effect size; M, medium effect size; S, small effect size; N, null. Post-traumatic growth levels and assessment time on coping strategies The influence of initial post-traumatic growth level on the use of coping strategies was studied. The total sample was divided into three groups according to their PGI-21 total score at T1: 85 patients with low post-traumatic growth (32.7%; 0-36 points), 84 with medium posttraumatic growth (32.3%; 37-60 points) and 91 with high post-traumatic growth (35%; 61-97 points). One way ANOVA and Chi square test analysis did not find any significant difference in sociodemographic and clinical sample characteristics between the three post-traumatic growth groups levels (Table 1). Effects of post-traumatic growth and assessment time on coping strategies As it is presented in Table 2 and Figure 2, interaction effects between initial level of posttraumatic growth and assessment time were found for the following coping strategies: emotional support [F (4,514) = 4.252, p < 0.0001], positive reframing [F (4,514) = 4.395, p < 0.0001], active coping [F (4,514) = 6.612, p < 0.0001], and planning [F (4,514) = 5.107, p <
117 0.0001]. Simple effects showed that these four coping strategies are used less frequently at T1 than T2 and T3 (ds from 0.58 to 1.03, medium to big effect size), and were more frequently used by patients in the high PTG than in the low PTG group (ds from 0.29 to 0.43, small effect size). See in Table 3. Comparisons between different follow-ups in the three PTG groups are reported in Table 4. The variable time showed significant effects on these coping strategies in the low PTG group (ds from 0.45 to 1.24, small to large effect size), except for positive reframing from T2 to T3 (p = 0.529, d = 0.15). In addition, time also had an effect in the medium PTG group (ds from 0.61 to 1.06, medium to large effect size) and high PTG group (ds from 0.42 to 0.67) when comparing the use of the four strategies at T1 with T2 and T3. Apart from planning (p = 0.048, d = 0.29), no significant difference was found between T2 and T3 in the use of these strategies in the medium and high PTG groups. Main effects: post-traumatic growth level effect on coping strategies Regarding main effects, initial post-traumatic growth level was significant for instrumental support [F (2,257) = 5.107, p = 0.007] and self-distraction [F (2,257) = 3.319, p = 0.038] (Table 2). Simple effect results reported in table 3 indicated that instrumental support and self-distraction were significantly more frequent in the high PTG group compared to the low PTG group (ds 0.40 and 0.35, small effect size). Main effects: assessment time on coping strategies The time factor was also significant for acceptance [F (2,514) = 26.463, p < 0.0001], humor [F (2,514) = 82.970, p < 0.0001], religion [F (2,514) = 9.566, p < 0.0001], instrumental support [F (2,514) = 15.780, p < 0.0001], self-distraction [F (2,514) = 49.652, p < 0.0001], self-blame [F (2,514) = 109.678, p < 0.0001] and venting [F (2,514) = 24.560 , p < 0.0001] (Table 2). These coping strategies were used less at T1 compared to T2 and T3 (ds from 0.20 to 1.02, from small to large effect size). For self-distraction (p = 0.03, d = 0.26) and self-blaming (p < 0.0001, d = 0.28) the comparison between T2 and T3 was also significant with a small effect size (Table 3).
118 L, large effect size; M, medium effect size; S, small effect size; N, null effect size. Significance value, *p < 0.05, **p < 0.001. Table 2. Coping strategies: differences in coping strategies used by evaluation phase and initial post-traumatic growth (3x3 mixed factorial analysis of variance). Main effects (Cohen´s f) Phase Post-traumatic growth Interaction effects (Cohen´s f) F (2,514) F (2,257) F (4,514) Acceptance 26.463** 0.320 M 2.869 0.149 S 0.378 0.054 N Emotional support 82.898** 0.568 L 7.050** 0.234 S 4.252** 0.181 S Humor 82.970** 0.568 L 4.623 0.190 S 1.383 0.105 S Positive reframing 75.263** 0.541 L 5381** 0.204 S 4.395** 0.184 S Religion 9.566** 0.193 S 1.019 0.089 N 1.239 0.1 S Active coping 102.369** 0.631 L 11.2** 0.29 M 6.612** 0.226 S Instrumental support 15.780** 0.250 M 5.107** 0.198 S 0.923 0.083 N Planning 86.358** 0.580 L 3.239* 0.206 S 5.107** 0.198 S Behavioral disengagement 0.015 0 N 1.091 0.008 N 1.393 0.011 N Denial 0.325 0.031 N 1.298 0.100 S 3.035 0.153 S Self-distraction 49.652** 0.439 L 3.319* 0.160 S 0.780 0.077 N Self-blaming 109.678** 0.653 L 0.767 0.077 N 0.364 0.054 N Substance use 1.008 0.063 N 0.792 0.044 N 1.012 0.089 N Venting 24.560** 0.308 M 0.736 0.077 N 1.246 0.101 S
119 Figure 2. Interactive effects between initial level of post-traumatic growth and evaluation phases. Predictors of post-traumatic growth Total post-traumatic growth score was regressed on coping strategies at the three time points. As it is presented in Table 5 a higher score in positive reframing (β = 0.188, p < 0.001), emotional support (β = 0.152, p = 0.002), planning (β = 0.134, p = 0.008), religion means (β = 0.115, p = 0.017) and self-distraction (β = 0.098, p = 0.044) predicted greater PGI-21 total score levels at T1. These five coping strategies explained 20.1% of PGI-21 total score variance with a medium effect size (f2 = 0.251). In descending order of contribution, religion means (β = 0.281, p <0.001), emotional support (β = 0.255, p < 0.001), active coping (β = 0.164, p = 0.001), positive reframing (β = 0.119, p = 0.023) and self-distraction (β = 0.107, p = 0.037) positively predicted PGI-total score at T2. All variables included in the model accounted for a 29.4% of PGI-21 total score variance. Effect size coefficient indicates a large effect size (f2 = 0.416) (Table 6). At T3 the PGI-21 total score predicting coping strategies were positive reframing (β = 0.269, p < 0.001), instrumental support (β = 0.215, p < 0.001) and religion (β = 0.158, p = 0.006). A higher use of positive reframing, instrumental support and religion predicted higher PGI-21 levels. The three variables together accounted for 17.2% of PGI-21 total score variance at T3, with a medium effect size (f2 = 0.207) (Table 7).
120 L, large effect size; M, medium effect size; S, small effect size; N, null effect size. Table 3. Coping strategies: differences between coping strategies used according to post-traumatic growth level and evaluation phases. Post-traumatic growth level M (SD) Phases Comparisons p (Cohen’s d) Low (a) n = 85 Medium (b) n = 84 High (c) n = 91 1 2 3 Group levels Phases a-b a-c b-c 1-2 1-3 2-3 Acceptance 2.40 (0.81) 2.44 (0.72) 2.56 (0.57) 2.28 (0.73) 2.58 (0.56) 2.56 (0.60) 1 0.05 N 0.072 0.23 S 0.237 0.18 N <0.001 0.58 M <0.001 0.42 S 1 0.03 N Emotional support 1.75 (0.95) 1.96 (0.77) 2.15 (0.91) 1.52 (0.92) 2.07 (0.96) 2.26 (0.83) 0.178 0.24 S 0.001 0.43 S 0.211 0.23 S <0.0001 0.58 M <0.0001 0.88 L 0.005 0.21 S Humor 1.81 (1.05) 1.83 (1.04) 2.13 (1.10) 1.42 (1.10) 2.24 (0.98) 2.11 (1.02) 1 0.02 N 0.023 0.30 S 0.035 0.28 S <0.0001 0.79 M <0.0001 0.65 M 0.103 0.13 N Positive reframing 1.84 (0.95) 2.03 (0.81) 2.17 (0.93) 1.57 (0.94) 2.21 (0.88) 2.26 (0.87) 0.196 0.22 S 0.004 0.30 S 0.504 0.16 N <0.0001 0.70 M <0.0001 0.76 M 1 0.06 N Religion means 0.90 (0.88) 1.06 (0.73) 1.07 (0.94) 0.86 (0.98) 1.06 (1.07) 1.09 (1.08) 0.701 0.19 N 0.605 0.19 N 1 0.01 N 0.001 0.20 S <0.0001 0.22 S 1 0.03 N Active coping 2.25 (0.85) 2.36 (0.65) 2.52 (0.67) 1.94 (0.79) 2.50 (0.62) 2.67 (0.58) 0.121 0.15 N <0.0001 0.35 S 0.029 0.24 S <0.0001 0.78 M <0.0001 1.03 L 0.004 0.28 S Instrumental support 1.42 (0.78) 1.55 (0.84) 1.76 (0.90) 1.38 (0.86) 1.64 (0.98) 1.73 (0.96) 0.713 0.16 N 0.005 0.40 S 0.158 0.24 S <0.0001 0.28 S <0.0001 0.38 S 0.555 0.09 N Planning 1.78 (0.78) 1.94 (0.76) 2.01 (0.82) 1.47 (0.83) 1.99 (0.90) 2.27 (0.81) 0.293 0.20 S 0.039 0.29 S 1 0.09 N <0.0001 0.60 M <0.0001 0.98 L <0.001 0.33 S Behavioral disengagem ent 0.39 (0.57) 0.37 (0.52) 0.40 (0.66) 0.39 (0.59) 0.39 (0.61) 0.38 (0.54) 1 0.04 N 1 0.02 N 0.427 0.05 N 1 0 N 1 0.02 N 1 0.02 N Denial 0.46 (0.62) 0.45 (0.73) 0.36 (0.57) 0.41 (0.64) 0.42 (0.61) 0.45 (0.63) 1 0.02 N 0.469 0.17 N 0.535 0.14 N 1 0.02 N 1 0.06 N 1 0.05 N Selfdistraction 1.40 (1.05) 1.51 (0.92) 1.77 (1.07) 1.57 (1.02) 2.05 (0.94) 2.28 (0.78) 0.552 0.11 N 0.032 0.35 S 0.678 0.26 S <0.0001 0.50 M <0.0001 0.78 M 0.003 0.26 S Selfblaming 1.61 (0.99) 1.74 (0.88) 1.71 (1.02) 1.15 (0.97) 1.83 (0.90) 2.07 (0.81) 0.703 0.14 N 1 0.09 N 1 0.03 N <0.0001 0.77 M <0.0001 1.02 L <0.0001 0.28 S Substance use 0.10 (0.35) 0.113 (0.23) 0.13 (0.47) 0.11 (0.37) 0.14 (0.51) 0.10 (0.35) 1 0.04 N 0.742 0.07 N 1 0.05 N <0.0001 0.07 N <0.0001 0.02 N 0.705 0.09 N Venting 0.97 (0.83) 0.89 (0.68) 1.15 (0.81) 1.01 (0.78) 1.26 (0.80) 1.37 (0.64) 1 0.11 N 1 0.12 N 0.766 0.35 S <0.0001 0.32 S <0.0001 0.50 M 0.138 0.15 N
121 Table 4. Coping strategies: differences in coping strategies use between evaluation phases by initial posttraumatic growth level. L, large effect size; M, medium effect size; S, small effect size; N, null effect size. M, medium effect size; s, small effect size. Significance value, *p < 0.05; **p < 0.001. Comparison between phases, p (Cohen´s d) Low CP Medium CP High CP 1-2 1-3 2-3 1-2 1-3 2-3 1-2 1-3 2-3 Emotional support <0.0001 0.61 M <0.0001 1.06 L <0.0001 0.50 M <0.0001 0.72 M <0.0001 0.86 L 1 0.09 N <0.0001 0.49 S <0.0001 0.57 M 1 0.05 N Positive reframing <0.0001 0.85 L <0.0001 1.05 L 0.529 0.15 N <0.0001 0.87 L <0.0001 0.90 L 1 0.02 N 0.002 0.46 S 0.003 0.39 S 1 0.05 N Active coping <0.0001 0.85 L <0.0001 1.24 L <0.0001 0.45 S <0.0001 0.78 M <0.0001 1.06 L 0.318 0.23 S <0.0001 0.60 M <0.0001 0.65 M 1 0.08 N Planning <0.0001 0.79 M <0.0001 1.18 L <0.0001 0.50 M <0.0001 0.61 M <0.0001 1.01 L 0.048 0.29 S 0.003 0.42 S <0.0001 0.67 M 0.642 0.22 S Table 5. T1 Post-traumatic growth (CP-21) Multiple linear regression model. F R2 B SE.B β 1-β f2 Model 1 54.523 (1.405) 0.119** 34.292 2.159 0.99 0.135 (S) Positive reframing 8.798 1.192 0.344** Model 2 37.997 (2.404) 0.158** 28.824 2.456 0.99 0.187 (M) Positive reframing 7.001 1.236 0.274** Emotional support 5.402 1.238 0.211** Model 3 29.391 (3.403) 0.180** 25.331 2.659 0.99 0.222 (M) Positive reframing 5.869 1.272 0.230** Emotional support 4.604 1.248 0.180** Planning 4.471 1.386 0.158** Model 4 24.082 (4.402) 0.193* 24.812 2.647 0.99 0.239 (M) Positive reframing 5.306 1.281 0.208** Emotional support 3.791 1.277 0.148** Planning 4.457 1.376 0.158** Religion means 3.054 1.165 0.126** Model 5 20.227 (5.401) 0.201* 22.896 2.802 0.99 0.251 (M) Positive reframing 4.806 1.300 0.188** Emotional support 3.883 1.273 0.152** Planning 3.784 1.410 0.134** Religion means 2.789 1.168 0.115* Self-distraction 2.361 1.170 0.098*
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131 Schwartz, J. R., Thomas, E. B. K., Juckett, M. B., & Costanzo, E. S. (2022). Predictors of posttraumatic growth among hematopoietic cell transplant recipients. Psycho-Oncology, 31(6), 1013-1021. https://doi.org/10.1002/pon.5892 Şen, S. (2018). Neurostatus and EDSS calculation with cases. Noropsikiyatri Arsivi, 55, 80- 83. https://doi.org/10.29399/NPA.23412 Tedeschi, R. G., & Calhoun, L. G. (1996). The posttraumatic growth inventory: Measuring the positive legacy of trauma. Journal of Traumatic Stress, 9(3), 455-471. https://doi.org/10.1002/jts.2490090305 Tedeschi, R. G., & Calhoun, L. G. (2004). Posttraumatic growth: Conceptual foundations and empirical evidence. Psychological Inquiry, 15(1), 1-18. https://doi.org/10.1207/s15327965pli1501_01 Treder-Rochna, N. (2020). Adaptation to the disease - the psychological resources of families struggling with multiple sclerosis. Health Psychology Report, 8(2), 136-144. https://doi.org/ 10.5114/HPR.2020.94722 Wawrziczny, E., Corrairie, A., & Antoine, P. (2021). Relapsing-remitting multiple sclerosis: an interpretative phenomenological analysis of dyadic dynamics. Disability and Rehabilitation, 43(1), 76–84. https://doi.org/10.1080/09638288.2019.1617794 Weiss, T., & Berger, R. (2006). Reliability and validity of a Spanish version of the posttraumatic growth inventory. Research on Social Work Practice, 16(2), 191-199. https://doi.org/10.1177/1049731505281374
132 4.6. Sexto trabajo titulado “Significance of post-traumatic growth and mental health for coping in multiple sclerosis caregivers” Este trabajo corresponde al artículo publicado que se referencia a continuación: Gil-González, I., Pérez-San-Gregorio, M. Á., Funuyet-Salas, F., Conrad, R., & Martín- Rodríguez, A. (2023). Significance of post-traumatic growth and mental health for coping in multiple sclerosis caregivers. Healthcare, 11, 1390. https://doi.org/10.3390/healthcare11101390
133 Abstract We investigated the influence of post-traumatic growth (PTG) and mental health (MH) on multiple sclerosis (MS) caregivers’ use of coping strategies and identify biopsychosocial predictors of proactive or reactive coping. The Short Form Health Survey (SF-12), General Health Questionnaire (GHQ-28), Post-traumatic Growth Inventory (PGI-21), Brief COPE Questionnaire (COPE-28), and Multidimensional Scale of Perceived Social Support (MSPSS) were used to evaluate 209 caregivers. Higher PTG was related to greater use of emotional support, positive reframing, religion, active coping, instrumental support, planning, denial, self-distraction, self-blaming, and venting. Better MH was associated with a greater use of acceptance, while behavioral disengagement and self-distraction were associated with poorer MH. The PTG dimensions relating to others and new possibilities, SF-12 dimensions physical and emotional role as well as partnership, not living with the patient, and significant others social support were predictors of proactive coping. Reactive coping was positively predicted by PTG dimension relating to others, depression, vitality, other than partner relation, and physical role, and negatively predicted by mental health level and emotional role. In summary, higher MH was associated with proactive coping strategies, whereas post-traumatic growth was related to the use of a wide range of proactive coping as well as reactive coping strategies. Keywords: multiple sclerosis; caregivers; post-traumatic growth; mental health; coping strategies. Introduction Multiple sclerosis (MS) is a chronic neurodegenerative disease frequently beginning in young adulthood. Myelin sheaths covering nerve fibers are attacked by the immune system. The result is inflammation that can lead to lesions in the brain, optic nerve, and spinal cord. Thus, communication between the central nervous system and other parts of the body is impaired (García-López et al., 2022). The prevalence of MS is heterogeneous. The highest estimated prevalence is in Europe and America, exceeding 100 cases per 100.000 individuals, whereas the estimated prevalence in Africa and Southeast Asia is below 10 cases per 100.000 individuals. The number of people living with MS has increased significantly in recent decades, with a female predominance in the incidence of the disease worldwide (Atlas MS, 2023). Symptomatology associated with MS includes fatigue, chronic pain, balance problems, spasticity, cognitive impairment, visual disturbances, and bowel problems. In socio-economic terms, MS patients have been found to have 15-30% lower employment and earnings, as well as higher absenteeism and work
134 disability (Kavaliunas et al., 2021). A significant proportion of MS patients therefore require informal care from family or friends (Rajachandrakumar & Finlayson, 2021). In formal caregivers may experience distress and reduced well-being affecting physical and mental health (MH) (Di Cara et al., 2020; Hlabangana & Hearn 2020; Merali et al., 2020). All participants in a study by Gafari et al. (2017) suffered at some point from emotional symptoms including anxiety, depression, and stress. Participants identified hard and extended periods of care as the origin of their disturbance. Hlabangana & Hearn (2020) reported that half of the caregivers in their sample fulfilled the criteria for depression. They also scored lower on all quality of life domains compared to the general population. Although caregiving repercussions have been widely reported, MS care can also lead to beneficial consequences, for instance, becoming aware of personal resources and family support (Maguire & Maguire, 2020; Rajachandrakumar & Finlayson, 2021). Thus, complications in MS caring may coexist with positive outcomes, such as benefit finding or post-traumatic growth (PTG). PTG is generally defined as an experience of positive change that occurs as a result of the struggle with highly challenging life crises. “Post-traumatic” refers that growth occurs after an adverse crucial event in life. “Growth” denotes to a personal enhancement of capabilities and functioning due to a superior mental and emotional awareness resulted from a critical life (Kivi et al., 2019). Regarding PTG connection with MH, PTG has been shown to be positively related to anxiety and negatively related to depressive symptoms. The lack of energy from depression can hinder the inner drive needed for growth (Kivi et al., 2019), while anxiety might leave the subject in a state more favorable to respond actively to adversity. In the process of adapting to the caring role and struggling with its duties the use of a wide spectrum of coping strategies has been emphasized. Early research studies in this field emphasized that in some cases, the efforts of caregivers and patients, due to conflicting coping styles, may result in even more stress in an already stressful situation (O’Brien, 1993). Different coping styles shape the background of diverging types of communication when dealing with MS, such as talking about the illness or not, and communicating emotions or not (Montel & Bungener, 2007). An interesting finding is the observed gender-related use of coping styles, for example men tend to use more planning as a coping strategy, while women tend to designate their own physical space within the house (Courts et al., 2005). As far as the typical coping styles in MS caregivers are concerned, these have been investigated in order to identify the specific ways caregivers respond to stress and demand but no reasonable conclusions have been reached yet, probably because coping is a highly complex phenomenon
135 (Kouzoupis et al., 2010). In this context Lazarus and Folkman's Transaction-al Model of Stress and Coping (Lazarus & Folkman, 1984) may help to comprehend the connection between caregivers’ role and well-being. According to the model, caregiving consists of functional and emotional demands. Coping should keep caregiving demands and caregivers’ resources in balance to preserve their well-being (Bassi et al., 2016). There is evidence that stress management coping, health promoting behaviors, supporting engagement, and positive reframing are helpful strategies for caregivers, contrary to avoidance coping (Maguire & Maguire, 2020; Rajachandrakumar & Finlayson, 2021). Potentially, proactive coping strategies, which aim at controlling a situation in advance, are thought to be more adaptive compared to reactive strategies, which respond to a challenging situation (Aspinwall, 2010). Impaired physical and MH in caregivers has been proven to be an obstacle in caregiving clarifying the need for adaptive coping strategies (Di Cara et al., 2020; Gafari et al., 2017; Martindale-Adams et al., 2020; Penwell-Waines et al., 2016; Rajachandrakumar & Finlayson 2021). Various studies point out the beneficial effect of social support to preserve MS caregivers’ well-being and MH (Dȩbska et al., 2020; Maguire & Maguire, 2020; Treder- Rochna, 2020; van der Hiele, 2019). Against this backdrop the thorough investigation of factors influencing the use of specific coping strategies is of great importance (Di Cara et al., 2020). A deeper insight into the use of coping strategies in MS caregivers facilitates the understanding of their specific needs. In the long run it may help to tailor interventions to reduce caregiver strain and burden to corroborate quality of life and reduce poor outcomes among both caregivers and care recipients with MS. A recent comprehensive review on caregiving in MS comes to the conclusion that current knowledge on MS caregiving is limited particularly with regard to approaches remediating caregiver burden (Rajachandrakumar & Finlayson, 2021). The present study is aimed to explore the use of different coping strategies in MS caregivers with special emphasis on PTG and MH levels and identify potential biopsychosocial predictors of proactive and reactive coping. Methods Sample and procedure Principal caregivers of MS outpatients from Virgen Macarena University Hospital in Seville/Spain were asked to take part in the study, from June 2017 to May 2018. The inclusion criteria were as follows: (a) being the caregiver of a patient with a confirmed MS diagnosis; (b) aged over 18; and (c) being able to understand and fill in study
136 questionnaires. The participants answered the questionnaires themselves. The research psychologist was available for any questions or assistance required. Figure 1 summarizes the sample selection process. The research was authorized by the responsible Ethics Committee (0846-N-18). All participants gave their informed consent to participate in the study. Figure 1. Study flowchart. CIS, clinically isolated syndrome. Instruments Health related Quality of life The 12-Item Short Form Health Survey (SF-12) consists of 12 items. The SF-12 includes eight domains: physical functioning, role-physical, bodily pain, general health, vitality, social functioning, role-emotional and MH. Subscales scores vary from 0 (worst) to 100 (best). These subscales can result in two summary component scores: the Physical Component Summary Score (PCS) and the Mental Component Summary Score (MCS). PCS and MCS were calculated using QualityMetric SF Health Outcomes Scoring Software (Vilagut, 2008; Ware, 2002). In our sample, the Cronbach’s alpha for the mentioned dimensions ranged from 0.89 to 0.92. Cronbach’s alpha for PCS and MCS were 0.92 and 0.88, respectively (Maruish, 2012). Mental Health Questionnaire (GHQ-28) The General Health Questionnaire (GHQ-28) contains 28 items 4-point Likert scaled. The four subscales are somatic symptoms, anxiety/insomnia, social dysfunction, and depression. Subscale scores range from 0 (best) to 21 (worst). Total GHQ-28 score ranges from 0 (best) to 84 (worst) (Golberg et al., 1997; Willmott et al., 2004). The GHQ-28 Spanish version presents an acceptable degree of validity (Lobo et al., 1986) and reliability in the investigation of chronic medical conditions (Vallejo et al., 2014). In our sample Cronbach’s alpha for the subscales varied from 0.77 to 0.92. Cronbach’s alpha for the total scale was 0.92.
137 Post-traumatic Growth Perception of personal gain after MS was measured using the Spanish version of the Post- Traumatic Growth Inventory (PGI-21) (Tedeschi & Calhoun, 1996; Weiss & Berger, 2006). The PGI-21 contains 21 items scored on a 5-point Likert scale, a higher score indicating greater change. Test results provide five dimensions: relating to others, new possibilities, personal strength, spiritual change, and appreciation of life. Cronbach’s alpha in our study ranged from 0.77 to 0.88 for the subscales, and 0.94 for the total score. Coping Strategies The Spanish version of Brief COPE Questionnaire (COPE-28) assessed caregivers’ coping strategies (Carver, 1997; Morán et al., 2010). The COPE-28 comprises 28 items scored on a 4 points Likert scale from 0 (“I have not been doing this at all”) to 3 (“I have been doing this a lot”). Items can be collapsed in 14 subscales: (1) acceptance; (2) emotional support; (3) humor; (4) positive reframing; (5) religion; (6) active coping; (7) instrumental support; (8) planning; (9) behavioral disengagement; (10) denial; (11) self-distraction; (12) self-blaming; (13) substance use; and (14) venting. Cronbach’s alpha in our study varied from 0.65 to 0.84 for the subscales. Social support The Multidimensional Scale of Perceived Social Support (MSPSS) evaluated perceived social support from family, friends, and partner or significant others (Arechabala & Miranda, 2002; Zimet et al., 1988). The MSPSS consists of 12-items scored on a Likert scale, from 1 to 7. The total score ranges from 12 to 84. In the present sample Cronbach´s alpha varied from 0.91 to 0.95 for the subscales and 0.92 for the total score. Data Analysis Sample features were reported using descriptive analysis. A one-way analysis of variance (ANOVA) for quantitative variables (age, Expanded Disability Status Scale -EDSS-, months since diagnosis, and months since the outbreak) and a Chi-squared test for categorial variables (gender, partnership, occupation, educational level, MS subtype, family relation, and living together) explored possible differences in demographics and patients’ clinical characteristics between subgroups with low, medium, or high PTG or MH levels. A 3×3 factorial ANOVA with Bonferroni post-hoc tests was calculated to study the influence of post-traumatic growth level and MH level on the use of coping strategies.
144 These six variables accounted for 34.6% of proactive coping variance with a large effect size (Table 6). Reactive coping was positively predicted by relating to others (β = 0.327, p < 0.001), severe depression (β = 0.228, p < 0.001), vitality (β = 0.293, p < 0.001), other than partner relation (β = 0.134, p < 0.013), and physical role (β = 0.158, p < 0.001). Reactive coping was negatively predicted by MH (β = -0.289, p < 0.001) and emotional role (β = -0.241, p = 0.002). All variables introduced in the model explained 46.3% of variance. Effect size coefficient indicated a large effect size (Table 6). Discussion MS caregivers’ coping has shown to be a key factor in the patient-caregiver dyad’s adaptation to the disease and well-being. The present study explored the influence of PTG and MH levels on the use of different coping strategies as well as biopsychosocial predictors of proactive and reactive coping Influence of Post-Traumatic Growth and Mental Health on Coping Strategies No interaction effect was found between PTG or MH and coping strategies. Regarding main effects post-traumatic growth showed a significant effect on coping. Caregivers with higher PTG used more frequently coping strategies such as emotional Table 6. Proactive and reactive coping strategies multiple linear regression models. F R2 B SE.B β 1-β f2 Proactive coping strategies Model 17.831 (6,202) 0.346 0.203 0.189 0.99 0.529 (L) Relating to others 0.102 0.032 0.263* Cohabitation 0.289 0.070 0.244** Partner or other support 0.062 0.021 0.171* New possibilities 0.076 0.033 0.186* Physical role 0.005 0.002 0.257* Emotional role 0.004 0.002 0.223* Reactive coping strategies Model 24.718 (7,201) 0.463 0.319 0.119 0.99 0.862 (L) Relating to others 0.097 0.016 0.327** Severe depression 0.029 0.007 0.228** Mental health -0.006 0.001 -0.289** Vitality 0.005 0.001 0.293** Emotional role -0.004 0.001 -0.241* Other than partner relation 0.107 0.043 0.134* Physical role 0.002 0.001 0.158* L, large effect size. Significance value, *p < 0.05; **p < 0.001.
145 support, positive reframing, religion, active coping, instrumental support, planning, denial, self-distraction, self-blaming, and venting. In reference to emotional and instrumental support, our results are in line with previous research pointing out the connection between opening up in a secure social environment and trauma processing (Henson et al., 2021). The promotion of communication and connection between MS family members is related to disease adaptation and higher PTG (Neate et al., 2019; Treder-Rochna, 2020). Asking for practical help involves perceiving one’s own needs and expressing the emotional stress associated with it (Henson et al., 2021). Positive reframing has been extensively related to benefit and personal gain, as posttraumatic growth (Henson et al., 2021; Neate et al., 2019; Pérez-San-Gregorio et al., 2017). This strategy implies a positive cognitive reconstruction of a challenging situation. Interventions focused on re-evaluation of goals and values have demonstrated to support meaningfulness and well-being in caregivers (Azimian et al., 2021). In the same direction religious coping has been associated with PTG by reevaluating despairing situations into a challenging assignment. Moreover, religious communities can enhance relationships via participation in communal activities (Henson et al., 2021; Martin et al., 2021). From the caregivers’ perspective planning and confronting themselves with the fact of an unpredictable disease progression may increase a feeling of being in control and being able to pursue new goals, which may explain the association with PTG in our study. Even though denial and self-distraction are regarded as disadvantageous coping strategies in many circumstances, our results agree with earlier research arguing a potentially positive relationship between avoidance coping and personal growth in MS carers (Martindale-Adams et al., 2020; Pakenham et al., 2005; Wawrziczny et al., 2021). The capability to transiently divert one’s attention from an uncontrollable situation might be helpful to maintain emotional stability (Kunz et al., 2018). Pakenham (2005) distinguished between two types of avoidance or withdrawal: First, complete flight from stress, burying the head in the sand, resulting in distress and maladjustment. Second, transient “time out” to reconsider priorities, gather strengths, and plan, which can be related to personal growth and gain. Very little was found in the literature on self-blaming and PTG. Bearing in mind that posttraumatic growth as conceptualized by Calhoun and Tedeschi (2006) derives from an intense inner confrontation with challenging situations it involves self-reflection and at least transient self-criticism that puts the handling of previous situations and the application of familiar strategies into question. Thus, self-blaming could be understood in this context as a transient strategy fueling inner cognitive and emotional reorganization.
146 Concerning venting, COPE-28 conceptualized this coping strategy as an emotion-al reaction to liberate oneself from negative emotions. To accomplish venting and benefit from its cathartic effect caregivers need to recognize the unpleasant emotions and allow themselves to show their feelings (Pakenham, 2005). MH did also have an impact on caregivers coping. Better MH was related with acceptance, while behavioral disengagement and self-distraction were associated with worse MH. These results are supported by previous research by Penwell-Waines et al. (2016) who found that caregivers with lower levels of distress employed more self-care and stress management. The findings of Bassi et al. (2016) associated caregivers’ avoidance with poorer well-being. As outlined above, on the one hand the ability to transiently distract from a challenging task might be beneficial to spare resources. Thus, O´Brien (1993) reported that MS caregivers used regularly wishful thinking to modify the current situation and distract themselves from the harsh reality. However, in poor MH associated, for example, with depression or anxiety selfdistraction and avoidance may become the predominant strategies, resulting in maladaptation, and worsening of the situation. Obviously, the COPE-28 questionnaire does not distinguish between the transient or predominant use of specific strategies. Coping Strategies Predictors The first predictor of proactive coping as defined in our study was relating to others. This PGI-21 subscale appraises closeness to others, compassion for those who suffer, and the willingness to be helped and use social support. Caregivers relying on their social network rather solve problems in conjunction with others and communicate their personal experiences (O’Brien, 1993). Seeking emotional and instrumental support, and positive reframing, might be easier for those caregivers who have a supportive social environment. A lot of evidence connects caregivers’ adaptation and healthier coping with close family relationships and social support (Dȩbska et al., 2020; Maguire & Maguire 2020; Treder-Rochna, 2020; van der Hiele, 2019). Not living together with the patient predicted higher use of proactive coping. Caregivers not living apart may be overtaxed due to long periods of care without being able to distance themselves from the challenging task and take care of themselves. In most cases, if the principal caregiver is not living with the patient this indicates a lower level of disability not requiring assistance 24 hours daily (Hlabangana & Hearn, 2020). Not surprisingly, partner support was a positive predictor of proactive coping. Most study participants were in a partnership with the patient (64.6%). Couples’ satisfaction has
147 demonstrated to play an important role in patients-caregiver dyad’s adjustment. Prior research stated that couples reporting a high-quality partnership cope better with emotional stress (Santos et al., 2019; Treder-Rochna, 2020). In fact, intervention programs incorporating partner support in MS have shown to improve adaptation in both members (Merali et al., 2020; Neate et al., 2018, 2019). The PGI-21 dimension of new possibilities was also a positive predictor of proactive coping. It can be argued that meeting new people, engaging in new activities or life projects is closely associated with a tendency to tackle challenging situations in advance rather than merely reacting to them (Henson et al., 2021). In the same direction less physical and emotional limitations as measured by the SF-12 scales role-physical and role-emotional predict proactive coping as less health impairment facilitates handling and controlling a problematic situation. The relationship between greater well-being and proactive coping has been proven in previous studies (Bassi et al., 2016; Rajachandrakumar & Finlayson 2021). Unexpectedly, relating to others was also the strongest predictor of reactive coping. One might argue that social contacts and relations can also be used to self-distract oneself from an unbearable situation or their help may lead to behavioral disengagement. However, as outlined above, this reactive coping strategy, at least when transiently used may also be adaptive as they help to spare resources and gain strength. Higher depression predicted more frequent reactive coping whereas better MH predicted less. These findings are broadly supported by previous research connecting reactive coping with a higher probability of maladaptation to the caregiving situation (Bassi et al., 2016; Pakenham, 2005). Improvement of depression and distress led to more health promoting and stress management behavior (Martindale-Adams et al., 2020; Penwell-Waines et al., 2016) highlighting the relevance of stress prevention in MS caregiving. Another family relationship of the caregiver than partner also predicted reactive coping. Several reports have shown that a “communal sense of the illness” can foster positive outcomes in the MS patient-caregiver dyad (Neate et al., 2019; Treder-Rochna et al., 2020; Neate et al., 2018). Developing a communal sense of MS requires intense mutual communication involving patients’ and caregivers’ inner perspective, which may be easier in a partnership (Gil-González et al., 2022). Contrary to expectations vitality predicted higher use of reactive coping. One might argue that vitality may be associated with the intense wish to take part in social life and associated activities. If this desire is hampered by a particularly challenging caregiving situation reactive
148 strategy, self-distraction or denial may be necessary and at least transiently more frequently used. The major study weakness is the non-random selection of the sample, which limits its external validity. Also, only self-report instruments were applied, which there is scarce data on the validation of use in the population of MS caregivers. Furthermore, some factors should have been controlled, such as age, gender, or time since diagnosis, to avoid some possible interactions in the results. Considering certain variables, such as the level of dependency of the MS patient on the caregiver would have been very appropriate. Its major strength is the large and heterogeneous sample and the investigation of a variety of biopsychosocial variables. Conclusions The findings of this study have high relevance for the preservation and improvement of the well-being of MS caregivers in the clinical practice, and thus for the care of patients. MH and post-traumatic growth have an important impact on the use of coping strategies in MS caregivers. Poor MH is associated with less proactive coping, whereas post-traumatic growth corroborates the use of a wide range of proactive coping as well as reactive coping strategies. The results highlight the importance of multi-disciplinary interventions to preserve MH and enhance those strategies that are most adaptive for each caregiver. Self-distraction and other reactive coping strategies may at least transiently be adaptive in caregivers. Preventive measures regarding MH and support of post-traumatic growth may increase the probability of more frequent proactive coping and increase the probability of favorable and adaptive coping in the long run. In this line, Acceptance and Commitment Therapy (ACT) is intended to help in accepting uncomfortable feelings instead of eliminating them. Caregivers could obtain a more flexible approach to unpleasant feelings and avoid maladaptive coping (Gil-González et al., 2022). Moreover, supporting social environments and connections should be considered a priority to facilitate the adaptation in MS caregivers. A variety of therapies, including social skills training and self-help groups, are proven to promote the usage of social supports by promoting communication, strengthening social bonds, and facilitating sharing emotions in a safe social environment (Gil-González et al., 2022, 2023) Future research is recommended to longitudinally explore the impact of biopsychosocial factors on MS caregivers’ coping.
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lxiii 10.6. Separata del trabajo titulado: “Significance of post-traumatic growth and mental health for coping in multiple sclerosis caregivers”
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