Join Diseases and
Rela ed Su ge y
J Dis Rela Su g
2021;32(1):224-229
CASE SERIES
Recei ed: No embe 03, 2020
Accep ed: Decembe 02, 2020
Published online: Janua y 06, 2021
Co espondence: Ca men Ma ínez-Azna , MD. O hopedic and
T auma Su ge y Se ice, Miguel Se e Uni e si y Hospi al, Paseo
de Isabel la Ca ólica, 50009 Za agoza. Spain.
E-mail: cma inezazna @gmail.com
Doi: 10.5606/ehc.2021.79576
Congeni al pseuda h osis o he cla icle (CPC) is
a a e en i y. I was i s epo ed by Fi zwilliams[1]
in 1910 and i is di icul o know he ue incidence
wi h a ound 200 cases published in he li e a u e. The
exac e iology is s ill unknown. The mos accep ed
heo y s a es ha i is a ailu e o usion o he wo
diaphyseal memb anous ossi ica ion cen e s in he
six h in au e ine week o ges a ion[2] and seconda y
o ana omic abno mali ies which a ec he subcla ian
a e y, as in he p esence o a p ominen i s ib.[3,4] I
explains ha mos pa ien s p esen igh -sided CPC,
and cases o le -sided a e associa ed o dex oca dia.
Bila e al cases a e excep ional.[5] I seems o be mo e
common in emales. Congeni al pseuda h osis o
he cla icle can be diagnosed a bi h o la e in
childhood, as a swelling in he middle- hi d cla icle
bu which is no mally painless (Figu e 1), being
necessa y o es ablish a di e en ial diagnosis wi h
Objec i es: This s udy epo s he su gical ou comes in a pa ien
coho wi h congeni al pseuda h osis o he cla icle (CPC).
Pa ien s and me hods: A o al o nine pedia ic pa ien s (5 males,
4 emales; mean age: 4.43 yea s; ange, 2 o 12 yea s) who we e
diagnosed wi h CPC and ea ed su gically wi h a minimum
one-yea ollow-up be ween Janua y 1996 and Decembe 2018 we e
e ospec i ely analyzed. The pa ien s we e di ided in o wo g oups
acco ding o he ixa ion me hod as he Ki schne wi e (K-wi e;
G oup A, n=2) o s abiliza ion wi h a pla e (G oup B, n=7). The
physical unc ion and symp oms we e e alua ed using he Quick
Disabili ies o he A m, Shoulde and Hand (QuickDASH) scale.
Resul s: The mean age a he ime o diagnosis was 2.95 ( ange,
0 o 12) yea s. Six cases we e a ophic pseuda h osis and h ee
cases we e hype ophic. Radiog aphic consolida ion occu ed in
all cases wi h a mean du a ion o 103.8±39.1 days, indica ing no
signi ican di e ence be ween he g oups (p>0.05). Complica ions
egis e ed we e a K-wi e b eakage in a pa ien in G oup A and an
in e nal ixa ion pla e loosening in a pa ien in G oup B. The mean
pos ope a i e ollow-up was 2.98±1.82 yea s. The QuickDASH
sco e was 0 poin s in all pa ien s.
Conclusion: The ea ly su gical indica ion based on e eshmen o
he pseuda h osis ocus, wi h bone au og a in e posi ion when a
ailu e exis s o es o e adequa e cla icula leng h, and ixa ion a e
eliable al e na i es wi h a o able clinical and adiological esul s
in he mid- and long- e m wi h ewe complica ions.
Keywo ds: Cla icle, congeni al pseuda h osis o cla icle, quick
disabili ies o he a m, shoulde and hand, su gical p ocedu es.
ABSTRACT
Su gical ea men o congeni al pseudoa h osis o he
cla icle: Ou 22-yea , single-cen e expe ience
Ca men Ma ínez-Azna , MD1, Isabel Pa ada-A endaño, MD1, Vic o ia E Gómez-Palacio, MD, PhD1,
Sand a Abando-Ruiz, MD1, Jo ge Gil-Alba o a, MD, PhD1,2
1O hopedic and T auma Su ge y Se ice, Miguel Se e Uni e si y Hospi al, Za agoza, Spain
2Depa men o Su ge y, Facul y o Medicine, Uni e si y o Za agoza, Za agoza, Spain.
obs e ic ac u es, pos - auma ic nonunion,
neu o ib oma osis o cleidoc anial dysos osis.[6,7] I
is possible ha symp oms appea due o g ow h
o inc eased ac i i y.[8] In a e cases, ho acic ou le
synd ome o b achial plexus ne e comp ession
a e desc ibed.[9] The e is no clea consensus on he
op imal ea men o his en i y, a wha age o
wha su gical echnique should be pe o med. Some
au ho s ha e ad oca ed o a mo e conse a i e
Ci a ion: Ma ínez-Azna C, Pa ada-A endaño I, Gómez-
Palacio VE, Abando-Ruiz S, Gil-Alba o a J. Su gical ea men o
congeni al pseudoa h osis o he cla icle: Ou 22-yea , single-
cen e expe ience. J Dis Rela Su g 2021;32(1):224-229.
This is an open access a icle unde he e ms o he C ea i e
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dis ibu ion and ep oduc ion in any medium, p o ided he o iginal
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(h p://c ea i ecommons.o g/licenses/by-nc/4.0/).
©2021 All igh ese ed by he Tu kish Join Diseases Founda ion
Su gical ea men o congeni al pseudoa h osis o he cla icle
225
ea men , con ending ha he di icul y o achie e a
bony union and he ac ha pa ien s do no achie e
be e unc ional esul s wi h su ge y.[10,11] Howe e , a
su gical in e en ion is now widely accep ed. Some
au ho s who a gue in a o o su ge y conclude
good unc ional ou comes in pa ien s wi h e iden
de o mi y and/o ela ed symp oma ology.[12]
In he p esen s udy, we aimed o e alua e he
su gical ou comes in a pa ien coho wi h CPC
ea ed in ou cen e .
PATIENTS AND METHODS
This single-cen e , e ospec i e s udy was conduc ed
a O hopedic and T auma Su ge y Se ice, Miguel
Se e Uni e si y Hospi al, Za agoza, Spain be ween
Janua y 1996 and Decembe 2018. All child en
diagnosed wi h CPC and ea ed su gically using
one o wo ixa ion echniques, namely Ki schne
wi e (K-wi e) o pla e ixa ion,[13] and ollowed o
minimum one yea a e su ge y we e included. Those
ha ing associa ed o he pa hologies (i.e., obs e ic o
auma ic ac u es o he cla icle, neu omuscula
diso de s o cleidoc anial dysplasia) we e excluded.
A o al o 13 pa ien s we e diagnosed wi h CPC in ou
cen e ; howe e , only nine o hem (5 males, 4 emales;
mean age: 4.43 yea s; ange, 2 o 12 yea s) ul illed
he inclusion c i e ia. Two pa ien s did no unde go
su ge y due o associa ed ce eb al palsy and majo
neu osenso y disabili y. The o he wo pa ien s we e
admi ed o ou cen e a he age o h ee mon hs and
eigh mon hs, espec i ely. The da a we e ex ac ed
om an ins i u ional da abase and a w i en in o med
consen was ob ained om each pa en and/o legal
gua dian o he su ge y. The s udy was conduc ed
in acco dance wi h he p inciples o he Decla a ion
o Helsinki.
Two me hods o ixa ion du ing su ge y we e
pe o med: The K-wi e (G oup A, n=2) o ixa ion
wi h a pla e (G oup B, n=7). Fixa ion was pe o med
wi h a K-wi e in he wo oldes cases, while he mos
common echnique was a pla e wi h 4 o 6 co ices on
each side o he ocus (Figu e 2). Da a we e collec ed
om medical eco ds and adiological es s a he
ime o diagnosis and a six weeks, a h ee mon hs,
FIGURE 1. Clinical aspec o congeni al pseuda h osis o
he cla icle.
FIGURE 2. (a) An in aope a i e image o congeni al
pseuda h osis o he cla icle. (b) Focus o pseuda h osis
a e e eshmen and in amedulla y d illing. (c) Fixa ion wi h
a econs uc ion pla e.
(a)
(b)
(c)
J Dis Rela Su g226
and a one yea a e su ge y and a e emo al o he
os eosyn hesis ma e ial. Demog aphic and clinical
cha ac e is ics o he pa ien s such as sex, age a he
ime o diagnosis, a ec ed side, symp oms, ype o
pseuda h osis, age a he ime o in e en ion, ype
o su gical ea men , adiog aphic bone healing ime,
esul s, complica ions, implan emo al ime, and
ollow-up we e eco ded. In o de o measu e he
esul s o physical unc ion and symp oms, we used
he Quick Disabili ies o he A m, Shoulde and Hand
(QuickDASH) scale du ing he pos ope a i e isi ,
wo o h ee mon hs a e emo al o he ma e ial.[14]
The policy o ou hospi al a he ime o his s udy
was o o e su gical managemen o all pa ien s
wi h CPC who me inclusion c i e ia. The minimum
age o he child en a he ime o ope a ion was
wo yea s old and he easons we e cosme ic in
all o hem and/o discom o wi h he backpack
in he cla icula a ea in olde pa ien s. No pa ien
p esen ed pain a he ime o diagnosis. All pa ien s
p esen ed a swelling in he cla icula egion a o
soon a e bi h. Radiog aphically, we classi ied
hem as a ophic o hype ophic pseuda h osis.
The echnique consis ed o a longi udinal incision
o e he supe io su ace o he cla icle, ollowed by
e eshmen he ocus o pseuda h osis and d illing
he in amedulla y canal in all cases. An au og a
was used only in he a ophic pa ien s a he bony
edges and/o a gap a e excision o he non-union.
Pos ope a i e ca e was simila in all pa ien s; a sling
was kep o an a e age o ou weeks, allowing
ac i e shoulde mobiliza ion a e wa ds, wi hou
eques ing physio he apy. Spo s ac i i y was no
allowed o h ee mon hs. All pa ien s unde wen a
scheduled ex ac ion o he os eosyn hesis ma e ial.
S a is ical analysis
S a is ical analysis was pe o med using he IBM
SPSS e sion 19.0 so wa e (IBM Co p., A monk,
NY, USA). Con inuous a iables we e exp essed in
mean ± s anda d de ia ion (SD), median (min-max),
while ca ego ical a iables we e exp essed in numbe
and equency. The Kolmogo o -Smi no es , wi h
Lillie o s co ec ion, was pe o med o assess he
no mali y dis ibu ion. The Fishe ’s exac es and
he non-pa ame ic Mann-Whi ney U es we e used
o compa e he g oups o non-no mally dis ibu ed
a iables. A p alue o <0.05 was conside ed
s a is ically signi ican .
RESULTS
Baseline demog aphic and clinical cha ac e is ics o
he pa ien s a e shown in Table I.
TAbLE I
Baseline demog aphic and clinical cha ac e is ics o pa ien s
Pa ien No. 1 2345 6 789
Sex F M M M F M M F F
Age a diagnosis 12 days 2 yea s 25 days 3 yea s 12 yea s 1 mon h 2 yea s 4 yea s 22 mon hs
Side R RRRR R RRR
Age a su ge y (yea s) 4 2 2 4 12 3 3 4 4
Type o pseudoa h osis H A A A A A H A H
Fixa ion K-wi e K-wi e PPP P PPP
Complica ions K-wi e b oken No No No No Sc ew mobiliza ion+pla e
ailu e+delayed union
No No No
Radiological healing (days) 150 105 92 46 96 180 85 97 84
Ma e ial emo al (mon hs) 12 18 10 20 22 14 14 11 13
Follow-up (yea s) 41,75 71,9 24,3 2,6 21,3
Ou come (QuickDASH) (%) 0 0000 0 000
F: Female; M: Male; R: Righ ; H: Hype ophic; A: A ophic; K-wi e: Ki schne wi e; P: Pla e.
Su gical ea men o congeni al pseudoa h osis o he cla icle
227
In all pa ien s, he igh side was a ec ed.
The mean age a he ime o diagnosis was 2.95
( ange, 0 o 12) yea s. On physical examina ion,
all pa ien s had a isible de o mi y o e he
midcla icula line and we e neu o ascula ly
in ac . Hype ophic pseuda h osis was desc ibed
in se en pa ien s (53.84%), h ee o whom we e
ope a ed. The emaining six child en (46.15%) had
a ophic cha ac e is ics and all we e ope a ed
(Figu e 3). In all o hem, we used local au og a
and allog a . Consolida ion occu ed in all pa ien s
wi h no signi ican di e ences ega ding he ime
o achie e i (p>0.05).
In G oup A, one pa ien p esen ed a complica ion
due o needle b eakage one mon h a e he
in e en ion, which equi ed ea ly eope a ion
(Figu e 4). In G oup B, he e we e se en cases,
one o which, wi h a ophic CPC, su e ed om
mobiliza ion o he sc ews ha had an impac on
consolida ion. This case unde wen ein e en ion
wi hin wo mon hs h ough an open educ ion,
ocus e eshmen , and a new pla e ixa ion wi h he
addi ional co icocancellous iliac c es bone g a ing.
The esul was sa is ac o y wi h a subsequen union.
No dono si e mo bidi y was obse ed.
Two pa ien s p esen ing wi h complica ions
wi h he ma e ial had a adiog aphic delay in
FIGURE 3. (a) A ophic CPC; (b) Hype ophic CPC.
CPC: Congeni al pseuda h osis o he cla icle.
(a)
(b)
FIGURE 4. (a) Fixa ion wi h Ki schne wi e; (b) Ki schne wi e b eak; (c) Rein e en ion wi h ocus
e eshmen and ixa ion wi h a pla e adding co icocancellous iliac c es au og a ; (d) One-yea
ollow-up esul s.
(a)
(c)
(b)
(d)
J Dis Rela Su g228
consolida ion, eaching a mean o 165±21.2
( ange, 150 o 180) days. Good adiological
consolida ion was achie ed in he es o pa ien s on
86.4±19.2 ( ange, 46 o 105) days wi h no s a is ically
signi ican di e ences be ween he g oups.
Rega ding sex, he e we e no signi ican
di e ences in consolida ion (p=0.858), as well as
in he ype o pseuda h osis (p=0.43). The mean
pos ope a i e ollow-up was 2.98±1.82 yea s.
The QuickDASH sco e was 0 poin s in all pa ien s.
Taking in o accoun he i s eigh i ems abou he
abili y o do ac i i ies, all pa ien s p esen ed comple e
shoulde mobili y and abili y o do hem. None o he
pa ien s had pain.
The emo al o he ma e ial in G oup A occu ed
a a mean du a ion o ou ( ange, 3 o 5) weeks,
while he pa ien s e u ned o he ope a ing oom
a a mean du a ion o 14.88 ( ange, 10 o 22) mon hs
in G oup B, as long as bone healing was obse ed
on X- ay. The unc ional and quali y o li e esul s
du ing ollow-up we e also sa is ac o y.
DISCUSSION
Congeni al pseuda h osis o he cla icle has a e y
low incidence. In he li e a u e, he e a e ew s udies
wi h long- e m se ies[12,15,16] and he a icle by Kim
e al.[15] is one o hem wi h he longes ollow-up in
47 cases.
Cu en ly, he e is no consensus on he mos
op imal me hod and iming o ea men . Some
au ho s ha e sugges ed a conse a i e ea men , i
he e a e no symp oms,[10,11] while o he s ecommend
su ge y, ega dless o he absence o symp oms.[17,18]
Su gical ea men can be es ablished o cosme ic
appea ance, d ooping o he shoulde , discom o
in he a ea, pain o unc ional symp oms[7] o o
a oid ho acic ou le synd ome o o he ascula
pa hologies.[19] In gene al, in all s udies, he
pa ien - epo ed su ge y ou comes a e a o able.
The mean age o in e en ion may be delayed
in some cases due o he pa en ’s decision who a e
unwilling o an ea ly su gical ea men (<6 yea s
o age) o hei asymp oma ic child en;[18] howe e ,
o e all, many au ho s ecommend pe o ming he
ope a ion a he age o h ee o six yea s.[4,7,8,15,20,21] In
ou s udy, he mean age was 4.43 yea s, consis en
wi h p e ious indings.[2-12] One o he easons o
his is he low a e o nonunion in his g oup o age,
compa ed o child en 18 mon hs o olde .[15]
The e a e di e en ixa ion me hods used in he
ea men including p ese a ion o he pe ios al
slee e and ixa ion wi h non-abso bable su u e
alone in child en unde 18 mon hs o age,[15] sc ews
alone o ully h eaded pins.[22] Howe e , pla es
( econs uc ion, comp ession o ana omical) and
K-wi es a e he mos equen ly used me hods.
Al hough he e a e se e al epo s ad oca ing
K-wi e ixa ion,[8,12,17] many o hem we e w i en be o e
he mos common use o ana omical and locking
pla es. Complica ions wi h K-wi es ixa ion include
pin- ac in ec ions, b eakages, delayed unions[12]
and mig a ion aking in o accoun he p oximi y o
subcla ian a e y and b achial plexus.[22] The e a e
no published cases in which he subcla ian a e y
o b achial plexus ha e been inju ed wi h he use o
pla e ixa ion.
The use o bone g a is conside ed impo an in
he ea men o CPC o inc ease he union a e.[16,21]
The use o allogenic g a has been desc ibed,[12,18]
and a combina ion o local au og a and allog a
has been epo ed, as well.[22] Howe e , i is p e e ed
au ologous iliac c es bone hanks o i s g ea e
biological bene i s,[1,16] aking in o accoun a emained
and p olonged pain in he dono a ea. To sol e
his complica ion, Abdellaoui e al.[23] p oposed a
modi ied Masquele echnique, as an al e na i e,
o ob ain comple e union o he ocus wi hou g a
in e posi ion and wi h minimal aes he ic de ec .
In ou s udy, he adiological e idence o
consolida ion was ob ained a a mean du a ion
o 103.8±39.1 days, consis en wi h he s udies o
Chand an e al.[20] and E l e al.[24]
Sal age p ocedu es a e su gical ea men
complica ions consis in mos o he cases o e ision
su ge y, epea ing esec ion o he pseuda h osis,
and placemen o bone g a and in e nal ixa ion;
howe e , he e a e some epo s using ascula ized
bone g a ing[25] o ascula ized medial emo al
condyle lap,[26] al hough hey can ne e be conside ed
he i s choice in he managemen o CPC.
The main limi a ion o ou s udy lies in i s
e ospec i e design wi h Le el o E idence IV,
wi hou a con ol g oup o conse a i e ea men .
In addi ion, he small sample size did no allow o
many s a is ical compa isons o subg oup analyses.
Despi e hese limi a ions, he cu en s udy de ails
ea men op ions o hese pa ien s and imp o es ou
unde s anding o he esul s o su gical ea men .
Ou case se ies is also compa able wi h p e ious
published s udies.
As a esul o ou expe ience and he li e a u e
e iew, we sugges ha he e is no clinical di e ence
be ween ea lie e sus la e ope a ion; howe e , we
ad ise an ea ly su gical ea men pe o ming a

Su gical ea men o congeni al pseudoa h osis o he cla icle
229
e eshmen o he pseuda h osis ocus, using
always au ologous bone g a in a ophic CPC o
when a gap appea s be ween he agmen s, and
s able ixa ion. Be ween ou o six yea s o age, i
seems o be easonable o achie e a good ixa ion
and adap a ion o a pla e acco ding o he size o
he cla icle. I can also a oid di icul ies which
may occu in olde child en wi h a la ge size o
he gap and a possible longe ime o heal. This
app oach may p e en he p oblems associa ed wi h
an abno mal ana omy o he cla icle. Rega ding
ixa ion, based on ou small coho , i is di icul o
each a i m conclusion; howe e , ou p ac ice has
changed om he K-wi es o pla e ixa ion as he
i s choice o ea men .
In conclusion, he scheduled ea ly su gical
indica ion (<6 yea s o age) based on e eshmen
o he pseuda h osis ocus, wi h bone au og a
in e posi ion when a gap exis s, and ixa ion a e
eliable al e na i es wi h a o able clinical and
adiological esul s in he mid- and long- e m. I
also yields minimal complica ions and imp o es
aes he ics, a oiding possible u u e p oblems. We
ecommend su gical ea men as o ou yea s o age
using pla e ixa ion as he i s op ion.
Decla a ion o con lic ing in e es s
The au ho s decla ed no con lic s o in e es wi h espec o
he au ho ship and/o publica ion o his a icle.
Funding
The au ho s ecei ed no inancial suppo o he esea ch
and/o au ho ship o his a icle.
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