Folia Mo phol.
Vol. 79, No. 4, pp. 823–828
DOI: 10.5603/FM.a2019.0138
Copy igh © 2020 Via Medica
ISSN 0015–5659
jou nals. iamedica.pl
O R I G I N A L A R T I C L E
823
Add ess o co espondence: D . A.I. Cisne os, Ana omy and His ology Depa men , School o Medicine, Za agoza Uni e si y, Spain,
e-mail: aicisne @uniza .es
Associa ion be ween supe io semici cula
canal dehiscence and o he dehiscences
in empo al bone
J. Why e1, A.I. Cisne os1, A. Ga cia-Ba ios1, J. F aile2, A. Why e3, R. C o e o4, M. Lahoz1
1Ana omy and His ology Depa men , School o Medicine, Za agoza Uni e si y, Spain
2Pa hology Depa men , Miguel Se e Hospi al, Spain
3Pa hology Depa men , School o Ve e ina y, Za agoza Uni e si y, Spain
4Bilbao Clinic, Spain
[Recei ed: 14 Oc obe 2019; Accep ed: 3 Decembe 2019]
Backg ound: The s udy o he associa ion be ween supe io semici cula canal
and o he dehiscences in he empo al bone.
Ma e ials and me hods: We ha e s udied compu ed omog aphy o adiologically
diagnosed people wi h supe io o pos e io semici cula canal dehiscences, in ou
heal h cen es. In addi ion, we ha e s udied one isola ed human empo al bone,
one skull and one cada e head belonging o he collec ion o he Depa men o
Human Ana omy and His ology o he Uni e si y o Za agoza ha had dehiscence
in he supe io semici cula canal.
Resul s: The mos equen associa ion ha we obse ed was be ween supe io
semici cula canal dehiscence and egmen ympani dehiscence (37.33%). Th ee
cases ( wo clinical cases and one isola ed empo al bone) showed mul iple associ-
a ed dehiscences ( egmen ympani, mas oid an um, pos e io semici cula canal,
in e nal audi o y canal, glenoid ca i y, ympanum bone and genicula e ganglion)
associa ed wi h supe io semici cula canal dehiscence
Conclusions: When he supe io semici cula canal dehiscence is associa ed o
o he in he pe ous bone ( egmen ympani, mas oid an um, pos e io semici cula
canal, in e nal audi o y canal) could be g ouped in o he same synd ome called
“o ic capsule synd ome”, since hey ha e he same o igin and common ae iology
(o ic capsule). (Folia Mo phol 2020; 79, 4: 823–828)
Key wo ds: bony laby in h, o ic capsule, bony de ec s g oup,
hi d window
INTRODUCTION
A miles one in he s udy o supe io semici cu-
la canal (SSC) pa hology was he manusc ip by
Mino [22] in 1998, who i s desc ibed he p es-
ence o dehiscence in he canal. This dehiscence is
a a e and uncommon pe iphe al es ibulopa hy,
which is cha ac e ised by a lack o bone co e age
o he SSC in he a ea closes o he du a o he
middle ce eb al ossa. Since ha da e, s udies
ha e been conduc ed o in es iga e i s p e alence,
pa hophysiology, clinical a iables, diagnosis and
su gical p ocedu es.
The e a e cu en ly wo heo ies abou he gen-
esis o dehiscence: congeni al (s op o delay in he
ossi ica ion o he canal du ing i s de elopmen ) and
acqui ed (bone laye up u e co e ing he canal as
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Folia Mo phol., 2020, Vol. 79, No. 4
a esul o ano he e en , as inc eased in ac anial
p essu e, auma, e en a e childbi h o coi us).
The associa ion o SSC dehiscence wi h o he ea
diseases [4, 9, 27] and o he dehiscences p oduced
in he bone laby in h is cu en ly being in es iga ed.
In his line, associa ions be ween dehiscence o
he SSC and dehiscence o he pos e io semici cu-
la canal ha e been desc ibed [6, 15, 18, 20, 21].
Bo h dehiscences show a simila , nonspeci ic clini-
cal p esen a ion, some imes he audi o y symp oms
(hypoacusis, inni us) and some imes he es ibula
ones (dizziness, nys agmus, Tullio phenomenon) p e-
domina e; hey may no e en p esen any.
The e may also be cases whe e he e a e dehis-
cences in o he s uc u es such as he egmen ym-
pani [1, 5, 24], genicula e ganglion [10, 13, 17] and
in e nal audi o y canal [19].
The objec i e o his s udy is o de e mine adio-
logically on he one hand, he possible associa ions
be ween dehiscence in he supe io and pos e io
semici cula canal, and on he o he , bo h dehiscences
wi h o he mo phological al e a ions in he empo al
bone (glenoid ca i y and in e nal audi o y canal).
MATERIALS AND METHODS
We ha e s udied compu ed omog aphy (CT) o
adiologically diagnosed people wi h supe io o pos-
e io semici cula canal dehiscences, in ou heal h
cen es: Basu o and C uces Hospi al, Gene al Hos-
pi al o he De ence and Miguel Se e Hospi al in
Za agoza. An in o med consen o m was signed
by e e y pa icipa ing pa ien . All he pa ien s ac-
knowledged ha hey canno be iden i ied ia he
pape ; he esea che s ully anonymised hem. The
e hics commi ee o each cen e app o ed his s udy
in acco dance wi h he guidelines o he Helsinki
Decla a ion o 1983.
The s udies ha e been pe o med wi h mul i-splice
helical CT equipmen (Philips B illiance 6), ob aining
he images on he axial plane and wi h he pa ien ’s
neck in hype ex ension o a oid di ec adia ion dam-
age o c ys alline. La e , co onal econs uc ions ha e
been ca ied ou in all he cases, in he plane o he
SSC o each ea (Pöschl plane) and in axial planes
o he pos e io semici cula canals. The oo o he
glenoid ca i y has been s udied wi h sagi al plane
econs uc ions.
The “ aw da a” ha e been econs uc ed using
a bone algo i hm.
The ollowing adiological p o ocols o he acqui-
si ion and o ma ing o he images ha e been used:
2 × 0.6 mm collima ion, 0.65 mm splice hickness,
0.32 mm splice inc ease, 0.75 s o a ion ime, 0.38 pi ch,
120 CV, 300 mAs, 1024 × 1024 ma ix, 180 mm
ield o ision, 0.5 mm econs uc ion hickness and
0.5 mm econs uc ion inc ease.
The CT scans we e upda ed by 3 adiologis s wi h
mo e han 10 yea s o expe ience in neu o adiology
and e iewed by he signa o ies o he manusc ip ,
all o hem wi h ex ensi e expe ience in dehiscence
o semici cula canals.
RESULTS
We ha e pe o med a adiological s udy on a o al
o 61 pa ien s (20 men, 41 women) wi h an a e age
age o 55.98 yea s ( anging 2–89), in e ms o he
possible associa ions be ween (supe io and pos e i-
o ) e ical semici cula canal dehiscence, and o he
dehiscences loca ed in he empo al bone. Two o he
pa ien s, who p esen ed unila e al SSC dehiscence,
we e sis e s.
Se en y one cases o SSC dehiscence and 4 o pos-
e io semici cula canal dehiscence we e de ec ed.
Thi y-one dehiscences we e de ec ed on he igh
side in he SSC, 20 on he le side, and bila e al
dehiscences (28.16%) we e obse ed in 10 pa ien s
(20 cases) (Table 1). Two dehiscences we e de ec ed
on he igh side in he pos e io semici cula canal,
and ano he wo on he le side, and he e was one
bila e al case.
The mos equen associa ion ha we obse ed
was be ween SSC dehiscence and egmen ympa-
ni dehiscence, which was e i ied in a o al o 28
(39.44%) cases.
Two cases p esen ed mul iple associa ed dehis-
cences (Fig. 1). Thus, he i s case p esen ed bila e al
supe io semici cula dehiscences (Fig. 1A), bila e al
egmen ympani dehiscences, and dehiscence in he
glenoid ca i y o he empo omandibula join on he
Table 1. Dis ibu ion o supe io semici cula canal (SSC)
dehiscence and pos e io semici cula canal (PSC)
dehiscence
Righ side Le side Bila e al
SSC 31/71 (43.67%) 20/71 (28.17%) 20/71 (28.16%)
PSC 1/4 (25%) 1/4 (25%) 2/4 (50%)
825
J. Why e e al., Associa ion be ween SSC dehiscence and o he s
igh side (Fig. 1B). The o he cases showed bila e al
supe io and pos e io semici cula canal dehiscences
(Fig. 1D), bila e al egmen ympani dehiscences, and
dehiscence o he oo o he in e nal audi o y canal
(Fig. 1C) on he igh side (Table 2).
DISCUSSION
Supe io semici cula canal dehiscence, as a s u-
c u al cause o dizziness and hypoacusis, is only
20 yea s old [22], so he e a e s ill knowledge gaps
ela ed o his pa hology. Fi e yea s la e i would be
K ombach e al. [14] who desc ibed pos e io semi-
ci cula canal dehiscence.
The i s au ho s who desc ibed associa ions be-
ween de ec s o he bony co e age o he SSC and
o he pa s o he bony laby in h was Mino [23] who
obse ed an associa ion be ween egmen dehiscence
and he SSC. One yea la e , Gianoli [10] unde lined
he high incidence o pa ien s who also p esen ed a-
diog aphic supe io canal dehiscence wi h genicula e
ganglion dehiscence. La e , Manza i and Modugno
[18] and Chen e al. [3] desc ibed isola ed cases o SSC
dehiscences associa ed wi h pos e io canal dehis-
cences, he o me in adul s, and he la e in child en.
In la ge se ies o pe sons wi h SSC dehiscence, he
a ec a ion was ound o be bila e al, in pe cen ages
ha a ied be ween 16% and 37%. These da a a e
e y simila o hose obse ed by us, as we ha e eg-
is e ed an incidence o 34.48%.
Figu e 1. Di e en associa ions be ween supe io semici cula canal dehiscence wi h o he s in he empo al bone; A. Supe io semici cula
canal dehiscence (black a ow); B. Tegmen ympani (black a ow), glenoid ca i y (whi e a ow); C. Tegmen ympani (black a ow), in e nal
audi o y canal (IAC; whi e a ow); D. Pos e io semici cula canal dehiscence (PSCD; whi e a ow).
Table 2. Associa ion o empo al bone dehiscences
TTD IACD GFD
SSCD 28/71 (39.44%) 1/71 (1.41%) 1/71 (1.41%)
PSCD 2/4 (50%) 1/4 (25%) 0/4 (0%)
SSCD — supe io semici cula canal dehiscence; PSCD — pos e io semici cula canal
dehiscence; TTD — egmen ympani dehiscence; IACD — in e nal audi o y canal dehi-
scence; GFD — glenoid ca i y dehiscence
A B
C D
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Folia Mo phol., 2020, Vol. 79, No. 4
The p esence o bila e al pos e io semici cula ca-
nal dehiscences is no e y equen , and hey a e lim-
i ed o isola ed cases such as hose desc ibed by Saxby
e al. [30] and Bea e al. [2]. We ha e also obse ed
one single isola ed case o bila e al dehiscence, bu
we mus conside ha pos e io semici cula dehis-
cence is much less equen han supe io semici cula
dehiscence, being 3.6% and 0.6%, espec i ely [5].
The associa ion be ween supe io and pos e io ca-
nal dehiscence has been desc ibed by K ombach e al.
[14], who obse ed 8 pa ien s wi h pos e io and SSC
dehiscences. Chen e al. [3] also ob ained 1 case wi h
bo h dehiscences. Gopen e al. [11] obse ed ha
2 o he 12 pa ien s wi h pos e io semici cula canal
dehiscences also p esen ed SSC dehiscences. Russo
e al. [29] desc ibed ha in 3 o he 5 cases wi h pos-
e io semici cula canal dehiscence we e associa ed
o dehiscences in he supe io canal. Saxby e al. [30]
communica ed 1 case o a child wi h bila e al supe io
unila e al and pos e io canal dehiscence. Kundaga i
e al. [15] also p esen ed a case o a pa ien wi h bila -
e al pos e io semici cula canal dehiscence, oge he
wi h igh supe io canal dehiscence. We ound only
1 case in which he 2 dehiscences we e associa ed,
among he 61 pa ien s s udied, which deno es he
low equency o his associa ion since hey we e
selec ed pa ien s.
The mos equen associa ion ound, which has
clinical epe cussion, was wi h he egmen ympani.
C o e o e al. [5] ound a 36.4% incidence o dehis-
cen egmens associa ed wi h SSC dehiscence, and
Nada aja e al. [24] ound 76%. In he e e se s udy,
El Hadi e al. [7] ound a 56.5% incidence o dehis-
cen semici cula canals in pa ien s who p esen ed
e i ied dehiscence o he egmen ympani. We ha e
obse ed his associa ion in 28 o he cases s udies
(37.33%) and we hink ha he explana ion would
be, as p oposed by F aile Rod igo e al. [8], ha bo h
s uc u es ha e a common o igin, he o ic capsule,
and ha he p ima y cen es o he ex e nal o la e al
SSCs, which, when hey g ow, ex end o he base o he
egmen al p olonga ion, collabo a e in he ossi ica ion
o he egmen ympani. In addi ion o his, he ex e nal
pe ios ium laye , which sepa a es he egmen om he
middle c anial ossa, is a con inua ion o he laye ha
co e s he SSC. We ha e no ound in he li e a u e any
manusc ip ha ela es he pos e io semici cula canal
dehiscence wi h he absence o egmen ympani, while
we ha e ound i in in 2 o he 4 cases o pos e io
semici cula canal dehiscence (50%).
The associa ion o SSC dehiscences wi h in e nal
audi o y canal dehiscences has only been desc ibed
in li e a u e by Manza i and Scagnelli [19]. We ha e
obse ed 1 case in which SSC dehiscences we e as-
socia ed wi h in e nal audi o y canal and pos e io
canal dehiscence.
In he li e a u e consul ed, we ha e no obse ed any
associa ion be ween SSC dehiscence and glenoid ca i y
dehiscence o he empo omandibula join o o he
ympanic bone, and only Ku e al. [16] associa es SSC
dehiscence wi h symp oms in he empo omandibula
join , desc ibing he la ening o he condyle, i egu-
la i ies on he su ace and o ma ion o os eophy es, as
adiological indings. We ha e seen hese associa ions
in a adiological s udy which leads us o conside ha
one same cause could ac du ing de elopmen , bo h
in he squamous po ion o he empo al bone and in
he o ic capsule, al hough he o igin is no ye clea .
The e o e, we ha e obse ed ha he cases o
SSC dehiscence associa ed wi h o he empo al bone
dehiscences a e ew and a be ween, ei he loca ed
in he pe ous apophysis, in he squamous po ion o
he empo al bone o in he ympanic bone.
To unde s and he ae iology o mul iple dehiscenc-
es, an emb yological cause ha ac s upon he o ic cap-
sule mus be sough . Thus, Isaacson and V abec [13]
and Pa k e al. [26] obse ed ha he bony o ic cap-
sule was signi ican ly hinne in pa ien s wi h associ-
a ed SSC dehiscence. G acia-Tello e al. [12] concludes
ha he exis ence o supe io semici cula dehiscence
is associa ed wi h he hinning o he bone o he ca-
nal on he opposi e side, and Manza i and Modugno
[18] hink ha he associa ion be ween wo bone
s uc u es ha p esen dehiscences, and de i e om
he o ic capsule, is p esen ed as suppo o he idea
ha i is a congeni al ype diso de .
Nies en e al. [25] in o m o h ee amilies in which
he i s -deg ee ela i es show SSC dehiscences, and
El Hadi e al. [7] egis e ed wo wins in he se ies
ha hey s udied, wi h bo h SSC dehiscence and
egmen ympani dehiscence. These e en s sugges
gene ic ae iology. In ou s udy, we ha e ound SSC
dehiscence in 2 sis e s, which sugges s, in ha case,
a gene ic componen . Howe e , in ou s udies abou
COACH synd ome and in he s udies conduc ed by
Roknic e al. [28], demons a ing a cons an gene ic
diso de associa ed wi h hese dehiscences has no
been possible.
Wackym e al. [31] coins a new e m, “o ic capsule
synd ome”, o de ine he exis ence o associa ed de-
827
J. Why e e al., Associa ion be ween SSC dehiscence and o he s
hiscences be ween he di e en semici cula canals,
and he bases his on he ac ha hese pa hologies
ha e e y simila symp oms, whe he implan ed in
(supe io and pos e io ) e ical canals o in he (la e -
al) ho izon al canal. We would use his e m o when
in he CT scans ind mul iple dehiscences o s uc u es
de i ed om he o ic capsule ( egmen ympani, mas-
oid an um, pos e io semici cula conduc , in e nal
audi o y canal).
We ha e no ound clinical ea u es o dis inguish
pa ien s wi h SSC dehiscence om he pos e io , and
bo h wi h o he empo al bone dehiscences.
CONCLUSIONS
The e o e, and in conclusion, we hink ha SSC
dehiscence may ha e a congeni al cause and be as-
socia ed wi h o he dehiscences, and in ou clinical
p ac ice, we mus seek hese associa ions whene e
we encoun e a diso de in he bony co e age o
he empo al bone, as al eady ecommended when
a egmen ympani o SSC dehiscence is diagnosed.
In ag eemen wi h ou obse a ions, e iews and
a e analysing he emb yological de elopmen , we
conside ha he exis ence o mal o ma ion syn-
d omes in which he e is an associa ion o mul iple
s uc u e dehiscences, bo h o he o ic capsule and
o he empo al bone, is e y likely.
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