Co esponding au ho : Beni Kuyukunu
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Occipi al En hesophy e in Axial Spondyloa h i is: Repo o wo cases
Beni Kuyukunu *, Soukaina Zahe , Kaw a Nassa , Wassima Ridah, Ahlam Aje ouassi, Wa ae Rachidi and
Saadia Janani
Depa men o Rheuma ology, Ibn Rochd Uni e si y Hospi al, Facul y o Medicine and Pha macy, Hassan II Uni e si y,
Casablanca, Mo occo.
Wo ld Jou nal o Ad anced Resea ch and Re iews, 2025, 27(02), 1730-1732
Publica ion his o y: Recei ed on 12 July 2025; e ised on 23 Augus 2025; accep ed on 25 Augus 2025
A icle DOI: h ps://doi.o g/10.30574/wja .2025.27.2.2938
Abs ac
Axial spondyloa h i is (axSpA) is a ch onic in lamma o y condi ion p ima ily a ec ing he sac oiliac join s and spine,
wi h en hesi is as i s hallma k ea u e. While en hesi is commonly in ol es lowe limb si es, c anial localiza ions, such
as he ex e nal occipi al p o ube ance, a e excep ionally a e. We epo wo cases o young male pa ien s wi h long-
s anding axSpA p esen ing wi h occipi al en hesophy es. A 34-yea -old male wi h ju enile-onse axSpA exhibi ed
bila e al sac oilii is, coxi is, and ce ical s i ness. A 33-yea -old male wi h ch onic polya h i is since adolescence
showed ad anced axial and pe iphe al s uc u al damage. Ce ical adiog aphs in bo h e ealed an en hesophy e a he
ex e nal occipi al p o ube ance. Labo a o y indings indica ed ele a ed in lamma o y ma ke s, wi h HLA-B27 posi i i y
in one pa ien . T ea men wi h TNF inhibi o s (e ane cep and golimumab) esul ed in clinical imp o emen . These
cases highligh he impo ance o comp ehensi e imaging o iden i y a e en heseal si es in axSpA, enabling ailo ed
he apeu ic s a egies.
Keywo ds: Axial Spondyloa h i is; Occipi al En hesophy e; Ra e Localiza ion; Radiog aphy; Ce ical Spine
1. In oduc ion
Axial spondyloa h i is (axSpA) is a ch onic in lamma o y heuma ic disease p ima ily a ec ing he sac oiliac join s and
spine. En hesi is, in lamma ion a he si e o endon o ligamen a achmen o bone, is a hallma k o he disease and
may lead o new bone o ma ion such as syndesmophy es o en hesophy es En hesophy es a e mos equen ly
desc ibed a pe iphe al si es including he Achilles endon, plan a ascia, and pa ella endon [1,2]. Axial en hesophy es
occu p edominan ly a e eb al endpla es and spinal ligamen s. In ol emen o he ex e nal occipi al p o ube ance,
co esponding o he inse ion o he ligamen um nuchae and neck ex enso muscles, is excep ionally a e and sca cely
documen ed in he li e a u e [3,4]. We desc ibe wo cases o occipi al en hesophy e in pa ien s wi h long-s anding
axSpA, illus a ing he clinical p esen a ion, imaging ea u es, and he apeu ic esponse. This epo desc ibes wo cases
o axSpA wi h occipi al en hesophy es, emphasizing he need o ecognize a ypical en heseal localiza ions o enhance
diagnosis and managemen .
2. Case Repo s
2.1. Case 1
A 34-yea -old male, ex-smoke (7 pack-yea s, qui 2 yea s p io ), wi h a his o y o hepa ic hyda id cys su ge y in 2010,
p esen ed wi h in lamma o y ce ico-lumba pain, hip pain, and bu ock pain since age 11. Examina ion e ealed
es ic ed hip mobili y and incomple e ce ical spine synd ome. Labo a o y es s showed ele a ed CRP (25 mg/L) and
Wo ld Jou nal o Ad anced Resea ch and Re iews, 2025, 27(02), 1730-1732
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ESR (52 mm/h). Pel ic adiog aphy con i med bila e al s age IV sac oilii is and coxi is. Ce ical spine adiog aphy
demons a ed loss o ce ical lo dosis, C7 e eb al squa ing, and an en hesophy e a he ex e nal occipi al
p o ube ance (Figu e 1). Ju enile-onse axSpA wi h bila e al coxi is was diagnosed. E ane cep (50 mg/week) was
ini ia ed a e NSAID ailu e, esul ing in clinical imp o emen .
Figu e 1 La e al ce ical spine adiog aph showing an en hesophy e a he ex e nal occipi al p o ube ance
2.2. Case 2
A 33-yea -old male, ollowed o ch onic polya h i is since age 16, p esen ed wi h in lamma o y back pain, bu ock
pain, and heel pain. Examina ion e ealed di use spinal s i ness, sac oiliac ende ness, and a h i is o he hips, knees,
ankles, and p oximal in e phalangeal join s. Imaging showed bila e al s age IV sac oilii is, hip join na owing, and
en hesopa hies a he ischial ube osi ies, pubic symphysis, and g ea e ochan e s. Ce ical spine adiog aphy
e ealed an occipi al en hesophy e (Figu e 2). Labo a o y es s indica ed CRP a 12.8 mg/L and HLA-B27 posi i i y.
Golimumab was ini ia ed o ad anced s uc u al damage and pe sis en in lamma ion, wi h a a o able esponse.
Figu e 2 La e al ce ical spine adiog aph demons a ing an en hesophy e a he ex e nal occipi al p o ube ance
3. Discussion
En hesophy es de elop a endon o ligamen inse ion si es as a consequence o ch onic en hesi is ollowed by
abno mal bone epai . In axSpA, his p ocess is d i en by pe sis en in lamma ion a he bone–en hesis in e ace,
leading o ib oca ilage me aplasia, calci ica ion, and ossi ica ion.
Wo ld Jou nal o Ad anced Resea ch and Re iews, 2025, 27(02), 1730-1732
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Pe iphe al en heses a e mo e o en a ec ed han axial o c anial si es. Occipi al localiza ion is a e, possibly due o he
lowe mechanical and in lamma o y bu den compa ed o weigh -bea ing en heses [1,2]. The ex e nal occipi al
p o ube ance se es as he inse ion poin o he ligamen um nuchae and mul iple ce ical muscles ( apezius, splenius
capi is, occipi alis), which may be subjec ed o combined in lamma o y and mechanical s ess.
The li e a u e con ains e y ew epo s o occipi al en hesophy e in axSpA [3,4]. In mos cases, i occu s in ad anced
disease s ages o in pa ien s wi h insu icien con ol o in lamma ion. In ou pa ien s, bo h had mo e han 10 yea s o
disease e olu ion and se e e s uc u al changes, sugges ing ha occipi al in ol emen may be a la e mani es a ion.
De ec ion equi es a ge ed imaging, as s anda d spinal adiog aphs may no include he skull base. Occipi al pain,
ende ness, o swelling in axSpA pa ien s should p omp la e al ce ical o skull-base imaging.
Bo h o ou pa ien s esponded symp oma ically o an i-TNF he apy, consis en wi h he known e icacy o biologics on
en heseal in lamma ion [5,6]. Howe e , es ablished ossi ied lesions did no eg ess, unde sco ing he impo ance o
ea ly diagnosis and agg essi e in lamma ion con ol o p e en i e e sible damage. This epo emphasizes he need
o conside a e en heseal si es in axSpA e alua ion, bo h o accu a e disease assessmen and o ailo ed imaging
s a egies.
4. Conclusion
Occipi al en hesophy es a e a a e mani es a ion o axial spondyloa h i is, ypically obse ed in ad anced s ages o he
disease. Thei de ec ion, o en inciden al, highligh s he impo ance o comp ehensi e imaging including he uppe
ce ical spine and skull base o ully assess disease ex en
Compliance wi h e hical s anda ds
Acknowledgmen s
We hank all who con ibu ed o his wo k. No unding was ecei ed.
Disclosu e o con lic o in e es
The au ho decla es no con lic o in e es .
S a emen o in o med consen
In o med consen was ob ained om all indi idual pa icipan s included in he s udy.
Re e ences
[1] Benjamin M, McGonagle D. The en hesis o gan concep and i s ele ance o he spondyloa h opa hies. Ad Exp
Med Biol. 2009;649:57–70.
[2] Tubbs RS, Sal e EG, Oakes WJ. The ex e nal occipi al p o ube ance: an ana omical s udy. Clin Ana .
2007;20(6):611–4.
[3] Lo ies RJ, Ha oon N, McInnes IB. En hesi is: pa hophysiology and ea men . Na Re Rheuma ol.
2020;16(12):731–41.
[4] Ba aliakos X, B aun J. Imaging ools in spondyloa h i is: a e iew o he li e a u e. Na Re Rheuma ol.
2015;11(7):431–44.
[5] Ba aliakos X, e al. An i-TNF he apy in ankylosing spondyli is and axial spondyloa h i is. Ann Rheum Dis.
2014;73(4):784–790.
[6] B aun J, Siepe J. The apy o spondyloa h i is: Biological and non-biological op ions. A h i is Res The .
2009;11(3):221
