Atypical Cogan Syndrome Featuring Orbital Myositis and Dacryoadenitis.
Journal
Ophthalmic plastic and reconstructive surgery
ISSN: 1537-2677
Titre abrégé: Ophthalmic Plast Reconstr Surg
Pays: United States
ID NLM: 8508431
Informations de publication
Date de publication:
Historique:
pubmed:
30
9
2020
medline:
25
5
2021
entrez:
29
9
2020
Statut:
ppublish
Résumé
A 39-year-old male presented with bilateral hearing loss and progressive left eye vision loss over a 14-month period. The development of systemic symptoms including arthralgias, enlarged lymph nodes, and profound leg weakness, prompted a workup for lymphoproliferative disease, infection, and autoimmune inflammatory conditions which was unrevealing. Subsequently, the right visual acuity declined from 20/25 to 20/70 and the left to hand motions due to corneal interstitial keratitis. There was limitation of left infraduction. Neuroimaging revealed dural thickening of the internal auditory canals, cavernous sinuses, cerebellum, and along the optic nerves. There was fusiform enhancing enlargement of the left inferior and medial rectus muscles and pathologic enlargement of the left lacrimal gland. Biopsy of the left lacrimal gland and left inferior rectus revealed fibrosis and lymphocytic infiltration. The patient was diagnosed with atypical Cogan syndrome and treated with oral prednisone, with improvement in visual acuity of the right eye, motility of the left eye, and systemic weakness.
Identifiants
pubmed: 32991499
pii: 00002341-202105001-00050
doi: 10.1097/IOP.0000000000001835
doi:
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
S160-S162Informations de copyright
Copyright © 2021 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.
Déclaration de conflit d'intérêts
The authors have no financial or conflicts of interest to disclose.
Références
Cogan DG.Syndrome of nonsyphilitic interstitial keratitis and vestibuloauditory symptoms. Arch Ophthalmol. 1945; 33:144–149
Haynes BF, Kaiser-Kupfer MI, Mason P, et al.Cogan syndrome: Studies in thirteen patients, long-term follow-up, and a review of the literature. Medicine (Baltimore). 1980; 59:426–441
Grasland A, Pouchot J, Hachulla E, et al.; Study Group for Cogan’s Syndrome. Typical and atypical Cogan’s syndrome: 32 cases and review of the literature. Rheumatology (Oxford). 2004; 43:1007–1015
Gluth MB, Baratz KH, Matteson EL, et al.Cogan syndrome: A retrospective review of 60 patients throughout a half century. Mayo Clin Proc. 2006; 81:483–488
Shahid FL, Mukherjee R, Knapp C.Cogan’s syndrome associated with orbital inflammation. Orbit. 2013; 32:206–207
McNab AA.Orbital myositis: A comprehensive review and reclassification. Ophthalmic Plast Reconstr Surg. 2020; 36:109–117
Shibuya M, Fujio K, Morita K, et al.Successful treatment with tocilizumab in a case of Cogan’s syndrome complicated with aortitis. Mod Rheumatol. 2013; 23:577–581
Vollertsen RS, McDonald TJ, Younge BR, et al.Cogan’s syndrome: 18 cases and a review of the literature. Mayo Clin Proc. 1986; 61:344–361
Espinoza GM, Wheeler J, Temprano KK, Keller AP.Cogan’s syndrome: Clinical presentation and update on treatment. Curr Allergy Asthma Rep. 2020; 20:46
Durtette C, Hachulla E, Resche-Rigon M, et al.; SNFMI and CRI. Cogan syndrome: characteristics, outcome and treatment in a French nationwide retrospective study and literature review. Autoimmun Rev. 2017; 16:1219–1223