Granulomatosis with Polyangiitis Presenting with Bilateral Orbital Apex Syndrome.

Granulomatosis with polyangiitis Myeloperoxidase-anti-neutrophil cytoplasmic antibody Orbital apex syndrome

Journal

Case reports in ophthalmology
ISSN: 1663-2699
Titre abrégé: Case Rep Ophthalmol
Pays: Switzerland
ID NLM: 101532006

Informations de publication

Date de publication:
Historique:
received: 21 05 2020
accepted: 20 07 2020
entrez: 22 2 2021
pubmed: 23 2 2021
medline: 23 2 2021
Statut: epublish

Résumé

We report a rare case of granulomatosis with polyangiitis (GPA) presenting with bilateral orbital apex syndrome (OAS). A 73-year-old woman with a history of endoscopic sinus surgery for ethmoidal sinusitis experienced a sudden decrease in visual acuity (VA) of both eyes. At the initial examination, her VA had decreased to 0.01 in the right eye and 0.03 in the left eye, and eye movement in both eyes was mildly limited in all directions. Visual field tests of both eyes showed a large central scotoma. Laboratory tests revealed an elevation of myeloperoxidase-anti-neutrophil cytoplasmic antibody. Facial computed tomography demonstrated a thickened mucosal membrane in the entire ethmoidal sinus, and the posterosuperior walls of Onodi cells filled with infiltrative lesions had thinned. Orbital magnetic resonance imaging showed severe inflammation in the orbital apex. From these clinical findings, the patient was diagnosed with GPA presenting with OAS associated with ethmoid sinusitis. Emergent endoscopic sinus surgery was performed for biopsy and debridement of the ethmoidal and sphenoid sinusitis to decompress the optic nerve. One day after endoscopic sinus surgery, the patient's VA and visual field were improved, and steroid pulse therapy was commenced postoperatively. Four days later, VA had recovered to 1.0 in both eyes, and eye movement and visual field had were improved. Although OAS is a rare manifestation, early surgical treatment should be considered when the orbital lesion presents as risk of rapid deterioration of visual function in patients with GPA.

Identifiants

pubmed: 33613248
doi: 10.1159/000510329
pii: cop-0012-0032
pmc: PMC7879294
doi:

Types de publication

Case Reports

Langues

eng

Pagination

32-40

Informations de copyright

Copyright © 2021 by S. Karger AG, Basel.

Déclaration de conflit d'intérêts

None of the authors have any conflicts of interest.

Références

Ophthalmol Ther. 2019 Jun;8(2):227-234
pubmed: 30875067
J Laryngol Otol. 2007 Jul;121(7):653-8
pubmed: 17140461
Ann Rheum Dis. 2007 Feb;66(2):222-7
pubmed: 16901958
BMJ Case Rep. 2013 Dec 05;2013:
pubmed: 24311409
Am J Med. 1977 Jul;63(1):131-41
pubmed: 327802
Rheumatol Int. 2017 Nov;37(11):1779-1788
pubmed: 28956128
Medicine (Baltimore). 2007 Sep;86(5):269-277
pubmed: 17873756
Singapore Med J. 2008 Oct;49(10):e259-62
pubmed: 18946592
J Laryngol Otol. 2008 Nov;122(11):1257-9
pubmed: 18397545
Ann Intern Med. 1992 Mar 15;116(6):488-98
pubmed: 1739240
Ann Rheum Dis. 2016 Sep;75(9):1583-94
pubmed: 27338776
Eye Brain. 2019 Dec 12;11:63-72
pubmed: 31849556
Ann Intern Med. 1983 Jan;98(1):76-85
pubmed: 6336643
Otolaryngol Head Neck Surg. 1995 Jun;112(6):758-62
pubmed: 7777366
Ophthalmology. 1983 Mar;90(3):279-90
pubmed: 6602963
Orbit. 2011 Jan;30(1):24-6
pubmed: 21281075
Ocul Immunol Inflamm. 2013 Apr;21(2):115-8
pubmed: 23252657
J Neurol Sci. 2016 Sep 15;368:175-7
pubmed: 27538627

Auteurs

Yusuke Murai (Y)

Division of Ophthalmology, Department of Surgery, Kobe University Graduate School of Medicine, Kobe, Japan.

Takuji Kurimoto (T)

Division of Ophthalmology, Department of Surgery, Kobe University Graduate School of Medicine, Kobe, Japan.

Sotaro Mori (S)

Division of Ophthalmology, Department of Surgery, Kobe University Graduate School of Medicine, Kobe, Japan.

Kaori Ueda (K)

Division of Ophthalmology, Department of Surgery, Kobe University Graduate School of Medicine, Kobe, Japan.

Mari Sakamoto (M)

Division of Ophthalmology, Department of Surgery, Kobe University Graduate School of Medicine, Kobe, Japan.

Kengo Akashi (K)

Division of Rheumatology and Clinical Immunology, Department of Internal Medicine, Kobe University Graduate School of Medicine, Kobe, Japan.

Yuko Yamada-Nakanishi (Y)

Division of Ophthalmology, Department of Surgery, Kobe University Graduate School of Medicine, Kobe, Japan.

Makoto Nakamura (M)

Division of Ophthalmology, Department of Surgery, Kobe University Graduate School of Medicine, Kobe, Japan.

Classifications MeSH