Unusual orbital, scleral and choroidal findings in Erdheim-Chester disease: a case report.

Erdheim-Chester disease choroid histiocytosis orbit scleral infiltration

Journal

Orbit (Amsterdam, Netherlands)
ISSN: 1744-5108
Titre abrégé: Orbit
Pays: England
ID NLM: 8301221

Informations de publication

Date de publication:
08 Jul 2024
Historique:
medline: 8 7 2024
pubmed: 8 7 2024
entrez: 8 7 2024
Statut: aheadofprint

Résumé

This case report highlights a unique presentation of Erdheim-Chester Disease (ECD) with bilateral scleral lesions, choroidal infiltration, and extensive sinus involvement. It is the first case report where the diagnosis was confirmed through a scleral biopsy after an initial presentation of a unilateral nodular scleritis. There was a gradual disease progression and ocular examination later revealed bilateral subconjunctival hyperemic lesions and mild exophthalmos, ophthalmoplegia, and extensive choroidal infiltration. Infiltration of the frontal and maxillary sinus was present and extended into the nasal cavity and both orbits. The diagnostic work-up is described in detail. Current treatment options are analyzed. It is emphasized that the ophthalmologist can play a crucial role in the diagnosis of ECD, given the substantial prevalence of orbital and ocular symptoms. The overall prognosis for ECD remains unfavorable, particularly in cases with orbital involvement. This case underscores the complexity and importance of a multidisciplinary approach in managing ECD.

Identifiants

pubmed: 38976616
doi: 10.1080/01676830.2024.2373791
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

1-6

Auteurs

M Meyers (M)

Department of Ophthalmology, Antwerp University Hospital, Edegem, Belgium.

I Vermeiren (I)

Department of Ophthalmology, Antwerp University Hospital, Edegem, Belgium.

A Dendooven (A)

Department of of Anatomical Pathology, Antwerp University Hospital, Edegem, Belgium.

N Lauwers (N)

Department of Ophthalmology, Antwerp University Hospital, Edegem, Belgium.

J Goemaere (J)

Department of Ophthalmology, Antwerp University Hospital, Edegem, Belgium.

Classifications MeSH