Osteopathia striata with cranial sclerosis causing a compressive optic neuropathy.

AMER1 mutation Osteopathia striata with cranial sclerosis compressive optic neuropathy disc edema skeletal dysplasia

Journal

Ophthalmic genetics
ISSN: 1744-5094
Titre abrégé: Ophthalmic Genet
Pays: England
ID NLM: 9436057

Informations de publication

Date de publication:
10 2023
Historique:
medline: 22 9 2023
pubmed: 30 11 2022
entrez: 29 11 2022
Statut: ppublish

Résumé

Osteopathia striata combined with cranial sclerosis (OS-CS) is an inherited skeletal dysplasia that manifests with macrocephaly, orofacial abnormalities, thickened craniofacial bones, and vertically oriented radiodensities of the long bones. Here, we present a severe case of OS-CS in a 4-year-old girl causing optic neuropathy as shown by radiographic evidence, ophthalmic findings, and histopathology. Previous genetic testing in this patient revealed a

Sections du résumé

BACKGROUND
Osteopathia striata combined with cranial sclerosis (OS-CS) is an inherited skeletal dysplasia that manifests with macrocephaly, orofacial abnormalities, thickened craniofacial bones, and vertically oriented radiodensities of the long bones.
CASE REPORT
Here, we present a severe case of OS-CS in a 4-year-old girl causing optic neuropathy as shown by radiographic evidence, ophthalmic findings, and histopathology. Previous genetic testing in this patient revealed a

Identifiants

pubmed: 36446546
doi: 10.1080/13816810.2022.2144902
doi:

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

496-500

Auteurs

Jennifer C W Hu (JCW)

Harkness Eye Institute, Department of Ophthalmology, Columbia University Irving Medical Center, New York, NY, USA.

Aliaa H Abdelhakim (AH)

Harkness Eye Institute, Department of Ophthalmology, Columbia University Irving Medical Center, New York, NY, USA.

Victoria S North (VS)

Harkness Eye Institute, Department of Ophthalmology, Columbia University Irving Medical Center, New York, NY, USA.

Maria D Garcia (MD)

Harkness Eye Institute, Department of Ophthalmology, Columbia University Irving Medical Center, New York, NY, USA.

Marc J Lustig (MJ)

Pediatric Ophthalmic Consultants of New York, New York, New York, USA.

Michael Kazim (M)

Harkness Eye Institute, Department of Ophthalmology, Columbia University Irving Medical Center, New York, NY, USA.

Jeffrey G Odel (JG)

Harkness Eye Institute, Department of Ophthalmology, Columbia University Irving Medical Center, New York, NY, USA.

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