Bilateral dendriform ulcer leading to an early diagnosis of tyrosinemia type 2.


Journal

Journal of AAPOS : the official publication of the American Association for Pediatric Ophthalmology and Strabismus
ISSN: 1528-3933
Titre abrégé: J AAPOS
Pays: United States
ID NLM: 9710011

Informations de publication

Date de publication:
04 2020
Historique:
received: 18 09 2019
revised: 16 12 2019
accepted: 21 12 2019
pubmed: 6 2 2020
medline: 27 7 2021
entrez: 5 2 2020
Statut: ppublish

Résumé

A 2-year-old girl with a 1-year history of photophobia presented with bilateral eye redness and was found to have dendriform corneal ulcers, which were initially managed with acyclovir ointment, without improvement. Clinical inspection revealed small cutaneous lesions on the fingers and toes. The association of pseudodendritic keratitis with palmo-plantar hyperkeratotic lesions was suspicious for tyrosinemia type 2, which was confirmed by high levels of plasmatic tyrosine (2134 μmol/L). A protein-restricted diet led to the complete resolution of the corneal and cutaneous lesions and avoided the potential toxicity of tyrosine on central nervous system.

Identifiants

pubmed: 32018000
pii: S1091-8531(20)30023-9
doi: 10.1016/j.jaapos.2019.12.007
pii:
doi:

Types de publication

Case Reports

Langues

eng

Sous-ensembles de citation

IM

Pagination

120-122

Informations de copyright

Copyright © 2020 American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved.

Auteurs

Gilles C Martin (GC)

Ophthalmology Department, Hôpital-Fondation A. de Rothschild, Paris, France. Electronic address: gillesmartin88@gmail.com.

Pascale de Lonlay (P)

Necker-Enfants Malades University Hospital, Reference Center of Metabolic Diseases, Paris, France; Paris-Descartes University, Sorbonne-Paris-Cité, Paris, France.

Thibaut Chapron (T)

Ophthalmology Department, Hôpital-Fondation A. de Rothschild, Paris, France.

Isabelle Cochereau (I)

Ophthalmology Department, Hôpital-Fondation A. de Rothschild, Paris, France; Paris-Diderot University, Paris, France.

Georges Caputo (G)

Ophthalmology Department, Hôpital-Fondation A. de Rothschild, Paris, France.

Eric Gabison (E)

Ophthalmology Department, Hôpital-Fondation A. de Rothschild, Paris, France.

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Classifications MeSH