Evanescent white linear flecks and posterior microphthalmos: new features of a recently established disease

Détails

ID Serval
serval:BIB_299763CD25BB
Type
Article: article d'un périodique ou d'un magazine.
Sous-type
Etude de cas (case report): rapporte une observation et la commente brièvement.
Collection
Publications
Titre
Evanescent white linear flecks and posterior microphthalmos: new features of a recently established disease
Périodique
Klinische Monatsblatter fur Augenheilkunde
Auteur(s)
Tran  H. V., Borruat  F. X., Reymond-Gruber  S., Schorderet  D., Munier  F.
ISSN
0023-2165 (Print)
Statut éditorial
Publié
Date de publication
05/2006
Volume
223
Numéro
5
Pages
397-9
Notes
Case Reports
Journal Article --- Old month value: May
Résumé
BACKGROUND: Posterior microphthalmos is a rare autosomal recessive condition with normal anterior segment and small posterior segment resulting in axial hyperopia and retinal folding. HISTORY AND SIGNS: The proband from a consanguineous Turkish family was clinically investigated at 5 months of age and followed for the next 8 years. At five months of age, refraction revealed a severe hyperopia of + 10 diopters in both eyes (OU) with a mean axial length of 16.5 mm. Fundus examination showed a dystrophic maculo-papillar bundle. Multiple whitish deep intraretinal linear lesions sparing the macula were present in the mid-periphery with no preferential orientation. THERAPY AND OUTCOME: By 8 years of age, visual acuity was 20/25 in the right eye (OD) and 20/30 in the left eye (OS) with S + 14 OU. The whitish linear lesions had disappeared and were replaced by retinal pigment epithelium alterations. Optical coherence tomography (OCT) revealed a thickened retina with normal appearance at the site of the linear lesions. CONCLUSIONS: This is the first report of long-term follow-up and OCT findings in this rare ocular developmental abnormality.
Mots-clé
Child Child, Preschool Humans Infant Male Microphthalmos/*classification/complications/*diagnosis Retinal Diseases/classification/*congenital/*diagnosis Vision Disorders/classification/*congenital/*diagnosis
Pubmed
Web of science
Création de la notice
28/01/2008 12:58
Dernière modification de la notice
20/08/2019 13:09
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