Common clinical features of unilateral retinal pigment epithelium dysgenesis and combined hamartoma of the retina and retinal pigment epithelium.
Case report
Combined hamartoma of the retina and the retinal pigment epithelium
Unilateral retinal pigment epithelium dysgenesis
Journal
BMC ophthalmology
ISSN: 1471-2415
Titre abrégé: BMC Ophthalmol
Pays: England
ID NLM: 100967802
Informations de publication
Date de publication:
15 Jan 2022
15 Jan 2022
Historique:
received:
31
07
2021
accepted:
31
12
2021
entrez:
16
1
2022
pubmed:
17
1
2022
medline:
19
1
2022
Statut:
epublish
Résumé
Herein, we report two cases of unilateral retinal pigment epithelium dysgenesis (URPED) in Chinese patients and explore the relationship between URPED and combined hamartoma of the retina and retinal pigment epithelium (CHRRPE). The lesion margins in the two cases showed pathognomonic clinical features of URPED, namely, a scalloped reticular margin in hyperplastic retinal pigment epithelium and mild fibrosis. The hypoautofluorescence observed by fundus autofluorescence was inverted compared with that observed by fundus fluorescence angiography. A large amount of fibroglial proliferation and disorganization of the retina involving the whole layer, which are also found in peripapillary CHRRPE, were found in the lesions. URPED appears to share some clinical features with CHRRPE, and the relationship between URPED and CHRRPE needs further study.
Sections du résumé
BACKGROUND
BACKGROUND
Herein, we report two cases of unilateral retinal pigment epithelium dysgenesis (URPED) in Chinese patients and explore the relationship between URPED and combined hamartoma of the retina and retinal pigment epithelium (CHRRPE).
CASE PRESENTATION
METHODS
The lesion margins in the two cases showed pathognomonic clinical features of URPED, namely, a scalloped reticular margin in hyperplastic retinal pigment epithelium and mild fibrosis. The hypoautofluorescence observed by fundus autofluorescence was inverted compared with that observed by fundus fluorescence angiography. A large amount of fibroglial proliferation and disorganization of the retina involving the whole layer, which are also found in peripapillary CHRRPE, were found in the lesions.
CONCLUSIONS
CONCLUSIONS
URPED appears to share some clinical features with CHRRPE, and the relationship between URPED and CHRRPE needs further study.
Identifiants
pubmed: 35033040
doi: 10.1186/s12886-022-02244-x
pii: 10.1186/s12886-022-02244-x
pmc: PMC8760665
doi:
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
24Informations de copyright
© 2022. The Author(s).
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