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一种新型的与PGAP3基因突变相关的大角膜可能被误诊为原发性先天性青光眼。

A Novel PGAP3 Gene Mutation-Related Megalocornea Can Be Misdiagnosed as Primary Congenital Glaucoma.

作者信息

Alhaidari Abdulmajeed I, Albakri Amani S, Alhumaidi Suzan S

机构信息

Ophthalmology, King Khaled Eye Specialist Hospital, Riyadh, SAU.

Division of Pediatric Ophthalmology, King Khaled Eye Specialist Hospital, Riyadh, SAU.

出版信息

Cureus. 2022 Sep 21;14(9):e29387. doi: 10.7759/cureus.29387. eCollection 2022 Sep.

Abstract

Hyperphosphatasia with mental retardation syndrome 4 (HPMRS4) is a rare autosomal recessive disorder caused by glycosylphosphatidylinositol (GPI) deficiency. GPI deficiency results from a mutation in one of six known genes. Mutation in post-GPI attachment to protein phospholipase 3 gene (PGAP3) is linked to HPMRS4. Patients usually present with dysmorphic features, developmental delay, central hypotonia, and seizure. However, in our case, we report a novel homozygous missense mutation of PGAP3 gene in a female child who presented with megalocornea, which is an unusual clinical presentation for HPMRS4. Megalocornea, in her first days of life, led to a misdiagnosis of primary congenital glaucoma. Later, other common clinical features of HPMRS4 became apparent.

摘要

高磷酸酶血症伴智力发育迟缓综合征4(HPMRS4)是一种由糖基磷脂酰肌醇(GPI)缺乏引起的罕见常染色体隐性疾病。GPI缺乏是由六个已知基因之一的突变导致的。GPI后附着蛋白磷脂酶3基因(PGAP3)的突变与HPMRS4相关。患者通常表现为畸形特征、发育迟缓、中枢性肌张力减退和癫痫发作。然而,在我们的病例中,我们报告了一名患有巨角膜的女童中PGAP3基因的一种新的纯合错义突变,这是HPMRS4不寻常的临床表现。在她出生后的头几天,巨角膜导致了原发性先天性青光眼的误诊。后来,HPMRS4的其他常见临床特征变得明显。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/167a/9585391/ae33f972aeaf/cureus-0014-00000029387-i01.jpg

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