Armentano Marta, Alisi Ludovico, Giovannetti Francesca, Iannucci Valeria, Lucchino Luca, Bruscolini Alice, Lambiase Alessandro
Department of Sense Organs, Sapienza University of Rome, 00185 Rome, Italy.
Life (Basel). 2024 Aug 13;14(8):1006. doi: 10.3390/life14081006.
22q11.2 deletion syndrome (22q11.2DS) is a genetic disorder caused by the deletion of the q11.2 band of chromosome 22. It may affect various systems, including the cardiovascular, immunological, gastrointestinal, endocrine, and neurocognitive systems. Additionally, several ocular manifestations have been described.
We report a case of a 34-year-old female diagnosed with 22q11.2DS who presented with visual discomfort and foreign body sensation in both eyes. She had no history of recurrent ocular pain. A comprehensive ophthalmological examination was performed, including anterior segment optical coherence tomography and in vivo confocal microscopy. Overall, the exams revealed bilateral corneal map-like lines, dots, and fingerprint patterns, consistent with a diagnosis of epithelial basement membrane dystrophy (EBMD). In addition to presenting with this novel corneal manifestation for 22q11.2 DS, we review the ocular clinical features of 22q11.2DS in the context of our case.
The EBMD may represent a new corneal manifestation associated with 22q11.2 syndrome, although the link between these conditions is unknown. Further research is warranted to investigate potentially shared genetic or molecular pathways to the understanding of the phenotypic variety observed among this rare syndrome.
22q11.2缺失综合征(22q11.2DS)是一种由22号染色体q11.2带缺失引起的遗传性疾病。它可能影响多个系统,包括心血管、免疫、胃肠、内分泌和神经认知系统。此外,还描述了几种眼部表现。
我们报告一例34岁女性诊断为22q11.2DS,双眼出现视觉不适和异物感。她没有复发性眼痛病史。进行了全面的眼科检查,包括眼前节光学相干断层扫描和活体共聚焦显微镜检查。总体而言,检查发现双眼角膜有地图样线条、点状和指纹状图案,符合上皮基底膜营养不良(EBMD)的诊断。除了呈现这种22q11.2DS新的角膜表现外,我们结合病例回顾了22q11.2DS的眼部临床特征。
EBMD可能代表一种与22q11.2综合征相关的新的角膜表现,尽管这些情况之间的联系尚不清楚。有必要进一步研究,以调查潜在的共同遗传或分子途径,从而了解这种罕见综合征中观察到的表型多样性。