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家族性视盘玻璃疣的患病率筛查:一项横断面研究。

Prevalence Screening for Familial Optic Disc Drusen: A Cross-Sectional Study.

作者信息

Steensberg Alvilda H, Malmqvist Lasse, Bertelsen Mette, Grønskov Karen, Hamann Steffen

机构信息

Department of Ophthalmology, Copenhagen University Hospital - Rigshospitalet, Glostrup, Denmark.

Department of Clinical Genetics, Copenhagen University Hospital-Rigshospitalet, Copenhagen, Denmark.

出版信息

Neuroophthalmology. 2024 Jul 17;49(1):43-50. doi: 10.1080/01658107.2024.2372624. eCollection 2025.

Abstract

Optic disc drusen (ODD) may present in multiple individuals and generations within a family, indicating hereditary predisposition. Individuals are often unaware of their ODD, and consequently, the prevalence of ODD within families remains largely unknown. The aim of this study was to estimate the prevalence and consider the inheritance pattern of familial ODD and to investigate ODD-related symptoms and their association to ODD location and size. In this cross-sectional study, 22 ODD patients, aged 10 years or older, were included. Of these, 13 ODD probands had 24 family members participating. The presence, size, and anatomical location of ODDs and the presence of hyperreflective lines were ascertained using enhanced depth imaging optical coherence tomography (EDI-OCT) scan. Visual symptoms were ascertained, and these were correlated with ODD burden in terms of ODD location and size. Familial ODD was found in eight of the 13 screened families. Hyperreflective lines were present in all individuals with ODD, including both probands and their family members, and in seven out of the 14 family members without ODD. Visual symptoms were reported in 14-50% and associated significantly with superficial ODD location. We found familial ODD in eight of the 13 screened families. Our results aligned with the previously suggested autosomal dominant inheritance pattern with incomplete penetrance, assuming that hyperreflective lines represent a less expressed form of ODD. This emphasizes that ODD runs in families and supports the hypothesis that genetic factors may contribute to the etiology.

摘要

视盘玻璃疣(ODD)可能在一个家族的多个个体及几代人中出现,提示存在遗传易感性。个体往往 unaware of their ODD,因此,家族中ODD的患病率在很大程度上仍不为人知。本研究的目的是估计家族性ODD的患病率并考虑其遗传模式,以及调查与ODD相关的症状及其与ODD位置和大小的关联。在这项横断面研究中,纳入了22名年龄在10岁及以上的ODD患者。其中,13名ODD先证者有24名家庭成员参与。使用增强深度成像光学相干断层扫描(EDI-OCT)确定ODD的存在、大小和解剖位置以及高反射线的存在。确定视觉症状,并将这些症状与ODD位置和大小方面的ODD负担相关联。在13个筛查家族中的8个家族中发现了家族性ODD。所有患有ODD的个体,包括先证者及其家庭成员,以及14名无ODD的家庭成员中的7名,均出现了高反射线。14%至50%的患者报告有视觉症状,且与浅表ODD位置显著相关。我们在13个筛查家族中的8个家族中发现了家族性ODD。我们的结果与先前提出的常染色体显性遗传模式且具有不完全外显率一致,假设高反射线代表ODD的一种表达较少的形式。这强调了ODD在家族中具有遗传性,并支持遗传因素可能导致其病因的假说。 (注:原英文文本中“unaware of their ODD”表述不太准确规范,推测可能是“unaware of having ODD”之类,但按要求未作修改。)

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/542c/12409904/2f42c087d721/IOPH_A_2372624_F0001_OC.jpg

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