先天性遗传性内皮营养不良伴进行性感音神经性耳聋:哈博扬综合征病例报告。

IF 1.1 4区 医学 Q3 OPHTHALMOLOGY Arquivos brasileiros de oftalmologia Pub Date : 2024-09-23 eCollection Date: 2024-01-01 DOI:10.5935/0004-2749.2023-0078
Ezgi Karataş, Canan Aslı Utine
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引用次数: 0

摘要

我们为您介绍一例 37 岁女性的病例,她在 10 岁时因先天性遗传性内皮营养不良症接受了双侧穿透性角膜移植术。在随后的 27 年中,患者的视力逐渐恶化。我们的检查发现,右侧角膜移植出现了失代偿。我们通过再植手术解决了这一问题。随后,我们了解到患者自青春期起就患有渐进性听力损失。音调测听显示其听力损失为 25 分贝,左耳更为严重。由于进行性感音神经性听力损失和先天性遗传性内皮营养不良的模式都与同一个基因 slc4a11 有关,我们对患者进行了该基因突变的检测。检测结果呈阳性,患者染色体 20p13-p12 上的 slc4a11 基因第五外显子发生了杂合突变,导致了框架移位。经临床和基因综合评估,确诊为哈博扬综合征。在对该病进行基因诊断后,对她的听力损失是否需要助听器进行了评估。患者还被告知要接受遗传咨询。
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Congenital hereditary endothelial dystrophy with progressive sensorineural deafness: a case report of Harboyan syndrome.

We present the case of a 37-year-old woman who underwent bilateral penetrating keratoplasty for congenital hereditary endothelial dystrophy at the age of 10 years. Over the subsequent 27 years, the patient's vision slowly deteriorated. Our examination revealed decompensation of the right corneal graft. We addressed this with regraft surgery. We then learned that the patient had been suffering from progressive hearing loss since adolescence. Tonal audiometry revealed hearing per ceptive deafness of 25 dB, which was more prominent in the left ear. Because the patterns of progressive sensorineural hearing loss and congenital hereditary endothelial dystrophy have both been linked to the same gene, slc4a11, we tested our patient for mutations in this gene. The test was positive for a heterozygous slc4a11 gene fifth exon mutation on chromosome 20p13-p12, which causes a frameshift. A combined clinical and genetic evaluation confirmed a diagnosis of Harboyan syndrome. After the genetic diagnosis of the disease, she was evaluated for the need for a hearing aid due to her hearing loss. The patient was also informed about genetic counseling.

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来源期刊
CiteScore
1.60
自引率
0.00%
发文量
200
审稿时长
6-12 weeks
期刊介绍: The ABO-ARQUIVOS BRASILEIROS DE OFTALMOLOGIA (ABO, ISSN 0004-2749 - print and ISSN 1678-2925 - (ABO, ISSN 0004-2749 - print and ISSN 1678-2925 - electronic version), the official bimonthly publication of the Brazilian Council of Ophthalmology (CBO), aims to disseminate scientific studies in Ophthalmology, Visual Science and Health public, by promoting research, improvement and updating of professionals related to the field.
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