{"title":"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。","authors":"Ezgi Karataş, Canan Aslı Utine","doi":"10.5935/0004-2749.2023-0078","DOIUrl":null,"url":null,"abstract":"<p><p>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.</p>","PeriodicalId":8397,"journal":{"name":"Arquivos brasileiros de oftalmologia","volume":"88 2","pages":"e20230078"},"PeriodicalIF":1.1000,"publicationDate":"2024-09-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Congenital hereditary endothelial dystrophy with progressive sensorineural deafness: a case report of Harboyan syndrome.\",\"authors\":\"Ezgi Karataş, Canan Aslı Utine\",\"doi\":\"10.5935/0004-2749.2023-0078\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>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.</p>\",\"PeriodicalId\":8397,\"journal\":{\"name\":\"Arquivos brasileiros de oftalmologia\",\"volume\":\"88 2\",\"pages\":\"e20230078\"},\"PeriodicalIF\":1.1000,\"publicationDate\":\"2024-09-23\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Arquivos brasileiros de oftalmologia\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.5935/0004-2749.2023-0078\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2024/1/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q3\",\"JCRName\":\"OPHTHALMOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Arquivos brasileiros de oftalmologia","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.5935/0004-2749.2023-0078","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/1/1 0:00:00","PubModel":"eCollection","JCR":"Q3","JCRName":"OPHTHALMOLOGY","Score":null,"Total":0}
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.
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.