Department of Neurology, Pauls Stradins Clinical University Hospital, Riga, Latvia.
Department of Medical Genetics and Prenatal Diagnostics, Child's Clinical University Hospital, Riga, Latvia.
Am J Case Rep. 2023 May 22;24:e939217. doi: 10.12659/AJCR.939217.
BACKGROUND Ververi-Brady syndrome (VEBRAS) is an autosomal dominant condition associated with short stature, microcephaly, mild dysmorphic features, and learning disabilities. It was first described in 2018, and only 38 cases have been reported since then. All patients have mutation in the Glutamine-rich protein 1 (QRICH1) gene, yet clinical presentation has a broad spectrum and continues to expand. This report is of a mother and daughter pair with VEBRAS, associated with a new variant of the QRICH1 gene, NM_017730.3: c.337C>T; p.(Gln113*), and few previously undescribed phenotypic features. CASE REPORT We present 2 new cases, a mother and daughter, with novel heterozygous nonsense variant NM_017730.3: c.337C>T; p.(Gln113*). The daughter was referred to a geneticist at the age of 17 years because of seizures, dysmorphic features, and magnetic resonance imaging suggestive of leukodystrophy. In addition to already described clinical features, she had diffuse infantile hemangiomatosis and occipital balding. She was accompanied by her mother, who shared similar phenotypic features, raising suspicion for a similar genetic condition. Unlike the daughter, the mother never had any significant health problems or concerns and described herself as perfectly healthy. Genetic testing was performed in both individuals, and a novel pathogenic QRICH1 variant was discovered. CONCLUSIONS Considering the novelty of VEBRAS, every new clinical case contributes to the enlargement of the VEBRAS cohort, expanding the phenotypical and mutational spectrum, with potential improvement in the further care and observation of probands and their offspring. This report has highlighted the importance of clinical genetics in the identification of familial genetic disorders with complex phenotypes.
Ververi-Brady 综合征(VEBRAS)是一种常染色体显性遗传病,与身材矮小、小头畸形、轻度发育不良和学习障碍有关。它于 2018 年首次被描述,此后仅报道了 38 例病例。所有患者均携带 Glutamine-rich protein 1(QRICH1)基因突变,但临床表现谱广泛且不断扩大。本报告介绍了一对 VEBRAS 母女病例,伴有 QRICH1 基因的新变异,NM_017730.3:c.337C>T;p.(Gln113*),以及一些以前未描述的表型特征。
我们介绍了 2 例新病例,一对母女,均携带新的杂合性无义变异 NM_017730.3:c.337C>T;p.(Gln113*)。女儿因癫痫发作、发育不良和磁共振成像提示白质营养不良而在 17 岁时被转介给遗传学家。除了已经描述的临床特征外,她还患有弥漫性婴儿血管瘤和枕部脱发。她的母亲也有类似的表型特征,这引起了对类似遗传疾病的怀疑。与女儿不同的是,母亲从未有过任何重大健康问题或担忧,并形容自己身体非常健康。对两人均进行了基因检测,发现了一种新的致病性 QRICH1 变异。
鉴于 VEBRAS 的新颖性,每一个新的临床病例都有助于扩大 VEBRAS 队列,扩大表型和突变谱,从而有可能改善先证者及其后代的进一步护理和观察。本报告强调了临床遗传学在识别具有复杂表型的家族性遗传疾病中的重要性。