骺软骨发育不良:两个胎儿病例的诊断线索和文献综述。

IF 2.7 2区 医学 Q2 GENETICS & HEREDITY
Prenatal Diagnosis Pub Date : 2024-08-01 Epub Date: 2024-06-23 DOI:10.1002/pd.6631
Vivien Cuvelier, Detlef Trost, Morgane Stichelbout, Caroline Michot, Valérie Cormier-Daire, Nathalie Boutry, Elise Machet, Catherine Vincent-Delorme
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引用次数: 0

摘要

本文介绍了两例胎儿骺端发育不良(GDD)病例(一种罕见的常染色体显性遗传疾病),并回顾了相关文献。这两个病例中的两个胎儿表现出骨弓形,从而被诊断为 GDD。基因检测发现了 ANO5 基因的两个新变异,从而确诊了该病症。我们进行了文献综述,以探讨 GDD 的临床和辅助临床表现、诊断和管理。GDD 是一种罕见但常见的遗传性骨脆弱和颌骨病变,其特征是 ANO5 基因的功能增益变异。临床表现多种多样,从伴有轻微颌骨病变的反复牙科感染,到伴有多处骨折的严重骨脆性,以及需要进行毁容手术的大面积颌骨病变。诊断技术取决于具体情况,包括 ANO5 基因的靶向基因检测、全外显子组测序的非靶向分子分析或全基因组测序。本病例报告强调了认识到 GDD 是导致孕期骨弯曲和骨折的新原因的重要性。通过总结文献,本文有助于提高医护人员的知识水平,改善对 GDD 患者的识别、诊断和护理。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Gnathodiaphyseal dysplasia: Diagnostic clues from two fetal cases and literature review.

This article presents two fetal cases of gnathodiaphyseal dysplasia (GDD), a rare autosomal dominant disorder, and reviews the relevant literature. The cases involved two fetuses exhibiting bone bowing, which led to the diagnosis of GDD. Genetic testing revealed two de novo variants of the ANO5 gene, confirming the diagnosis. A literature review was conducted to explore GDD's clinical and paraclinical presentation, diagnosis, and management. GDD is a rare but frequently inherited cause of bone fragility and jaw lesions characterized by a gain-of-function variant within the ANO5 gene. Clinical manifestations range from recurrent dental infections with mild jaw lesions to severe bone fragility with several fractures associated with large jaw lesions requiring disfiguring surgeries. Diagnostic techniques depend on the context and include targeted genetic testing of ANO5, untargeted molecular analysis with whole-exome sequencing, or whole-genome sequencing. This case report highlights the importance of recognizing GDD as a novel cause of bone bowing and fractures during pregnancy. By summarizing the literature, this article contributes to healthcare professionals' knowledge and improves the recognition, diagnosis, and care of patients with GDD.

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来源期刊
Prenatal Diagnosis
Prenatal Diagnosis 医学-妇产科学
CiteScore
5.80
自引率
13.30%
发文量
204
审稿时长
2 months
期刊介绍: Prenatal Diagnosis welcomes submissions in all aspects of prenatal diagnosis with a particular focus on areas in which molecular biology and genetics interface with prenatal care and therapy, encompassing: all aspects of fetal imaging, including sonography and magnetic resonance imaging; prenatal cytogenetics, including molecular studies and array CGH; prenatal screening studies; fetal cells and cell-free nucleic acids in maternal blood and other fluids; preimplantation genetic diagnosis (PGD); prenatal diagnosis of single gene disorders, including metabolic disorders; fetal therapy; fetal and placental development and pathology; development and evaluation of laboratory services for prenatal diagnosis; psychosocial, legal, ethical and economic aspects of prenatal diagnosis; prenatal genetic counseling
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