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一名14岁男性骨髓衰竭综合征患者,无耳聋,由从父亲遗传的新型SRP72突变引起:病例报告

A 14-Year-Old Male Patient With Bone Marrow Failure Syndrome, Without Deafness, Caused by a Novel SRP72 Mutation Inherited From His Father: A Case Report.

作者信息

Sun Dan, Luo Quanfang, Wu Chunlin, Deng Hui, Li Miao

机构信息

Clinical Genomics Center, Wuhan Kingmed Medical Laboratory Co. Ltd., Wuhan, CHN.

Hematology, Nanxishan Hospital of Guangxi Zhuang Autonomous Region, Guilin, CHN.

出版信息

Cureus. 2025 Aug 7;17(8):e89574. doi: 10.7759/cureus.89574. eCollection 2025 Aug.

Abstract

Inherited bone marrow failure syndrome 1 (IBMFS1) is a rare autosomal dominant disorder associated with mutations in the SRP72 gene. However, mutations in this gene are exceedingly rare, and the clinical manifestations are often nonspecific, leading to delayed or misdiagnosed cases. The incidence, lifetime risk, and clinical management guidelines for SRP72-related IBMFS1 are poorly understood due to its rarity. Molecular diagnosis is essential for accurate diagnosis, treatment, and prognosis. Here, we report a novel frameshift mutation in the SRP72 gene identified in a 14-year-old patient presenting with pancytopenia. Sanger sequencing revealed that the mutation was inherited from the patient's asymptomatic father. In vitro functional studies indicated a loss-of-function mechanism. Our case expands the mutation spectrum of the SRP72 gene and highlights the importance of genetic testing in identifying hematologic disorders.

摘要

遗传性骨髓衰竭综合征1(IBMFS1)是一种罕见的常染色体显性疾病,与SRP72基因突变有关。然而,该基因突变极为罕见,且临床表现往往不具特异性,导致病例延误诊断或误诊。由于其罕见性,SRP72相关IBMFS1的发病率、终生风险及临床管理指南尚不清楚。分子诊断对于准确诊断、治疗及预后至关重要。在此,我们报告在一名出现全血细胞减少的14岁患者中鉴定出的SRP72基因的一种新型移码突变。桑格测序显示该突变遗传自患者无症状的父亲。体外功能研究表明存在功能丧失机制。我们的病例扩展了SRP72基因的突变谱,并突出了基因检测在识别血液系统疾病中的重要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/be9f/12413996/a15a1d2ca7dd/cureus-0017-00000089574-i01.jpg

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