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一例长期皮肤结节性多动脉炎患者中导致腺苷脱氨酶2酶缺乏的罕见基因突变:病例报告

A Rare Genetic Mutation Leading to a Deficiency of Adenosine Deaminase 2 Enzyme in a Long-Standing Case of Cutaneous Polyarteritis Nodosa: A Case Report.

作者信息

Contractor Rutu A, Bhavsar Yash D, Joshi Arpit P, Pujara Niyati N, Shukla Dhaiwat M

机构信息

Medical Student, Smt. Nathiba Hargovandas Lakhmichand Municipal Medical College, Ahmedabad, IND.

Internal Medicine, Vadilal Sarabhai General Hospital, Ahmedabad, IND.

出版信息

Cureus. 2022 Oct 14;14(10):e30295. doi: 10.7759/cureus.30295. eCollection 2022 Oct.

Abstract

Vasculitis is an inflammatory disorder of blood vessels affecting multiple organs. A deficiency of adenosine deaminase enzyme type 2 (DADA2) is a novel condition identified as a monogenic cause of cutaneous vasculitis. Since its first description in 2014, numerous case series and literature across several countries have expanded the scope of our understanding of this rare genetic condition. However, due to a scarcity of reported cases in adults, little is known regarding its full spectrum of clinical presentations, treatment guidelines, or outcomes in the adult patient population. It is established that it commonly affects multiple organ systems, such as the skin, musculoskeletal, neurological, hematological, gastrointestinal, and renal systems. It presents with a wide range of clinical manifestations, including fever, Livedoid rash, cutaneous polyarteritis nodosa, polyneuropathy, and immunodeficiency. Such a varied clinical spectrum opens an opportunity for discussion to list some of the differential signs of DADA2. In this article, we report a unique case of a 26-year-old male with a delay of nine years in diagnosing a genetic mutation that led to DADA2. In addition, a 10-year history of recurring cutaneous ulcers and peripheral neuropathy makes this case a noteworthy addition to the literature on cutaneous vasculitis and its miscellaneous causes.

摘要

血管炎是一种影响多个器官的血管炎症性疾病。2型腺苷脱氨酶(DADA2)缺乏是一种新发现的单基因导致的皮肤血管炎病因。自2014年首次被描述以来,多个国家的众多病例系列和文献扩展了我们对这种罕见遗传病的认识范围。然而,由于成人病例报告较少,对于其在成年患者群体中的全部临床表现范围、治疗指南或预后了解甚少。已知它通常会影响多个器官系统,如皮肤、肌肉骨骼、神经、血液、胃肠道和肾脏系统。它具有广泛的临床表现,包括发热、类紫癜样皮疹、皮肤结节性多动脉炎、多发性神经病和免疫缺陷。如此多样的临床谱为讨论列出一些DADA2的鉴别体征提供了机会。在本文中,我们报告了一例独特的病例,一名26岁男性,其导致DADA2的基因突变诊断延迟了九年。此外,10年复发性皮肤溃疡和周围神经病变病史使该病例成为皮肤血管炎及其多种病因文献中值得关注的补充。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b637/9658489/bf64aa047649/cureus-0014-00000030295-i01.jpg

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