腺苷脱氨酶2缺乏:结节性多动脉炎的鉴别诊断具有挑战性。

IF 1.4 Q3 RHEUMATOLOGY Reumatologia Pub Date : 2023-01-01 DOI:10.5114/reum.2023.124878
Shakiba Hassanzadeh, Mohammad Bahadoram, Karim Mowla
{"title":"腺苷脱氨酶2缺乏:结节性多动脉炎的鉴别诊断具有挑战性。","authors":"Shakiba Hassanzadeh,&nbsp;Mohammad Bahadoram,&nbsp;Karim Mowla","doi":"10.5114/reum.2023.124878","DOIUrl":null,"url":null,"abstract":"<p><p>Deficiency of adenosine deaminase 2 (DADA2) is an autosomal recessive disorder that was first described in 2014. It is a monogenic disease that is caused by loss-of-function variants in the <i>ADA2</i> gene. Deficiency of adenosine deaminase 2 involves small- and medium-sized vessels and its clinical presentations include polyarteritis nodosa (PAN)-like features such as livedoid rash, early-onset stroke, hypogammaglobulinemia, hematological abnormalities, and systemic inflammation. Early diagnosis and treatment of DADA2 are crucial as the clinical features could be potentially life-threatening but might be treatable. The first-line treatment of choice in DADA2 is tumor necrosis factor α inhibitors. We aimed to provide an overview of the known pathophysiology, clinical presentations, diagnosis, and treatment of DADA2. A clearer knowledge of DADA2 may help to better diagnose, manage, and improve the clinical outcome of DADA2 patients. However, further studies are required to investigate the genotype-phenotype associations and exact pathophysiology of DADA2.</p>","PeriodicalId":21312,"journal":{"name":"Reumatologia","volume":null,"pages":null},"PeriodicalIF":1.4000,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/28/1f/RU-61-152721.PMC10044036.pdf","citationCount":"1","resultStr":"{\"title\":\"Deficiency of adenosine deaminase 2: a challenging differential diagnosis of polyarteritis nodosa.\",\"authors\":\"Shakiba Hassanzadeh,&nbsp;Mohammad Bahadoram,&nbsp;Karim Mowla\",\"doi\":\"10.5114/reum.2023.124878\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Deficiency of adenosine deaminase 2 (DADA2) is an autosomal recessive disorder that was first described in 2014. It is a monogenic disease that is caused by loss-of-function variants in the <i>ADA2</i> gene. Deficiency of adenosine deaminase 2 involves small- and medium-sized vessels and its clinical presentations include polyarteritis nodosa (PAN)-like features such as livedoid rash, early-onset stroke, hypogammaglobulinemia, hematological abnormalities, and systemic inflammation. Early diagnosis and treatment of DADA2 are crucial as the clinical features could be potentially life-threatening but might be treatable. The first-line treatment of choice in DADA2 is tumor necrosis factor α inhibitors. We aimed to provide an overview of the known pathophysiology, clinical presentations, diagnosis, and treatment of DADA2. A clearer knowledge of DADA2 may help to better diagnose, manage, and improve the clinical outcome of DADA2 patients. However, further studies are required to investigate the genotype-phenotype associations and exact pathophysiology of DADA2.</p>\",\"PeriodicalId\":21312,\"journal\":{\"name\":\"Reumatologia\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":1.4000,\"publicationDate\":\"2023-01-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/28/1f/RU-61-152721.PMC10044036.pdf\",\"citationCount\":\"1\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Reumatologia\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.5114/reum.2023.124878\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"RHEUMATOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Reumatologia","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.5114/reum.2023.124878","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"RHEUMATOLOGY","Score":null,"Total":0}
引用次数: 1

摘要

腺苷脱氨酶2缺乏症(DADA2)是一种常染色体隐性遗传病,于2014年首次发现。这是一种单基因疾病,由ADA2基因的功能丧失变异引起。腺苷脱氨酶2缺乏累及中小型血管,其临床表现包括结节性多动脉炎(PAN)样特征,如样皮疹、早发性卒中、低γ球蛋白血症、血液学异常和全身性炎症。早期诊断和治疗DADA2至关重要,因为其临床特征可能危及生命,但可能是可治疗的。DADA2的一线治疗选择是肿瘤坏死因子α抑制剂。我们旨在概述DADA2的已知病理生理、临床表现、诊断和治疗。了解DADA2可能有助于更好地诊断、管理和改善DADA2患者的临床结果。然而,需要进一步研究DADA2的基因型-表型关联和确切的病理生理。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
查看原文
分享 分享
微信好友 朋友圈 QQ好友 复制链接
本刊更多论文
Deficiency of adenosine deaminase 2: a challenging differential diagnosis of polyarteritis nodosa.

Deficiency of adenosine deaminase 2 (DADA2) is an autosomal recessive disorder that was first described in 2014. It is a monogenic disease that is caused by loss-of-function variants in the ADA2 gene. Deficiency of adenosine deaminase 2 involves small- and medium-sized vessels and its clinical presentations include polyarteritis nodosa (PAN)-like features such as livedoid rash, early-onset stroke, hypogammaglobulinemia, hematological abnormalities, and systemic inflammation. Early diagnosis and treatment of DADA2 are crucial as the clinical features could be potentially life-threatening but might be treatable. The first-line treatment of choice in DADA2 is tumor necrosis factor α inhibitors. We aimed to provide an overview of the known pathophysiology, clinical presentations, diagnosis, and treatment of DADA2. A clearer knowledge of DADA2 may help to better diagnose, manage, and improve the clinical outcome of DADA2 patients. However, further studies are required to investigate the genotype-phenotype associations and exact pathophysiology of DADA2.

求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
Reumatologia
Reumatologia Medicine-Rheumatology
CiteScore
2.70
自引率
0.00%
发文量
44
审稿时长
10 weeks
期刊最新文献
Bilateral rapidly destructive coxopathy in rheumatoid arthritis. Level of glial cell derived neurotrophic factor in the blood plasma of rheumatoid arthritis patients and its relationship with alexithymia. Patterns and prevalence of psychiatric morbidity among individuals with rheumatoid arthritis. Assessment of neurocognitive function in patients with Behçet's disease. Early predictive factors in routine clinical practice for rituximab therapy response in patients with rheumatoid arthritis.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
现在去查看 取消
×
提示
确定
0
微信
客服QQ
Book学术公众号 扫码关注我们
反馈
×
意见反馈
请填写您的意见或建议
请填写您的手机或邮箱
已复制链接
已复制链接
快去分享给好友吧!
我知道了
×
扫码分享
扫码分享
Book学术官方微信
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术
文献互助 智能选刊 最新文献 互助须知 联系我们:info@booksci.cn
Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。
Copyright © 2023 Book学术 All rights reserved.
ghs 京公网安备 11010802042870号 京ICP备2023020795号-1