LPIN2相关Majeed综合征:两例印度LPIN2新变异患者的报告和文献综述。

IF 0.4 4区 医学 Q4 GENETICS & HEREDITY Clinical Dysmorphology Pub Date : 2024-01-01 Epub Date: 2023-10-12 DOI:10.1097/MCD.0000000000000476
Vaishnavi Ashok Badiger, Suma Balan, Sumanth Madan, Kishore Sai Gogineni, Hitesh Shah, Dhanya Lakshmi Narayanan
{"title":"LPIN2相关Majeed综合征:两例印度LPIN2新变异患者的报告和文献综述。","authors":"Vaishnavi Ashok Badiger, Suma Balan, Sumanth Madan, Kishore Sai Gogineni, Hitesh Shah, Dhanya Lakshmi Narayanan","doi":"10.1097/MCD.0000000000000476","DOIUrl":null,"url":null,"abstract":"<p><p>LPIN2 -related Majeed syndrome (MIM# 609628) is a rare non-inflammasome autoinflammatory disease, caused due to biallelic variants in LPIN2 (MIM* 605519). To date, only 31 individuals from 18 families have been reported with this rare condition. Exome sequencing was done in two affected individuals from two unrelated families. Additionally, phenotypic, and genotypic information from the literature was reviewed. Two novel homozygous missense variants, c.2207G>A p. (Arg736His) and c.1157C>G p. (Ser386Ter) in LPIN2 , were identified in family 1 and family 2 respectively. Chronic recurrent osteomyelitis involving the lower extremities was the most common clinical presentation. LPIN2 -related Majeed syndrome should be considered as a differential diagnosis in an individual with clinical or radiological evidence of recurrent sterile osteomyelitis and chronic anaemia.</p>","PeriodicalId":50682,"journal":{"name":"Clinical Dysmorphology","volume":" ","pages":"27-30"},"PeriodicalIF":0.4000,"publicationDate":"2024-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"LPIN2 -related Majeed syndrome: report of two Indian patients with novel variants in LPIN2 and review of literature.\",\"authors\":\"Vaishnavi Ashok Badiger, Suma Balan, Sumanth Madan, Kishore Sai Gogineni, Hitesh Shah, Dhanya Lakshmi Narayanan\",\"doi\":\"10.1097/MCD.0000000000000476\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>LPIN2 -related Majeed syndrome (MIM# 609628) is a rare non-inflammasome autoinflammatory disease, caused due to biallelic variants in LPIN2 (MIM* 605519). To date, only 31 individuals from 18 families have been reported with this rare condition. Exome sequencing was done in two affected individuals from two unrelated families. Additionally, phenotypic, and genotypic information from the literature was reviewed. Two novel homozygous missense variants, c.2207G>A p. (Arg736His) and c.1157C>G p. (Ser386Ter) in LPIN2 , were identified in family 1 and family 2 respectively. Chronic recurrent osteomyelitis involving the lower extremities was the most common clinical presentation. LPIN2 -related Majeed syndrome should be considered as a differential diagnosis in an individual with clinical or radiological evidence of recurrent sterile osteomyelitis and chronic anaemia.</p>\",\"PeriodicalId\":50682,\"journal\":{\"name\":\"Clinical Dysmorphology\",\"volume\":\" \",\"pages\":\"27-30\"},\"PeriodicalIF\":0.4000,\"publicationDate\":\"2024-01-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Clinical Dysmorphology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1097/MCD.0000000000000476\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2023/10/12 0:00:00\",\"PubModel\":\"Epub\",\"JCR\":\"Q4\",\"JCRName\":\"GENETICS & HEREDITY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Clinical Dysmorphology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1097/MCD.0000000000000476","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2023/10/12 0:00:00","PubModel":"Epub","JCR":"Q4","JCRName":"GENETICS & HEREDITY","Score":null,"Total":0}
引用次数: 0

摘要

LPIN2相关的Majeed综合征(MIM#609628)是一种罕见的非炎症性自身炎症性疾病,由LPIN2的双等位基因变异引起(MIM*605119)。迄今为止,只有来自18个家庭的31人被报告患有这种罕见的疾病。对来自两个不相关家族的两名受影响个体进行了外显子组测序。此外,还对文献中的表型和基因型信息进行了综述。在家族1和家族2中分别鉴定了LPIN2中的两个新的纯合错义变体,即c.2207G>A.(Arg736His)和c.1157C>G.(Ser386Ter)。累及下肢的慢性复发性骨髓炎是最常见的临床表现。LPIN2相关的Majeed综合征应被视为具有复发性无菌性骨髓炎和慢性贫血临床或放射学证据的个体的鉴别诊断。
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
LPIN2 -related Majeed syndrome: report of two Indian patients with novel variants in LPIN2 and review of literature.

LPIN2 -related Majeed syndrome (MIM# 609628) is a rare non-inflammasome autoinflammatory disease, caused due to biallelic variants in LPIN2 (MIM* 605519). To date, only 31 individuals from 18 families have been reported with this rare condition. Exome sequencing was done in two affected individuals from two unrelated families. Additionally, phenotypic, and genotypic information from the literature was reviewed. Two novel homozygous missense variants, c.2207G>A p. (Arg736His) and c.1157C>G p. (Ser386Ter) in LPIN2 , were identified in family 1 and family 2 respectively. Chronic recurrent osteomyelitis involving the lower extremities was the most common clinical presentation. LPIN2 -related Majeed syndrome should be considered as a differential diagnosis in an individual with clinical or radiological evidence of recurrent sterile osteomyelitis and chronic anaemia.

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
Clinical Dysmorphology
Clinical Dysmorphology 医学-遗传学
CiteScore
1.20
自引率
0.00%
发文量
64
审稿时长
6-12 weeks
期刊介绍: Clinical Dysmorphology publishes succinct case reports on the etiology, clinical delineation, genetic mapping, and molecular embryology of birth defects. This journal covers such topics as multiple congenital anomaly syndromes - with particular emphasis on previously undescribed conditions, rare findings, ethnic differences in existing syndromes, fetal abnormalities, and cytogenetic aberrations that might give clues to the localization of developmental genes. Regular features include original, peer-reviewed articles, conference reports, book and software reviews, abstracts and summaries from the UK Dysmorphology Club, and literature summaries. Submitted articles undergo a preliminary review by the editor. Some articles may be returned to authors wihtout further consideration. Those being considered for publication will undergo further assessment and peer-review by the editors and those invited to do so from a reviewer pool.
期刊最新文献
Biallelic variants in AGRN with recurrent pregnancy losses in a family with a fetal akinesia deformation sequence. Targeted genetic testing approach in a case with characteristic clinical and radiographic findings of Roberts phocomelia syndrome. Immune dysregulation in a dysmorphic child with 6q23.3 deletion: a single case report. Nephrocalcinosis, distal renal tubular acidosis and skeletal abnormality in two siblings with ROGDI -related Kohlschütter-Tönz syndrome. A girl with a de novo PPP2R5D W207R pathogenic variant was also born with an occipital encephalocele.
×
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