1型糖尿病与色素性干皮病罕见关联的临床、免疫学和遗传学研究。

Ach Taieb, Imen Nacef, Nadia Ghariani, Nadia Nabli, Yosra Hasni, Maha Kacem, Molka Chaieb, Amel Maaroufi, Mohamed Denguezli, Koussay Ach
{"title":"1型糖尿病与色素性干皮病罕见关联的临床、免疫学和遗传学研究。","authors":"Ach Taieb,&nbsp;Imen Nacef,&nbsp;Nadia Ghariani,&nbsp;Nadia Nabli,&nbsp;Yosra Hasni,&nbsp;Maha Kacem,&nbsp;Molka Chaieb,&nbsp;Amel Maaroufi,&nbsp;Mohamed Denguezli,&nbsp;Koussay Ach","doi":"10.5114/pedm.2022.118318","DOIUrl":null,"url":null,"abstract":"<p><p>Xeroderma pigmentosum (XP) is a rare genodermatosis predisposing to skin cancers. Autoimmune diseases related to XP are rarely discussed in the literature. Type 1 diabetes (T1D) has been associated with other genodermatoses like Cockayne syndrome, but it has never been described in XP. In the present study, we report the rare occurrence of T1D in XP patients. Five XP patients belonging to 4 consanguineous families originating from different regions of Tunisia were investigated. Their ages ranged between 8 and 18 years. All the patients had a severe hypovitaminosis D. All the patients had positive GAD antibody levels, and 4 of them had familial history of other autoimmune diseases. The spectrum of XP was variable in all the patients, with dermatological and neurological symptoms, and the occurrence of some cancers. Various hypotheses have been proposed to explain this association, among of which we cite the role of immunomodulation and down-regulation of ATP-dependent DNA excision repair protein genes, implying that impaired DNA repair may contribute to the development of some autoimmune diseases. Vitamin D3 deficiency secondary to sun protective measures was found in all patients and thus may play a role in increasing T1D risk in these patients.</p>","PeriodicalId":39165,"journal":{"name":"Pediatric Endocrinology, Diabetes and Metabolism","volume":"28 3","pages":"233-237"},"PeriodicalIF":0.0000,"publicationDate":"2022-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/87/45/PEDM-28-47552.PMC10214951.pdf","citationCount":"0","resultStr":"{\"title\":\"Clinical, immunological, and genetic investigations in a rare association of type 1 diabetes with xeroderma pigmentosum.\",\"authors\":\"Ach Taieb,&nbsp;Imen Nacef,&nbsp;Nadia Ghariani,&nbsp;Nadia Nabli,&nbsp;Yosra Hasni,&nbsp;Maha Kacem,&nbsp;Molka Chaieb,&nbsp;Amel Maaroufi,&nbsp;Mohamed Denguezli,&nbsp;Koussay Ach\",\"doi\":\"10.5114/pedm.2022.118318\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Xeroderma pigmentosum (XP) is a rare genodermatosis predisposing to skin cancers. Autoimmune diseases related to XP are rarely discussed in the literature. Type 1 diabetes (T1D) has been associated with other genodermatoses like Cockayne syndrome, but it has never been described in XP. In the present study, we report the rare occurrence of T1D in XP patients. Five XP patients belonging to 4 consanguineous families originating from different regions of Tunisia were investigated. Their ages ranged between 8 and 18 years. All the patients had a severe hypovitaminosis D. All the patients had positive GAD antibody levels, and 4 of them had familial history of other autoimmune diseases. The spectrum of XP was variable in all the patients, with dermatological and neurological symptoms, and the occurrence of some cancers. Various hypotheses have been proposed to explain this association, among of which we cite the role of immunomodulation and down-regulation of ATP-dependent DNA excision repair protein genes, implying that impaired DNA repair may contribute to the development of some autoimmune diseases. Vitamin D3 deficiency secondary to sun protective measures was found in all patients and thus may play a role in increasing T1D risk in these patients.</p>\",\"PeriodicalId\":39165,\"journal\":{\"name\":\"Pediatric Endocrinology, Diabetes and Metabolism\",\"volume\":\"28 3\",\"pages\":\"233-237\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2022-01-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/87/45/PEDM-28-47552.PMC10214951.pdf\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Pediatric Endocrinology, Diabetes and Metabolism\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.5114/pedm.2022.118318\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"Medicine\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Pediatric Endocrinology, Diabetes and Metabolism","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.5114/pedm.2022.118318","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"Medicine","Score":null,"Total":0}
引用次数: 0

摘要

摘要色素性干皮病是一种罕见的遗传性皮肤病,易致皮肤癌。文献中很少讨论与XP相关的自身免疫性疾病。1型糖尿病(T1D)与其他遗传性皮肤病(如柯凯因综合征)有关,但在XP中从未描述过。在本研究中,我们报告了XP患者中罕见的T1D。调查了来自突尼斯不同地区的4个近亲家庭的5名XP患者。他们的年龄在8到18岁之间。所有患者均患有严重维生素d缺乏症,GAD抗体均呈阳性,其中4例有其他自身免疫性疾病家族史。所有患者的XP频谱都是可变的,有皮肤和神经症状,并发生一些癌症。人们提出了各种假设来解释这种关联,其中我们引用了免疫调节和atp依赖性DNA切除修复蛋白基因下调的作用,这意味着DNA修复受损可能导致一些自身免疫性疾病的发展。所有患者均发现继发于防晒措施的维生素D3缺乏症,因此可能在这些患者中增加T1D风险。
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
Clinical, immunological, and genetic investigations in a rare association of type 1 diabetes with xeroderma pigmentosum.

Xeroderma pigmentosum (XP) is a rare genodermatosis predisposing to skin cancers. Autoimmune diseases related to XP are rarely discussed in the literature. Type 1 diabetes (T1D) has been associated with other genodermatoses like Cockayne syndrome, but it has never been described in XP. In the present study, we report the rare occurrence of T1D in XP patients. Five XP patients belonging to 4 consanguineous families originating from different regions of Tunisia were investigated. Their ages ranged between 8 and 18 years. All the patients had a severe hypovitaminosis D. All the patients had positive GAD antibody levels, and 4 of them had familial history of other autoimmune diseases. The spectrum of XP was variable in all the patients, with dermatological and neurological symptoms, and the occurrence of some cancers. Various hypotheses have been proposed to explain this association, among of which we cite the role of immunomodulation and down-regulation of ATP-dependent DNA excision repair protein genes, implying that impaired DNA repair may contribute to the development of some autoimmune diseases. Vitamin D3 deficiency secondary to sun protective measures was found in all patients and thus may play a role in increasing T1D risk in these patients.

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
Pediatric Endocrinology, Diabetes and Metabolism
Pediatric Endocrinology, Diabetes and Metabolism Medicine-Pediatrics, Perinatology and Child Health
CiteScore
2.00
自引率
0.00%
发文量
36
期刊最新文献
Elevated level of prenatal testosterone and vitamin D3 deficiency during pregnancy, in the presence of prenatal maternal stress, and their association with the development of attention deficit hyperactivity disorder (ADHD)-like symptoms in toddlers. Hereditary vitamin D resistant rickets (HVDRR) case series: phenotype, genotype, conventional treatment, and adjunctive cinacalcet therapy. Human growth hormone therapy - in three stages: past, present, and future. Importance of diet in children and adolescents with obesity and asthma. Indications for genetic diagnosis in children with growth hormone deficiency and born small for gestational age.
×
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