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。

Q3 Medicine
Ach Taieb, Imen Nacef, Nadia Ghariani, Nadia Nabli, Yosra Hasni, Maha Kacem, Molka Chaieb, Amel Maaroufi, Mohamed Denguezli, Koussay Ach
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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。
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.

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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
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发文量
36
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