异质性
先证者
线粒体DNA
医学
线粒体肌病
粒线体疾病
基因检测
糖尿病
遗传学
精密医学
生物信息学
症候群
感音神经性聋
2型糖尿病
遗传诊断
肌肉无力
内科学
儿科
队列
遗传倾向
突变
人类线粒体遗传学
作者
Kaylee R Oppenheimer,Nava T Himelhoch,Michael E. McCullough,TIANA L. BOWDEN,Balamurugan Kandasamy,Lisa R. Letourneau,Rochelle N. Naylor,Siri Atma W. Greeley,Louis H Philipson
标识
DOI:10.1210/clinem/dgaf698
摘要
Maternally inherited diabetes and deafness (MIDD) syndrome is a rare form of monogenic diabetes most often caused by the pathogenic m.3243A > G mutation in the mitochondrial tRNALeu (UUR) gene, MT-TL1. Mutations causing MIDD are also associated with mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes. This paper analyzes the data of 15 probands with mitochondrial diabetes enrolled in the University of Chicago Monogenic Diabetes Registry, all of whom have confirmed pathogenic variants, primarily m.3243A > G. Three of these probands (3/15) were selected for detailed case studies and pedigree analysis. Among the total cohort, sensorineural hearing loss (80%) and muscle weakness (53%) were frequent comorbidities, and all tested individuals were negative for islet autoantibodies. Treatment regimens included insulin and sulfonylureas, with some reporting use of biguanides despite safety concerns related to mitochondrial dysfunction. Three probands noted subjective improvement with mitochondrial cocktail supplementation. Familial heteroplasmy testing revealed significant inter- and intrafamilial variability. This cohort represents 1 of the largest clinically characterized US populations with mitochondrial diabetes and underscores the importance of urine-based heteroplasmy testing and personalized management strategies informed by mitochondrial pathophysiology.
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