MedElevator Войти в исследования
← Поиск публикаций

DOI 10.1002/adbi.202200301

Медицинская библиотека · Advanced biology · 2023

The Clinical Characteristics and Potential Molecular Mechanism of LMNA Mutation-Related Lipodystrophy.

Xiao Cheng, Liu Jieying, Yang Chunru, Zhai Xiaojun, Liu Peng, Xiao Xinhua, Yu Miao

СтатьяПоддержка исследования: вне правительства СШАПеревод пока не подготовлен

Аннотация

This study aimed to enhance understanding of LMNA mutation-related lipodystrophy by elucidating genotype-phenotype correlations and potential molecular mechanisms. Clinical data from six patients with LMNA mutation-related lipodystrophy are analyzed, and four distinct LMNA mutations are identified. Associations between mutations and lipodystrophy phenotypes are assessed. Three LMNA mutation plasmids are constructed and transfected into HEK293 cells. Protein stability, degradation pathways, and binding proteins of mutant Lamin A/C are examined using Western blotting, co-immunoprecipitation, and mass spectrometry. Confocal microscopy is employed to observe nuclear structure. Four different LMNA mutations are identified in the six patients, all exhibiting lipodystrophy and metabolic disorders. Cardiac dysfunction is observed in two out of six patients. Metformin and pioglitazone are the primary treatments for glucose control. Confocal microscopy revealed nuclear blebbing and irregular cell membranes. Mutant Lamin A/C stability is significantly decreased, and degradation occurred primarily via the ubiquitin-proteasome system (UPS). Potential binding ubiquitination-related proteins of mutant Lamin A/C are identified. This study investigated LMNA mutation-related lipodystrophy, identifying four unique mutations and their connections to specific phenotypes. It is found to decreased mutant Lamin A/C stability and degradation primarily through the UPS, offering new insights into molecular mechanisms and potential therapeutic targets.

Для цитирования: Xiao Cheng, Liu Jieying, Yang Chunru, Zhai Xiaojun, Liu Peng, Xiao Xinhua, Yu Miao. The Clinical Characteristics and Potential Molecular Mechanism of LMNA Mutation-Related Lipodystrophy.. Advanced biology. 2023. DOI: 10.1002/adbi.202200301.

Предметные рубрики MeSH

Подготовка и перевод публикации
Обработка статьи · этапы, ошибки и управление
Загрузка состояния…

История · было → стало

Состояние обновляется каждые 5 секунд, пока блок открыт. Процент показывается только там, где известен объём работы. Пауза останавливает продолжение этапа; текущий запрос к источнику может завершиться.

Тип публикации указан по данным источника. Уровень доказательности автоматически не присваивается.