MedElevator Войти в исследования
← Поиск публикаций

DOI 10.1002/ajmg.a.38619

Медицинская библиотека · American journal of medical genetics. Part A · 2018

CHILD syndrome: A modified pathogenesis-targeted therapeutic approach.

Bergqvist Christina, Abdallah Bilal, Hasbani Divina-Justina, Abbas Ossama, Kibbi Abdul Ghani, Hamie Lamiaa, Kurban Mazen, Rubeiz Nelly

Клинический случайСтатьяПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

Congenital Hemidysplasia with Ichthyosiform nevus and Limb Defects (CHILD syndrome) is a rare X-linked dominant genodermatosis caused by mutations in the NAD(P) dependent steroid dehydrogenase-like protein gene. Its defect leads to accumulation of toxic metabolic intermediates upstream from the pathway block and to the deficiency of bulk cholesterol, probably leading to altered keratinocyte membrane function, resulting in the phenotype seen in CHILD syndrome. Symptomatic treatment using emollients and retinoids to reduce scaling has long been used until recently, whereby new therapeutic means based on the pathogenesis-targeted therapy have been developed. We subsequently chose to use the same pathogenesis-based therapy using a 2% cholesterol and 2% lovastatin cream with or without glycolic acid in two of our patients. Improvement in CHILD skin lesions was seen as early as 4 weeks after initiation. The addition of glycolic acid helped improve the penetrance of the cholesterol and lovastatin cream into the thick waxy scales. Our study confirms the efficacy of the pathogenesis-targeted therapy and introduces the possibility of modifying its formula by adding glycolic acid in order to improve the treatment.

Для цитирования: Bergqvist Christina, Abdallah Bilal, Hasbani Divina-Justina, Abbas Ossama, Kibbi Abdul Ghani, Hamie Lamiaa, Kurban Mazen, Rubeiz Nelly. CHILD syndrome: A modified pathogenesis-targeted therapeutic approach.. American journal of medical genetics. Part A. 2018. DOI: 10.1002/ajmg.a.38619.

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.