库伦-德-弗里斯综合征合并垂体功能减退和1型糖尿病1例。

IF 1 4区 医学 Q3 MEDICINE, GENERAL & INTERNAL
Mafalda Félix Cabral, Francisco Branco Caetano, Carla Conceição, Diana Oliveira Antunes, Lurdes Lopes
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引用次数: 0

摘要

复杂的疾病是遗传和环境因素相互作用的结果。我们提出了一个复杂疾病似乎共存的案例。一名12个月大的女孩因身材矮小和张力不足而被转诊。初步评估显示中枢性甲状腺功能减退,生长激素缺乏和小垂体与异位神经垂体。替代疗法改善了生长,但发育迟缓和斜视随之而来。10岁时,她第一次用左乙拉西坦治疗癫痫发作。12岁时,她出现糖尿病酮症酸中毒,并开始功能性胰岛素治疗;自身抗体阳性确认自身免疫性病因通过微阵列分析进行的初始基因检测显示正常结果,但外显子组测序显示KANSL1基因的杂合致病性变异,允许诊断Koolen-de - Vries综合征。在这个病人中,Koolen-de - Vries综合征最初表现为垂体功能减退,后来才出现癫痫。之后,1型糖尿病接踵而至,凸显了交织在一起的疾病的复杂性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
A Complex Case of Koolen-De Vries Syndrome Associated with Hypopituitarism and Type 1 Diabetes Mellitus.

Complex diseases arise from the interplay of genetic and environmental factors. We present a case where complex diseases seem to coexist. A 12-month-old girl was referred for short stature and hypotonia. Initial evaluation revealed central hypothyroidism, growth hormone deficiency and a small pituitary gland with ectopic neurohypophysis. Replacement therapy improved growth, but developmental delay and strabismus ensued. At age 10, she experienced a first seizure treated with levetiracetam. At age 12, she presented diabetic ketoacidosis and functional insulin therapy was started; positive autoantibodies confirmed autoimmune etiology. Initial genetic testing performed by microarray analysis retrieved normal results, but exome sequencing revealed a heterozygous pathogenic variant in KANSL1 gene, allowing for the diagnosis of Koolen-de Vries syndrome. In this patient, Koolen-de Vries syndrome presented initially as hypopituitarism and only later epilepsy. Afterwards, type 1 diabetes mellitus ensued, highlighting the complexity of intertwined conditions.

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来源期刊
Acta medica portuguesa
Acta medica portuguesa MEDICINE, GENERAL & INTERNAL-
CiteScore
1.90
自引率
16.70%
发文量
256
审稿时长
6-12 weeks
期刊介绍: The aim of Acta Médica Portuguesa is to publish original research and review articles in biomedical areas of the highest standard, covering several domains of medical knowledge, with the purpose to help doctors improve medical care. In order to accomplish these aims, Acta Médica Portuguesa publishes original articles, review articles, case reports and editorials, among others, with a focus on clinical, scientific, social, political and economic factors affecting health. Acta Médica Portuguesa will be happy to consider manuscripts for publication from authors anywhere in the world.
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