遗传性或熟悉形式的原发性甲状旁腺功能亢进:家族性孤立性甲状旁腺功能亢进的病例系列描述和文献回顾。

IF 2.3 4区 医学 Q4 ENDOCRINOLOGY & METABOLISM
Miguel Madeira, Maria Caroline Alves Coelho, Leandro Kasuki, Filipe Barbosa Linhares, Isabel Sampaio Tostes, Rafael Mazzutti Dutra Santana, Raquel Beatriz Gonçalves Muniz, Maria Lucia Fleiuss de Farias, Fernanda Vaisman
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引用次数: 0

摘要

原发性甲状旁腺功能亢进(PHPT)是一种由甲状旁腺激素分泌不当或过量引起的矿物质代谢紊乱。约95%的病例零星发生,但也可能与复杂综合征和/或家族(即遗传)史有关。我们报告了一个家族性孤立性甲状旁腺功能亢进病例系列的临床、实验室和遗传概况。确诊患者年龄22-41岁(中位32岁),4例患者复发(3例为腺瘤,1例为增生及甲状旁腺癌)。6名家庭成员出现CDC73基因杂合突变,1名患者在同一基因中出现拷贝数变异,其临床意义尚不确定。此外,我们回顾了与PHPT相关的每种疾病的特殊性,遗传评估的适应症,以及随访和治疗的建议。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Genetic or familiar forms of primary hyperparathyroidism: description of a case series with familial isolated hyperparathyroidism and review of the literature.

Genetic or familiar forms of primary hyperparathyroidism: description of a case series with familial isolated hyperparathyroidism and review of the literature.

Primaryhyperparathyroidism (PHPT) is a disorder of mineral metabolism caused by inappropriate or excessive secretion of parathyroid hormone. It occurs sporadically in approximately 95% of cases but may also be associated with complex syndromes and/or a familial (i.e., hereditary) history. We report the clinical, laboratory, and genetic profiles of a case series with familial isolated hyperparathyroidism. Diagnosis was established in patients aged 22-41 years (median = 32), and recurrence was identified in four patients (three with adenoma and one with hyperplasia and parathyroid carcinoma). Six family members presented with a heterozygous mutation in the CDC73 gene, and one patient had a copy number variation of undetermined clinical significance in the same gene. In addition, we review the particularities of each condition associated with PHPT, indications for genetic evaluation, and recommendations for follow-up and treatment.

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来源期刊
Archives of Endocrinology Metabolism
Archives of Endocrinology Metabolism Medicine-Endocrinology, Diabetes and Metabolism
CiteScore
2.90
自引率
5.90%
发文量
107
审稿时长
7 weeks
期刊介绍: The Archives of Endocrinology and Metabolism - AE&M – is the official journal of the Brazilian Society of Endocrinology and Metabolism - SBEM, which is affiliated with the Brazilian Medical Association. Edited since 1951, the AE&M aims at publishing articles on scientific themes in the basic translational and clinical area of Endocrinology and Metabolism. The printed version AE&M is published in 6 issues/year. The full electronic issue is open access in the SciELO - Scientific Electronic Library Online e at the AE&M site: www.aem-sbem.com. From volume 59 on, the name was changed to Archives of Endocrinology and Metabolism, and it became mandatory for manuscripts to be submitted in English for the online issue. However, for the printed issue it is still optional for the articles to be sent in English or Portuguese. The journal is published six times a year, with one issue every two months.
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