Suppr超能文献

患有X连锁先天性肾上腺发育不全男孩的睾丸微石症。

Testicular microlithiasis in a boy with X-linked adrenal hypoplasia congenita.

作者信息

Serbis Anastasios, Tsinopoulou Vassiliki Regina, Mouzaki Konstantina, Kotanidou Eleni P, Giza Styliani, Galli-Tsinopoulou Assimina

机构信息

4th Department of Pediatrics, School of Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, Papageorgiou General Hospital, Thessaloniki, Greece.

出版信息

Ann Pediatr Endocrinol Metab. 2018 Sep;23(3):162-165. doi: 10.6065/apem.2018.23.3.162. Epub 2018 Sep 28.

Abstract

X-linked adrenal hypoplasia congenita (AHC) is a rare disorder that usually presents clinically as adrenal insufficiency in early infancy. It is caused by mutations in the NR0B1 gene which is located on the short arm of chromosome X (Xp21). The NR0B1 gene plays an important role in normal development and function of both the adrenal and gonadal axes and some patients with the disease can present in adolescence with hypogonadotropic hypogonadism. Testicular microlithiasis is an ultrasonographic finding of unknown etiology that has been associated with several benign conditions such as cryptorchidism, congenital adrenal hyperplasia, varicoceles, and testicular malignancy. We report the case of an 11-year-old boy who was diagnosed at the age of 8 months with X-linked AHC due to adrenal failure and presented testicular microlithiasis during follow-up. To the best of our knowledge, this is the first case of an X-linked AHC patient diagnosed with testicular microlithiasis in follow-up.

摘要

X连锁先天性肾上腺发育不全(AHC)是一种罕见疾病,通常在婴儿早期临床上表现为肾上腺功能不全。它由位于X染色体短臂(Xp21)上的NR0B1基因突变引起。NR0B1基因在肾上腺和性腺轴的正常发育及功能中起重要作用,一些患有该疾病的患者在青春期可出现低促性腺激素性性腺功能减退。睾丸微结石症是一种病因不明的超声检查发现,与隐睾、先天性肾上腺增生、精索静脉曲张和睾丸恶性肿瘤等多种良性疾病有关。我们报告一例11岁男孩,其8个月大时因肾上腺功能衰竭被诊断为X连锁AHC,随访期间出现睾丸微结石症。据我们所知,这是首例随访中被诊断为睾丸微结石症的X连锁AHC患者。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5b43/6177664/8caaf2b5e284/apem-2018-23-3-162f1.jpg

相似文献

1
Testicular microlithiasis in a boy with X-linked adrenal hypoplasia congenita.
Ann Pediatr Endocrinol Metab. 2018 Sep;23(3):162-165. doi: 10.6065/apem.2018.23.3.162. Epub 2018 Sep 28.
3
Identification and Analysis of a Novel NR0B1 Mutation in Late-Onset Adrenal Hypoplasia Congenita and Hypogonadism.
J Endocr Soc. 2020 Nov 13;5(2):bvaa176. doi: 10.1210/jendso/bvaa176. eCollection 2021 Feb 1.
5
9
A novel stop mutation (p.(Gln22*)) of DAX1 (NR0B1) results in late-onset X-linked adrenal hypoplasia congenita.
Endocrinol Diabetes Metab Case Rep. 2017 Sep 4;2017. doi: 10.1530/EDM-17-0054. eCollection 2017.
10
Identification of a novel point mutation in DAX-1 gene in a patient with adrenal hypoplasia congenita.
Ann Pediatr Endocrinol Metab. 2021 Jun;26(2):126-129. doi: 10.6065/apem.2040088.044. Epub 2021 Jun 30.

引用本文的文献

1
X-linked congenital adrenal hypoplasia: a case presentation.
BMC Endocr Disord. 2021 Jun 15;21(1):118. doi: 10.1186/s12902-021-00785-8.

本文引用的文献

1
How should we monitor boys with testicular microlithiasis?
Pediatr Hematol Oncol. 2016 Apr;33(3):171-7. doi: 10.3109/08880018.2016.1156203. Epub 2016 Mar 23.
2
Clinical manifestations of testicular adrenal rest tumor in males with congenital adrenal hyperplasia.
Ann Pediatr Endocrinol Metab. 2015 Sep;20(3):155-61. doi: 10.6065/apem.2015.20.3.155. Epub 2015 Sep 30.
3
DAX-1 (NR0B1) and steroidogenic factor-1 (SF-1, NR5A1) in human disease.
Best Pract Res Clin Endocrinol Metab. 2015 Aug;29(4):607-19. doi: 10.1016/j.beem.2015.07.004. Epub 2015 Jul 14.
5
Clinical aspects of testicular microlithiasis in boys: a review.
J Pediatr Urol. 2012 Oct;8(5):459-69. doi: 10.1016/j.jpurol.2011.07.003. Epub 2011 Aug 19.
7
Clinical and genetic heterogeneity of congenital adrenal hypoplasia due to NR0B1 gene mutations.
Clin Endocrinol (Oxf). 2010 Apr;72(4):448-54. doi: 10.1111/j.1365-2265.2009.03652.x. Epub 2009 Jun 8.
8
Testicular microlithiasis: our experience of 10 years.
J Ultrasound Med. 2007 Jul;26(7):867-73. doi: 10.7863/jum.2007.26.7.867.
9
Testicular calcification and microlithiasis: association with primary intra-testicular malignancy in 3,477 patients.
Eur Radiol. 2007 Feb;17(2):363-9. doi: 10.1007/s00330-006-0296-0. Epub 2006 May 18.
10
Analysis of DAX1 (NR0B1) and steroidogenic factor-1 (NR5A1) in children and adults with primary adrenal failure: ten years' experience.
J Clin Endocrinol Metab. 2006 Aug;91(8):3048-54. doi: 10.1210/jc.2006-0603. Epub 2006 May 9.

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验