Serbis Anastasios, Tsinopoulou Vassiliki Regina, Mouzaki Konstantina, Kotanidou Eleni P, Giza Styliani, Galli-Tsinopoulou Assimina
4th Department of Pediatrics, School of Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, Papageorgiou General Hospital, Thessaloniki, Greece.
Ann Pediatr Endocrinol Metab. 2018 Sep;23(3):162-165. doi: 10.6065/apem.2018.23.3.162. Epub 2018 Sep 28.
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.
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。