Starzyk J, Starzyk B, Bartnik-Mikuta A, Urbanowicz W, Dziatkowiak H
Department of Pediatric Endocrinology, Polish-American Children's Hospital, Collegium Medicum, Jagiellonian University, Kraków.
J Pediatr Endocrinol Metab. 2001 Jun;14(6):789-96. doi: 10.1515/jpem.2001.14.6.789.
A 5 year-old girl presented with typical features of isosexual precocity with breast and pubic hair development (Tanner stage 3) and menarche, following a few months history of hirsutism of the back and thighs. Stimulation testing revealed GnRH-independent precocious puberty, tertiary hypothyroidism, hyperprolactinemia and mild testosteronemia. The ovaries in ultrasound examination were prepubertal. Tumor markers beta-hCG and AFP were markedly elevated and a 2.5 x 1.5 cm suprasellar germ cell tumor (GCT) was visualized by MRI. Combined chemotherapy followed by radiotherapy resulted in normalization of pubertal features along with estrogen and marker levels. Our observations support the possibility of hCG-dependent precocious puberty (PP) in girls caused by suprasellar hCG-secreting tumor. We emphasize the need of diagnostic management of hCG-dependent PP not only in boys, but also in girls, especially when they present even slight features of androgenization. We hypothesize that the rarity of isosexual PP in girls with hCG-secreting suprasellar GCT results not only from the lower occurrence of these tumors in girls than in boys, but above all from a rare simultaneous concomitant incidence of both high tumor aromatase activity and hCG secreting potency.
一名5岁女孩出现同性性早熟的典型特征,有乳房和阴毛发育(坦纳3期)及初潮,此前有几个月背部和大腿多毛的病史。刺激试验显示为GnRH非依赖性性早熟、三级甲状腺功能减退、高催乳素血症和轻度睾酮血症。超声检查显示卵巢处于青春期前状态。肿瘤标志物β-hCG和AFP显著升高,MRI显示鞍上有一个2.5×1.5 cm的生殖细胞瘤(GCT)。联合化疗后放疗使青春期特征以及雌激素和标志物水平恢复正常。我们的观察结果支持鞍上分泌hCG的肿瘤导致女孩出现hCG依赖性性早熟(PP)的可能性。我们强调,不仅对男孩,而且对女孩,尤其是当她们出现哪怕轻微的雄激素化特征时,都需要对hCG依赖性PP进行诊断管理。我们推测,鞍上分泌hCG的GCT女孩中同性PP罕见,不仅是因为这些肿瘤在女孩中的发生率低于男孩,更主要是因为肿瘤高芳香化酶活性和hCG分泌能力同时并存的情况罕见。