Stecchini Monica F, Macedo Delanie B, Reis Ana Claudia S, Abreu Ana Paula, Moreira Ayrton C, Castro Margaret, Kaiser Ursula B, Latronico Ana Claudia, Antonini Sonir R
Departments of Pediatrics and Internal Medicine, Ribeirão Preto Medical School, University of São Paulo, Ribeirão Preto, Brazil.
Horm Res Paediatr. 2016;86(2):126-130. doi: 10.1159/000447515. Epub 2016 Jul 16.
Loss-of-function mutations in the imprinted gene MKRN3 represent the most common known genetic defects associated with central precocious puberty (CPP).
We report the first case of a girl carrying an MKRN3 mutation detected in childhood and followed until the development of pubertal signs.
The girl was screened at the age of 4 years because of a positive family history; her sister had developed CPP at 6 years of age and was found to harbor the MKRN3 p.Pro161Argfs*16 mutation, inherited from their asymptomatic father. During close follow-up, she initially developed increased growth velocity at 6 years (9 cm/year), followed by a slightly increased basal luteinizing hormone level (0.4 mIU/ml) and, ultimately, clinical thelarche with rapid progression (Tanner stage 1-3) between 6.3 and 6.7 years. In the context of a loss-of-function MKRN3 mutation and a positive family history, these features established the diagnosis of CPP and supported the initiation of treatment with a gonadotropin-releasing hormone analog. The absence of significant bone age advancement, pubic or axillary hair, or behavioral or social problems could be ascribed to the early diagnosis.
The identification of carriers of MKRN3 mutations may contribute to early diagnosis of CPP, facilitating treatment decisions and guiding genetic counseling and prompt intervention in familial cases.
印记基因MKRN3的功能丧失突变是已知与中枢性性早熟(CPP)相关的最常见遗传缺陷。
我们报告了首例在儿童期检测到携带MKRN3突变并随访至青春期体征出现的女孩病例。
该女孩因家族史阳性在4岁时接受筛查;她的姐姐6岁时患中枢性性早熟,被发现携带从无症状父亲遗传而来的MKRN3 p.Pro161Argfs*16突变。在密切随访期间,她最初在6岁时生长速度加快(9厘米/年),随后基础促黄体生成素水平略有升高(0.4 mIU/ml),最终在6.3至6.7岁之间出现临床乳房发育并迅速进展(坦纳分期1 - 3期)。在存在MKRN3功能丧失突变和家族史阳性的情况下,这些特征确立了中枢性性早熟的诊断,并支持开始使用促性腺激素释放激素类似物进行治疗。骨龄无明显提前、无阴毛或腋毛,以及无行为或社会问题可归因于早期诊断。
识别MKRN3突变携带者可能有助于中枢性性早熟的早期诊断,有助于治疗决策,指导遗传咨询,并对家族性病例进行及时干预。