Bertelloni Silvano, Tyutyusheva Nina, Valiani Margherita, D'Alberton Franco, Baldinotti Fulvia, Caligo Maria Adelaide, Baroncelli Giampiero I, Peroni Diego G
Paediatric and Adolescent Endocrinology, Division of Pediatrics, Department of Clinical and Experimental Medicine, University of Pisa, Pisa, Italy.
Clinical Psychologist, Bologna, Italy.
Front Pediatr. 2021 Apr 22;9:627281. doi: 10.3389/fped.2021.627281. eCollection 2021.
Differences/disorders of sex development (DSD) are a heterogeneous group of congenital conditions, resulting in discordance between an individual's sex chromosomes, gonads, and/or anatomic sex. The management of a newborn with suspected 46,XY DSD remains challenging. Newborns with 46,XY DSD may present with several phenotypes ranging from babies with atypical genitalia or girls with inguinal herniae to boys with micropenis and cryptorchidism. A mismatch between prenatal karyotype and female phenotype is an increasing reason for presentation. Gender assignment should be avoided prior to expert evaluation and possibly until molecular diagnosis. The classic diagnostic approach is time and cost-consuming. Today, a different approach may be considered. The first line of investigations must exclude rare life-threatening diseases related to salt wasting crises. Then, the new genetic tests should be performed, yielding increased diagnostic performance. Focused imaging or endocrine studies should be performed on the basis of genetic results in order to reduce repeated and invasive investigations for a small baby. The challenge for health professionals will lie in integrating specific genetic information with better defined clinical and endocrine phenotypes and in terms of long-term evolution. Such advances will permit optimization of counseling of parents and sex assignment. In this regard, society has significantly changed its attitude to the acceptance and expansion beyond strict binary male and female sexes, at least in some countries or cultures. These management advances should result in better personalized care and better long-term quality of life of babies born with 46,XY DSD.
性发育差异/障碍(DSD)是一组异质性先天性疾病,导致个体的性染色体、性腺和/或解剖学性别之间不一致。对疑似46,XY DSD的新生儿进行管理仍然具有挑战性。患有46,XY DSD的新生儿可能表现出多种表型,从生殖器不典型的婴儿、患有腹股沟疝的女孩到患有小阴茎和隐睾症的男孩。产前核型与女性表型不匹配是就诊的一个日益常见的原因。在专家评估之前,甚至可能在分子诊断之前,应避免进行性别指定。传统的诊断方法既耗时又费钱。如今,可以考虑采用不同的方法。首先必须排除与失盐危象相关的罕见危及生命的疾病。然后,应进行新的基因检测,以提高诊断效能。应根据基因检测结果进行针对性的影像学或内分泌学检查,以减少对小婴儿进行的重复和侵入性检查。卫生专业人员面临的挑战在于将特定的基因信息与定义更明确的临床和内分泌表型相结合,并考虑到长期发展情况。这些进展将有助于优化对父母的咨询和性别指定。在这方面,社会对接受和超越严格的二元男女性别观念的态度已经发生了显著变化,至少在一些国家或文化中是如此。这些管理方面的进展应能为患有46,XY DSD的新生儿带来更好的个性化护理和更高的长期生活质量。