Centre for Clinical Genetics, Sydney Children's Hospital Randwick, New Sout Wales, Australia.
Hereditary Cancer Centre, Prince of Wales Hospital Randwick, New Sout Wales, Australia.
Pediatrics. 2024 Sep 1;154(3). doi: 10.1542/peds.2022-059517.
Early onset medullary thyroid carcinoma, later pheochromocytomas, and nonspecific extra-endocrine features (hypermobility and persistent constipation) are part of the clinical phenotype of Multiple Endocrine Neoplasia type 2B (MEN2B). A de novo pathogenic M918T variant in the rearranged during transfection proto-oncogene is usually identified. Affected children are often seen by multiple clinicians over a long period before consideration of a diagnosis of MEN2B, with metastatic medullary thyroid carcinoma often the precipitator. We describe the clinical presentation and course of 5 children ultimately diagnosed with MEN2B in New South Wales and the Australian Capital Territory, Australia between 1989 and 2021. All cases had intestinal ganglioneuromatosis that could have prompted an earlier diagnosis. Population wide newborn genomic screening for rare diseases is on the horizon. We propose that MEN2B genomic screening should be included in newborn screening programs and that careful exclusion of intestinal ganglioneuromatosis would allow earlier identification leading to improved clinical outcomes.
早发性甲状腺髓样癌、后期嗜铬细胞瘤和非特异性内分泌外特征(高活动度和持续性便秘)是多发性内分泌肿瘤 2B 型(MEN2B)的临床表型的一部分。通常会发现重排期间的转染原癌基因中的从头致病性 M918T 变异。在考虑 MEN2B 诊断之前,受影响的儿童通常会在很长一段时间内由多名临床医生就诊,转移性甲状腺髓样癌通常是诱因。我们描述了在 1989 年至 2021 年间在澳大利亚新南威尔士州和澳大利亚首都领地最终诊断为 MEN2B 的 5 名儿童的临床表现和病程。所有病例均有肠神经节瘤病,这可能促使更早地做出诊断。针对罕见疾病的人群新生儿基因组筛查即将出现。我们建议将 MEN2B 基因组筛查纳入新生儿筛查计划,并仔细排除肠神经节瘤病,以便更早发现,从而改善临床结局。