Mierzwa Magdalena, Blaska Małgorzata, Hamm Marek, Czarniecka Agnieszka, Krajewska Jolanta, Taczanowska-Niemczuk Anna, Zachurzok Agnieszka
Department of Pediatrics, Faculty of Medical Sciences in Zabrze, Medical University of Silesia in Katowice, 3 Maja 13-15, 41-800 Zabrze, Poland.
Nuclear Medicine and Endocrine Oncology Department, M. Sklodowska-Curie National Research Institute of Oncology, Gliwice Branch, 44-101 Gliwice, Poland.
Children (Basel). 2023 Dec 12;10(12):1916. doi: 10.3390/children10121916.
Multiple endocrine neoplasia 2B (MEN2B) is a rare syndrome with prevalence estimated at approximately 0.2 per 100,000; it is caused by mutation of the RET proto-oncogene. MEN2B is characterized by early-onset medullary thyroid carcinoma (MTC), ganglioneuromatosis of the aerodigestive tract, marfanoid habitus, ophthalmologic abnormalities, and pheochromocytoma in adulthood. Mutations in the RyR1 gene manifest clinically in congenital myopathies and/or malignant hyperthermia susceptibility. We present a case of a 4-year-old boy with an accidentally detected RET and RyR1 mutations in the course of diagnostic approach of short stature and delayed motor development. Due to a poor and blurred clinical picture of MEN2B syndrome, accompanied by RyR1 mutation symptoms, the diagnostic path was extended. Our patient had no family history of MTC. In the imaging studies of the thyroid gland, no abnormalities were found, whereas the serum level of calcitonin was elevated to 34 pg/mL (N < 5.0). The patient qualified for total thyroidectomy, and the histopathological examination confirmed the diagnosis of MTC. The postoperative serum calcitonin level dropped to normal ranges. This case shows how new genetic diagnostic procedures could be crucial in accidentally diagnosing rare endocrine disease with atypical symptoms, giving an opportunity for relatively early intervention.
多发性内分泌腺瘤病2B型(MEN2B)是一种罕见综合征,估计患病率约为每10万人中有0.2例;它由RET原癌基因突变引起。MEN2B的特征包括早发性甲状腺髓样癌(MTC)、气消化道神经节瘤病、类马凡体型、眼科异常以及成年期嗜铬细胞瘤。RyR1基因突变在临床上表现为先天性肌病和/或恶性高热易感性。我们报告一例4岁男孩,在对身材矮小和运动发育迟缓进行诊断评估过程中意外检测到RET和RyR1基因突变。由于MEN2B综合征的临床表现不佳且不明确,并伴有RyR1突变症状,诊断过程有所延长。我们的患者没有MTC家族史。在甲状腺的影像学检查中未发现异常,而血清降钙素水平升高至34 pg/mL(正常范围<5.0)。该患者符合甲状腺全切术条件,组织病理学检查确诊为MTC。术后血清降钙素水平降至正常范围。该病例表明,新的基因诊断程序对于意外诊断具有非典型症状的罕见内分泌疾病可能至关重要,为相对早期的干预提供了机会。