Slouma Maroua, Abbes Maissa, Dhahri Rim, Litaiem Noureddine, Gueddiche Nour, Mansouri Nada, Msekni Issam, Gharsallah Imen, Metoui Leila, Louzir Bassem
Department of Internal Medicine, Military Hospital, 1007, Tunis, Tunisia.
Faculty of Medicine of Tunis, University of Tunis El Manar, Tunis, Tunisia.
Clin Rheumatol. 2021 Feb;40(2):775-782. doi: 10.1007/s10067-020-05281-3. Epub 2020 Jul 14.
Multiple endocrine neoplasia type 1 is a rare autosomal inherited syndrome that affects a variety of endocrine tissues such as the parathyroid, endocrine pancreas, and anterior pituitary. Osseous complications are often misdiagnosed. We presented a case of a 46-year-old woman with pathological fractures of the lower limb. She had a history of type 1 diabetes and galactorrhea. Laboratory examinations showed hypercalcemia and an increased level of parathyroid hormone related to hyperparathyroidism. Serum chromogranin A level was increased at 9369 ng/mL (N < 102). A somatostatin receptor scintigraphy (octreoscan) revealed pathological uptake in the gastric wall, later cave adenopathy, and liver. The diagnosis of multiple endocrine neoplasia type 1 was made based on radiological and histological findings. The patient underwent a subtotal parathyroidectomy associated with somatostatin analog treatment leading to significant improvement. A literature review was conducted by searching PubMed using these following terms: multiple endocrine neoplasia type 1, hyperparathyroidism, fracture, menin, osteoporosis. We emphasized bone involvement related to multiple endocrine neoplasia type 1 syndrome. This diagnosis should be considered when pathological fractures occur in young patients with a history of endocrine disorder. We highlighted the importance of imaging features in making the diagnosis of multiple endocrine neoplasia type 1. Early management of this disease is necessary. Treatment including parathyroidectomy and somatostatin analogs leads to bone preservation and functional improvement.
1型多发性内分泌腺瘤病是一种罕见的常染色体显性遗传综合征,可累及多种内分泌组织,如甲状旁腺、内分泌胰腺和垂体前叶。骨并发症常被误诊。我们报告了一例46岁下肢病理性骨折的女性病例。她有1型糖尿病和溢乳病史。实验室检查显示高钙血症以及与甲状旁腺功能亢进相关的甲状旁腺激素水平升高。血清嗜铬粒蛋白A水平升高至9369 ng/mL(正常<102)。生长抑素受体闪烁扫描(奥曲肽扫描)显示胃壁、后纵隔淋巴结病和肝脏有异常摄取。根据影像学和组织学检查结果确诊为1型多发性内分泌腺瘤病。患者接受了甲状旁腺次全切除术并联合生长抑素类似物治疗,病情显著改善。通过在PubMed上使用以下关键词进行文献检索:1型多发性内分泌腺瘤病、甲状旁腺功能亢进、骨折、menin、骨质疏松症。我们强调了与1型多发性内分泌腺瘤病综合征相关的骨受累情况。对于有内分泌疾病史的年轻患者发生病理性骨折时,应考虑这一诊断。我们强调了影像学特征在1型多发性内分泌腺瘤病诊断中的重要性。对该疾病进行早期治疗很有必要。包括甲状旁腺切除术和生长抑素类似物在内的治疗可实现骨骼保护和功能改善。