{"title":"一名青少年在小肠卷曲并伴有进行性巨结肠后被诊断为多发性内分泌肿瘤 2B 型。","authors":"Yusuke Sakai, Yoshiko Nakayama, Shingo Kurasawa, Tomomitsu Sado, Sawako Kato, Nao Hidaka, Shigeru Takamizawa, Katsumi Yoshizawa, Koichiro Yoshimaru, Tomoaki Taguchi","doi":"10.1007/s12328-024-01979-y","DOIUrl":null,"url":null,"abstract":"<p><p>Multiple endocrine neoplasia type 2B is a rare autosomal dominant disease characterized by the presence of medullary thyroid carcinoma, pheochromocytoma, Marfan-like fatigue, a peculiar face with thickening of the lips, mucosal neuromas on the lips and tongue, and gastrointestinal phenomena. Most patients harbor pathological variants of the RET gene. Herein, we present the first case of a 14 year-old boy who experienced small intestinal volvulus along with a megacolon, and he was diagnosed with multiple endocrine neoplasia type 2B. The patient complained of constipation since he was 2 years old and slowly progressive abdominal distension at school age. At 14 years of age, he presented with remarkable megacolon mimicking Hirschsprung's disease and complicated with small intestinal volvulus. The volvulus was successfully repaired, and the particularly dilated transverse colon was resected following a rectal biopsy. Histopathological evaluation of the resected transverse colon revealed to be compatible with ganglioneuromatosis. After emergency surgery, the patient was diagnosed with multiple endocrine neoplasia type 2B with medullary thyroid carcinoma, and a de novo variant of RET was confirmed. Gastroenterologists should consider it when treating patients with constipation, especially those with megacolon. Therefore, timely diagnosis may lead to appropriate treatment of medullary thyroid carcinoma and improve mortality.</p>","PeriodicalId":10364,"journal":{"name":"Clinical Journal of Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.8000,"publicationDate":"2024-08-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11284186/pdf/","citationCount":"0","resultStr":"{\"title\":\"Multiple endocrine neoplasia type 2B diagnosed after small intestinal volvulus with progressive megacolon in an adolescent.\",\"authors\":\"Yusuke Sakai, Yoshiko Nakayama, Shingo Kurasawa, Tomomitsu Sado, Sawako Kato, Nao Hidaka, Shigeru Takamizawa, Katsumi Yoshizawa, Koichiro Yoshimaru, Tomoaki Taguchi\",\"doi\":\"10.1007/s12328-024-01979-y\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Multiple endocrine neoplasia type 2B is a rare autosomal dominant disease characterized by the presence of medullary thyroid carcinoma, pheochromocytoma, Marfan-like fatigue, a peculiar face with thickening of the lips, mucosal neuromas on the lips and tongue, and gastrointestinal phenomena. Most patients harbor pathological variants of the RET gene. Herein, we present the first case of a 14 year-old boy who experienced small intestinal volvulus along with a megacolon, and he was diagnosed with multiple endocrine neoplasia type 2B. The patient complained of constipation since he was 2 years old and slowly progressive abdominal distension at school age. At 14 years of age, he presented with remarkable megacolon mimicking Hirschsprung's disease and complicated with small intestinal volvulus. The volvulus was successfully repaired, and the particularly dilated transverse colon was resected following a rectal biopsy. Histopathological evaluation of the resected transverse colon revealed to be compatible with ganglioneuromatosis. After emergency surgery, the patient was diagnosed with multiple endocrine neoplasia type 2B with medullary thyroid carcinoma, and a de novo variant of RET was confirmed. Gastroenterologists should consider it when treating patients with constipation, especially those with megacolon. Therefore, timely diagnosis may lead to appropriate treatment of medullary thyroid carcinoma and improve mortality.</p>\",\"PeriodicalId\":10364,\"journal\":{\"name\":\"Clinical Journal of Gastroenterology\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":0.8000,\"publicationDate\":\"2024-08-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11284186/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Clinical Journal of Gastroenterology\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1007/s12328-024-01979-y\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2024/5/16 0:00:00\",\"PubModel\":\"Epub\",\"JCR\":\"Q4\",\"JCRName\":\"GASTROENTEROLOGY & HEPATOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Clinical Journal of Gastroenterology","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1007/s12328-024-01979-y","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/5/16 0:00:00","PubModel":"Epub","JCR":"Q4","JCRName":"GASTROENTEROLOGY & HEPATOLOGY","Score":null,"Total":0}
引用次数: 0
摘要
多发性内分泌肿瘤 2B 型是一种罕见的常染色体显性遗传病,其特征是出现甲状腺髓样癌、嗜铬细胞瘤、马凡样疲劳、嘴唇增厚的特殊面容、唇舌粘膜神经瘤和胃肠道现象。大多数患者携带 RET 基因的病理变异。在此,我们介绍了第一例 14 岁男孩的病例,他出现了小肠下垂和巨结肠,并被诊断为多发性内分泌肿瘤 2B 型。患者自 2 岁起就主诉便秘,在学龄期腹胀逐渐加重。14 岁时,他出现了类似赫氏巨结肠症的显著巨结肠,并伴有小肠肠套叠。他成功地修复了肠套叠,并在直肠活检后切除了特别扩张的横结肠。对切除的横结肠进行组织病理学评估后发现,该病与神经节瘤病相符。紧急手术后,患者被诊断为多发性内分泌肿瘤 2B 型伴甲状腺髓样癌,并确诊为 RET 新发变异体。消化科医生在治疗便秘患者,尤其是巨结肠患者时应考虑到这一点。因此,及时诊断可使甲状腺髓样癌得到适当治疗,并改善死亡率。
Multiple endocrine neoplasia type 2B diagnosed after small intestinal volvulus with progressive megacolon in an adolescent.
Multiple endocrine neoplasia type 2B is a rare autosomal dominant disease characterized by the presence of medullary thyroid carcinoma, pheochromocytoma, Marfan-like fatigue, a peculiar face with thickening of the lips, mucosal neuromas on the lips and tongue, and gastrointestinal phenomena. Most patients harbor pathological variants of the RET gene. Herein, we present the first case of a 14 year-old boy who experienced small intestinal volvulus along with a megacolon, and he was diagnosed with multiple endocrine neoplasia type 2B. The patient complained of constipation since he was 2 years old and slowly progressive abdominal distension at school age. At 14 years of age, he presented with remarkable megacolon mimicking Hirschsprung's disease and complicated with small intestinal volvulus. The volvulus was successfully repaired, and the particularly dilated transverse colon was resected following a rectal biopsy. Histopathological evaluation of the resected transverse colon revealed to be compatible with ganglioneuromatosis. After emergency surgery, the patient was diagnosed with multiple endocrine neoplasia type 2B with medullary thyroid carcinoma, and a de novo variant of RET was confirmed. Gastroenterologists should consider it when treating patients with constipation, especially those with megacolon. Therefore, timely diagnosis may lead to appropriate treatment of medullary thyroid carcinoma and improve mortality.
期刊介绍:
The journal publishes Case Reports and Clinical Reviews on all aspects of the digestive tract, liver, biliary tract, and pancreas. Critical Case Reports that show originality or have educational implications for diagnosis and treatment are especially encouraged for submission. Personal reviews of clinical gastroenterology are also welcomed. The journal aims for quick publication of such critical Case Reports and Clinical Reviews.