Interventional Radiology Center, Xiangya Hospital Central South University, .
Rev Esp Enferm Dig. 2024 May;116(5):291-292. doi: 10.17235/reed.2023.9896/2023.
Neurofibromatosis type I is a relatively rare inherited disorder. Simultaneous occurrence of abdominal tumors of three types in NF-I and causing intussusception is rare. We report a synchronous case of small bowel gastrointestinal stromal tumor, small bowel adenocarcinoma, and ganglioneuroma complicated by intussusception in an old woman with NF-I. The patient presented with a 2-month history of abdominal pain and multiple neurofibromas and café au lait macules was found all over the patient's body. The enhanced computed tomography revealed a terminal ileal tumor which was significantly enhanced with secondary intussusception. The tumor of the ileum was considered to be NF-I-associated intestinal neurofibroma.Postoperative pathology and immunohistochemistry showed that the terminal ileocecal mass was adenocarcinoma and ganglioneuroma and intraoperative exploration also revealed a gastrointestinal stromal tumor. In this patient, the preoperative imaging was not compatible with the pathological findings.Either neurofibromatosis involving the intestine or neurofibromatosis with small bowel tumors should be managed aggressively to improve the patient's survival and quality of life.
神经纤维瘤病 1 型是一种相对罕见的遗传性疾病。NF-I 同时发生三种类型的腹部肿瘤并引起肠套叠的情况很少见。我们报告了一例 NF-I 老年女性同时发生小肠胃肠道间质瘤、小肠腺癌和神经节细胞瘤合并肠套叠的病例。该患者表现为腹痛 2 个月,全身多发性神经纤维瘤和咖啡牛奶斑。增强 CT 显示末端回肠肿瘤明显强化,继发肠套叠。回肠肿瘤被认为是 NF-I 相关的肠神经纤维瘤。术后病理和免疫组化显示回盲部肿块为腺癌和神经节细胞瘤,术中探查还发现胃肠道间质瘤。在该患者中,术前影像学与病理结果不相符。无论是累及肠道的神经纤维瘤病还是伴有小肠肿瘤的神经纤维瘤病,都应积极治疗,以提高患者的生存率和生活质量。