Aggarwal Kunal, Fine Kerry, Wong David
Medical Education, Saint George's University School of Medicine, True Blue, GRD.
General Surgery, Arrowhead Regional Medical Center, Colton, USA.
Cureus. 2021 Oct 3;13(10):e18456. doi: 10.7759/cureus.18456. eCollection 2021 Oct.
Neurofibromatosis type 1 (NF1) is a multisystem genetic disorder characterized by café-au-lait macules on the skin, Lisch nodules of the iris, and predisposition to a wide array of tumors. These include neurofibromas, pheochromocytomas, and gastrointestinal stromal tumors (GIST). While there is documented evidence to suggest that the NF1 gene may play a role in the pathogenesis of transitional cell carcinoma (TCC) of the bladder, there is a paucity of documented cases of TCC in patients with NF1. Our patient is a 53-year-old male with a known diagnosis of NF1 and prior history of GIST who presented to the emergency department with lower abdominal pain, constipation, hematuria, and oliguria. The patient was found to have marked colonic distention prompting a decompressive cecostomy with subsequent return of bowel function. Cystoscopy was performed at this time for hematuria, which revealed a 9 cm bladder mass. Pathology showed a high-grade TCC of the bladder with nuclear pleomorphism and necrosis. The patient was treated with gemcitabine and cisplatin neoadjuvant chemotherapy, followed by cystoprostatectomy with bilateral pelvic lymphadenectomy and ileal conduit urinary diversion. Our case report is the first documented instance in the United States exhibiting an in vivo association of NF1 with the development of TCC of the bladder, an association previously identified in vitro. We hope our work inspires further investigation into this unique association.
1型神经纤维瘤病(NF1)是一种多系统遗传性疾病,其特征为皮肤上的咖啡斑、虹膜的Lisch结节,以及易患多种肿瘤。这些肿瘤包括神经纤维瘤、嗜铬细胞瘤和胃肠道间质瘤(GIST)。虽然有文献证据表明NF1基因可能在膀胱移行细胞癌(TCC)的发病机制中起作用,但NF1患者中记录在案的TCC病例很少。我们的患者是一名53岁男性,已知患有NF1,既往有GIST病史,因下腹痛、便秘、血尿和少尿就诊于急诊科。患者被发现有明显的结肠扩张,于是进行了减压性盲肠造口术,随后肠道功能恢复。此时因血尿进行了膀胱镜检查,发现膀胱有一个9厘米的肿块。病理显示为高级别膀胱TCC,伴有核多形性和坏死。患者接受了吉西他滨和顺铂新辅助化疗,随后进行了膀胱前列腺切除术、双侧盆腔淋巴结清扫术和回肠导管尿流改道术。我们的病例报告是美国第一例记录在案的NF1与膀胱TCC发生存在体内关联的实例,这种关联此前在体外已被确定。我们希望我们的工作能激发对这种独特关联的进一步研究。