Banks Kian C, Hsu Diana S, Velotta Jeffrey B
J Surg Case Rep. 2022 Jan 26;2022(1):rjab636. doi: 10.1093/jscr/rjab636. eCollection 2022 Jan.
A 50-year-old male with history of HIV, syphilis, paraneoplastic Morvan syndrome secondary to thymoma resected in 2013 presented recently with tachycardia, tremors, diarrhea, hyperhidrosis and bilateral lower extremity pain leading to the discovery of thymoma recurrence. He initially developed Morvan Syndrome after thymectomy in 2013 and gradually improved with negative anti-contactin-associated protein-like 2 antibody testing in 2017 and symptom resolution in 2018. Upon return of dysautonomia symptoms, subsequent imaging revealed widespread disease recurrence diffusely in the right lung parenchyma and pleura for which he underwent right extrapleural pneumonectomy. He was managed with low-dose prednisone perioperatively, but when his symptoms worsened, he was started on rituximab and methylprednisolone. Nearly 3 months from surgery, he died from urinary sepsis. This represents a unique case of recurrent paraneoplastic Morvan syndrome leading to the diagnosis of metastatic thymoma as well as the challenges of symptom control during the surgical management of the underlying disease.
一名50岁男性,有HIV、梅毒病史,2013年因胸腺瘤切除术后继发副肿瘤性莫旺综合征,近期出现心动过速、震颤、腹泻、多汗及双下肢疼痛,从而发现胸腺瘤复发。他最初在2013年胸腺切除术后患上莫旺综合征,并在2017年抗接触蛋白相关蛋白样2抗体检测呈阴性且症状于2018年缓解后逐渐好转。自主神经功能障碍症状复发后,后续影像学检查显示右肺实质和胸膜广泛复发,为此他接受了右胸膜外全肺切除术。围手术期给予低剂量泼尼松治疗,但症状恶化时,开始使用利妥昔单抗和甲泼尼龙。术后近3个月,他死于尿脓毒症。这是一例复发性副肿瘤性莫旺综合征导致转移性胸腺瘤诊断的独特病例,以及基础疾病手术治疗期间症状控制的挑战。