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颊面部非霍奇金淋巴瘤误诊为炎性肌病六例临床特点分析

Clinical features of non-Hodgkin's lymphoma in cheek misdiagnosed as inflammatory myopathy

摘要:

目的 探讨误诊为炎性肌病的淋巴瘤患者的临床特点及误诊原因,以提高对此类病例的认识.方法 收集北京协和医院普通内科2010年1月至2014年12月期间临床首发表现为单侧颊面部肿胀且曾被误诊为炎性肌病,人院后最终确诊为非霍奇金淋巴瘤患者的临床资料,回顾性总结分析其临床表现、实验室检查及诊断经过.结果 符合筛选条件患者6例,其中男性3例,女性3例,中位年龄30岁.6例患者从发病到最终明确诊断经历中位诊断时间为13个月,平均转诊4家医院,经历3次活组织检查,病程中均应用半量或足量激素,但激素减量过程中均出现病情再次加重.6例患者均无肌无力、肌痛表现,2例患者出现单侧前臂肿胀,3例患者肌酸激酶轻度升高,4例患者肌电图为肌源性损害.6例患者颊面部病程早期组织活检为肌炎合并或不合并小血管炎表现.6例患者ESR及超敏C反应蛋白均无明显升高,抗Jo-1、ANA及抗U1RNP抗体均阴性.5例患者外周血EB病毒(EBV)-DNA显著升高,3例患者的病理组织进行了EBV原位杂交检测均为阳性.3例患者进行了PET-CT检查,面颊局部提示代谢增高,SUV值为2.0~8.5.对所有患者发病初期活检病理标本进行复习,均未发现淋巴瘤诊断证据.最终患者经颊面部(3/6)、唇部(2/6)及前臂皮肤(1/6)再次活组织病理检查确诊为非霍奇金淋巴瘤,2例为T细胞淋巴瘤,4例为NK/T细胞淋巴瘤.结论 临床出现单侧颊面部肿胀受累,如无皮肌炎或多发性肌炎近端肌无力的典型临床表现特点时,即使病理检查结果提示炎性肌病,亦应警惕淋巴瘤的可能,明确诊断可能需要多次活组织病理检查.

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abstracts:

Objective To assess the clinical features of non-Hodgkin's lymphoma in cheek misdiagnosed as inflammatory myopathy.Methods Six patients presenting with unilateral swollen cheek were admitted at the Department of General Internal Medicine,Peking Union Medical College Hospital between January 2010 and December 2014.These patients were misdiagnosed as inflammatory myopathy and finally confirmed as Non-Hodgkin's lymphoma.The clinical characteristics,findings of laboratory tests and the diagnostic procedures were retrospectively analyzed.Results The median age of 6 patients (3 males and 3 females) was 30 years.The median duration from onset of unilateral swollen cheek to the final diagnosis was 13 months;the patients were referred to average 4 hospitals and experienced average 3 biopsies before admission.All patients had been treated with prednisone at a dose of 1 mg/kg or 0.5mg/kg per day.However,the symptoms relapsed in all patients after glucocorticoid tapering.No patient presented with proximal muscle weakness or myalgia.Two of 6 patients presented with unilateral forearm swollen.Creatine kinase was mildly elevated in 3 patients.Electromyographic abnormalities were seen in 4 patients.The biopsy showed muscle fiber necrosis,degeneration and a large amount of lymphocytes infiltration with or without vessel wall involvement.Erythrocyte sedimentation rate and high-sensitive C-reactive protein levels were normal.Anti-Jo-1,ANA and anti-U1RNP were all negative.EBV-DNV was significantly elevated in peripheral blood in 5 patients and it was positive in biopsy specimen in 3 patients.PET-CT scan was performed in 3 patients and suggested high metabolic activity with a SUV of 2.0-8.5 in cheek region.The biopsy specimen taken before admission did not show evidence of lymphoma.Repeated biopsies from cheek regions (3/6),lips (2/6) and forearm confirmed the final diagnosis as Non-Hodgkin's lymphoma,including 2 cases of T-cell lymphoma and 4 cases of NK/T cell lymphoma.Conclusion When patients present with unilateral swollen cheek without proximal skeletal muscle weakness or myalgia,even the pathologic evaluation implies inflammatory myopathy,non-Hodgkin's lymphoma should be considered.Repeated biopsy may be necessary to confirm the diagnosis.

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