Takada Toshinori, Asakawa Katsuaki, Barrios Roberto
Division of Respiratory Medicine, Niigata University Medical and Dental Hospital, Niigata, Japan.
Uonuma Institute of Community Medicine, Niigata University Medical and Dental Hospital, Niigata, Japan.
Clin Rheumatol. 2021 Mar;40(3):1159-1165. doi: 10.1007/s10067-020-05292-0. Epub 2020 Jul 16.
Patients with clinically amyopathic dermatomyositis (CADM) have a risk of developing rapidly progressive interstitial lung disease (ILD). CADM-ILD is associated with the anti-MDA-5 antibody. In the USA, however, patients with CADM have these antibodies less frequently than those in Japan. In addition, those with this disorder are less often complicated with rapidly progressive ILD than those in Japan. We present a case of a 42-year-old Japanese-American female with a 3-month history of a rash on her hands and face with joint pain. Based on the negative results from lupus tests, her primary care provider and a rheumatologist treated her with steroids, hydroxychloroquine, and methotrexate. During treatment, the patient started noticing shortness of breath because of pneumonia, which was revealed by a CT scan. The woman was finally diagnosed with acute respiratory failure due to CADM with ILD. She underwent a double lung transplant as well as treatment with multiple immunosuppressive agents and repeated plasma exchange but died 15 days after transplantation. Her clinical course is similar to that of Japanese patients with CADM-ILD. Outside Japan, primary care providers, rheumatologists, and dermatologists, as well as pulmonary physicians, may be less familiar with this disorder than those in Japan. Since CADM-ILD progresses very quickly and could be fatal, these doctors should be aware of this disease to treat such patients as soon as possible, particularly when seeing a patient of Japanese descent.
临床上无肌病性皮肌炎(CADM)患者有发生快速进展性间质性肺病(ILD)的风险。CADM-ILD与抗MDA-5抗体相关。然而,在美国,CADM患者携带这些抗体的频率低于日本患者。此外,与日本患者相比,患有这种疾病的患者较少并发快速进展性ILD。我们报告一例42岁的日裔美国女性病例,她有手部和面部皮疹伴关节疼痛3个月的病史。基于狼疮检查的阴性结果,她的初级保健医生和风湿病学家用类固醇、羟氯喹和甲氨蝶呤对她进行治疗。治疗期间,患者因肺炎开始出现呼吸急促,CT扫描显示了这一情况。该女性最终被诊断为因CADM合并ILD导致的急性呼吸衰竭。她接受了双肺移植以及多种免疫抑制剂治疗和重复血浆置换,但在移植后15天死亡。她的临床病程与日本CADM-ILD患者相似。在日本以外地区,初级保健医生、风湿病学家、皮肤科医生以及肺科医生可能比日本同行对这种疾病了解较少。由于CADM-ILD进展非常迅速且可能致命,这些医生应该了解这种疾病,以便尽快治疗此类患者,尤其是在诊治日裔患者时。