Azuma Kota, Tamura Masao, Kurajoh Masafumi, Hosono Yuji, Nakajima Ran, Tsuboi Kazuyuki, Abe Takeo, Ogita Chie, Yokoyama Yuichi, Furukawa Tetsuya, Yoshikawa Takahiro, Saito Atsushi, Nishioka Aki, Sekiguchi Masahiro, Azuma Naoto, Kitano Masayasu, Tsunoda Shinichiro, Omura Koichiro, Koyama Hidenori, Matsui Kiyoshi, Mimori Tsuneyo, Sano Hajime
Division of Rheumatology, Department of Internal Medicine, Hyogo College of Medicine.
Nihon Rinsho Meneki Gakkai Kaishi. 2016;39(6):538-544. doi: 10.2177/jsci.39.538.
A 48-year-old woman had suffered from a fever and general fatigue, and visited the other hospital for fever elevation in November 2013, at which time interstitial lung disease was revealed. In January 2014, she experienced an eruption in the hand and developed peripheral blood flow damage. Under a diagnosis of adult Still's disease, the patient was administered 0.5 mg of betamethasone as well as cyclosporin at 75 mg/day. In November 2014, general fatigue, fever, and headache were noted, while MRI revealed an enlarged hypophysis and laboratory findings were positive for the anti-pituitary cell antibody, thus a diagnosis of autoimmune hypophysitis was made. Although disease activity was low, she requested hospitalization and was admitted by the Division of Endocrinology and Metabolism at our hospital in May 2015, though only observed. Fever developed again, along with interstitial lung disease, Raynaud's phenomenon, and pain in the crural area again, and we considered the possibility of another disease. After stopping administration of betamethasone and cyclosporin, we made a diagnosis of anti-aminoacyl tRNA synthetase antibody syndrome, and administered methylprednisolone at 500 mg for 3 days as well as prednisolone at 35 mg/day following steroid pulse therapy. Although her condition soon improved, fever, muscle pain, and pancytopenia returned after 3 days. Bone marrow findings revealed the existence of hemophagocytosis, for which we again gave methylprednisolone at 500 mg for 3 days and cyclosporin at 125 mg/day. Thereafter, the patient recovered and was discharged from the hospital.
一名48岁女性曾出现发热和全身乏力症状,2013年11月因发热加剧前往其他医院就诊,当时被诊断为间质性肺病。2014年1月,她手部出现皮疹,并伴有外周血流损伤。在诊断为成人斯蒂尔病后,患者接受了0.5毫克倍他米松以及每天75毫克环孢素的治疗。2014年11月,患者出现全身乏力、发热和头痛症状,磁共振成像显示垂体增大,抗垂体细胞抗体实验室检查结果呈阳性,因此诊断为自身免疫性垂体炎。尽管疾病活动度较低,但她要求住院治疗,于2015年5月被我院内分泌代谢科收治,仅进行观察。之后再次出现发热,同时伴有间质性肺病、雷诺现象和小腿疼痛,我们考虑可能患有其他疾病。停用倍他米松和环孢素后,诊断为抗氨酰tRNA合成酶抗体综合征,给予甲泼尼龙500毫克静脉滴注3天,之后给予泼尼松龙35毫克/天进行激素冲击治疗。尽管她的病情很快得到改善,但3天后发热、肌肉疼痛和全血细胞减少症再次出现。骨髓检查发现存在噬血细胞现象,为此我们再次给予甲泼尼龙500毫克静脉滴注3天,并给予环孢素125毫克/天。此后患者康复出院。