Takahashi Yoshiaki, Takamiya Motonori, Ichimura Yuki, Okiyama Naoko, Nishino Ichizo, Morimoto Nobutoshi
Department of Neurology, Kagawa Prefectural Central Hospital.
Department of Neurology, Okayama National Hospital Medical Center.
Rinsho Shinkeigaku. 2023 Nov 23;63(11):737-742. doi: 10.5692/clinicalneurol.cn-001863. Epub 2023 Oct 25.
Case 1 involved a 68-year-old woman who was admitted to our hospital because of muscle weakness, diffuse subcutaneous edema, dysphagia, and an elevated serum creatine kinase level that had worsened within the previous month. Case 2 involved a 78-year-old woman who was admitted to our hospital because of muscle weakness, bilateral shoulder pain, diffuse subcutaneous edema, and dysphagia that had gradually worsened during the past 5 months. Both patients showed severe diffuse subcutaneous edema and dysphagia and underwent enteral tube feeding. Although they had no skin lesions consistent with dermatomyositis, muscle biopsies showed myxovirus resistance protein A (MxA) expansion, and blood tests showed positivity for anti-nuclear matrix protein 2 (anti-NXP-2) antibody. Therefore, both presents were diagnosed with anti-NXP-2 antibody-positive dermatomyositis sine dermatitis (DMSD). Anti-NXP-2 antibody-positive dermatomyositis has been reported to be closely associated with DMSD, severe edema and dysphagia. Differential diagnosis for patients who develop myositis with severe subcutaneous edema and dysphagia should include anti-NXP-2 antibody-positive dermatomyositis, and it is important to consider measurement of anti-NXP-2 antibody.
病例1为一名68岁女性,因肌肉无力、弥漫性皮下水肿、吞咽困难以及血清肌酸激酶水平升高(在前一个月内有所恶化)而入住我院。病例2为一名78岁女性,因肌肉无力、双侧肩部疼痛、弥漫性皮下水肿以及吞咽困难(在过去5个月中逐渐加重)而入住我院。两名患者均表现出严重的弥漫性皮下水肿和吞咽困难,并接受了肠内管饲。尽管她们没有与皮肌炎相符的皮肤病变,但肌肉活检显示黏液病毒抗性蛋白A(MxA)扩增,血液检查显示抗核基质蛋白2(抗NXP - 2)抗体呈阳性。因此,两名患者均被诊断为抗NXP - 2抗体阳性无皮疹皮肌炎(DMSD)。据报道,抗NXP - 2抗体阳性皮肌炎与DMSD、严重水肿和吞咽困难密切相关。对于出现伴有严重皮下水肿和吞咽困难的肌炎患者,鉴别诊断应包括抗NXP - 2抗体阳性皮肌炎,并且考虑检测抗NXP - 2抗体很重要。