Zarachi Athina, Pelechas Eleftherios, Tsikou Alkistis, Kalambokis Georgios N, Zioga Aikaterini, Panteli Katerina E, Rapti Iro, Filippas-Ntekouan Sempastian, Kastanioudakis Ioannis, Karassa Fotini B
Department of Otorhinolaryngology, Faculty of Medicine, School of Health Sciences, University of Ioannina, Ioannina, Greece.
Department of Rheumatology, Faculty of Medicine, School of Health Sciences, University of Ioannina, Ioannina, Greece.
Mediterr J Rheumatol. 2022 Sep 30;33(3):349-360. doi: 10.31138/mjr.33.3.349. eCollection 2022 Sep.
Autoimmune hepatitis (AIH) is characterized by elevated serum transaminase, increased immunoglobulin G levels, presence of autoantibodies, and hepatocellular damage. Coexistence with other autoimmune diseases has been reported in almost half of patients with AIH. Here, we report a 60-year-old man who developed rapidly progressive, bilateral, asymmetrical, and asynchronous sensorineural hearing loss that was consistent with immune-mediated inner ear disease (IMIED). This devastating presentation evolved as a late manifestation in the context of a six-month systemic illness that had previously resulted in type 1 AIH. A biochemical remission with normalization of aminotransferases achieved within two months after the initiation of corticosteroids with azathioprine. Further, an acceptable response has also been achieved at the patient regarding the right ear-hearing impairment; though, treatment could not reverse the substantial decrement in hearing capability of the left ear. To our knowledge, this is the first case report of the concurrent development of type 1 AIH and IMIED.
自身免疫性肝炎(AIH)的特征是血清转氨酶升高、免疫球蛋白G水平升高、存在自身抗体以及肝细胞损伤。几乎一半的AIH患者报告有与其他自身免疫性疾病共存的情况。在此,我们报告一名60岁男性,他出现了快速进展的双侧、不对称和不同步的感音神经性听力损失,这与免疫介导的内耳疾病(IMIED)一致。这种严重的表现是在先前导致1型AIH的为期6个月的全身性疾病背景下作为晚期表现出现的。在使用硫唑嘌呤的皮质类固醇治疗开始后两个月内,转氨酶恢复正常,生化指标缓解。此外,患者右耳听力障碍也取得了可接受的反应;然而,治疗未能逆转左耳听力能力的大幅下降。据我们所知,这是1型AIH和IMIED同时发生的首例病例报告。