Thamprajamchit Sriurai, Jariengprasert Chanchai, Rajatanavin Rajata
Department of Medicine, Faculty of Medicine, Ramathibodi Hospital, Mahidol University, Bangkok, Thailand.
Endocr Pract. 2004 Sep-Oct;10(5):432-7. doi: 10.4158/EP.10.5.432.
To report the case of a young woman with Graves' disease in whom ototoxicity developed because of propylthiouracil (PTU)-induced antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis.
We present the clinical findings, pertinent laboratory data, and follow-up course of a patient with Graves' disease and bilateral sensorineural hearing loss. We also provide a literature review regarding thionamide-induced ANCA-associated vasculitis.
In a 22-year-old Thai woman with Graves' disease, tinnitus, hearing impairment in the left ear (with progression to the right ear), and vertigo developed after 3 years of therapy with PTU. Audiograms showed bilateral sensorineural hearing loss with cochlear lesions. She also had low-grade fever, intermittent chronic abdominal pain, weight loss, polyarthralgia, and conjunctivitis. An indirect immunofluorescence test for ANCA was positive. The hearing impairment and tinnitus were gradually reduced after PTU withdrawal and corticosteroid and azathioprine treatment. Definitive therapy by subtotal thyroidectomy was subsequently performed and yielded good results.
This rare case of PTU-induced ANCA-associated vasculitis manifested with ototoxicity in combination with systemic involvement.
报告1例年轻女性格雷夫斯病患者,其因丙硫氧嘧啶(PTU)诱发抗中性粒细胞胞浆抗体(ANCA)相关血管炎而发生耳毒性。
我们介绍了1例格雷夫斯病合并双侧感音神经性听力损失患者的临床发现、相关实验室数据及随访过程。我们还提供了关于硫代酰胺诱发ANCA相关血管炎的文献综述。
1名22岁患格雷夫斯病的泰国女性,在接受PTU治疗3年后出现耳鸣、左耳听力障碍(后发展至右耳)及眩晕。听力图显示双侧感音神经性听力损失伴耳蜗病变。她还伴有低热、间歇性慢性腹痛、体重减轻、多关节痛及结膜炎。ANCA间接免疫荧光试验呈阳性。停用PTU并给予皮质类固醇和硫唑嘌呤治疗后,听力障碍和耳鸣逐渐减轻。随后进行了甲状腺次全切除术的确定性治疗,效果良好。
这例罕见的PTU诱发ANCA相关血管炎表现为耳毒性并伴有全身受累。