Division of Nephrology, Department of Internal Medicine, Showa University Northern Yokohama Hospital, Chigasaki-chuo 35-1, Tsuzuki, Yokohama, Kanagawa, 224-8503, Japan.
Pathology and Laboratory Medicine, Showa University Northern Yokohama Hospital, Kanagawa, Japan.
CEN Case Rep. 2023 May;12(2):189-194. doi: 10.1007/s13730-022-00741-y. Epub 2022 Oct 18.
Anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) is a relatively rare form of autoimmune disease. Diabetes insipidus (DI) is characterized by diluted polyuria and thirstiness, and is clinically categorized into central and nephrogenic DI depending on damaged organs. In most previously reported cases, ANCA-related disorders have been implicated in central DI, which is attributed to impaired secretion of arginine vasopressin (AVP) from the posterior pituitary. However, no previous case of AAV-related nephrogenic DI has been reported in the English literature. Herein, we report a case of nephrogenic DI likely caused by AAV. A 76-year-old man was admitted to our hospital for acute kidney injury. He showed dehydration, polyuria, and polydipsia. Laboratory tests demonstrated elevated levels of serum urea and creatinine and a high myeloperoxidase ANCA titer. In the present case, both plasma AVP concentration and response of AVP secretion to 5% saline load test were normal. In addition, 1-desmino-8-arginine vasopressin administration could not increase urinary osmolarity. Kidney biopsy specimen revealed tubulointerstitial nephritis with findings that appeared to indicate peritubular capillaritis. Therefore, the patient was diagnosed with nephrogenic DI likely owing to ANCA-associated tubulointerstitial nephritis. Immediately after prednisolone administration, urinary volume decreased, urinary osmolarity increased, and kidney function was improved. This case demonstrates that AAV that extensively affects the tubulointerstitial area can result in nephrogenic DI.
抗中性粒细胞胞质抗体(ANCA)相关性血管炎(AAV)是一种相对罕见的自身免疫性疾病。尿崩症(DI)的特征是稀释性多尿和口渴,并根据受损器官的不同,临床上分为中枢性和肾性 DI。在大多数先前报道的病例中,与 ANCA 相关的疾病与中枢性 DI 有关,这归因于垂体后叶精氨酸血管加压素(AVP)分泌受损。然而,在英语文献中,以前没有报道过与 AAV 相关的肾性 DI 病例。在此,我们报告一例可能由 AAV 引起的肾性 DI。一名 76 岁男性因急性肾损伤入院。他表现为脱水、多尿和多饮。实验室检查显示血清尿素和肌酐水平升高,髓过氧化物酶 ANCA 滴度升高。在本病例中,血浆 AVP 浓度和 AVP 分泌对 5%盐水负荷试验的反应均正常。此外,1-去氨基-8-精氨酸血管加压素给药不能增加尿渗透压。肾活检标本显示肾小管间质性肾炎,表现为小管间毛细血管炎。因此,患者被诊断为可能因 ANCA 相关性肾小管间质性肾炎引起的肾性 DI。泼尼松龙给药后,尿量减少,尿渗透压增加,肾功能改善。该病例表明,广泛影响肾小管间质区域的 AAV 可导致肾性 DI。