Arman Farid, Shakeri Hania, Nobakht Niloofar, Rastogi Anjay, Kamgar Mohammad
Department of Medicine, University of California, Los Angeles, CA, USA.
Department of Pathology, Shahid Beheshti University of Medical Sciences, Tehran, Iran.
Am J Case Rep. 2017 Jun 3;18:622-626. doi: 10.12659/ajcr.903476.
BACKGROUND Sjögren's syndrome is an autoimmune disorder caused by the infiltration of monocytes in epithelial glandular and extra-glandular tissues. Hallmark presentations include mouth and eye dryness. Although renal involvement is uncommon in primary Sjögren's syndrome (pSS), patients may experience renal tubular acidosis type I (RTA I), tubulointerstitial nephritis, diabetes insipidus (DI), nephrolithiasis, and Fanconi syndrome. However, it is atypical to see more than 1 of these manifestations in a single patient. CASE REPORT We present the case of a 24-year-old woman with polyuria and polydipsia, who was initially diagnosed with nephrogenic diabetes insipidus. She also had chronic hypokalemia and nephrolithiasis. Based on clinical presentation and work up, she was diagnosed with pSS and treated accordingly. CONCLUSIONS This was a pSS patient with tubulointerstitial nephritis, diabetes insipidus, renal tubular acidosis, hypokalemia, and nephrolithiasis, who was receiving symptomatic treatment for diabetes insipidus. Diagnosis and treatment of pSS led to significant improvement in systemic and renal presentations of the patient. pSS should be considered as one of the differential diagnoses in patients with diabetes insipidus and renal tubular acidosis.
干燥综合征是一种自身免疫性疾病,由单核细胞浸润上皮腺性和腺外组织引起。标志性表现包括口干和眼干。虽然肾脏受累在原发性干燥综合征(pSS)中并不常见,但患者可能会出现I型肾小管酸中毒(RTA I)、肾小管间质性肾炎、尿崩症(DI)、肾结石和范科尼综合征。然而,在单一患者中出现超过1种这些表现是不典型的。病例报告:我们报告一例24岁多尿、烦渴的女性患者,最初被诊断为肾性尿崩症。她还患有慢性低钾血症和肾结石。根据临床表现和检查,她被诊断为pSS并接受了相应治疗。结论:这是一名患有肾小管间质性肾炎、尿崩症、肾小管酸中毒、低钾血症和肾结石的pSS患者,正在接受尿崩症的对症治疗。pSS的诊断和治疗使患者的全身和肾脏表现有了显著改善。pSS应被视为尿崩症和肾小管酸中毒患者的鉴别诊断之一。