Shibata Shigeru, Ubara Yoshifumi, Sawa Naoki, Tagami Tetsuo, Hosino Junichi, Yokota Masafumi, Katori Hideyuki, Takemoto Fumi, Hara Shigeko, Takaichi Kenmei, Fujii Akiko, Murata Hirokatu, Nishi Tadahiro
Nephrology Center, Toranomon Hospital, Tokyo.
Intern Med. 2004 Mar;43(3):248-52. doi: 10.2169/internalmedicine.43.248.
A 53-year-old woman presented with oliguria, urinary frequency, abdominal pain and severe edema of the lower extremities. Her serum creatinine was 8.1 mg/dl. Computed tomographic and ultrasonographic studies showed a severely dilated urinary bladder, and bilateral hydroureteronephrosis. Examination of a urinary bladder biopsy specimen showed subepithelial edema and infiltration by lymphocytes and plasmacytes. However, the patient complainted of dry mouth and dry eyes. Ophthalmologically, the Schirmer test was positive. A biopsy of the minor salivary glands in the lip showed chronic sialoadenitis. A diagnosis of Sjögren's syndrome complicated by interstitial cystitis was made. Since she had been anuric, secondary to urinary obstruction, intermittent self-catheterization was started. Combination of corticosteroid and cyclosporin therapy was initiated. Spontaneous urination began, and gradually the patient's symptoms remitted. After 8 months of therapy, bladder capacity increased from 140 ml to 350 ml, and she voided approximately 1,200 ml by herself and 600 ml by catheterization daily. This case suggests that when severe interstitial cystitis is associated with Sjögren's syndrome, a therapeutic trial of corticosteroids and cyclosporin may be beneficial.
一名53岁女性出现少尿、尿频、腹痛及双下肢严重水肿。她的血清肌酐为8.1mg/dl。计算机断层扫描和超声检查显示膀胱严重扩张，双侧肾盂输尿管积水。膀胱活检标本检查显示上皮下水肿以及淋巴细胞和浆细胞浸润。然而，患者主诉口干、眼干。眼科检查显示泪液分泌试验阳性。唇部小唾液腺活检显示慢性涎腺炎。诊断为干燥综合征合并间质性膀胱炎。由于她因尿路梗阻而无尿，遂开始间歇性自我导尿。开始使用皮质类固醇和环孢素联合治疗。患者开始自主排尿，症状逐渐缓解。治疗8个月后，膀胱容量从140ml增加到350ml，她每天自主排尿约1200ml，通过导尿排出600ml。该病例表明，当严重间质性膀胱炎与干燥综合征相关时，皮质类固醇和环孢素的治疗性试验可能有益。
