Interstitial cystitis: a rare manifestation of primary Sjögren's syndrome, successfully treated with low dose cyclosporine.
Chronic interstitial cystitis (IC), mostly affecting middle-aged women, is a very rare manifestation of primary Sjögren's syndrome (pSS). Hereby, we report a 42-year-old woman with pSS, presenting with dysuria, urinary frequency, and suprapubic pain. She was diagnosed to have chronic IC, based upon the cystoscopic biopsy finding of chronic inflammation in the bladder wall. Systemic corticosteroid and azathioprine treatments together with local intravesical therapies were not effective. Therefore, cyclosporine (CSA) therapy was initiated. Initial low dose of CSA (1.5 mg/kg/d) improved the symptoms of the patient, with no requirement for dose increment. After 4 months of therapy, control cystoscopic biopsy showed that bladder inflammation regressed and IC improved. This case suggests that even low doses of CSA may be beneficial for treating chronic IC associated with pSS syndrome.
Department of Internal Medicine, Division of Rheumatology, Ege University School of Medicine Hospital, 35100, Bornova, Izmir, Turkey. hakan.emmungil@ege.edu.tr
Hakan
H
Emmungil
Melike
M
Kalfa
Figen Yargucu
FY
Zihni
Gonca
G
Karabulut
Gokhan
G
Keser
Sait
S
Sen
Kenan
K
Aksu
D000328:Adult; D001706:Biopsy; D002908:Chronic Disease; D016572:Cyclosporine / Q000008:administration & dosage*; D018856:Cystitis, Interstitial / Q000175:diagnosis / Q000188:drug therapy* / Q000209:etiology / Q000276:immunology; D003558:Cystoscopy; D004359:Drug Therapy, Combination; D005260:Female; D006801:Humans; D007166:Immunosuppressive Agents / Q000008:administration & dosage*; D011241:Prednisone / Q000008:administration & dosage / Q000031:analogs & derivatives; D012859:Sjogren's Syndrome / Q000150:complications / Q000175:diagnosis / Q000188:drug therapy* / Q000276:immunology; D016896:Treatment Outcome
D007166:Immunosuppressive Agents; D016572:Cyclosporine; D011241:Prednisone
J Rheumatol. 1993 Jun;20(6):962-6
8350331
Urology. 1997 May;49(5A Suppl):58-63
9146003
Ann Rheum Dis. 2002 Jun;61(6):554-8
12006334
Curr Opin Rheumatol. 1996 Sep;8(5):438-45
8941447
Int Urogynecol J Pelvic Floor Dysfunct. 2000;11(2):84-6
10805264
J Urol. 2005 Nov;174(5):1837-40
16217303
Intern Med. 2004 Mar;43(3):248-52
15098610
J Urol. 2005 Dec;174(6):2235-8
16280777
Clin Nephrol. 1996 Mar;45(3):205-8
8706364
J Urol. 2004 Jun;171(6 Pt 1):2138-41
15126772
J Allergy Clin Immunol. 1996 Aug;98(2):389-99
8757216
Clin Exp Rheumatol. 1995 Sep-Oct;13(5):645-8
8575146
Clin Nephrol. 1992 May;37(5):239-44
1606774