Interstitial Cystitis in Sjögren’s Syndrome

dok_zlo — 09.02.2024

A 66-year-old woman with primary Sjögren’s syndrome presented to the rheumatology clinic with a 1-month history of lower abdominal pain and urinary urgency. One year before this presentation, she had received a diagnosis of Sjögren’s syndrome after presenting with sicca symptoms and arthralgias. Subsequent oral administration of pilocarpine had controlled her symptoms. The physical examination was notable for suprapubic pain and proximal interphalangeal joint tenderness. Laboratory testing showed elevated inflammatory markers, mild acute kidney injury, and mild hematuria and pyuria on urinalysis. A urine culture was negative. Computed tomography of the abdomen showed bladder-wall thickening (Panel A, arrow) and hydronephrosis in both kidneys without nephrolithiasis (Panel B, asterisks). The results of bladder biopsy were negative for cancer. Histopathological analysis showed lymphocytic and plasmacytic infiltration, ulceration, and fibrin deposition of the bladder wall, as well as lymphoid follicle formation (Panel C, hematoxylin and eosin stain). A diagnosis of interstitial cystitis associated with Sjögren’s syndrome — a rare extraglandular feature of the autoimmune condition — was made. Oral prednisolone and mycophenolate mofetil were initiated. The patient’s renal function improved after 3 months of treatment, and her urinary symptoms had abated by 4 months. Repeat imaging that was performed 6 months after the initial presentation showed resolution of the hydronephrosis.Hirotaka Yamamoto, M.D. Yoshinori Taniguchi, M.D. Kochi Medical School, Nankoku, Japan February 8, 2024 N Engl J Med 2024; 90:548 DOI: 10.1056/NEJMicm2308925

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