A 73-year-old woman with chronic pelvic pain, burning toes, and an eighty-pound weight loss.

Abstract

A 73-year-old woman developed malaise and a wasting illness that led to a weight loss of 80 pounds over the 5 years before presentation. During this time, her body mass index declined from 30.3 to 17.0 kg/m (1). At approximately the same time her weight loss began, the patient began to experience chronic pelvic and bladder pain. These symptoms, particularly uncomfortable when she was sitting, were partly relieved by micturition. The pain was associated with intermittent dysuria, severe urinary urgency, and frequency. She urinated approximately 9 times a day, and also awoke to urinate approximately 9 times at night. She denied incontinence and did not have recurrent urinary tract infections. However, her symptoms were associated with dyspareunia. She was not sexually active because of pain. Gynecologic and urologic evaluations revealed that the external genitalia, perineum, anus, and rectum were normal, as was the strength of the pubococcygeal and external anal sphincter muscles. Her gynecologist had detected mild urethral tenderness on palpation, but there was no urethral or bladder prolapse. The cul-desac between the posterior vagina and anterior rectum was normal, without nodularity or an enterocele. Catheterized urine specimens showed reactive uroepithelial cells, many neutrophils, and some red blood cells, suggesting abundant acute inflammation. A computed tomography (CT) scan of the pelvis showed generalized thickening of the bladder wall (Figure 1). A cystoscopic examination revealed no uroepithelial masses, but demonstrated a trabeculated bladder surface. Biopsies obtained at cystoscopy revealed nonspecific bladder inflammation. No neoplasm was identified. Following these urologic evaluations, the patient was diagnosed with interstitial cystitis/painful bladder syndrome (IC/PBS) (2). Over the next 5 years, she was treated with a variety of medications, including neurontin, pentosan polysulfate sodium, oxybutynin, and dimethyl sulfoxide. She also underwent cystoscopy with hydrodistention. None of these interventions provided more than mild, temporary relief of her bladder symptoms. In this same 5-year period, the patient had a variety of persistent abdominal symptoms. She reported loose bowel movements but denied diarrhea, constipation, hematochezia, fevers, arthritis, or eye problems. She noted consistent anorexia and early satiety, and reported a band-like abdominal pain that started just above her umbilicus. She had grown increasingly weak since becoming ill, and usually required assistance for ambulation. Over the course of her abdominal symptoms, she had undergone upper and lower endoscopy, as well as sonograms and CT scans of her abdomen. Biopsies of the small intestine had been negative for Tropheryma whipplei. Assays for IgA anti-endomysial antibodies, IgA antibodies to transglutaminase, and IgG and IgA directed against gliadin were all negative. A variety of diagnoses for her gastrointestinal symptoms had been entertained, including the postcholecystectomy syndrome, lactose intolerance, and Julius Birnbaum, MD, Michael Polydefkis, MD, MHS: The Johns Hopkins University School of Medicine, Baltimore, Maryland; Toby C. Chai, MD, Tehmina Z. Ali, MD: University of Maryland School of Medicine, Baltimore; John H. Stone, MD, MPH: Massachusetts General Hospital, Boston. Address correspondence to John H. Stone, MD, MPH, Massachusetts General Hospital, 55 Fruit Street, Yawkey 2, Boston, MA 02114. E-mail: jhstone@partners.org. Submitted for publication April 2, 2008; accepted in revised form July 31, 2008. Figure 1. Computed tomography scan of the bladder. Arthritis & Rheumatism (Arthritis Care & Research) Vol. 59, No. 12, December 15, 2008, pp 1825–1831 DOI 10.1002/art.24051 © 2008, American College of Rheumatology