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The gut as a causative factor in rheumatic disease has fascinated many researchers and physicians. We present an unexpected link between gut and arthritis and report an exceptional case of inflammatory seronegative arthritis that happened to be associated with chronic appendicitis.
A 52 year old, otherwise healthy woman presented with three months of migratory arthritis, which turned during the fourth month into an additive course involving symmetrically the wrists, ankles, metacarpophalangeal (MCP) joints, and left shoulder. She also reported morning stiffness and fatigue of more than one hour. She denied that she had had fever, skin eruption, psoriasis, aphtae, gastrointestinal or genitourinary symptoms or a familial history of rheumatic conditions, psoriasis or inflammatory bowel diseases. Physical examination was normal except for symmetric synovitis involving both ankles and wrists, the left 3rd and 4th and the right 2nd MCP joints. Laboratory tests disclosed an erythrocyte sedimentation rate (ESR) of 25 mm 1st h; serum C reactive protein, haemoglobin and thrombocytes count were normal; white blood cell count was 13 700/mm3, with 79% neutrophils, 13.8 % lymphocytes, 5.6% monocytes, 0.8 % eosinophils and 0.8% basophils. The results of blood chemistry tests including uric acid, calcium, phosphorus, liver enzymes, and creatinine were normal. Urine analysis disclosed mild microhaematuria. Rheumatoid factor and antinuclear antibodies were negative. Radiographs of the hands and feet showed soft tissue swelling and a chest radiograph was normal.
The patient was treated with naproxen with partial improvement of her arthritis. Ten days after admission, she complained of severe sudden abdominal pain spreading within an hour from the epigastrium to the entire abdomen. Her fever rose to 38.5°C. The abdomen was slightly more tender in the right lower quadrant without defence or rebound. Normal peristalsis sounds were heard. Abdominal plain radiography was normal. ESR increased to 60 mm 1st h and the white blood cell count to 29 000/mm3. Urine analysis showed microhaematuria. Blood cultures were later found to be sterile. The surgeon did not diagnose an acute surgical abdomen; gynaecological examination was normal. The patient recalled a similar episode of transient abdominal pain two months previously. Cefuroxime was prescribed. Within 24 hours, her abdominal symptoms and fever resolved completely. An abdominal ultrasound study and a cystoscopy—because of persistent microhaematuria—were normal. Repeated urine cultures were found to be sterile. Beside the above mentioned brief episode, the patient remained completely asymptomatic regarding the abdominal symptoms, but despite naproxen, she still had active polyarthritis. Weekly methotrexate at a dose of 7.5 mg was started with the working diagnosis of seronegative rheumatoid arthritis (RA). After receiving five doses of methotrexate, in a routine outpatient visit, her joint symptoms were slightly improved but she reported another episode of acute abdominal pain two weeks previously that resolved spontaneously within hours. A gastrointestinal barium study was ordered. A week later, the patient was admitted to the emergency ward with severe abdominal pain and fever of 39°C. On laparotomy a perforated appendix with local adhesions was identified and removed. Pathological studies confirmed acute suppurative appendicitis without evidence of Crohn’s disease or granulomatous lesions. The day after the operation, the patient experienced a dramatic resolution of her polyarthritis. Methotrexate was stopped at the time of appendicectomy. Since then and during two years of further follow up the patient is still free of any musculoskeletal symptoms.
Although the relation between an acute to chronic appendicitis and polyarthritis in this patient could have been serendipitous, the fact that the established RA-like syndrome resolved immediately, completely, and permanently after the appendicectomy strongly suggests a causative link between the two entities.
Retrospectively, the patient had at least four episodes of acute appendicitis—the fourth one evolving into perforation and acute localised appendicitis. During the past 15 years several convincing reviews have established that besides acute appendicitis, the “appendicitis repertoir” also includes recurrent bouts of acute as well as longstanding appendicitis.1 2 Nevertheless, our awareness of atypical forms of appendicitis is still low. Appendicitis as a cause of rheumatic symptoms has been described in two cases.3 4 In both of them, the abdominal presentation was atypical while the rheumatic symptoms were prominent. In one case, appendicitis, later found to be granulomatous, was found on abdominal computed tomography examination of a patient with a three month duration migratory arthritis and septicaemia—without abdominal symptoms. The second case was a 10 year old girl with 19 months of episodic colicky abdominal pain and nine months migratory polyarthritis. The underlying process was found to be an acute on chronic appendicitis that finally perforated. Appendicectomy in both these cases resulted in prompt resolution of the rheumatic symptoms.
The flora of appendicitis does not qualitatively differ from that of the normal appendix, Escherichia coli andBacteroides fragilis being the most commonly encountered organism.5 A possible link between appendicitis and arthritis could be Yersinia. Although pseudoappendicular syndrome is a prevalent manifestation ofYersinia in adults,5 occasional reports of true appendicitis do exist.6 On the other hand, a known complication of yersiniosis is reactive arthritis7typically persisting 1–4 months after resolution of the gastrointestinal symptoms.8
Sometimes, trivial diseases may offer a diagnostic challenge. Polyarthritis is a frequent medical problem and acute appendicitis is one of the most banal surgical conditions. The lesson to be learnt from this case report is the challenge to the physician of recognising this unexpected yet possible encounter between the two.
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