Rheumatology 2001; 40: 351-353
© 2001 British Society for Rheumatology
Letters to the Editor |
First presentation of intestinal bypass syndrome 18 yr after initial surgery
University Department of Dermatology, Kantonsspital Basel, Petersgraben 4–6, 4031 Basel and
1 University Department of Rheumatology, Felix-Platter-Spital, Burgfelder Strasse 101, 4055 Basel, Switzerland
SIR, From the 1950s to the 1980s, intestinal bypass operations were used to reduce weight in morbidly obese patients [1]. Loss of weight was often successful but bacterial overgrowth of the blind loop caused secondary, extraintestinal complications such as arthritis and vasculitis. The so-called intestinal bypass syndrome affected about 20% of the surgically treated patients [2]. We describe a patient with therapy-refractory oligoarthritis and necrotizing cutaneous vasculitis that appeared for the first time 18 yr after an intestinal bypass operation.
The patient, a 40 yr old male weighing 140 kg, with a history of a jejuno-ileostomy in 1979 for morbid obesity and no other history of note, developed arthritis of the right knee in 1997 without antecedent trauma. Six weeks prior to the onset of symptoms, a diagnostic laparoscopy with division of adhesions had been carried out due to the development of acute intestinal colic. He then developed repeated knee effusions requiring repeated aspiration, intra-articular steroids and athroscopic synovectomy. In January 1999, an open synovectomy was performed which showed a chronic, fibrinous, purulent synovitis in the histology. In March 1999, during his rehabilitation, he developed a bilateral plantar fasciitis more marked on the left foot, pustulous erythema nodosum-like skin lesions on both lower legs and arthritis of his left elbow. Synovial fluid aspirates from the right knee and the left elbow were sterile and without evidence of crystal arthropathy, but the leucocyte count was elevated to 70 000 cells/µl. Cultures of blood, urine and stool were negative and a transoesophageal echocardiography showed no evidence of endocarditis. A throat swab was negative for haemolytic streptococci, as was serology for Chlamydia, Brucella, Yersinia and Salmonella, hepatitis viruses, HIV, cytomegalovirus, Epstein–Barr virus and toxoplasmosis. Rheumatoid factor, antinuclear antibodies, antineutrophil cytoplasmic antibodies and cryoglobulins were also negative. A duodenal biopsy revealed no evidence of Whipple's disease. A biopsy of one of the skin lesions showed subacute inflammation of the subcutaneous tissue with fat necrosis and a florid focal endovasculitis. Under therapy with high-dose prednisone (50 mg/day) and oral cyclophosphamide (150 mg/day) for 2 weeks, the patient showed rapid improvement, but after stopping the cyclophosphamide and reduction of the prednisone, weekly methotrexate 20 mg i.m. and from July 1999, sulphasalazine 2 g per day had to be introduced to stabilize the arthritis.
In January 2000 the patient was again admitted to hospital with a large cutaneous ulcer of the lateral border of the left foot, which had formed within a few weeks (Fig. 1
). In addition, he developed an active synovitis of the left elbow, the right knee and the right hip. The biopsy of the cutaneous ulcer showed a necrotizing leucocytoclastic vasculitis with obstruction of the small vessels and terminal endarteritis of the medium-sized vessels (Fig. 2). In an immunofluorescence investigation, deposits of complement and some IgM were evident in the small vessels, but no IgA or IgG. There was no evidence for arteriosclerosis or a cardiac source of emboli. Microscopy of the urine was unremarkable, a urethral swab excluded gonococci and Chlamydia trachomatis and a polymerase chain reaction test for the latter in the early morning urine was also negative. The C-reactive protein concentration was within the normal range but there was slight leucocytosis (12.65x109/l, 87.6% neutrophils). Due to the non-responsiveness of the cutaneous ulcer or arthritis to an increase of the methotrexate dose to 30 mg i.m. weekly and prednisone to 50 mg daily, we now considered the intestinal bypass syndrome as a possible cause for the refractory arthritis and vasculitic ulcer. After treatment with doxycycline 100 mg daily for 2 weeks to reduce the intestinal flora, the arthritis improved rapidly and the ulcer started to heal. The ulcer healed completely within a few weeks after application of an Apligraft® (cultured skin cells), but there was a flare of the arthritis in the same joints several weeks later, which responded to a further 2-week course of doxycycline. A further flare of the arthritis occurred in June 2000 after an episode of culture-positive Campylobacter enteritis, but this settled spontaneously. Subsequently it was possible to reduce the prednisone to 7.5 mg/day, but methotrexate and sulphasalazine were still required at the time of writing (August 2000).
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In 1971 the first reports of polyarthralgia, polyarthritis, tenosynovitis and myalgia following intestinal bypasses were published [2]. Up to 20% of the patients who had had bowel bypass surgery for morbid obesity suffered from these complications [2, 3]. In 1976, cutaneous manifestations were first described in association with bypass enteropathy [4]. These included pustular vasculitis, erythema nodosum, spontaneous ecchymoses, nodular erythematous plaques and urticaria [5]. Among the histopathological features that have been described are leucocytoclastic vasculitis with pericapillary inflammation, necrotizing angiitis, nodular vasculitis, folliculitis and septal panniculitis. The immunofluorescence is often positive and resembles a positive lupus band [5, 6]. The term ‘bowel bypass syndrome’ was first used in 1979 [7]. It includes the above cutaneous and articular lesions as well as Raynaud's phenomenon, paraesthesias, pericarditis or liver disease. The bowel bypass syndrome is presumed to be due to bacterial overgrowth that causes an immunological reaction. It is thought that circulating serum immune complexes play a role in the pathogenesis of this complication following intestinal bypass operations [8], and our patient showed deposition of complement and IgM on immune staining of the skin biopsy, which is in keeping with this theory. Reduction of the intestinal flora with tetracycline or metronidazole has been proposed as therapy for the intestinal bypass syndrome [9].
Our patient showed an asymmetrical, non-erosive oligoarthritis and plantar fasciitis, an erythema nodosum-like rash on both lower legs and a necrotic ulcer on his left foot, with a necrotizing, leucocytoclastic vasculitis on histology. These symptoms were refractory to immunosuppressive therapy but regressed rapidly after the introduction of doxycycline, so that we made the presumptive diagnosis of an intestinal bypass syndrome. It is interesting to speculate that the laparoscopic division of adhesions, which had been carried out approximately 6 weeks prior to the onset of the first symptoms, might have triggered the process.
In the 1980s this technique to reduce obesity was abandoned and the picture of the bowel bypass or arthritis–dermatitis syndrome has become rare. Our case shows, however, that this syndrome can appear for the first time as long as 18 yr after bypass surgery. Jejuno-ileostomy was previously a quite common treatment for obesity. As long as people live with their blind loops, it is important to think of the possibility of a bowel bypass syndrome when we see patients with arthritis, vasculitis and a history of surgical treatment for obesity.
Notes
Correspondence to: D. Maclachlan. 
References
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