Rheumatology and Rehabilitation, 1977, 16, 152
ORIGINAL PAPER
ARTHRITIS WITH SIGMOID VOLVULUS AND HL-A B27 BY H. A. BIRD AND J. A. COSH Royal National Hospital for Rheumatic Diseases, Bath
has been described in association with ulcerative colitis (Wright and Watkinson, 1965), Crohn's Disease (Haslock, 1973) and bacterial infections of the bowel including Shigella (Chaudhuri, 1951) and Yersinia (Ahvonen et al., 1969). In patients with chronic inflammatory bowel disease, the HL-A B27 antigen is found in association with those who have spondylitis but not those with peripheral arthritis alone (Bluestone et al., 1975). In the reactive arthritis following bowel infection, HL-A B27 antigen was found in 94 % of patients with Salmonella arthritis, 88 % with Yersinia arthritis and 85% with Shigella arthritis (Aho et al., 1975). We here describe a patient with HL-A B27 antigen in which a polyarthritis of the feet occurred simultaneously with a chronic recurrent sigmoid volvulus in the absence of demonstrable infection. ACUTE POLYARTHRITIS
CASE HISTORY A 43-year-old priest, J.N., was first seen as a domiciliary consultation with his practitioner in December 1975 with progressive bilateral foot oedema of 14 days' duration. Six months before, his right big toe had been swollen and painful after a short attack of diarrhoea and vomiting. The diarrhoea improved but joint pain and stiffness, greatest in the metatarsophalangeal joints of the feet, forced him to relinquish his parochial duties in spite of rest and treatment with phenylbutazone. There was no family history of arthritis, spondylitis or back pain. On examination there was diffuse oedema of both feet extending up above the ankles with pain and tenderness in the metatarsophalangeal joints and also, less acutely in the midtarsal joints. No other joints were affected and he had no back pain. There was no conjunctivitis, urethritis or rash though he had had a further episode of diarrhoea without blood or mucus, attributed by his practitioner to phenylbutazone. His haemoglobin was 11.3 g/dl and his viscosity slightly raised at 1.75 though Rose latex was negative. A diagnosis of polyarthritis, possibly rheumatoid, was made and a period of three weeks' rest with gentle active physiotherapy and soluble aspirin was prescribed. As there was no improvement, he was admitted to hospital in January 1976. He looked ill and had lost weight. Diarrhoea had occurred with colicky abdominal pain and distension. He now admitted that for two years he had been subject to intermittent attacks of abdominal distension and colicky pain, lasting two days on each occasion, recurring about three times a year. There was constipation with each attack but they were not associated with diarrhoea. On examination, the foot oedema and tenderness in the metatarsophalangeal joints persisted, both ankles were now painful and there was slight limitation of movement with pain in the right shoulder, though all other joints were normal. His abdomen was distended and he had obstructed bowel sounds. No mass was palpable and there was no rigidity. Rectal examination was normal and sigmoidoscopy showed no evidence of colitis. His haemoglobin was 11.0 g/dl, white-cell count 6.7 x 10° and plasma viscosity 2.16 (normal 1.5-1.72). Electrolytes, urea and liver function Accepted for publication February 1977. Requests for reprints to Dr. H. A. Bird. 152
Downloaded from http://rheumatology.oxfordjournals.org/ at University of Arizona on September 5, 2015
SUMMARY A 43-year-old male developed seronegative polyarthritis after a short attack of diarrhoea. For about two years before he had experienced intermittent abdominal pain, distension and constipation. Laparotomy showed a chronic sigmoid volvulus for which sigmoid colectomy was performed. Post-operatively he had no further bowel symptoms, no further arthritis and there was radiological improvement of involved joints. Tissue typing showed HL-A B27 antigen.
Downloaded from http://rheumatology.oxfordjournals.org/ at University of Arizona on September 5, 2015
PLATE
XIV
FIG. 2.—Radiograph of feet eight months after colectomy showing improvement. [facing page 153}
Downloaded from http://rheumatology.oxfordjournals.org/ at University of Arizona on September 5, 2015
FIG. 1.—Radiograph of feet before operation, showing severe osteopenia.
BIRD AND COSH: ARTHRITIS WITH SIGMOID VOLVULUS
153
DISCUSSION We believe this man had a polyarthritis involving the ankles, tarsi and metatarsophalangeal joints. Although the radiographs showed considerable osteopenia, we think this was secondary to his arthritis rather than caused by Sudeck's Atrophy or post-traumatic osteodystrophy. Sudeck's Atrophy tends to be unilateral with less constitutional upset and always follows trauma (Plewes, 1956). In the published series of volvulus of the intestine we can find no previous mention of an association with arthritis (Bhansali, 1969; Arnold and Nance, 1973). It has recently been stressed that recurrent attacks of volvulus in young people may be more common than is usually realized (Sturzaker et ah, 1975). Recurrent attacks of abdominal pain with abdominal distension and constipation are the most common presentation though diarrhoea is said to occur in 30 % of cases. It is not certain whether this man's arthritis was a direct result of his mechanical bowel problem, whether it was coincidental, or whether it followed an alteration in his bowel microflora secondary to the recurrent volvulus. The history suggests intermittent intestinal torsion may have preceded the arthritis by one year. However, the simultaneous improvement post-operatively of both bowel symptoms and arthritis is striking, even though there are no previous reports of arthritis secondary to intestinal obstruction in patients with HL-A B27 antigen. The alternative explanation of a change in bowel microflora causing arthritis in a patient with HL-A B27 antigen is perhaps more attractive. Although stool cultures showed no pathogens, serial antibody titres to rarer organisms such as Yersinia were not performed and therefore infection cannot be excluded. The high occurrence of HL-A B27 in individuals with reactive arthritis following bowel infection has already been noted by Aho et al. (1975) and here too HL-A 2 was found as frequently as HL-A B27 in patients with reactive arthritis. HL-A 2 is more common throughout the general population than HL-A B27, so the association does not reach the same level of significance as that with HL-AB27. ACKNOWLEDGMENT
We wish to thank Mr. F. Keane and Mr. P. Sames for the surgical management of this case. REFERENCES
K., AHVONEN, P., LASSUS, A., SAIRENEN, E., SIEVERS, K. and TILIKAINEN, A. (1975). "HLA 27 in Reactive Arthritis Following Infection". Ann. Rheum. Dis. 34, Suppl. 1,29.
AHO,
Downloaded from http://rheumatology.oxfordjournals.org/ at University of Arizona on September 5, 2015
tests and serum calcium were all normal. Uric acid was normal, Rose latex negative and immunoglobulins normal. Stool culture and MSU were normal and occult bloods were negative. On radiography, the chest, hands, shoulders, ankles and sacroiliac joints were normal, though radiographs of the feet (Fig. 1, Plate XIV) showed generalized osteopenia without erosions. His persistent abdominal distension was now the main problem. Straight abdominal radiography showed dilated loops of large bowel and a barium enema suggested that a sigmoid volvulvus rather than stricture or neoplasm was the cause of his intestinal obstruction. On 3rd February he had a laparotomy which showed a sigmoid volvulus, apparently chronic, for which a sigmoid colectomy was performed. There was no evidence of colitis and the intraabdominal organs looked normal. Histology of the resected colon showed abundant ganglionic areas and no evidence of malignancy. He made a routine post-operative recovery and his joint symptoms have improved. He no longer has foot oedema and the metatarsophalangeal joints and ankles are no longer tender. Radiographs of the feet eight months after operation (Fig. 2) show the osteopenia has improved. There are no erosions and his viscosity and Rose latex are both normal. He has had no further bowel symptoms and is now back at work. The results of tissue typing are HL-A A2, 29; B12, 27.
154
RHEUMATOLOGY AND REHABILITATION VOL. XVI NO. 3
P., SIEVERS, K. and AHO, K. (1969) "Arthritis Associated with Yersinia enterocolitica Infection". Acta. Rheum. Scand. 15,232. ARNOLD, G. J. and NANCE, F. C. (1973) "Volvulus of the Sigmoid Colon". Ann. Surg. 177,527. BHANSALI, S. K., PATH,, S. D. and DESAI, V. K. (1969) "Volvulus". / . Postgrad. Med. 3,120. BLUESTONE, R., MORRIS, R., METZGER, A. and TERASAKI, P. (1975) "HLA- 27 and the SpondyJitis of Chronic Inflammatory Bowel Disease and Psoriasis". Ann. Rheum. Dis. 34, Suppl. 1, 31. AHVONEN,
CHAUDHURI, R. N., CHAKRAVARTI, H. and RAI CHAUDHURI, M. N. (1951) "Streptomycin in
Postdysenteric Arthritis". Lancet i, 510.
Volvulus in Young People: A Missed Diagnosis". Br. Med. J. 1,338. WRIGHT, V. and WATKINSON, G. (1965) "The Arthritis of Ulcerative Colitis". Br. Med. J. 2, 670.
Downloaded from http://rheumatology.oxfordjournals.org/ at University of Arizona on September 5, 2015
HASLOCK, I. (1973), "Arthritis and Crohn's Disease". Ann. Rheum. Dis. 32,479. PLEWES, L. W. (1956) "Sudeck's Atrophy in the Hand". / . BoneJt. Surg. 38B, 195. STURZAKER, H. G., LAWRIE, R. S. and JOINER, C. L. (1975) "Recurrent Sigmoid
ORIGINAL PAPER
ARTHRITIS WITH SIGMOID VOLVULUS AND HL-A B27 BY H. A. BIRD AND J. A. COSH Royal National Hospital for Rheumatic Diseases, Bath
has been described in association with ulcerative colitis (Wright and Watkinson, 1965), Crohn's Disease (Haslock, 1973) and bacterial infections of the bowel including Shigella (Chaudhuri, 1951) and Yersinia (Ahvonen et al., 1969). In patients with chronic inflammatory bowel disease, the HL-A B27 antigen is found in association with those who have spondylitis but not those with peripheral arthritis alone (Bluestone et al., 1975). In the reactive arthritis following bowel infection, HL-A B27 antigen was found in 94 % of patients with Salmonella arthritis, 88 % with Yersinia arthritis and 85% with Shigella arthritis (Aho et al., 1975). We here describe a patient with HL-A B27 antigen in which a polyarthritis of the feet occurred simultaneously with a chronic recurrent sigmoid volvulus in the absence of demonstrable infection. ACUTE POLYARTHRITIS
CASE HISTORY A 43-year-old priest, J.N., was first seen as a domiciliary consultation with his practitioner in December 1975 with progressive bilateral foot oedema of 14 days' duration. Six months before, his right big toe had been swollen and painful after a short attack of diarrhoea and vomiting. The diarrhoea improved but joint pain and stiffness, greatest in the metatarsophalangeal joints of the feet, forced him to relinquish his parochial duties in spite of rest and treatment with phenylbutazone. There was no family history of arthritis, spondylitis or back pain. On examination there was diffuse oedema of both feet extending up above the ankles with pain and tenderness in the metatarsophalangeal joints and also, less acutely in the midtarsal joints. No other joints were affected and he had no back pain. There was no conjunctivitis, urethritis or rash though he had had a further episode of diarrhoea without blood or mucus, attributed by his practitioner to phenylbutazone. His haemoglobin was 11.3 g/dl and his viscosity slightly raised at 1.75 though Rose latex was negative. A diagnosis of polyarthritis, possibly rheumatoid, was made and a period of three weeks' rest with gentle active physiotherapy and soluble aspirin was prescribed. As there was no improvement, he was admitted to hospital in January 1976. He looked ill and had lost weight. Diarrhoea had occurred with colicky abdominal pain and distension. He now admitted that for two years he had been subject to intermittent attacks of abdominal distension and colicky pain, lasting two days on each occasion, recurring about three times a year. There was constipation with each attack but they were not associated with diarrhoea. On examination, the foot oedema and tenderness in the metatarsophalangeal joints persisted, both ankles were now painful and there was slight limitation of movement with pain in the right shoulder, though all other joints were normal. His abdomen was distended and he had obstructed bowel sounds. No mass was palpable and there was no rigidity. Rectal examination was normal and sigmoidoscopy showed no evidence of colitis. His haemoglobin was 11.0 g/dl, white-cell count 6.7 x 10° and plasma viscosity 2.16 (normal 1.5-1.72). Electrolytes, urea and liver function Accepted for publication February 1977. Requests for reprints to Dr. H. A. Bird. 152
Downloaded from http://rheumatology.oxfordjournals.org/ at University of Arizona on September 5, 2015
SUMMARY A 43-year-old male developed seronegative polyarthritis after a short attack of diarrhoea. For about two years before he had experienced intermittent abdominal pain, distension and constipation. Laparotomy showed a chronic sigmoid volvulus for which sigmoid colectomy was performed. Post-operatively he had no further bowel symptoms, no further arthritis and there was radiological improvement of involved joints. Tissue typing showed HL-A B27 antigen.
Downloaded from http://rheumatology.oxfordjournals.org/ at University of Arizona on September 5, 2015
PLATE
XIV
FIG. 2.—Radiograph of feet eight months after colectomy showing improvement. [facing page 153}
Downloaded from http://rheumatology.oxfordjournals.org/ at University of Arizona on September 5, 2015
FIG. 1.—Radiograph of feet before operation, showing severe osteopenia.
BIRD AND COSH: ARTHRITIS WITH SIGMOID VOLVULUS
153
DISCUSSION We believe this man had a polyarthritis involving the ankles, tarsi and metatarsophalangeal joints. Although the radiographs showed considerable osteopenia, we think this was secondary to his arthritis rather than caused by Sudeck's Atrophy or post-traumatic osteodystrophy. Sudeck's Atrophy tends to be unilateral with less constitutional upset and always follows trauma (Plewes, 1956). In the published series of volvulus of the intestine we can find no previous mention of an association with arthritis (Bhansali, 1969; Arnold and Nance, 1973). It has recently been stressed that recurrent attacks of volvulus in young people may be more common than is usually realized (Sturzaker et ah, 1975). Recurrent attacks of abdominal pain with abdominal distension and constipation are the most common presentation though diarrhoea is said to occur in 30 % of cases. It is not certain whether this man's arthritis was a direct result of his mechanical bowel problem, whether it was coincidental, or whether it followed an alteration in his bowel microflora secondary to the recurrent volvulus. The history suggests intermittent intestinal torsion may have preceded the arthritis by one year. However, the simultaneous improvement post-operatively of both bowel symptoms and arthritis is striking, even though there are no previous reports of arthritis secondary to intestinal obstruction in patients with HL-A B27 antigen. The alternative explanation of a change in bowel microflora causing arthritis in a patient with HL-A B27 antigen is perhaps more attractive. Although stool cultures showed no pathogens, serial antibody titres to rarer organisms such as Yersinia were not performed and therefore infection cannot be excluded. The high occurrence of HL-A B27 in individuals with reactive arthritis following bowel infection has already been noted by Aho et al. (1975) and here too HL-A 2 was found as frequently as HL-A B27 in patients with reactive arthritis. HL-A 2 is more common throughout the general population than HL-A B27, so the association does not reach the same level of significance as that with HL-AB27. ACKNOWLEDGMENT
We wish to thank Mr. F. Keane and Mr. P. Sames for the surgical management of this case. REFERENCES
K., AHVONEN, P., LASSUS, A., SAIRENEN, E., SIEVERS, K. and TILIKAINEN, A. (1975). "HLA 27 in Reactive Arthritis Following Infection". Ann. Rheum. Dis. 34, Suppl. 1,29.
AHO,
Downloaded from http://rheumatology.oxfordjournals.org/ at University of Arizona on September 5, 2015
tests and serum calcium were all normal. Uric acid was normal, Rose latex negative and immunoglobulins normal. Stool culture and MSU were normal and occult bloods were negative. On radiography, the chest, hands, shoulders, ankles and sacroiliac joints were normal, though radiographs of the feet (Fig. 1, Plate XIV) showed generalized osteopenia without erosions. His persistent abdominal distension was now the main problem. Straight abdominal radiography showed dilated loops of large bowel and a barium enema suggested that a sigmoid volvulvus rather than stricture or neoplasm was the cause of his intestinal obstruction. On 3rd February he had a laparotomy which showed a sigmoid volvulus, apparently chronic, for which a sigmoid colectomy was performed. There was no evidence of colitis and the intraabdominal organs looked normal. Histology of the resected colon showed abundant ganglionic areas and no evidence of malignancy. He made a routine post-operative recovery and his joint symptoms have improved. He no longer has foot oedema and the metatarsophalangeal joints and ankles are no longer tender. Radiographs of the feet eight months after operation (Fig. 2) show the osteopenia has improved. There are no erosions and his viscosity and Rose latex are both normal. He has had no further bowel symptoms and is now back at work. The results of tissue typing are HL-A A2, 29; B12, 27.
154
RHEUMATOLOGY AND REHABILITATION VOL. XVI NO. 3
P., SIEVERS, K. and AHO, K. (1969) "Arthritis Associated with Yersinia enterocolitica Infection". Acta. Rheum. Scand. 15,232. ARNOLD, G. J. and NANCE, F. C. (1973) "Volvulus of the Sigmoid Colon". Ann. Surg. 177,527. BHANSALI, S. K., PATH,, S. D. and DESAI, V. K. (1969) "Volvulus". / . Postgrad. Med. 3,120. BLUESTONE, R., MORRIS, R., METZGER, A. and TERASAKI, P. (1975) "HLA- 27 and the SpondyJitis of Chronic Inflammatory Bowel Disease and Psoriasis". Ann. Rheum. Dis. 34, Suppl. 1, 31. AHVONEN,
CHAUDHURI, R. N., CHAKRAVARTI, H. and RAI CHAUDHURI, M. N. (1951) "Streptomycin in
Postdysenteric Arthritis". Lancet i, 510.
Volvulus in Young People: A Missed Diagnosis". Br. Med. J. 1,338. WRIGHT, V. and WATKINSON, G. (1965) "The Arthritis of Ulcerative Colitis". Br. Med. J. 2, 670.
Downloaded from http://rheumatology.oxfordjournals.org/ at University of Arizona on September 5, 2015
HASLOCK, I. (1973), "Arthritis and Crohn's Disease". Ann. Rheum. Dis. 32,479. PLEWES, L. W. (1956) "Sudeck's Atrophy in the Hand". / . BoneJt. Surg. 38B, 195. STURZAKER, H. G., LAWRIE, R. S. and JOINER, C. L. (1975) "Recurrent Sigmoid
