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Home , Arthralgia, Fatigue, Malaise

502 Annals ofthe Rheumatic 1996; 55: 502-503

LESSON OF THE MONTH Ann Rheum Dis: first published as 10.1136/ard.55.8.502 on 1 August 1996. Downloaded from Chronic , arthralgia, and

MM Gompels, G P Spickett

Case report There was no deformity, swelling, or A 25 year old female veterinary nurse restriction of movement, and no lymphadeno- presented with a six year history of general pathy or splenomegaly. Investigations showed malaise and severe fatigue. Associated with this normal haematology except for a persistently she described frequent (monthly) episodes of raised international normalised ratio (INR) at polyarthralgia affecting all but with a 1-2-1-4, normal range <1-14 (lupus predilection for the small joints of the hands anticoagulant was not detected), normal and the wrists. When present this was biochemistry including creatinine kinase, cor- accompanied by mild morning stiffness. In tisol, negative porphyria screen, and serum addition she experienced colicky abdominal angiotensin converting enzyme. She had nega- , sometimes with diarrhoea, occasionally tive stool cultures (these had been negative with blood mixed with her faeces. Other consistently as had sigmoidoscopy, endos- complaints consisted of low , sore copy, and associated biopsies six months gritty eyes, and an inability to perform any previously) and a negative glycocholate breath physical at the time of these symp- test. Serology to borrelia, brucella, and toxo- toms. Her symptoms had been remarkably plasmosis was negative as was serology to consistent, with no recent change to their herpes , B, , and pattern. parvovirus. Her HLA tissue tissue type was Six years ago she had been on a working A2,B7,B50(2 1),DR1 5(2),DR4 (not normally holiday at a veterinary practice situated in associated with autoimmune ). Her New York state, USA. After eating a dish made chest radiograph was normal. A full autoanti- with "blue fish" she had immediately devel- body screen, including extractable nuclear oped severe nausea, vomiting, and malaise. antigens, was negative. An ophthalmological Although all her acute symptoms resolved, her review showed no evidence of an abnormality. other symptoms started on return to the Yersinia serology was requested and was nega- United Kingdom. She was investigated twice, tive to Y enterocolitica and Y pseudotuberculosis http://ard.bmj.com/ at different hospitals, before being referred to I, II, III, and V. Serology to Ypseudotuberculosis this department. It had been found that her type IV was positive for IgM at a titre of 1:320. symptoms were helped by treatment with 30 There had been no history of recent typhoid mg prednisolone daily for the severe episodes vaccination. and a maintenance dose of 5 mg daily. Severe Treatment was commenced with high dose episodes were occurring three to four times a tetracyclines for one month. After three year. Non-steroidal anti-inflammatory drugs, months the serology had reverted to normal, as on September 27, 2021 by guest. Protected copyright. sulphasalazine, and other treatments of inflam- had her abnormal INR. Over this period the matory bowel disease had not helped her patient required no steroid , worked symptoms. On all occasions the examination full time, and became able to exercise without and investigations had been reported as normal restriction. She experienced occasional mild including markers of , connective arthralgia, which resolved spontaneously. tissue disease, and radiological and histological gastrointestinal studies. No blood had been seen in her faeces. No diagnosis was made Discussion other than a seronegative arthralgia. Yersinia pseudotuberculosis is usually found in Regional Department She had managed to continue her job rodents or birds with spread to other debili- ofImmunology, throughout this period but was becoming tated animals through contamination of their Newcastle General increasingly unable to perform other physical food by faeces. of humans is Hospital, rested at to occur of food or Westgate Road, activities and completely weekends. by ingestion contaminated Newcastle-upon-Tyne She was well supported by her partner and from handling infected animals. Diagnosis is NE4 6BZ, family and had no features or history to suggest made by serological means as only during the United Kingdom a illness. She had no other relevant acute episode is isolation 2 In M M Gompels depressive possible.1 G P Spickett medical history and there were no familial humans, like the more common Y causes an with Correspondence to: illnesses. Foreign travel before the illness had enterocolitica, it enterocolitis Dr MM Gompels, only been to the United States. Subsequently , diarrhoea, and inflammation of the Regional Department of Immunology, Newcastle she had been to Ireland, where she had also mesenteric lymph nodes3 and sometimes a General Hospital, Westgate worked, and on holiday to India. pseudoappendicitis,6 presumably accounting Road, Newcastle-upon-Tyne NE4 6BZ, United Kingdom. On examination she was a thin but fit 25 year for the abnormal INR. It has also been as an acute arthritis7'- Accepted for publication old with no features of connective tissue documented producing 6 February 1996 disease, , or Sjogren's syndrome. which is ofthe classic sacroiliac pattern in those Chronicfatigue, arthralgia, and malaise 503

who are HLA-B27 positive.9 '1 In most ofthese all suggest that this case represented persistent cases it has been described as producing a rather than an acute infection, although we

clearly defined, acute inflammatory , cannot entirely exclude the second possibility. Ann Rheum Dis: first published as 10.1136/ard.55.8.502 on 1 August 1996. Downloaded from sacroilitis, or tenosynovitis around the time of This case highlights the importance of a the initial infection. It can persist, particularly thorough occupational history and the fact that in those who are HLA-B27 positive. Y pseudotuberculosis can cause a chronic low We are not aware of any studies on the long grade infection but with severe consequences. term follow up of patients with Y The response to treatment we obtained pseudotuberculosis. However, in subjects with Y suggests that its diagnosis was worthwhile. It is enterocolitica there are data on the long term therefore important that Yersinia infection is sequelae of infection. In one study 31 patients screened for in the investigation ofunexplained were followed up because they developed acute persisting arthralgia and arthritis. This, and arthritis immediately after infection with Y other treatable infection, may be responsible enterocolitica." After five years, 10 out of 31 for a proportion of those cases of seronegative patients still had arthralgia, six were asympto- arthritis. matic, and the remaining 15 had ongoing and arthritis. Of those with arthralgia five out of 10 had not had antibiotic The lesson treatment when compared with one of the six * Careful consideration of occupation may be patients who had no symptoms. The HLA important status ofthose who had an active arthritis is not * A long history and previous investigation recorded. does not always indicate an untreatable Our case highlights several important as- condition pects. It shows the importance of an occu- * A number of can cause persis- pational history. There would have been a tent arthralgia - namely, yersinia, borrelia, strong possibility that the infection was parvovirus, brucella, campylobacter, shigella, acquired at work. In such patients with a chlamydia, and C, persistent history of arthralgia, abdominal pain, and diar- Epstein-Barr , and cytomegalovirus. rhoea Yersinia infections should be considered to the same extent as ; particularly 1 Carniel E, Mollaret H H. [review]. Comp as it is well documented that acute Y pseudo- Immunol Micorbiol Infect Dis 1990; 13: 51-8. infection can be asymptomatic. 2 2 Georgescu C, Dan E, Cojocaru M, Plosceanu L. The role of infections with Yersinia pseudotuberculosis and Presumably what happened in New York was Yersinia enterocolitica in the pathogenesis of chronic an episode ofgastroenteritis and the "blue fish" inflammatory, rheumatic and collagen diseases. Med Interne 1986; 24: 217-20. was a red herring. What is interesting is that 3 Rich E J, McDonald R A, Christie D L. Yersinia subsequently there were no features to suggest pseudotuberculosis: report of a case with endoscopic findings. Jf Pediatr Gastroenterol Nutr 1990; 10: 413-5. an acute inflammation, a fact that made 4 Leino R, Granfors K, Havia T, Heinonen R, Lampinen M, diagnosis more difficult. Although it is not Toivanen A. Yersiniosis as a . ScandlInfect Dis 1987; 19: 63-8. http://ard.bmj.com/ widely described, in common with the Y 5 Tobin M V, Meigh R E, Smith C L, Gilmore I T. Yersinia enterocolitica study, Ypseudotuberculosis can give pseudotuberculosis ileitis presenting with severe intestinal haemorrhage. _J R Soc Med 1988; 81: 423-4. persistent infection manifesting as joint 6 Grant H, Rode H, Cywes S. Yersinia pseudotuberculosis and arthralgia. " If such infections are not affecting the appendix. J Pediatr Surg 1994; 29: 1621. 7 Bignardi G E. Yersinia pseudotuberculosis and arthritis. sought they will remain undiagnosed. Ann Rheum Dis 1989; 48: 518-9. The serology is the most important diag- 8 Lindley R I, Pattman R S, Snow M H. Yersinia pseudo- tuberculosis infection as a cause of as nostic tool as cultures can be negative even seen in a genitourinary clinic: case report. Genitourin Med during acute infection.12 Although typhoid 1989; 65: 255-6. on September 27, 2021 by guest. Protected copyright. 9 Chalmers A, Kaprove R E, Reynolds W J, Urowitz M B. vaccination and salmonella may cross react Postdiarrheal arthropathy of Yersinia pseudotuberculosis. with the serology for Ypseudotuberculosis IV, in Can MedAssocJ 1978; 118: 515-6. 10 Magub M, SanyJ, Le Peu G, Bertrand A, Blanc F, Bertrand L. this case there was no history of recent typhoid Articular manifestations ofYersinia infections. Apropos of vaccination. Salmonella would have been 4 personal cases. Review of the literature [review]. Rev Rhum Mal Osteo-artic 1983; 50: 115-24. (In French). isolated from the intensive microbiology that 11 Saari K M, Maki M, Paivonsalo T, Leino R, Toivanen A. was performed and the loss of IgM sero- Acute anterior uveitis and conjunctivitis following Yersinia infection in children. Int Ophthalmol 1986; 9: positivity on specific treatment all strongly 237-41. suggests a diagnosis of Yersinia. The clinical 12 Tertti R, Vuento R, Mikkola P, Granfors K, Makela A L, Toivanen A. Clinical manifestations of Yersinia pseudo- features of this illness, the resolution of these tuberculosis infection in children. Eur J Clin Microbiol on treatment, and the loss ofIgM seropositivity Infect Dis 1989; 8: 587-91.