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Rheumatology 2001; 40: 704-706
© 2001 British Society for Rheumatology
Letters to the Editor |
Focal myositis due to Campylobacter infection
Rheumatology Research Group, University of Liverpool and
1 Department of Paediatric Rheumatology, Alder Hey, Royal Liverpool Children's NHS Trust, Liverpool, UK
SIR, We read with interest the recent article on focal myositis and pregnancy [1]. We recently had a case of focal myositis that had several similarities with the case reported, but in our patient the cause was found to be Campylobacter infection.
A 14-yr-old girl presented with a 5-day history of bloody diarrhoea followed by bilateral calf pain and swelling. On examination she was pyrexial and had swelling and tenderness of the calves with erythema over the left calf. Her temperature and diarrhoea persisted for 3 days and her right calf symptoms settled soon after. Left calf pain and swelling persisted. The investigations revealed a leucocytosis with neutrophilia, erythrocyte sedimentation rate (ESR) 100 mm/h, C-reactive protein (CRP) 169 mg/l, normal electrolytes, and creatinine phosphokinase (CPK) 694 U/l (normal range 24195 U/l); no antibodies to antinuclear or extractable nuclear antigens were detected and immunoglobulin and complement levels were normal. Deep vein thrombosis was excluded. Stool culture isolated Campylobacter species.
Left calf symptoms continued despite erythromycin. There was persistent elevation of CPK with focal myositis demonstrated on magnetic resonance imaging (Fig. 1
). The girl was treated with naproxen alone and the symptoms settled. Calf swelling, muscle enzymes and acute-phase reactants returned to normal after 6 weeks of treatment. No features of polymyositis or dermatomyositis have developed.
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Clinically, the presentation with erythema and swelling of focal lower limb muscles followed by spontaneous resolution with minimal treatment, was very similar to the case described by Hepburn et al. [1]. Elevation of ESR, CRP and CPK and MRI appearances were also in keeping with their case.
There is already a lengthy list of infectious causes for focal myositis [2]. As far as we are aware, this is the first report of infectious myositis caused by Campylobacter infection.
Campylobacter infection caused a non-suppurative focal myositis in our patient that could not be differentiated from Hepburn et al.'s [1] case of idiopathic focal myositis occurring in pregnancy, in which there was no history of diarrhoea. It should be remembered in pregnancy that non-specific immune suppression can occur [3]. We would like to reinforce the recommendation of Hepburn et al. to investigate carefully for bacterial and viral infection before diagnosing idiopathic focal myositis in pregnancy. When considering bacterial causes, stool culture for Campylobacter species should be included, as infection with these bacteria can have a disastrous effect on the mother and fetus [4, 5].
Notes
Correspondence to: J. K. Dawson, Department of Rheumatology, Southport NHS Trust Hospital, Town Lane, Southport, Merseyside PR8 6 PN, UK. ![]()
Accepted 27 November 2000
References
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Hepburn A, Damani N, Sandison A, Pandit N. Idiopathic focal myositis in pregnancy. Rheumatology2000;39:2113.
[Free Full Text] - Maddison PJ, Isenberg DA, Woo P, Glass DN. Oxford textbook of rheumatology, Vol. 2. Oxford: Oxford University Press, 1993:8113.
- Chapel H, Haeney M. Essentials of clinical immunology. Oxford: Blackwell Scientific Publications, 1984:3589.
- Meyer A, Stallmach T, Goldenberger D, Altwegg M. Lethal maternal sepsis caused by Campylobacter jejuni: pathogen preserved in placenta and identified by molecular methods. Modern Pathol1997;10:12536.[Web of Science][Medline]
- Simor AE, Ferro S. Campylobacter jejuni infection occurring during pregnancy. Eur J Clin Microbiol Infect Dis1990;9:1424.[Medline]
Reply
Rheumatology Section, Division of Medicine, Imperial College School of Medicine, Hammersmith Hospital, London W12 0NN and
1 Department of Rheumatology, Watford General Hospital, Watford, Hertfordshire WD1 8HB, UK
We read the letter from Dawson and Davidson with great interest. Their case was clearly very similar to ours in several respects, such as the spontaneous resolution of symptoms, the moderate increase in the acute phase response and creatine kinase and the magnetic resonance imaging findings. Our patient did not report diarrhoea at any stage in her illness and cultures of blood, urine, synovial fluid and skeletal muscle were all sterile.
Interestingly, the reported case demonstrated bilateral lower limb involvement in this rare syndrome. Unilateral lower limb involvement is the most frequently encountered form of focal myositis [1], but bilateral involvement has also been reported [2]. Most cases appear to resolve spontaneously in 46 weeks, but patients with prolonged symptoms of pain and swelling lasting up to 3 yr have been reported.
As Dawson and Davidson point out, numerous infections are associated with myositis, although more usually in a generalized form. Viruses such as HIV, coxsackievirus and, in particular, influenza are all frequent causes [3]. A viral aetiology for focal myositis has also been postulated, but a study using the polymerase chain reaction failed to find a number of viral agents in a series of seven cases [4]. The temporal relationship in their case suggests that campylobacter may have been directly involved in the development of the patient's lower limb myositis. It was unfortunate that a muscle biopsy was not performed to assess this further, but we appreciate that this is not a light undertaking in a patient of this age. This might be considered should a relapse occurthis can clearly occur with focal myositis [2]. Reinfection with campylobacter would obviously need excluding should the reported patient develop further rheumatic symptoms. Furthermore, it would be interesting to know this patient's HLA B27 status.
Some form of reactive phenomenon is clearly a possibility in this case. Campylobacter jejuni is an established cause of reactive arthritis [5]. Focal myositis secondary to pyogenic infection elsewhere might therefore appear plausible. Other rheumatic manifestations of infection with this organism include septic arthritis, erythema nodosum and osteomyelitis, although these are usually seen in immunocompromised hosts. We are not aware of focal lower limb myositis occurring secondary to confirmed bacterial enterocolitis. The majority of previously reported cases of focal myositis have been carefully screened for an infectious cause. Bacteria, such as staphylococci and ß-haemolytic streptococci, are known to cause localized muscle infection, but more systemic reactions may also be seen. A generalized reactive myalgia following group A streptococcal infection has also been described [6]. More localized disease is much less common.
All cases of focal myositis should be thoroughly investigated to exclude underlying infection, not only in the muscle itself but also at distant sites, including the gastrointestinal tract. A muscle biopsy for histological examination is also necessary, except in the most transient of cases, to exclude a mitotic process. MRI is diagnostically very helpful, but a biopsy should be considered in such cases to exclude more sinister pathology.
Notes
Correspondence to: A. Hepburn. ![]()
References
- Heffner RR Jr, Armbrustmacher VW, Earle KM. Focal myositis. Cancer1977;40:3016.[Web of Science][Medline]
- Naughton M, Jessop JD, Williams BD. Idiopathic recurrent non-suppurative focal myositis: a report of two cases. Br J Rheumatol1993;22:11014.
- Nicholson KJ. Clinical features of influenza. Semin Resp Infect1992;7:2637.[Medline]
- Toti P, Romano L, Villanova M, Zazzi M, Luzi P. Focal myositis: a polymerase chain reaction analysis for a viral aetiology. Human Pathol1997;28:1113.[Medline]
- Peterson MC. Rheumatic manifestations of Campylobacter jejuni and C. fetus infection in adults. Scand J Rheumatol1994;23:16770.[Medline]
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Jansen TL, Janssen M, Macfarlane JD, De Long AJ. Post-streptococcal reactive myalgia: a novel syndrome secondary to infection with group A or G streptococci. Br J Rheumatol1998;37:13438.
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