| ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
Comment on: Adult-onset Still's disease and myocarditis: successful treatment with intravenous immunoglobulin and maintenance of remission with etanercept
Department of Rheumatology, Southport and Ormskirk NHS Trust, Liverpool University, UK
Correspondence to: K. A. Binymin, Southport, DGH Kew Southport, Mersyside PR8 6PN, UK. E-mail: Khalid.binymin{at}southportandormskirk.nhs.uk
 |
Introduction |
|---|
 Top Introduction AcknowledgementsReferences |
|---|
SIR, We read with interest the case of dilated cardiomyopathy complicating adult-onset Still's disease (AOSD) by Kuek et al. [1]. The firm diagnosis of myocarditis was established on clinical grounds and was substantiated by the raised cardiac specific muscle enzymes. I, however, think that the cause of recurrent and partially responsive myocarditis in an immunocompromised individual who has a long-standing chronic inflammatory illness, merits further study to exclude other causes for myocarditis. This is especially true when an acclaim for a successful therapeutic regimen is being expressed and its wider use is encouraged.
The negative viridae serology does not exclude viral myocarditis (by far the commonest cause of myocarditis) since tests of many of the viruses implicated in the causation of myocarditis are not part of the routine virology screen. Furthermore, the fact the patient suffers with AOSD does not rule out other specific forms of idiopathic or viral myocarditis [2, 3]. Chronic bacterial, fungal or infiltrative cardiomyopathies are possibilities needing also to be explored [4].
All these disorders would have similar initial presentation of dilated cardiomyopathy but would greatly differ in the natural course and prognosis of the disease [5, 6]. Many warrant additional or different modalities of therapy and may require cardiac transplant much earlier than cases with inflammatory myocarditis [7].
The positive response to intravenous immunoglobulin is well reported in various forms of myocarditis, especially with viral myocarditis [8–10]. Giving anti-tumour necrosis factor (TNF) therapy is yet another reason for being more diligent in excluding cardiac infective process before the inception in this particular case [11]. This patient already had three attacks of acute myocarditis with dramatic course.
For all of the above, I think a histological diagnosis using transvenous cardiac biopsy would be the appropriate step at the outset of any future relapse. The procedure is relatively safe and can be done as a day case [12]. The information gained will be invaluable. Polymerase chain reaction (PCR) for specific viruses and bacterial studies for chronic infective myocarditis can be readily tested [13]. The various histological examinations and immunohistochemical studies will exclude other primary forms and infiltrative disorders. The histopathological picture would after all confirm the diagnosis of inflammatory myocarditis. A biopsy directed therapy would present a solid cause and effect evidence. It would also substantiate the clinical decision to use other targeted therapy.
|
Acknowledgements |
|---|
|
TopIntroductionAcknowledgementsReferences |
|---|
The author would like to thank Dr K Pulya, consultant cardiologist, Southport District General Hospital, Southport.
Disclosure statement: The author has declared no conflicts of interest.
|
References |
|---|
|
TopIntroductionAcknowledgementsReferences |
|---|
- Kuek A, Weerakoon A, Ahmed K, Östör AJK. Adult-onset Still's disease and myocarditis: successful treatment with intravenous immunoglobulin and maintenance of remission with etanercept. Rheumatology (2007) 46:1043–4.
[Free Full Text] - Fett JD. Viral particles in endomyocardial biopsy tissue from peripartum cardiomyopathy patients. Am J Obstet Gynecol (2006) 195:330–1.[Web of Science][Medline]
- Kuhl U, Pauschinger M, Noutsias M, et al. High prevalence of viral genomes and multiple viral infections in the myocardium of adults with ‘idiopathic’ left ventricular dysfunction. Circulation (2005) 111:887–93.
[Abstract/Free Full Text] - Dec GW Jr, Palacios IF, Fallon JT, et al. Active myocarditis in the spectrum of acute dilated cardiomyopathies. Clinical features, histologic correlates, and clinical outcome. N Engl J Med (1985) 312:885–90.[Abstract]
- Cooper LT, Berry GJ, Shabetai R. Idiopathic giant-cell myocarditis–natural history and treatment. Multicenter Giant Cell Myocarditis Study Group Investigators. N Engl J Med (1997) 336:1860–6.
[Abstract/Free Full Text] - Rosenstein ED, Zucker MJ, Kramer N. Giant cell myocarditis: most fatal of autoimmune diseases. Semin Arthritis Rheum (2000) 30:1–16.[Web of Science][Medline]
- Pulerwitz TC, Cappola TP, Felker GM, et al. Mortality in primary and secondary myocarditis. Am Heart J (2004) 147:746–50.[CrossRef][Web of Science][Medline]
- Kato S, Morimoto S, Hiramitsu S, et al. Successful high-dose intravenous immunoglobulin therapy for a patient with fulminant myocarditis. Heart Vessels (2007) 22:48–51. Epub 26 January 2007.[CrossRef][Web of Science][Medline]
- McNamara DM, Starling RC, Dec GW. Intervention in myocarditis and acute cardiomyopathy with immune globulin: results from the randomized placebo controlled IMAC trial. Circulation (1999) 100(Suppl I):1211.
- Robinson J, Hartling L, Vandermeer B, Crumley E, Klassen TP. Intravenous immunoglobulin for presumed viral myocarditis in children and adults. Cochrane Database of Systemic Reviews 2005. Art. No.: CD004370. DOI: 10.1002/14651858.CD004370.pub2.
- Wada H, Saito K, Kanda T, et al. Tumor necrosis factor-alpha (TNF-alpha) plays a protective role in acute viralmyocarditis in mice: a study using mice lacking TNF-alpha. Circulation (2001) 103:743–9.
[Abstract/Free Full Text] - Caforio AL, Calabrese F, Angelini A, et al. A prospective study of biopsy-proven myocarditis: prognostic relevance of clinical and aetiopathogenetic features at diagnosis. Eur Heart J (2007) 28:1326–33. Epub 9 May 2007.
[Abstract/Free Full Text] - Cunningham KS, Veinot JP, Butany J. An approach to endomyocardial biopsy interpretation. J Clin Pathol (2006) 59:121–9.
[Abstract/Free Full Text]
CiteULike 
Connotea 
Del.icio.us What's this?
This article has been cited by other articles:
|
|
 |
|
  A. J. K. Ostor Comment on: Adult-onset Still's disease and myocarditis: successful treatment with intravenous immunoglobulin and maintenance of remission with etanercept: reply Rheumatology, November 1, 2007; 46(11): 1742 - 1743. [Full Text] [PDF]
|
|
| ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||