Abstract
A 64-year-old woman was admitted to hospital due to protracted diarrhea and liver dysfunction. The patient was diagnosed as Churg-Strauss syndrome (CSS) due to asthma, paranasal sinusitis, hypereosinophilia, and polyneuropathy. There was a history of taking montelukast, a leukotriene receptor antagonist (LTRA), which is thought to have some relationship with CSS. The liver biopsy specimen showed eosinophilic infiltration and centrolobular fatty change. In this paper, we review the relationship between LTRA and CSS. Several lines of evidence suggest that leukotriene plays an important role in maintaining neural tissues. We also review the potential relationship between centrolobular fatty change and pivoxil-containing antibiotics, which was prescribed for sinusitis before admission. Carnitine deficiency induced by pivoxil-containing agents may cause impaired fatty acid oxidation in mitochondria.
Highlights
Churg-Strauss syndrome (CSS) was first described in 1951 by Churg and Strauss
Asthma is one of the presenting symptoms in CSS, asthma symptom was well controlled in this case at the onset of the neuropathy
Hattori et al reported that, in most cases, asthma is well controlled when the neurological symptoms develop [2]. This finding suggests the difference in the mechanisms of the asthma and the neuropathy
Summary
Churg-Strauss syndrome (CSS) was first described in 1951 by Churg and Strauss. CSS is a systemic eosinophilic vasculitis with unknown etiology [1]. There are some reports of LTRA-induced CSS without history of systemic corticosteroids [5,6,7]. We present a case of montelukast-induced CSS associated with liver dysfunction without a previous use of systemic corticosteroids. This is the first case report of CSS presented with a liver dysfunction and confirmed by the liver biopsy. A 64-year-old woman suffered from bronchial asthma for ten years and was followed up by a family physician. She had been prescribed montelukast 4 months before. Diarrhea and anorexia occurred in the patient a month earlier The patient noticed her grip strength getting weaker and felt an abnormal sensation in her planters 3 weeks earlier. The patient was referred to our hospital because of the lasting diarrhea and liver dysfunction in laboratory tests
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