Abstract
BackgroundEosinophilic gastrointestinal disorders are an emerging disease entity characterized by eosinophilic infiltration of the intestinal wall. Oral steroids can be still considered as first line treatment. Unfortunately relapses are quite common. Usually long term low-dose prednisone or immunosuppressive therapy is required, which is especially problematic in young patients. Thus a reliable steroid sparing agent with low side effects suitable for long term use is needed. There are strong hints to a similar pathophysiology of eosinophilic gastrointestinal disorders to that of asthma. Indeed leukotriene D4 plays an important role in the recruitment of eosinophils into the intestinal tissue causing damage. This patho-mechanism provides the rationale for the treatment with a leukotriene D4 receptor antagonist. Recently there have been first reports about successful short term use of Montelukast in eosinophilic gastrointestinal disorders.Case presentationWe report the case of a 17 year old girl with a long history of severe abdominal complaints leading to several hospitalizations in the past. Mimicking the picture of an intestinal tuberculosis she received an anti mycobacterial treatment without any success. Marked eosinophilia in blood, ascites and tissue samples of the intestinal tract finally lead to the diagnosis eosinophilic gastroenteritis. Tapering off prednisone caused another severe episode of abdominal pain. At that point leukotriene antagonist Montelukast was started at a dose of 10 mg once daily. Steroids could be tapered off completely within six weeks. The patient has been free of symptoms for over two years by now. Routine examinations, blood tests and endoscopy have rendered regular results. So far no side effects were noted.ConclusionHere report about successful long term remission of eosinophilic gastroenteritis under Montelukast. Further randomized control trials are required to asses the full benefits of Montelukast therapy in the whole spectrum of eosinophilic gastrointestinal disorders.
Highlights
Eosinophilic gastrointestinal disorders are an emerging disease entity characterized by eosinophilic infiltration of the intestinal wall
Eosinophilic gastroenteritis is a rare disease characterized by eosinophilic infiltration of the intestinal wall
The clinical presentation depends on the gastrointestinal division affected and the wall layers infiltrated
Summary
Eosinophilic gastroenteritis is a rare disease, but should be considered as differential diagnosis in cases of recurrent abdominal pain. Failure to diagnose this disorder often relates to reluctance to biopsy an apparently normal intestinum. Montelukast therapy seems promising to be an effective long term treatment in these patients, when they are steroid dependent. No side effects were noted in our patient. Further randomized trials are required to asses the full benefits of Montelukast therapy in eosinophilic gastrointestinal disorders. CSS, Churg-Strauss-syndrome; EG, eosinophilic gastroenteritis; EGID, eosinophilic gastrointestinal disorder; HES, hypereosinophilic syndrome; LTRA, leukotriene receptor antagonist; FIP1L1-PDGFRA, FIP1-like-1-platelet-derivedgroth-factor-receptor-alpha fusion kinase
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