A case of hypereosinophilic syndrome presenting with chronic cough successfully treated with imatinib.
Chronic cough is caused by a wide variety of disease conditions, including asthma, rhino-sinusitis and gastro-oesophageal reflux. We describe the case of a 42-year-old man with hypereosinophilic syndrome presenting with chronic dry cough. The cough did not respond to inhaled corticosteroid or leucotriene receptor antagonists. Hepatosplenomegaly was noted and the patient became anaemic and thrombocytopenic. He was refractory to treatment with hydroxyurea and interferon-alpha. Administration of imatinib resulted in complete resolution of eosinophilia and cough, without the use of anti-asthma drugs. Analysis of RNA from this patient demonstrated expression of the Fip1-like 1/platelet-derived growth factor receptor-alpha (FIP1L1-PDGFRA) fusion gene. The myeloproliferative variant of hypereosinophilic syndrome may cause chronic intractable cough, and a trial of imatinib treatment may be warranted.
Kobayashi
Makoto
M
Department of Hematology and Respiratory Medicine, Kochi Medical School, Kochi University, Nankoku, Kochi, Japan. kmakoto@jinseikai-group.or.jp
Kubota
Tetsuya
T
Uemura
Yoshiki
Y
Taguchi
Hirokuni
H
D000328:Adult; D001549:Benzamides; D002908:Chronic Disease; D003371:Cough / Q000188:drug therapy* / Q000209:etiology*; D006801:Humans; D017681:Hypereosinophilic Syndrome / Q000150:complications* / Q000188:drug therapy* / Q000235:genetics; D000068877:Imatinib Mesylate; D008297:Male; D015514:Oncogene Proteins, Fusion / Q000235:genetics; D010879:Piperazines / Q000627:therapeutic use*; D047428:Protein Kinase Inhibitors / Q000627:therapeutic use*; D011743:Pyrimidines / Q000627:therapeutic use*; D020796:Receptor, Platelet-Derived Growth Factor alpha / Q000235:genetics; D016896:Treatment Outcome; D039221:mRNA Cleavage and Polyadenylation Factors / Q000235:genetics
D001549:Benzamides; D015514:Oncogene Proteins, Fusion; D010879:Piperazines; D047428:Protein Kinase Inhibitors; D011743:Pyrimidines; D039221:mRNA Cleavage and Polyadenylation Factors; D000068877:Imatinib Mesylate; C479819:FIP1L1-PDGFRA fusion protein, human; D020796:Receptor, Platelet-Derived Growth Factor alpha