Clonal Hypereosinophilic Syndrome: Two Cases Report in Black Men from Sub-Saharan Africa and Literature Reviews

The first case is about a man of 60 years old suffering of hypereosinophilic syndrome (HES) developed since 1998. He presented chronic cough, insomnia, and negative parasitical test. We observed hypereosinophilia and fibroblastic hyperplasia at the bone marrow biopsy. Initially, hydroxyurea and α-interferon treatment failed. We proposed to him imatinib mesylate in May 2003. The FIP1L1-PDGFRA gene was detected. The second case is about a man of 34 years old seen in March 2002. First investigation concluded to CML. Progressively, eosinophil cells increased, and complications occurred as oedema syndrome, dyspnoea, and parietal chronic endocarditic fibrosis associated with pericarditis. In addition, a bowel obstruction happened and was cured by surgery. Bcr-abl fusion was negative, and FIP1L1-PDGFRA gene was detected after and imatinib mesylate was given. Actually, endocarditic fibrosis decreased. The two patients are in haematological and cytogenetic remission. We concluded that clonal HES is present in Africa, and imatinib mesylate is effective.

[1]  S. Kische,et al.  Cardiac manifestation of the hypereosinophilic syndrome: new insights , 2010, Clinical Research in Cardiology.

[2]  H. Murata,et al.  Oral mucosa lesions in hypereosinophilic syndrome--an update. , 2008, Oral diseases.

[3]  J. Cools,et al.  Molecular mechanisms underlying FIP1L1-PDGFRA-mediated myeloproliferation. , 2007, Cancer research.

[4]  C. Haferlach,et al.  Characterization of three new imatinib-responsive fusion genes in chronic myeloproliferative disorders generated by disruption of the platelet-derived growth factor receptor beta gene. , 2007, Haematologica.

[5]  B. Bain,et al.  Diagnosis and treatment of hypereosinophilic syndromes , 2007, Current opinion in hematology.

[6]  C. Peschel,et al.  Myeloid blast crisis evolving during imatinib treatment of an FIP1L1-PDGFR alpha-positive chronic myeloproliferative disease with prominent eosinophilia , 2005, Leukemia.

[7]  J. Conklin,et al.  Eosinophilic cholecystitis, appendiceal inflammation, pericarditis, and cephalosporin-associated eosinophilia , 1994, Digestive Diseases and Sciences.

[8]  B. Bain Relationship between idiopathic hypereosinophilic syndrome, eosinophilic leukemia, and systemic mastocytosis , 2004, American journal of hematology.

[9]  H. Klues,et al.  Idiopathische Hypereosinophilie mit kardialer Beteiligung , 2004 .

[10]  D. Metcalfe,et al.  Molecular remission and reversal of myelofibrosis in response to imatinib mesylate treatment in patients with the myeloproliferative variant of hypereosinophilic syndrome. , 2004, Blood.

[11]  A. Patchefsky,et al.  Case report 3 , 1976, Skeletal Radiology.

[12]  M. Caligiuri,et al.  Discovery of a fusion kinase in EOL-1 cells and idiopathic hypereosinophilic syndrome , 2003, Proceedings of the National Academy of Sciences of the United States of America.

[13]  F. Brito-Babapulle The eosinophilias, including the idiopathic hypereosinophilic syndrome , 2003, British journal of haematology.

[14]  Peter Marynen,et al.  A tyrosine kinase created by fusion of the PDGFRA and FIP1L1 genes as a therapeutic target of imatinib in idiopathic hypereosinophilic syndrome. , 2003, The New England journal of medicine.

[15]  P. Zalloua,et al.  Effective treatment of hypereosinophilic syndrome with imatinib mesylate. , 2003, The hematology journal : the official journal of the European Haematology Association.

[16]  A. Tefferi,et al.  Treatment of hypereosinophilic syndrome with imatinib mesilate , 2002, The Lancet.

[17]  N. Cross,et al.  Myeloproliferative disorders with translocations of chromosome 5q31-35: role of the platelet-derived growth factor receptor Beta. , 2002, Acta haematologica.

[18]  B. Bain,et al.  Hypereosinophilia. , 2009, Blood.

[19]  A. Cuneo,et al.  Cytogenetic and molecular cytogenetic characterization of 6 new cases of idiopathic hypereosinophilic syndrome. , 2000, Haematologica.

[20]  M. Arquati,et al.  Autoimmune hemolytic anemia in a patient with idiopathic hypereosinophilia , 1995, American journal of hematology.

[21]  F. Talarmin,et al.  [Difficulties in diagnosing idiopathic hypereosinophilic syndrome in Black Africa. A case report observed in Senegal]. , 1994, Medecine tropicale : revue du Corps de sante colonial.

[22]  M. Horimoto,et al.  [A case of hypereosinophilic syndrome exhibiting left ventricular systolic dysfunction]. , 1992, Kokyu to junkan. Respiration & circulation.

[23]  M. Pérez,et al.  Ecchymosis and hepatomegaly as the presenting form of an idiopathic hypereosinophilic syndrome , 1988 .

[24]  J. Alijotas Reig,et al.  [Ecchymosis and hepatomegaly as the presenting form of an idiopathic hypereosinophilic syndrome]. , 1988, Revista clinica espanola.

[25]  H. Liaño,et al.  Neurologic dysfunction in the idiopathic hypereosinophilic syndrome. , 1986, Annals of internal medicine.

[26]  H. Schumacher,et al.  Philadelphia chromosome-negative CML with eosinophils? , 1984, American journal of clinical pathology.

[27]  O. Blétry,et al.  [Cardiac manifestations of the hypereosinophilic syndrome. The value of 2-dimensional echography (12 cases)]. , 1984, Archives des maladies du coeur et des vaisseaux.

[28]  S. M. Sumi,et al.  Neurotoxicity of human eosinophils. , 1979, Proceedings of the National Academy of Sciences of the United States of America.

[29]  S. Wolff,et al.  THE HYPEREOSINOPHILIC SYNDROME: Analysis of Fourteen Cases With Review of The Literature , 1975, Medicine.

[30]  R. Myerson,et al.  Hypereosinophilic syndrome. Report of two cases, with prolonged courses. , 1971, The American journal of medicine.