PURPOSE: Intensification of antileukemic treatment and progress in supportive management have improved the survival rates of children with acute myeloid leukemia (AML). However, morbidity and early mortality in these patients are still very high, especially in children with acute monoblastic leukemia (AML FAB M5). Inflammatory syndromes complicating the management of these children after application of cytosine arabinoside and due to hyperleukocytosis at initial presentation have been reported. Hemophagocytic lymphohistiocytosis (HLH) has been described as a serious and life-threatening acute complication during treatment of different oncologic entities; however, data on HLH in children with AML FAB M5 are extremely rare. METHODS: A retrospective study of all children with AML FAB M5 treated at our institution between 1993 and 2013 was performed to describe the clinical characteristics of patients who developed an inflammatory syndrome with HLH during oncologic treatment. RESULTS: Three of 10 children developed an inflammatory syndrome with fever, elevation of C-reactive protein, hyperferritinemia, elevation of soluble interleukin-2, and hemophagocytosis during prolonged aplasia following the first cycle of chemotherapy not responding to broad-spectrum antibiotics. No infectious agents could be identified; the initial symptoms occurred 17, 18, and 28 days after diagnosis of AML, respectively. The children immediately responded to dexamethasone; however, the same syndrome was observed again after the second cycle of chemotherapy and, in one patient, also after the third cycle. CONCLUSIONS: Treating physicians should be aware of an inflammatory syndrome resembling HLH in children with monoblastic leukemia since this problem might extremely complicate management and supportive care of these children. The co-incidence of monoblastic leukemia with HLH might be explained by cytokines released from the monoblastic leukemic cells themselves.
PURPOSE: Intensification of antileukemic treatment and progress in supportive management have improved the survival rates of children with acute myeloid leukemia (AML). However, morbidity and early mortality in these patients are still very high, especially in children with acute monoblastic leukemia (AML FAB M5). Inflammatory syndromes complicating the management of these children after application of cytosine arabinoside and due to hyperleukocytosis at initial presentation have been reported. Hemophagocytic lymphohistiocytosis (HLH) has been described as a serious and life-threatening acute complication during treatment of different oncologic entities; however, data on HLH in children with AML FAB M5 are extremely rare. METHODS: A retrospective study of all children with AML FAB M5 treated at our institution between 1993 and 2013 was performed to describe the clinical characteristics of patients who developed an inflammatory syndrome with HLH during oncologic treatment. RESULTS: Three of 10 children developed an inflammatory syndrome with fever, elevation of C-reactive protein, hyperferritinemia, elevation of soluble interleukin-2, and hemophagocytosis during prolonged aplasia following the first cycle of chemotherapy not responding to broad-spectrum antibiotics. No infectious agents could be identified; the initial symptoms occurred 17, 18, and 28 days after diagnosis of AML, respectively. The children immediately responded to dexamethasone; however, the same syndrome was observed again after the second cycle of chemotherapy and, in one patient, also after the third cycle. CONCLUSIONS: Treating physicians should be aware of an inflammatory syndrome resembling HLH in children with monoblastic leukemia since this problem might extremely complicate management and supportive care of these children. The co-incidence of monoblastic leukemia with HLH might be explained by cytokines released from the monoblastic leukemic cells themselves.
Authors: Jan-Inge Henter; Annacarin Horne; Maurizio Aricó; R Maarten Egeler; Alexandra H Filipovich; Shinsaku Imashuku; Stephan Ladisch; Ken McClain; David Webb; Jacek Winiarski; Gritta Janka Journal: Pediatr Blood Cancer Date: 2007-02 Impact factor: 3.167
Authors: Herwig Lackner; Christian Urban; Petra Sovinz; Martin Benesch; Andrea Moser; Wolfgang Schwinger Journal: Haematologica Date: 2008-02 Impact factor: 9.941
Authors: Ursula Creutzig; Martin Zimmermann; Jean-Pierre Bourquin; Michael N Dworzak; Christine von Neuhoff; Annette Sander; André Schrauder; Andrea Teigler-Schlegel; Jan Stary; Selim Corbacioglu; Dirk Reinhardt Journal: Blood Date: 2011-09-26 Impact factor: 22.113
Authors: B Stark; P Resnitzky; M Jeison; D Luria; O Blau; S Avigad; D Shaft; Y Kodman; R Gobuzov; S Ash Journal: Leuk Res Date: 1995-06 Impact factor: 3.156
Authors: Hiroto Inaba; Ying Fan; Stanley Pounds; Terrence L Geiger; Jeffrey E Rubnitz; Raul C Ribeiro; Ching-Hon Pui; Bassem I Razzouk Journal: Cancer Date: 2008-08-01 Impact factor: 6.860
Authors: Nobuko Hijiya; Monika L Metzger; Stan Pounds; Jeffrey E Schmidt; Bassem I Razzouk; Jeffrey E Rubnitz; Scott C Howard; Cesar A Nunez; Ching-Hon Pui; Raul C Ribeiro Journal: Pediatr Blood Cancer Date: 2005-01 Impact factor: 3.167
Authors: Kai Lehmberg; Kim E Nichols; Jan-Inge Henter; Michael Girschikofsky; Tatiana Greenwood; Michael Jordan; Ashish Kumar; Milen Minkov; Paul La Rosée; Sheila Weitzman Journal: Haematologica Date: 2015-08 Impact factor: 9.941
Authors: Volker Strenger; Gerald Merth; Herwig Lackner; Stephan W Aberle; Harald H Kessler; Markus G Seidel; Wolfgang Schwinger; Daniela Sperl; Petra Sovinz; Anna Karastaneva; Martin Benesch; Christian Urban Journal: Ann Hematol Date: 2018-02-06 Impact factor: 3.673