Granjo Elisa, Lima Margarida, Fraga Manuela, Santos Filipe, Magalhães Conceição, Queirós Maria Luís, Moreira Ilidia, Rocha Sandra, Silva Alice Santos, Rebelo Irene, Quintanilha Alexandre, Ribeiro Maria Letícia, Candeias Jorge, Orfão Alberto
Department of Hematology, Hospital S. João, Porto, Portugal.
Int J Hematol. 2002 Jun;75(5):484-8. doi: 10.1007/BF02982110.
We report the case of a boy with hereditary spherocytosis who presented with mild microcytic hypochromic anemia and recurrent leg ulcers that had been present since childhood. Chronic natural killer (NK) cell and B-cell lymphocytosis was detected 1 year after therapeutic splenectomy during investigation of recurrent episodes of neutropenia and persistent lymphocytosis. NK cells proved to be abnormal at immunophenotyping studies, and B-cells were polyclonal and displayed a normal immunophenotype. Genotypic analysis of T-cell receptor (TCR)-beta and TCR-gamma genes showed a germ-line pattern. The clinical course of this patient was characterized by multiple pulmonary infections and amygdalitis. We discuss the potential roles of persistent immune stimulation due to chronic hemolysis and severe leg ulcers and of splenectomy in the origin of NK cell lymphocytosis and the relationship between NK cells and recurrent infections, relapsing neutropenia, and polyclonal B-cell response.