Abstract • A 10-year-old girl was first seen at age 5 years with pneumonia and neutropenia. Since then, she has remained leukopenic, although manifesting a leukocytosis only when she has pulmonary infection. A rapid fall in her peripheral WBC count occurs with initiation of antibiotic therapy. Despite her neutropenia, marked myeloid hyperplasia is evident on marrow smear examination; many cells being hypersegmented with fine intralobular bridging with chromatin strands and cytoplasmic vacuolation. The peripheral WBC response to epinephrine administration did not indicate a shift from the circulating to marginal neutrophil pool. Results from a Rebuck skin window test suggested poor neutrophil tissue migration. A defect in granulocyte release from the patient's marrow may explain these bizarre hematologic findings. (Am J Dis Child 131:655-658, 1977) References 1. Baehner RL: Disorders of leukocytes leading to recurrent infection . Pediatr Clin North Am 19:935-956, 1972. 2. Krill CE, Smith HD, Mauer AM: Chronic idiopathic granulocytopenia . N Engl J Med 270:973-979, 1964.Crossref 3. Zuelzer WW: "Myelokathexis": A new form of chronic granulocytopenia . N Engl J Med 270:699-704, 1964.Crossref 4. Polmar SM, Chase PA: Quantitation of the biosynthesis of immunoglobulin in peripheral blood lymphocytes of normal and immunodeficient patients . J Pediatr 87:545-549, 1975.Crossref 5. Page AR, Good RA: Studies on cyclic neutropenia: A clinical and experimental investigation . Am J Dis Child 94:623-661, 1957. 6. Schwachman M, Diamond LK, Oski FA, et al: The syndrome of pancreatic insufficiency and bone marrow dysfunction . J Pediatr 65:645-663, 1964.Crossref 7. Zuelzer WW, Bajoghl M: Chronic granulocytopenia in childhood . Blood 23:359-374, 1964. 8. Rothstein G, Hugh E, Bishop C, et al: Stimulation of granulocytopoiesis by a diffusible factor in vivo . J Clin Invest 50:2004-2007, 1971.Crossref 9. Boggs DR, Marsh JC, Chervenick PA, et al: Neutrophil releasing activity in plasma of normal human subjects injected with endotoxin . Proc Soc Exp Biol Med 127:689-693, 1968. 10. Greenberg PL, Schrier SL: Clinical utility of in vitro evaluation of granulopoiesis . Ann Rev Med 25:269-278, 1974.Crossref 11. Boggs DR, Cartwright GE, Wintrobe MM: Neutrophilia inducing activity in plasma of dogs recovering from drug induced myelotoxicity . Am J Physiol 211:51-60, 1968. 12. Marsh JC, Levitt M: Neutrophilia inducing activity in plasma of neutropenic human beings . Blook 37:647-656, 1971.
American Journal of Diseases of Children – American Medical Association
Published: Jun 1, 1977
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