Literature DB >> 868817

'Myelokathexis'. Neutropenia with marrow hyperplasia.

S O'Regan, A J Newman, R C Graham.   

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 adminstration 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.

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Year:  1977        PMID: 868817     DOI: 10.1001/archpedi.1977.02120190049011

Source DB:  PubMed          Journal:  Am J Dis Child        ISSN: 0002-922X


  9 in total

1.  WHIM syndrome myelokathexis reproduced in the NOD/SCID mouse xenotransplant model engrafted with healthy human stem cells transduced with C-terminus-truncated CXCR4.

Authors:  Toshinao Kawai; Uimook Choi; Lanise Cardwell; Suk See DeRavin; Nora Naumann; Narda L Whiting-Theobald; Gilda F Linton; Jaehyun Moon; Philip M Murphy; Harry L Malech
Journal:  Blood       Date:  2006-08-31       Impact factor: 22.113

2.  Myelokathexis: chronic neutropenia with hyperplastic bone marrow and hypersegmented neutrophils in two siblings.

Authors:  J Bohinjec
Journal:  Blut       Date:  1981-03

Review 3.  WHIM Syndrome: from Pathogenesis Towards Personalized Medicine and Cure.

Authors:  Lauren E Heusinkveld; Shamik Majumdar; Ji-Liang Gao; David H McDermott; Philip M Murphy
Journal:  J Clin Immunol       Date:  2019-07-16       Impact factor: 8.317

Review 4.  Infections in patients with inherited defects in phagocytic function.

Authors:  Timothy Andrews; Kathleen E Sullivan
Journal:  Clin Microbiol Rev       Date:  2003-10       Impact factor: 26.132

Review 5.  WHIM syndrome: congenital immune deficiency disease.

Authors:  Toshinao Kawai; Harry L Malech
Journal:  Curr Opin Hematol       Date:  2009-01       Impact factor: 3.284

6.  Pathogenesis, diagnosis and therapeutic strategies in WHIM syndrome immunodeficiency.

Authors:  Lauren E Heusinkveld; Erin Yim; Alexander Yang; Ari B Azani; Qian Liu; Ji-Liang Gao; David H McDermott; Philip M Murphy
Journal:  Expert Opin Orphan Drugs       Date:  2017-09-25       Impact factor: 0.694

Review 7.  WHIM syndrome: a defect in CXCR4 signaling.

Authors:  George A Diaz; A Virginia Gulino
Journal:  Curr Allergy Asthma Rep       Date:  2005-09       Impact factor: 4.919

8.  Familial occurrence of warts, hypogammaglobulinemia, infections, and myelokathexis (WHIM) syndrome.

Authors:  Maciej Siedlar; Zbigniew Rudzki; Magdalena Strach; Elzbieta Trzyna; Anna Pituch-Noworolska; Anita Błaut-Szlósarczyk; Karolina Bukowska-Strakova; Marzena Lenart; Tomasz Grodzicki; Marek Zembala
Journal:  Arch Immunol Ther Exp (Warsz)       Date:  2008-12-01       Impact factor: 4.291

9.  Description and outcome of a cohort of 8 patients with WHIM syndrome from the French Severe Chronic Neutropenia Registry.

Authors:  Sarah Beaussant Cohen; Odile Fenneteau; Emmanuel Plouvier; Pierre-Simon Rohrlich; Gerard Daltroff; Isabelle Plantier; Alain Dupuy; Delphine Kerob; Blandine Beaupain; Pierre Bordigoni; Fanny Fouyssac; Anne-Lise Delezoide; Gilles Devouassoux; Jean François Nicolas; Philippe Bensaid; Yves Bertrand; Karl Balabanian; Christine Bellanne Chantelot; Françoise Bachelerie; Jean Donadieu
Journal:  Orphanet J Rare Dis       Date:  2012-09-25       Impact factor: 4.123

  9 in total

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