Literature DB >> 25504888

Reversible pancytopenia and immunodeficiency in a patient with hereditary folate malabsorption.

Miriam Erlacher1, Sarah Catharina Grünert, Annamaria Cseh, Robert Steinfeld, Ulrich Salzer, Ekkehart Lausch, Ulrike Nosswitz, Gregor Dückers, Tim Niehues, Stephan Ehl, Charlotte Marie Niemeyer, Carsten Speckmann.   

Abstract

Mutations in SLC46A1 result in a defect of the proton coupled folate transporter (PCFT) and are the basis of hereditary folate malabsorption (HFM). Patients with HFM frequently present with neurodevelopmental delay and megaloblastic anemia. Some cases may be complicated by additional lymphopenia and immunodeficiency. We report a patient with a new homozygous mutation in the SLC46A1 gene. The boy presented with early-onset pancytopenia and secondary immunodeficiency. We provide clinical and molecular observations that extend the phenotypic description of HFM and highlight diagnostic as well as therapeutic pitfalls in this rare condition.
© 2014 Wiley Periodicals, Inc.

Entities:  

Keywords:  bone marrow failure; congenital (not HIV); hematology; immunodeficiency; immunology; infections in imunocompromised host non-malignant

Mesh:

Year:  2014        PMID: 25504888     DOI: 10.1002/pbc.25364

Source DB:  PubMed          Journal:  Pediatr Blood Cancer        ISSN: 1545-5009            Impact factor:   3.167


  7 in total

Review 1.  Folate nutrition and blood-brain barrier dysfunction.

Authors:  Patrick J Stover; Jane Durga; Martha S Field
Journal:  Curr Opin Biotechnol       Date:  2017-02-10       Impact factor: 9.740

Review 2.  The proton-coupled folate transporter (PCFT-SLC46A1) and the syndrome of systemic and cerebral folate deficiency of infancy: Hereditary folate malabsorption.

Authors:  Rongbao Zhao; Srinivas Aluri; I David Goldman
Journal:  Mol Aspects Med       Date:  2016-09-21

3.  Role of Intramuscular Levofolinate Administration in the Treatment of Hereditary Folate Malabsorption: Report of Three Cases.

Authors:  Emanuela Manea; Paul Gissen; Simon Pope; Simon J Heales; Spyros Batzios
Journal:  JIMD Rep       Date:  2017-07-07

4.  Hereditary folate malabsorption due to a mutation in the external gate of the proton-coupled folate transporter SLC46A1.

Authors:  Srinivas Aluri; Rongbao Zhao; Charlotte Lubout; Susanna M I Goorden; Andras Fiser; I David Goldman
Journal:  Blood Adv       Date:  2018-01-05

5.  Abnormal folate metabolism causes age-, sex- and parent-of-origin-specific haematological defects in mice.

Authors:  Nisha Padmanabhan; Katerina Menelaou; Jiali Gao; Alexander Anderson; Georgina E T Blake; Tanya Li; B Nuala Daw; Erica D Watson
Journal:  J Physiol       Date:  2018-08-15       Impact factor: 5.182

Review 6.  Missing Cells: Pathophysiology, Diagnosis, and Management of (Pan)Cytopenia in Childhood.

Authors:  Miriam Erlacher; Brigitte Strahm
Journal:  Front Pediatr       Date:  2015-07-13       Impact factor: 3.418

Review 7.  The evolving biology of the proton-coupled folate transporter: New insights into regulation, structure, and mechanism.

Authors:  Zhanjun Hou; Aleem Gangjee; Larry H Matherly
Journal:  FASEB J       Date:  2022-02       Impact factor: 5.834

  7 in total

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