Literature DB >> 8669937

Severe combined immunodeficiency (SCID) associated neutropenia: a lesson from monozygotic twins.

T Niehues1, K Schwarz, M Schneider, H Schroten, E Schröder, V Stephan, V Wahn.   

Abstract

A case of severe combined immunodeficiency (SCID) in monozygotic twin sisters was detected at 3 months of age with neutropenia in one twin and a normal differential count in the other. The neutropenic twin, suffering from severe skin ulcers, was successfully treated with granulocyte colony stimulating factor (G-CSF). Discordant occurrence of neutropenia in identical twins shows that there may be a non-genetic cause for the neutropenia in SCID. Suppression of myelopoiesis was probably induced by activated maternal T cells. The neutropenia in this case may thus be classified as SCID associated neutropenia, as opposed to reticular dysgenesis, in which the neutropenia is G-CSF refractory and is most probably caused by a genetic stem cell defect. A response to G-CSF in a neutropenic child with SCID can be clinically beneficial and might help to distinguish between G-CSF unresponsive reticular dysgenesis and G-CSF responsive SCID associated neutropenia.

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Year:  1996        PMID: 8669937      PMCID: PMC1511478          DOI: 10.1136/adc.74.4.340

Source DB:  PubMed          Journal:  Arch Dis Child        ISSN: 0003-9888            Impact factor:   3.791


  8 in total

1.  Reticular dysgenesia.

Authors:  O de VAAL; V SEYNHAEVE
Journal:  Lancet       Date:  1959-12-19       Impact factor: 79.321

2.  Effect of recombinant human granulocyte colony-stimulating factor in reticular dysgenesis.

Authors:  W Bujan; A Ferster; E Sariban; W Friedrich
Journal:  Blood       Date:  1993-09-01       Impact factor: 22.113

3.  Severe combined immunodeficiency with leukopenia (reticular dysgenesis) in siblings: immunologic and histopathologic findings.

Authors:  D R Ownby; S Pizzo; L Blackmon; S A Gall; R H Buckley
Journal:  J Pediatr       Date:  1976-09       Impact factor: 4.406

4.  Characterization of a panel of highly variable minisatellites cloned from human DNA.

Authors:  Z Wong; V Wilson; I Patel; S Povey; A J Jeffreys
Journal:  Ann Hum Genet       Date:  1987-10       Impact factor: 1.670

5.  Use of recombinant human granulocyte-macrophage colony stimulating factor in an infant with reticular dysgenesis.

Authors:  C Azcona; V Alzina; P Barona; L Sierrasesúmaga; I Villa-Elízaga
Journal:  Eur J Pediatr       Date:  1994-03       Impact factor: 3.183

6.  Severe congenital leukopenia (reticular dysgenesis). Immunologic and morphologic characterizations of leukocytes.

Authors:  M Roper; R T Parmley; W M Crist; D R Kelly; M D Cooper
Journal:  Am J Dis Child       Date:  1985-08

7.  Successful bone-marrow transplantation for reticular dysgenesis.

Authors:  R J Levinsky; K Tiedeman
Journal:  Lancet       Date:  1983-03-26       Impact factor: 79.321

8.  Severe combined immunodeficiency: a retrospective single-center study of clinical presentation and outcome in 117 patients.

Authors:  J L Stephan; V Vlekova; F Le Deist; S Blanche; J Donadieu; G De Saint-Basile; A Durandy; C Griscelli; A Fischer
Journal:  J Pediatr       Date:  1993-10       Impact factor: 4.406

  8 in total

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