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American Academy of Pediatrics
Case Report

Multicentric Castleman Disease in an HHV8-Infected Child Born to Consanguineous Parents With Systematic Review

Sandrine Leroy, Despina Moshous, Olivier Cassar, Yves Reguerre, Minji Byun, Vincent Pedergnana, Danielle Canioni, Antoine Gessain, Eric Oksenhendler, Claire Fieschi, Nizar Mahlaoui, Jean-Pierre Rivière, Rose-Marie Herbigneaux, Matthias Muszlak, Jean-Pierre Arnaud, Alain Fischer, Capucine Picard, Stéphane Blanche, Sabine Plancoulaine and Jean-Laurent Casanova
Pediatrics January 2012, 129 (1) e199-e203; DOI: https://doi.org/10.1542/peds.2010-2739
Sandrine Leroy
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Despina Moshous
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Olivier Cassar
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Yves Reguerre
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Minji Byun
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Vincent Pedergnana
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Danielle Canioni
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Antoine Gessain
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Eric Oksenhendler
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Claire Fieschi
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Nizar Mahlaoui
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Jean-Pierre Rivière
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Rose-Marie Herbigneaux
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Matthias Muszlak
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Jean-Pierre Arnaud
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Alain Fischer
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Capucine Picard
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Stéphane Blanche
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Sabine Plancoulaine
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Jean-Laurent Casanova
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Abstract

Childhood multicentric Castleman disease (MCD) is a rare and unexplained lymphoproliferative disorder. We report a human herpesvirus-8 (HHV-8)-infected child, born to consanguineous Comorian parents, who displayed isolated MCD in the absence of any known immunodeficiency. We also systematically review the clinical features of the 32 children previously reported with isolated and unexplained MCD. The characteristics of this patient and the geographic areas of origin of most previous cases suggest that pediatric MCD is associated with HHV-8 infection. Moreover, as previously suggested for Kaposi sarcoma, MCD in childhood may result from inborn errors of immunity to HHV-8 infection.

KEY WORDS
  • Children
  • human herpes virus 8
  • multicentric Castleman disease
  • systematic review
  • Abbreviations:
    CD —
    Castleman disease
    HHV-8 —
    human herpes virus 8
    Ig —
    immunoglobulin
    KS —
    Kaposi sarcoma
    MCD —
    multicentric Castleman disease
    PCR —
    polymerase chain reaction
    SNP —
    single-nucleotide polymorphism
    • Accepted September 8, 2011.
    • Copyright © 2012 by the American Academy of Pediatrics

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    Multicentric Castleman Disease in an HHV8-Infected Child Born to Consanguineous Parents With Systematic Review
    Sandrine Leroy, Despina Moshous, Olivier Cassar, Yves Reguerre, Minji Byun, Vincent Pedergnana, Danielle Canioni, Antoine Gessain, Eric Oksenhendler, Claire Fieschi, Nizar Mahlaoui, Jean-Pierre Rivière, Rose-Marie Herbigneaux, Matthias Muszlak, Jean-Pierre Arnaud, Alain Fischer, Capucine Picard, Stéphane Blanche, Sabine Plancoulaine, Jean-Laurent Casanova
    Pediatrics Jan 2012, 129 (1) e199-e203; DOI: 10.1542/peds.2010-2739

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    Multicentric Castleman Disease in an HHV8-Infected Child Born to Consanguineous Parents With Systematic Review
    Sandrine Leroy, Despina Moshous, Olivier Cassar, Yves Reguerre, Minji Byun, Vincent Pedergnana, Danielle Canioni, Antoine Gessain, Eric Oksenhendler, Claire Fieschi, Nizar Mahlaoui, Jean-Pierre Rivière, Rose-Marie Herbigneaux, Matthias Muszlak, Jean-Pierre Arnaud, Alain Fischer, Capucine Picard, Stéphane Blanche, Sabine Plancoulaine, Jean-Laurent Casanova
    Pediatrics Jan 2012, 129 (1) e199-e203; DOI: 10.1542/peds.2010-2739
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