Immunodeficiency in a Child with Rapadilino Syndrome: A Case Report and Review of the Literature

Rapadilino syndrome is a genetic disease characterized by a characteristic clinical tableau. It is caused by mutations in RECQL4 gene. Immunodeficiency is not described as a classical feature of the disease. We present a 2-year-old girl with Rapadilino syndrome with important lymphadenopathies and p...

Full description

Saved in:
Bibliographic Details
Main Authors: M. M. G. Vollebregt, A. Malfroot, M. De Raedemaecker, M. van der Burg, J. E. van der Werff ten Bosch
Format: Article
Language:English
Published: Wiley 2015-01-01
Series:Case Reports in Immunology
Online Access:http://dx.doi.org/10.1155/2015/137368
Tags: Add Tag
No Tags, Be the first to tag this record!
Description
Summary:Rapadilino syndrome is a genetic disease characterized by a characteristic clinical tableau. It is caused by mutations in RECQL4 gene. Immunodeficiency is not described as a classical feature of the disease. We present a 2-year-old girl with Rapadilino syndrome with important lymphadenopathies and pneumonia due to disseminated Mycobacterium lentiflavum infection. An immunological work-up showed several unexpected abnormalities. Repeated blood samples showed severe lymphopenia. Immunophenotyping showed low T, B, and NK cells. No Treg cells were seen. T cell responses to stimulations were insufficient. The IL12/IL23 interferon gamma pathway was normal. Gamma globulin levels and vaccination responses were low. With this report, we aim to stress the importance of screening immunodeficiency in patients with RECQL4 mutations for immunodeficiency and the need to further research into its physiopathology.
ISSN:2090-6609
2090-6617