Clinical Manifestations and Pathophysiological Mechanisms of the Wiskott-Aldrich Syndrome

被引:146
作者
Candotti, Fabio [1 ,2 ]
机构
[1] Univ Hosp Lausanne, Div Immunol & Allergy, Lausanne, Switzerland
[2] CHU Vaudois, IAL, BH10-527,Rue Bugnon 46, CH-1011 Lausanne, Switzerland
关键词
Thrombocytopenia; atopy; autoimmunity; immunodeficiency; X-chromosome; X-LINKED THROMBOCYTOPENIA; SYNDROME PROTEIN-DEFICIENCY; STEM-CELL TRANSPLANTATION; REGULATORY T-CELLS; IN-VIVO REVERSION; SOCIETY-FOR-IMMUNODEFICIENCIES; BONE-MARROW-TRANSPLANTATION; MEDIATED GENE-THERAPY; B-CELLS; SOMATIC MOSAICISM;
D O I
10.1007/s10875-017-0453-z
中图分类号
R392 [医学免疫学]; Q939.91 [免疫学];
学科分类号
100102 ;
摘要
The Wiskott-Aldrich syndrome (WAS) is a rare X-linked disorder originally described by Dr. Alfred Wiskott in 1937 and Dr. Robert Aldrich in 1954 as a familial disease characterized by infections, bleeding tendency, and eczema. Today, it is well recognized that the syndrome has a wide clinical spectrum ranging from mild, isolated thrombocytopenia to full-blown presentation that can be complicated by life-threatening hemorrhages, immunodeficiency, atopy, autoimmunity, and cancer. The pathophysiology of classic and emerging features is being elucidated by clinical studies, but remains incompletely defined, which hinders the application of targeted therapies. At the same time, progress of hematopoietic stem cell transplantation and gene therapy offer optimistic prospects for treatment options aimed at the replacement of the defective lymphohematopoietic system that have the potential to provide a cure for this rare and polymorphic disease.
引用
收藏
页码:13 / 27
页数:15
相关论文
共 140 条
[1]   Outcomes Following Gene Therapy in Patients With Severe Wiskott-Aldrich Syndrome [J].
Abina, Salima Hacein-Bey ;
Gaspar, H. Bobby ;
Blondeau, Johanna ;
Caccavelli, Laure ;
Charrier, Sabine ;
Buckland, Karen ;
Picard, Capucine ;
Six, Emmanuelle ;
Himoudi, Nourredine ;
Gilmour, Kimberly ;
McNicol, Anne-Marie ;
Hara, Havinder ;
Xu-Bayford, Jinhua ;
Rivat, Christine ;
Touzot, Fabien ;
Mavilio, Fulvio ;
Lim, Annick ;
Treluyer, Jean-Marc ;
Heritier, Sebastien ;
Lefrere, Francois ;
Magalon, Jeremy ;
Pengue-Koyi, Isabelle ;
Honnet, Geraldine ;
Blanche, Stephane ;
Sherman, Eric A. ;
Male, Frances ;
Berry, Charles ;
Malani, Nirav ;
Bushman, Frederic D. ;
Fischer, Alain ;
Thrasher, Adrian J. ;
Galy, Anne ;
Cavazzana, Marina .
JAMA-JOURNAL OF THE AMERICAN MEDICAL ASSOCIATION, 2015, 313 (15) :1550-1563
[2]   Impaired in vitro regulatory T cell function associated with Wiskott-Aldrich syndrome [J].
Adriani, Marsilio ;
Aoki, Joseph ;
Horai, Reiko ;
Thornton, Angela M. ;
Konno, Akihiro ;
Kirby, Martha ;
Anderson, Stacie M. ;
Siegel, Richard M. ;
Candotti, Fabio ;
Schwartzberg, Pamela L. .
CLINICAL IMMUNOLOGY, 2007, 124 (01) :41-48
[3]   Lentiviral Hematopoietic Stem Cell Gene Therapy in Patients with Wiskott-Aldrich Syndrome [J].
Aiuti, Alessandro ;
Biasco, Luca ;
Scaramuzza, Samantha ;
Ferrua, Francesca ;
Cicalese, Maria Pia ;
Baricordi, Cristina ;
Dionisio, Francesca ;
Calabria, Andrea ;
Giannelli, Stefania ;
Castiello, Maria Carmina ;
Bosticardo, Marita ;
Evangelio, Costanza ;
Assanelli, Andrea ;
Casiraghi, Miriam ;
Di Nunzio, Sara ;
Callegaro, Luciano ;
Benati, Claudia ;
Rizzardi, Paolo ;
Pellin, Danilo ;
Di Serio, Clelia ;
Schmidt, Manfred ;
Von Kalle, Christof ;
Gardner, Jason ;
Mehta, Nalini ;
Neduva, Victor ;
Dow, David J. ;
Galy, Anne ;
Miniero, Roberto ;
Finocchi, Andrea ;
Metin, Ayse ;
Banerjee, Pinaki P. ;
Orange, Jordan S. ;
Galimberti, Stefania ;
Valsecchi, Maria Grazia ;
Biffi, Alessandra ;
Montini, Eugenio ;
Villa, Anna ;
Ciceri, Fabio ;
Roncarolo, Maria Grazia ;
Naldini, Luigi .
SCIENCE, 2013, 341 (6148) :865-U71
[4]   X-linked thrombocytopenia (XLT) due to WAS mutations: clinical characteristics, long-term outcome, and treatment options [J].
Albert, Michael H. ;
Bittner, Tanja C. ;
Nonoyama, Shigeaki ;
Notarangelo, Lucia Dora ;
Burns, Siobhan ;
Imai, Kohsuke ;
Espanol, Teresa ;
Fasth, Anders ;
Pellier, Isabelle ;
Strauss, Gabriele ;
Morio, Tomohiro ;
Gathmann, Benjamin ;
Noordzij, Jeroen G. ;
Fillat, Cristina ;
Hoenig, Manfred ;
Nathrath, Michaela ;
Meindl, Alfons ;
Pagel, Philipp ;
Wintergerst, Uwe ;
Fischer, Alain ;
Thrasher, Adrian J. ;
Belohradsky, Bernd H. ;
Ochs, Hans D. .
BLOOD, 2010, 115 (16) :3231-3238
[5]  
ALDRICH RA, 1954, PEDIATRICS, V13, P133
[6]   Spontaneous in vivo reversion of an inherited mutation in the Wiskott-Aldrich syndrome [J].
Ariga, T ;
Kondoh, T ;
Yamaguchi, K ;
Yamada, M ;
Sasaki, S ;
Nelson, DL ;
Ikeda, H ;
Kobayashi, K ;
Moriuchi, H ;
Sakiyama, Y .
JOURNAL OF IMMUNOLOGY, 2001, 166 (08) :5245-5249
[7]  
BACH FH, 1968, LANCET, V2, P1364
[8]  
Badolato R, 1998, J IMMUNOL, V161, P1026
[9]   Human regulatory T cells and their role in autoimmune disease [J].
Baecher-Allan, Clare ;
Hafler, David A. .
IMMUNOLOGICAL REVIEWS, 2006, 212 :203-216
[10]   A rare case of Wiskott-Aldrich Syndrome with normal platelet size: A case report [J].
Baharin M.F. ;
Dhaliwal J.S. ;
Sarachandran S.V.V. ;
Idris S.Z. ;
Yeoh S.L. .
Journal of Medical Case Reports, 10 (1)