Literature DB >> 9989845

The spleen in the Wiskott-Aldrich syndrome: histopathologic abnormalities of the white pulp correlate with the clinical phenotype of the disease.

W Vermi1, L Blanzuoli, M D Kraus, P Grigolato, F Donato, G Loffredo, C E Marino, D Alberti, L D Notarangelo, F Facchetti.   

Abstract

The Wiskott-Aldrich syndrome (WAS) is a X-linked hematologic disorder characterized by thrombocytopenia, eczema, and immunodeficiency of variable severity. Reported here are the results of a morphologic, morphometric, and immunophenotypic analysis of splenic lymphoid tissue in 12 WAS patients with documented molecular defect and with different disease severity. Spleens from 29 age-matched patients with different diseases were used as controls. Paraffin-embedded tissue (from all cases) and fresh-frozen samples (from 5 WAS patients and 4 control subjects) were used to study the different white pulp compartments by classic morphologic, immunophenotyping, and image analysis techniques. Data were statistically analyzed by both parametric and nonparametric tests. Spleens from WAS patients showed a significant depletion of the total white pulp (p = 0.0008), T cell (p < 0.05), and B cell (p = 0.0002) areas and marginal zone (MZ) thickness (p < 0.0001). Among WAS patients, a negative correlation was found between the score of severity of the disease and all variables considered (Spearman's rank correlation coefficient, r = -0.79, r = -0.73, r = -0.68, and r = -0.56, respectively). In conclusion, this study shows that in WAS a general depletion of the splenic white pulp occurs, supporting the evidence that WAS is characterized by a combined immune defect. The significant reduction of the MZ may explain the inability of WAS patients to mount a response to T-independent antigens.

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Year:  1999        PMID: 9989845     DOI: 10.1097/00000478-199902000-00007

Source DB:  PubMed          Journal:  Am J Surg Pathol        ISSN: 0147-5185            Impact factor:   6.394


  14 in total

Review 1.  Wiskott-Aldrich syndrome: another piece in the puzzle.

Authors:  L D Notarangelo; L Mori
Journal:  Clin Exp Immunol       Date:  2005-02       Impact factor: 4.330

2.  WASP confers selective advantage for specific hematopoietic cell populations and serves a unique role in marginal zone B-cell homeostasis and function.

Authors:  Lisa S Westerberg; Miguel A de la Fuente; Fredrik Wermeling; Hans D Ochs; Mikael C I Karlsson; Scott B Snapper; Luigi D Notarangelo
Journal:  Blood       Date:  2008-09-04       Impact factor: 22.113

3.  Phenotypic perturbation of B cells in the Wiskott-Aldrich syndrome.

Authors:  J Y Park; A Shcherbina; F S Rosen; A P Prodeus; E Remold-O'Donnell
Journal:  Clin Exp Immunol       Date:  2005-02       Impact factor: 4.330

4.  Abnormalities of follicular helper T-cell number and function in Wiskott-Aldrich syndrome.

Authors:  Xuan Zhang; Rongxin Dai; Wenyan Li; Hongyi Zhao; Yongjie Zhang; Lina Zhou; Hongqiang Du; Guangjin Luo; Junfeng Wu; Linlin Niu; Yunfei An; Zhiyong Zhang; Yuan Ding; Wenxia Song; Chaohong Liu; Xiaodong Zhao
Journal:  Blood       Date:  2016-05-11       Impact factor: 22.113

5.  Wiskott-Aldrich syndrome protein (WASP) and N-WASP are critical for peripheral B-cell development and function.

Authors:  Lisa S Westerberg; Carin Dahlberg; Marisa Baptista; Christopher J Moran; Cynthia Detre; Marton Keszei; Michelle A Eston; Frederick W Alt; Cox Terhorst; Luigi D Notarangelo; Scott B Snapper
Journal:  Blood       Date:  2012-03-12       Impact factor: 22.113

Review 6.  Hematopoietic cell transplantation for Wiskott-Aldrich syndrome: advances in biology and future directions for treatment.

Authors:  Sung-Yun Pai; Luigi D Notarangelo
Journal:  Immunol Allergy Clin North Am       Date:  2010-05       Impact factor: 3.479

7.  B-cell intrinsic TLR7 signals promote depletion of the marginal zone in a murine model of Wiskott-Aldrich syndrome.

Authors:  Nikita S Kolhatkar; Nicole E Scharping; Jenna M Sullivan; Holly M Jacobs; Marc A Schwartz; Socheath Khim; Luigi D Notarangelo; Adrian J Thrasher; David J Rawlings; Shaun W Jackson
Journal:  Eur J Immunol       Date:  2015-08-31       Impact factor: 5.532

Review 8.  The thrombocytopenia of WAS: a familial form of ITP?

Authors:  Ted S Strom
Journal:  Immunol Res       Date:  2009       Impact factor: 2.829

9.  WASP and Mst1 coregulate B-cell development and B-cell receptor signaling.

Authors:  Lu Huang; Xiaoyu Sun; Di Yang; Xin Dai; Panpan Jiang; Xiaoming Bai; Yongjie Zhang; Jinzhi Wang; Wenyan Li; Heather Miller; Wenxia Song; Bebhinn Treanor; Xiaodong Zhao; Chaohong Liu
Journal:  Blood Adv       Date:  2020-02-11

Review 10.  Wiskott-Aldrich Syndrome: Immunodeficiency resulting from defective cell migration and impaired immunostimulatory activation.

Authors:  Gerben Bouma; Siobhan O Burns; Adrian J Thrasher
Journal:  Immunobiology       Date:  2009-07-22       Impact factor: 3.144

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