Pradeep Deivasigamani, Suprit Basu, Rashmi Rikhi, Kanika Arora, Vibhu Joshi, Richa Jain, Amit Rawat, Surjit Singh, Deepti Suri
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Screening for Wiskott-Aldrich Syndrome in Chronic Idiopathic Thrombocytopenic Purpura.
The clinical spectrum of X-linked thrombocytopenia (XLT)/ Wiskott-Aldrich syndrome (WAS) is broad, ranging from mild, intermittent thrombocytopenia to the classical severe phenotype characterized by eczema, infections, and thrombocytopenia. Patients with XLT often lack the complete triad but manifest with thrombocytopenia-associated bleeding and are hence misdiagnosed as chronic immune thrombocytopenia (ITP). Moreover, the clinical picture is further complicated by the development of autoimmune cytopenia. Early identification is critical, as definitive treatment with hematopoietic stem cell transplantation (HSCT) can be offered, and unnecessary immunosuppressive therapies, including corticosteroids, can be avoided. In this study, the authors screened a cohort of boys with chronic ITP using flow cytometry-based WASp protein expression analysis, followed by genetic testing. Among 38 patients evaluated, 5 (13.5%) had pathogenic variants in the WAS gene. Clinical features such as X-linked family history, eczema, low mean platelet volume (MPV), or reduced WASp expression in chronic ITP patients with suboptimal response to immunomodulatory drugs should prompt genetic evaluation for WAS.
期刊介绍:
Indian Journal of Pediatrics (IJP), is an official publication of the Dr. K.C. Chaudhuri Foundation. The Journal, a peer-reviewed publication, is published twelve times a year on a monthly basis (January, February, March, April, May, June, July, August, September, October, November, December), and publishes clinical and basic research of all aspects of pediatrics, provided they have scientific merit and represent an important advance in knowledge. The Journal publishes original articles, review articles, case reports which provide new information, letters in relation to published articles, scientific research letters and picture of the month, announcements (meetings, courses, job advertisements); summary report of conferences and book reviews.