Evaluation of immunophenotypic and molecular biomarkers for Sézary syndrome using standard operating procedures: multicenter study of 59 cases

Stephanie E. Boonk, Willem H. Zoutman, Anne Marie-Cardine, Leslie van der Fits, Jacoba J. Out-Luiting, Tracey J. Mitchell, Isabella Tosi, Stephen L. Morris, Blaithin Moriarty, Nina Booken, Moritz Felcht, Pietro Quaglino, Renata Ponti, Emanuela Barberio, Caroline Ram-Wolff, Kirsi Jäntti, Annamari Ranki, Maria Grazia Bernengo, Claus-Detlev Klemke, Armand BensussanLaurence Michel, Sean Whittaker, Martine Bagot, Cornelis P. Tensen, Rein Willemze, Maarten H. Vermeer

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Abstract

Abstract Differentiation between Sézary syndrome (SS) and erythrodermic inflammatory dermatoses (EID) can be challenging and a number of studies have attempted to identify characteristic immunophenotypic changes and molecular biomarkers in Sézary cells that could be useful as additional diagnostic criterion. In this European multicenter study the sensitivity and specificity of these immunophenotypic and recently proposed but unconfirmed molecular biomarkers in SS was investigated. Peripheral blood CD4+ T-cells from 59 SS and 19 EID patients were analyzed for cell surface proteins by flow cytometry, and for copy number alterations and differential gene expression using custom made qPCR plates. Experiments were performed in duplicate in two independent centers using standard operating procedures with almost identical results. Sézary cells showed MYC gain (40%) and MNT loss (66%), upregulation of DNM3 (75%), TWIST1 (69%), EPHA4 (66%) and PLS3 (66%) and downregulation of STAT4 (91%). Loss of CD26 (≥ 80% CD4+ T-cells) and/ or CD7 (≥ 40% CD4+ T-cells) and combination of altered expression of STAT4, TWIST1 and DNM3 or PLS3, could distinguish respectively 83% and 98% of SS patients from EID cases with 100% specificity. These additional diagnostic panels will be useful adjuncts in the differential diagnosis of SS versus EID.
Original languageEnglish
JournalJournal of Investigative Dermatology
Early online date28 Feb 2016
DOIs
Publication statusE-pub ahead of print - 28 Feb 2016

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