Germinotropic lymphoproliferative disorder: a systematic review
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REVIEW ARTICLE
Germinotropic lymphoproliferative disorder: a systematic review Magda Zanelli 1 & Maurizio Zizzo 2,3 & Alessandra Bisagni 1 & Elisabetta Froio 1 & Loredana De Marco 1 & Riccardo Valli 1 & Alessandra Filosa 4 & Stefano Luminari 5 & Giovanni Martino 6 & Fulvio Massaro 5 & Stefano Fratoni 7 & Stefano Ascani 8 Received: 18 November 2019 / Accepted: 4 April 2020 # Springer-Verlag GmbH Germany, part of Springer Nature 2020
Abstract Germinotropic lymphoproliferative disorder is a rare and rather enigmatic novel entity with distinctive clinicopathological features, one of which is the typical co-infection by Human herpesvirus 8 and Epstein-Barr virus. Human herpesvirus 8 is a lymphotropic virus detected in Kaposi sarcoma, multicentric Castleman disease, primary effusion lymphoma, Human herpesvirus 8-positive diffuse large B cell lymphoma not otherwise specified, and germinotropic lymphoproliferative disorder. Co-infection by Human herpesvirus 8 and Epstein-Barr virus is identified only in two lymphoproliferative diseases: germinotropic lymphoproliferative disorder and primary effusion lymphoma, which are otherwise diseases with totally different clinical presentations and outcomes. Unlike primary effusion lymphoma mostly occurring in immunocompromised individuals and following an aggressive course, germinotropic lymphoproliferative disorder usually presents with single or multiple lymphadenopathy affecting mainly immunocompetent individuals and mostly follows an indolent course. Based on the PRISMA guidelines, we carried out a systematic search on PubMed/MEDLINE, Web of Science, Scopus, EMBASE, and Cochrane Library using the search terms “germinotropic” and “lymphoproliferative disorder.” Current scientific literature reports just 19 cases of germinotropic lymphoproliferative disorder. The purpose of our systematic review is to improve our understanding of the disease, focusing on epidemiology, clinical presentation, pathological features, treatment, and outcome. In addition, we discuss the differential diagnosis with the other Human herpesvirus 8-related lymphoproliferative diseases as currently recognized in the World Health Organization classification, adding a focus on lymphoproliferative disorders showing overlapping features. Keywords Germinotropic . Lymphoproliferative . Disorder . Epstein-Barr virus . Human herpesvirus 8 * Magda Zanelli [email protected] 1
Pathology Unit, Azienda USL-IRCCS di Reggio Emilia, Reggio Emilia, Italy
2
Surgical Oncology Unit, Azienda USL-IRCCS di Reggio Emilia, Reggio Emilia, Italy
3
Clinical and Experimental Medicine PhD Program, University of Modena and Reggio Emilia, Modena, Italy
4
Pathology Unit, Area Vasta 5 (Ascoli Piceno-San Benedetto del Tronto), Ascoli Piceno, Italy
5
Hematology, Azienda USL-IRCCS di Reggio Emilia, Reggio Emilia, Italy
6
Hematology Unit, CREO, Azienda Ospedaliera di Perugia, University of Perugia, Perugia, Italy
7
Pathology Unit, St. Eugenio Hospital, Rome, Italy
8
Pathology Unit, Azienda Ospedaliera Santa Maria di Terni,
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