Literature DB >> 33616851

Myeloid Neoplasm with PDGFRA Rearrangement Manifesting as a Retromolar Pad Mass.

Amy S Duffield1,2,3, Jonathan Webster2, B Douglas Smith2, Julius S Necciai4, Austin McCuiston1,5, Alisha D Ware6.   

Abstract

Myeloid neoplasms with PDGFRA rearrangement are rare, and most commonly present with features of chronic eosinophilic leukemia; however, they rarely manifest as acute myeloid or lymphoblastic leukemia. Patients typically present with symptoms of hypereosinophilia including cardiovascular and pulmonary symptoms. An increase in mast cells is also a common feature of this disease, and there may be elevated serum tryptase with significant clinical overlap with systemic mastocytosis. Here, we present an unusual case of a myeloid neoplasm with PDGFRA rearrangement manifesting as a retromolar pad mass in a patient with a prior diagnosis of systemic mastocytosis. This case highlights the possibility of soft tissue involvement by myeloid neoplasms with PDGFRA rearrangement in the oral cavity. The identification of this entity is of significant clinical importance because many patients can be effectively treated with tyrosine kinase inhibitors.
© 2021. The Author(s), under exclusive licence to Springer Science+Business Media, LLC part of Springer Nature.

Entities:  

Keywords:  Eosinophilia; Gingiva; Mastocytosis; Oral pathology; PDGFRA-associated myeloproliferative neoplasm; Tyrosine kinase inhibitor

Mesh:

Substances:

Year:  2021        PMID: 33616851      PMCID: PMC8633353          DOI: 10.1007/s12105-021-01305-9

Source DB:  PubMed          Journal:  Head Neck Pathol        ISSN: 1936-055X


  17 in total

1.  Myeloid blast crisis evolving during imatinib treatment of an FIP1L1-PDGFR alpha-positive chronic myeloproliferative disease with prominent eosinophilia.

Authors:  N von Bubnoff; M Sandherr; G Schlimok; R Andreesen; C Peschel; J Duyster
Journal:  Leukemia       Date:  2005-02       Impact factor: 11.528

2.  Immunohistochemical study of calretinin in normal skin and cutaneous adnexal proliferations.

Authors:  Elena González-Guerra; Heinz Kutzner; Arno Rutten; Luis Requena
Journal:  Am J Dermatopathol       Date:  2012-07       Impact factor: 1.533

Review 3.  How I treat hypereosinophilic syndromes.

Authors:  Amy D Klion
Journal:  Blood       Date:  2015-05-11       Impact factor: 22.113

4.  A tyrosine kinase created by fusion of the PDGFRA and FIP1L1 genes as a therapeutic target of imatinib in idiopathic hypereosinophilic syndrome.

Authors:  Jan Cools; Daniel J DeAngelo; Jason Gotlib; Elizabeth H Stover; Robert D Legare; Jorges Cortes; Jeffrey Kutok; Jennifer Clark; Ilene Galinsky; James D Griffin; Nicholas C P Cross; Ayalew Tefferi; James Malone; Rafeul Alam; Stanley L Schrier; Janet Schmid; Michal Rose; Peter Vandenberghe; Gregor Verhoef; Marc Boogaerts; Iwona Wlodarska; Hagop Kantarjian; Peter Marynen; Steven E Coutre; Richard Stone; D Gary Gilliland
Journal:  N Engl J Med       Date:  2003-03-27       Impact factor: 91.245

Review 5.  Myeloid neoplasms with eosinophilia.

Authors:  Andreas Reiter; Jason Gotlib
Journal:  Blood       Date:  2016-12-27       Impact factor: 22.113

6.  FIP1L1-PDGFRalpha D842V, a novel panresistant mutant, emerging after treatment of FIP1L1-PDGFRalpha T674I eosinophilic leukemia with single agent sorafenib.

Authors:  E Lierman; L Michaux; E Beullens; P Pierre; P Marynen; J Cools; P Vandenberghe
Journal:  Leukemia       Date:  2009-02-12       Impact factor: 11.528

7.  The efficacy of imatinib mesylate in patients with FIP1L1-PDGFRalpha-positive hypereosinophilic syndrome. Results of a multicenter prospective study.

Authors:  Michele Baccarani; Daniela Cilloni; Michela Rondoni; Emanuela Ottaviani; Francesca Messa; Serena Merante; Mario Tiribelli; Francesco Buccisano; Nicoletta Testoni; Enrico Gottardi; Antonio de Vivo; Emilia Giugliano; Ilaria Iacobucci; Stefania Paolini; Simona Soverini; Gianantonio Rosti; Francesca Rancati; Cinzia Astolfi; Fabrizio Pane; Giuseppe Saglio; Giovanni Martinelli
Journal:  Haematologica       Date:  2007-08-01       Impact factor: 9.941

8.  Relapse following discontinuation of imatinib mesylate therapy for FIP1L1/PDGFRA-positive chronic eosinophilic leukemia: implications for optimal dosing.

Authors:  Amy D Klion; Jamie Robyn; Irina Maric; Weiming Fu; Laura Schmid; Steven Lemery; Pierre Noel; Melissa A Law; Marilyn Hartsell; Cheryl Talar-Williams; Michael P Fay; Cynthia E Dunbar; Thomas B Nutman
Journal:  Blood       Date:  2007-08-20       Impact factor: 22.113

Review 9.  Chronic eosinophilic leukemias and the myeloproliferative variant of the hypereosinophilic syndrome.

Authors:  Barbara J Bain; Sarah H Fletcher
Journal:  Immunol Allergy Clin North Am       Date:  2007-08       Impact factor: 3.479

10.  Identification of Ponatinib as a potent inhibitor of growth, migration, and activation of neoplastic eosinophils carrying FIP1L1-PDGFRA.

Authors:  Irina Sadovnik; Els Lierman; Barbara Peter; Harald Herrmann; Verena Suppan; Gabriele Stefanzl; Oskar Haas; Thomas Lion; Winfried Pickl; Jan Cools; Peter Vandenberghe; Peter Valent
Journal:  Exp Hematol       Date:  2014-01-06       Impact factor: 3.084

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