Literature DB >> 34845303

Molecular assessment of testicular adult granulosa cell tumor demonstrates significant differences when compared to ovarian counterparts.

Stephanie Siegmund1, Lynette M Sholl1, Kristine M Cornejo2, Ankur R Sangoi3, Christopher N Otis4, Rohit Mehra5, Michelle S Hirsch1, Andres M Acosta6.   

Abstract

Testicular adult granulosa cell tumor (AGCT) is a rare type of sex-cord stromal tumor that affects patients of a wide age range and has the potential for late metastasis. In the testis, the diagnosis of AGCTs often requires the exclusion of other more common types of sex-cord stromal tumors. Immunohistochemistry is of limited utility, being used mostly to confirm sex-cord lineage and to exclude other entities when morphology is not typical. Unlike ovarian AGCTs, which are molecularly homogeneous and harbor a specific activating FOXL2 mutation (c.7558C > T p.C134W) in >90% of cases, the molecular characteristics of testicular AGCTs remain largely unknown. In the current study, we analyzed 13 testicular AGCTs diagnosed at multiple institutions using massively parallel DNA sequencing to evaluate single nucleotide variants, copy number alterations, and structural variants. In all, 10/13 cases were sequenced successfully. Notably, the FOXL2 c.7558C > T (p.C134W) mutation was identified in only a single case (1/10, 10%). The remaining cases were molecularly heterogeneous, with largely nonrecurrent genetic variants. Putative driver events in individual cases included a well-characterized gain-of-function NRAS mutation, as well as inactivation of ATM and TP53, among others. The only highly recurrent finding was single copy loss of 22q (7/10 cases, 70%). Comparatively, the frequencies of FOXL2 c.7558C > T (p.C134W) and 22q loss in 12 metastatic ovarian AGCTs identified in our database were 92% (11/12) and 42% (5/12), respectively. The results of the present study suggest that testicular AGCTs are different from their ovarian counterparts in that they appear to be molecularly heterogeneous and only rarely harbor FOXL2 mutations.
© 2021. The Author(s), under exclusive licence to United States & Canadian Academy of Pathology.

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Year:  2021        PMID: 34845303     DOI: 10.1038/s41379-021-00977-6

Source DB:  PubMed          Journal:  Mod Pathol        ISSN: 0893-3952            Impact factor:   8.209


  3 in total

1.  Adult granulosa cell tumor of the testicle.

Authors:  Richard W Norman; Anna Sheridan-Jonah; Jennifer Merrimen; Rekha Gupta
Journal:  Can J Urol       Date:  2013-02       Impact factor: 1.344

2.  A novel mouse model of testicular granulosa cell tumors.

Authors:  Xin Fang; Nan Ni; Yang Gao; David F Vincent; Laurent Bartholin; Qinglei Li
Journal:  Mol Hum Reprod       Date:  2018-07-01       Impact factor: 4.025

3.  KMT2D/MLL2 inactivation is associated with recurrence in adult-type granulosa cell tumors of the ovary.

Authors:  R Tyler Hillman; Joseph Celestino; Christopher Terranova; Hannah C Beird; Curtis Gumbs; Latasha Little; Tri Nguyen; Rebecca Thornton; Samantha Tippen; Jianhua Zhang; Karen H Lu; David M Gershenson; Kunal Rai; Russell R Broaddus; P Andrew Futreal
Journal:  Nat Commun       Date:  2018-06-27       Impact factor: 14.919

  3 in total

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