Myoid gonadal tumor. Case series, systematic review, and Bayesian analysis

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ORIGINAL ARTICLE

Myoid gonadal tumor. Case series, systematic review, and Bayesian analysis Salvatore Lorenzo Renne 1,2 & Marina Valeri 1,2 Giuseppe Renne 5 & Maurizio Colecchia 4

&

Antonella Tosoni 3 & Alessia Bertolotti 4 & Roberta Rossi 3 &

Received: 16 September 2020 / Revised: 18 October 2020 / Accepted: 23 October 2020 # Springer-Verlag GmbH Germany, part of Springer Nature 2020

Abstract Myoid gonadal stromal tumor represents a rare testicular neoplasm displaying smooth muscular and gonadal stromal differentiation. This entity has very few cases reported in the literature that describe heterogeneous clinical and pathological characteristics. Bayesian statistics provides a useful framework to combine information from diverse sources. We here presented a case series—the largest so far reported—of myoid gonadal stromal tumor (4 cases) with extensive morphologic, immunohistochemical, and molecular characterization, performed a systematic review of the literature (that identified 9 papers), and used a Bayesian data analysis to understand the characteristics of this disease. Our study collectively described 16 cases. This neoplasm is mainly found in adults (mean age about 40 years) and often has a size of about 3 cm. By morphology, the tumor can infiltrate testicular tubules and is composed of spindle cells; few mitoses can be seen (usually 2/10 HPF). Neoplastic cells are diffusely positive with α-smooth muscle actin with a tram-track staining pattern. S100 protein, FOXL2, and SF1 are also characteristically positive. Moreover, this neoplasm can display epithelial differentiation, in about half of the cases. In conclusion, we foresee the use of this statistical approach in pathology: our analysis allowed a more precise description of this rare entity. Keywords Testicular neoplasm . Myoid gonadal stromal tumor . Bayesian analysis . Probabilistic modeling

Introduction Spindle cell tumor of the testis represents an uncommon entity, and its differential diagnosis is often troublesome due to

Supplementary Information The online version contains supplementary material available at https://doi.org/10.1007/s00428-02002957-8. * Maurizio Colecchia [email protected] 1

Department of Pathology, Humanitas Clinical and Research Center – IRCCS, via Manzoni 56, 20089 Rozzano, MI, Italy

2

Department of Biomedical Sciences, Humanitas University, Via Rita Levi Montalcini 4, 20090 Pieve Emanuele, MI, Italy

3

Department of Pathology, ASST Fatebenefratelli Sacco, via Grassi 74, 20157 Milano, MI, Italy

4

Department of Pathology, Fondazione IRCCS Istituto Nazionale dei Tumori, via Venezian 1, 20133 Milano, MI, Italy

5

Division of Uropathology and Intraoperative Diagnostic Division, European Institute of Oncology, IRCCS, via Ripamonti 435, 20141 Milan, MI, Italy

overlapping morphological and immunohistochemical features with gonadal stromal tumors and other mesenchymal low-grade spindle cell neoplasms. As an emerging distinct entity, the myoid gonadal stromal tumor was reported in the latest update of