Skip to main content

Advertisement

SpringerLink
Log in

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

  • Original Article
  • Published:
Virchows Archiv Aims and scope Submit manuscript

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.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3
Fig. 4

Similar content being viewed by others

Data availability

All the data are available in the public repository https://github.com/slrenne/Myoid_Tumor_Testis.

References

  1. International Agency for Research on Cancer, Moch H, Reuter VE (2016) WHO classification of tumours of the urinary system and male genital organs. IARC WHO Classification of Tumours. International Agency for Research on Cancer. https://books.google.it/books?id=qxQyjgEACAAJ

  2. Weidner N (1991) Myoid gonadal stromal tumor with epithelial differentiation (? testicular myoepithelioma). Ultrastruct Pathol 15:403–408

    Article  Google Scholar 

  3. Evans HL (1977) Unusual gonadal stromal tumor of the testis. Case report with ultrastructural observations. Arch Pathol Lab Med 101:317–320

    CAS  PubMed  Google Scholar 

  4. Greco MA, Feiner HD, Theil KS, Mufarrij AA (1984) Testicular stromal tumor with myofilaments: ultrastructural comparison with normal gonadal stroma. Hum Pathol 15:238–243

    Article  CAS  Google Scholar 

  5. Du S, Powell J, Hii A, Weidner N (2012) Myoid gonadal stromal tumor: a distinct testicular tumor with peritubular myoid cell differentiation. Hum Pathol 43:144–149

    Article  CAS  Google Scholar 

  6. Renshaw AA, Gordon M, Corless CL (1997) Immunohistochemistry of unclassified sex cord-stromal tumors of the testis with a predominance of spindle cells. Mod Pathol 10:693–700

    CAS  PubMed  Google Scholar 

  7. Kao CS, Ulbright TM (2014) Myoid gonadal stromal tumor: a clinicopathologic study of three cases of a distinctive testicular tumor. Am J Clin Pathol 142:675–682

    Article  Google Scholar 

  8. Nistal M, Puras A, Perna C, Guarch R, Paniagua R (1996) Fusocellular gonadal stromal tumour of the testis with epithelial and myoid differentiation. Histopathology 29:259–264

    Article  CAS  Google Scholar 

  9. Allen PR, King AR, Sage MD, Sorrell VF (1990) A benign gonadal stromal tumor of the testis of spindle fibroblastic type. Pathology 22:227–229

    Article  CAS  Google Scholar 

  10. Miettinen M, Salo J, Virtanen I (1986) Testicular stromal tumor: ultrastructural, immunohistochemical, and gel electrophoretic evidence of epithelial differentiation. Ultrastruct Pathol 10:515–528

    Article  CAS  Google Scholar 

  11. McElreath R (2020) Statistical rethinking: a Bayesian course with examples in R and Stan. Chapman & Hall/CRC Texts in Statistical Science. CRC Press

  12. Colecchia M et al (2017) Myoid gonadal stromal tumor of the testis. An immunohistochemical and ultrastructural study of three cases. Mod Pathol 30:539–568

  13. Carpenter B, Gelman A, Hoffman MD, Lee D, Goodrich B, Betancourt M, Brubaker M, Guo J, Li P, Riddell A (2017) Stan: A probabilistic programming language. J Stat Softw 76(1). https://doi.org/10.18637/jss.v076.i01

  14. R Core Team (2019) R: a language and environment for statistical computing

  15. Duane S, Kennedy AD, Pendleton BJ, Roweth D (1987) Hybrid Monte Carlo. Phys Lett B 195:216–222

    Article  CAS  Google Scholar 

  16. Metropolis N, Rosenbluth AW, Rosenbluth MN, Teller AH, Teller E (1953) Equation of state calculations by fast computing machines. J Chem Phys 21:1087–1092

    Article  CAS  Google Scholar 

  17. Hoffman MD, Gelman A (2014) The no-U-turn sampler: adaptively setting path lengths in Hamiltonian Monte Carlo. J Mach Learn Res 15:1593–1623

    Google Scholar 

  18. Gelman A, Carlin JB, Stern HS, Dunson DB, Vehtari A, Rubin DB (2013) Bayesian data analysis, 3rd edition. Chapman \& Hall/CRC Texts in Statistical Science. CRC Press. https://books.google.it/books?id=ZXL6AQAAQBAJ

  19. Gelman A (2005) Discussion paper analysis of variance - why it is more important than ever. Ann Stat 33:1–53

    Article  Google Scholar 

  20. Papaspiliopoulos O, Roberts GO, Sköld M (2007) A general framework for the parametrization of hierarchical models. Stat Sci 22:59–73

    Article  Google Scholar 

  21. Wenger SJ, Freeman MC (2008) Estimating species occurrence, abundance, and detection probability using zero-inflated distributions. Ecology 89:2953–2959

    Article  Google Scholar 

  22. Box GEP (1976) Science and statistics. J Am Stat Assoc 71:791–799

    Article  Google Scholar 

  23. Sawcer S, Wason J (2012) Risk in complex genetics: “all models are wrong but some are useful”. Ann Neurol 72:502–509

    Article  Google Scholar 

  24. Stout A, Lattes R (1967) Tumors of the soft tissues. Armed Forces Institute of Pathology. https://doi.org/10.17226/18647

  25. Joensuu H, Vehtari A, Riihimäki J, Nishida T, Steigen SE, Brabec P, Plank L, Nilsson B, Cirilli C, Braconi C, Bordoni A, Magnusson MK, Linke Z, Sufliarsky J, Federico M, Jonasson JG, Dei Tos AP, Rutkowski P (2012) Risk of recurrence of gastrointestinal stromal tumour after surgery: An analysis of pooled population-based cohorts. Lancet Oncol 13:265–274

    Article  Google Scholar 

  26. Maclehose RF, Hamra GB (2014) Applications of Bayesian methods to epidemiologic research. https://doi.org/10.1007/s40471-014-0019-z

  27. Held L (2014) Handbook of epidemiology. Springer New York. https://doi.org/10.1007/978-0-387-09834-0

  28. Gelman A, Weakliem D (2009) Of Beauty Sex and Power: Too little attention has been paid to the statistical challenges in estimating small effects. Am Sci 4:310–316

    Article  Google Scholar 

Download references

Author information

Authors and Affiliations

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

    Salvatore Lorenzo Renne & Marina Valeri

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

    Salvatore Lorenzo Renne & Marina Valeri

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

    Antonella Tosoni & Roberta Rossi

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

    Alessia Bertolotti & Maurizio Colecchia

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

    Giuseppe Renne

Authors
  1. Salvatore Lorenzo Renne
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Marina Valeri
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Antonella Tosoni
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Alessia Bertolotti
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Roberta Rossi
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Giuseppe Renne
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Maurizio Colecchia
    View author publications

    You can also search for this author in PubMed Google Scholar

Contributions

SLR conceived the study; SLR, MV, AB, AT, RR, GR, and MC collected the data; SLR analyzed the data; SLR and MC interpreted the data; SLR and MV searched the literature; SLR generated the figures; SLR and MV wrote the manuscript; AB, AT, RR, GR, and MC corrected and reviewed the manuscript. All authors approved the final version of the manuscript.

Corresponding author

Correspondence to Maurizio Colecchia.

Ethics declarations

Conflicts of interest

The authors declare that they have no conflict of interest.

Ethics approval

Waived.

Consent to participate

Not applicable.

Consent for publication

Not applicable.

Code availability

All the custom code used for this paper is available in the public repository https://github.com/slrenne/Myoid_Tumor_Testis.

Additional information

Publisher’s note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Supplementary Information

ESM 1

(PDF 992 kb)

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Renne, S.L., Valeri, M., Tosoni, A. et al. Myoid gonadal tumor. Case series, systematic review, and Bayesian analysis. Virchows Arch 478, 727–734 (2021). https://doi.org/10.1007/s00428-020-02957-8

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00428-020-02957-8

Keywords

Search

Navigation