Primary signet ring stromal tumor of the testis: a study of 13 cases indicating their phenotypic and genotypic analogy to pancreatic solid pseudopapillary neoplasm

Hum Pathol. 2017 Sep:67:85-93. doi: 10.1016/j.humpath.2017.07.010. Epub 2017 Jul 21.

Abstract

Primary signet ring stromal tumor of the testis (PSRSTT) is an extremely rare tumor described only twice in the literature. Pancreatic-analogue solid pseudopapillary neoplasm (SPN) of the testis is a recently reported entity with morphological overlap with PSRSTT. We reviewed our files to find all cases of PSRSTT to better characterize this entity. We studied 13 cases of PSRSTTs using histological, immunohistochemical (IHC), and molecular genetic methods and compared the results with pancreatic SPN. Grossly, the size of PSRSTTs ranged from 0.5 to 2 cm (mean 1.1). Microscopically, PSRSTTs predominantly showed a proliferation of low-grade epithelioid cells containing characteristic cytoplasmic vacuole dislodging the nucleus (signet ring cells) separated by fibrous septa into trabeculae and nests. The immunoprofile was characterized by immunoreactivity for β-catenin, cyclin D1 (nuclear positivity for both antibodies), CD10, vimentin, galectin-3, claudin 7, α-1-antitrypsin, CD56, and neuron-specific enolase and negativity for chromogranin, inhibin, calretinin, SF-1, NANOG, OCT3/4, and SALL4. In some cases, the IHC panel was restricted because of a limited amount of tissue. Molecular genetic analysis revealed mutations within exon 3 of the CTNNB1 encoding β-catenin in all analyzable cases. Based on histological similarities between pancreatic SPN and PSRSTT and their identical IHC and molecular genetic features, we assume that both neoplasms share the same pathogenesis, and thus, PSRSTT can be considered as a testicular analogue of pancreatic SPN.

Keywords: Analogue; Pancreas; Primary signet ring stromal tumor; Solid pseudopapillary neoplasm; Testis.

Publication types

  • Comparative Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Biomarkers, Tumor / analysis
  • Biomarkers, Tumor / genetics
  • Biopsy
  • Carcinoma, Signet Ring Cell* / chemistry
  • Carcinoma, Signet Ring Cell* / genetics
  • Carcinoma, Signet Ring Cell* / immunology
  • Carcinoma, Signet Ring Cell* / pathology
  • Cell Differentiation
  • Cell Lineage
  • Cell Proliferation
  • DNA Mutational Analysis
  • Genetic Predisposition to Disease
  • Humans
  • Immunohistochemistry
  • Male
  • Middle Aged
  • Mutation
  • Neoplasm Grading
  • Pancreatic Neoplasms* / chemistry
  • Pancreatic Neoplasms* / genetics
  • Pancreatic Neoplasms* / immunology
  • Pancreatic Neoplasms* / pathology
  • Phenotype
  • Sex Cord-Gonadal Stromal Tumors* / chemistry
  • Sex Cord-Gonadal Stromal Tumors* / genetics
  • Sex Cord-Gonadal Stromal Tumors* / immunology
  • Sex Cord-Gonadal Stromal Tumors* / pathology
  • Testicular Neoplasms* / chemistry
  • Testicular Neoplasms* / genetics
  • Testicular Neoplasms* / immunology
  • Testicular Neoplasms* / pathology
  • Young Adult
  • beta Catenin / analysis
  • beta Catenin / genetics

Substances

  • Biomarkers, Tumor
  • CTNNB1 protein, human
  • beta Catenin