Validation of pathological grading systems for predicting metastatic potential in pheochromocytoma and paraganglioma

PLoS One. 2017 Nov 8;12(11):e0187398. doi: 10.1371/journal.pone.0187398. eCollection 2017.

Abstract

Purpose: The Grading system for Adrenal Pheochromocytoma and Paraganglioma (GAPP) was proposed for predicting the metastatic potential of pheochromocytoma and paraganglioma to overcome the limitations of the Pheochromocytoma of the Adrenal Scaled Score (PASS). However, to date, no study validating the GAPP has been conducted, and previous studies did not include mutations in the succinate dehydrogenase type B (SDHB) gene in the score calculation. In this retrospective cohort study, we validated the prediction ability of GAPP and assessed whether it would be improved by inclusion of the loss of SDHB immunohistochemical staining.

Methods: We divided the tumors into non-metastatic and metastatic groups based on the presence of synchronous or metachronous metastases. The GAPP score and PASS at the initial operation were measured. Moreover, we combined some GAPP parameters with the immunohistochemical staining of SDHB to obtain a modified GAPP (M-GAPP) score.

Results: Metastasis occurred in 15/72 (20.8%) patients, with a mean follow-up of 43.5 months. Loss of SDHB staining was more frequent (P = 0.044) in the metastatic group. The GAPP score (P = 0.006), PASS (P = 0.003), and M-GAPP score (P<0.001) were all higher in the metastatic group. Twelve of 40 (30.0%) moderately or poorly differentiated tumors, as defined by the GAPP score, and 12/34 (35.3%) tumors with a PASS ≥4 were metastatic. Conversely, 10/19 (52.6%) tumors with an M-GAPP score ≥3 were metastatic. The area under the curve of the M-GAPP score (0.822) was significantly higher than that of the GAPP (0.728) (P = 0.012), but similar to that of the PASS (0.753) (P = 0.411). The GAPP (P = 0.032) and M-GAPP scores (P = 0.040), but not PASS (P = 0.200), negatively correlated with metastasis-free survival.

Conclusion: The GAPP was validated, and M-GAPP, a combination of some GAPP parameters and loss of SDHB staining, might be useful for the prediction of the metastatic potential of pheochromocytoma and paraganglioma.

Publication types

  • Validation Study

MeSH terms

  • Adrenal Gland Neoplasms / pathology*
  • Adrenal Gland Neoplasms / surgery
  • Adult
  • Female
  • Humans
  • Immunohistochemistry
  • Male
  • Middle Aged
  • Neoplasm Metastasis
  • Paraganglioma / pathology*
  • Paraganglioma / surgery
  • Pheochromocytoma / pathology*
  • Pheochromocytoma / surgery
  • ROC Curve
  • Reproducibility of Results
  • Succinate Dehydrogenase / genetics
  • Survival Analysis

Substances

  • SDHB protein, human
  • Succinate Dehydrogenase

Grants and funding

This study was supported by grants from the Asan Institute for Life Sciences, Seoul, Republic of Korea (project no. 2016-0840). The funder had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.