Description of longitudinal tumor evolution in a case of multiply relapsed clear cell sarcoma of the kidney

Cancer Rep (Hoboken). 2022 Feb;5(2):e1458. doi: 10.1002/cnr2.1458. Epub 2021 Dec 29.

Abstract

Background: Clear cell sarcoma of the kidney (CCSK) is the second most common pediatric renal tumor.

Case: A 2-year-old boy was diagnosed with CCSK, which relapsed four times until he yielded to the disease at the age of 7 years. To characterize the longitudinal genetic alterations occurring in the present case, we performed targeted-capture sequencing by pediatric solid tumors panel (381 genes) for longitudinally sampled tumors, including autopsy samples of metastasis. Internal tandem duplication of BCOR (BCOR-ITD) was the only truncal mutation, confirming the previously reported role of BCOR-ITD in CCSK.

Conclusion: Acquisition of additional mutations along tumor relapses and detection of metastasis-specific mutations were reminiscent of the tumor progression and therapeutic resistance of this case, leading to clonal selection and a dismal fate.

Keywords: BCOR-ITD; clear cell sarcoma of the kidney; clonal evolution; relapse.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Child
  • Fatal Outcome
  • Humans
  • Kidney Neoplasms / genetics
  • Kidney Neoplasms / pathology*
  • Male
  • Neoplasm Metastasis
  • Neoplasm Recurrence, Local / genetics
  • Neoplasm Recurrence, Local / pathology*
  • Proto-Oncogene Proteins
  • Repressor Proteins
  • Sarcoma, Clear Cell / genetics
  • Sarcoma, Clear Cell / pathology*

Substances

  • BCOR protein, human
  • Proto-Oncogene Proteins
  • Repressor Proteins