Primary cutaneous SMARCB1-deficient carcinoma

J Cutan Pathol. 2021 Aug;48(8):1051-1060. doi: 10.1111/cup.13996. Epub 2021 Mar 14.

Abstract

Background: SMARCB1-deficient malignancies can arise in various sites. We describe a novel primary SMARCB1-deficient carcinoma of skin (SDCS) and characterize SMARCB1 mutations in non-melanoma skin cancers (NMSC).

Methods: Cases underwent immunophenotyping and targeted exome sequencing (MSK-IMPACT) assay interrogating somatic mutations in 468 cancer-related genes. The MSK-IMPACT database from 2014 to 2020 encompassing 55, 000 cases was searched for NMSC with SMARCB1 mutations.

Results: SDCS arose on the scalp of an 18-year-old woman showing homozygous SMARCB1 deletion with a LATS2 G963E variant. Another case arose on the temple of a 76-year-old man harboring a SMARCB1 W206* mutation associated with loss of heterozygosity (LOH), 59 concurrent mutations, and a UV mutation signature (UV-MS). Both tumors exhibited INI1 loss, positive CK5/6, p40, p63, and claudin-4 with negative CD34. Of 378 NMSC cases, including 370 carcinomas, 7 SMARCB1-mutated tumors were identified: 3 squamous cell, 3 Merkel cell, and one basal cell carcinoma. Six showed UV-MS. Five INI1-interrogated cases retained protein expression suggesting they were SMARCB1-proficient.

Conclusions: SDCS can be clinically aggressive, harbor SMARCB1 homozygous deletions or truncating SMARCB1 mutations associated with LOH, and can occur with or without UV-MS. Overall, SMARCB1 mutations in NMSC are rare with most being of undetermined significance and associated with retained INI1 and UV-MS.

Keywords: BAF47; INI1; SMARCB1; epithelioid sarcoma; squamous cell carcinoma.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Adolescent
  • Aged
  • Aged, 80 and over
  • Carcinoma, Squamous Cell / diagnosis*
  • Carcinoma, Squamous Cell / genetics*
  • Carcinoma, Squamous Cell / metabolism
  • Carcinoma, Squamous Cell / therapy
  • Exome Sequencing / methods
  • Fatal Outcome
  • Female
  • Homozygote
  • Humans
  • Immunohistochemistry / methods
  • Immunophenotyping / methods
  • Immunotherapy / methods
  • Loss of Heterozygosity / genetics
  • Male
  • Middle Aged
  • Mutation / genetics
  • Protein Serine-Threonine Kinases / genetics
  • Proton Therapy / methods
  • SMARCB1 Protein / deficiency*
  • Scalp / pathology
  • Skin Neoplasms / genetics
  • Skin Neoplasms / pathology*
  • Tumor Suppressor Proteins / genetics

Substances

  • SMARCB1 Protein
  • SMARCB1 protein, human
  • Tumor Suppressor Proteins
  • LATS2 protein, human
  • Protein Serine-Threonine Kinases