BAP1 tumor predisposition syndrome case report: pathological and clinical aspects of BAP1-inactivated melanocytic tumors (BIMTs), including dermoscopy and confocal microscopy

BMC Cancer. 2019 Nov 9;19(1):1077. doi: 10.1186/s12885-019-6226-8.

Abstract

Background: BRCA1 associated-protein 1 (BAP1) tumor predisposition syndrome is associated with an increased risk for malignant mesotheliomas, uveal and cutaneous melanomas, renal cell carcinomas, and singular cutaneous lesions. The latter are referred to as BAP1-inactivated melanocytic tumors (BIMTs). When multiple BIMTs manifest, they are considered potential markers of germline BAP1 mutations.

Case presentation: Here, we report a novel pathogenic BAP1 germline variant in a family with a history of BIMTs, cutaneous melanomas, and mesotheliomas. We also describe singular pathological aspects of the patient's BIMT lesions and their correlation with dermoscopic and reflectance confocal microscopy findings.

Conclusions: This knowledge is crucial for the recognition of BIMTs by dermatologists and pathologists, allowing the determination of appropriate management for high-risk patients, such as genetic investigations and screening for potentially aggressive tumors.

Keywords: BAP1; BIMT; Confocal microscopy; Dermoscopy; Hereditary cancer syndromes.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Aged
  • Aged, 80 and over
  • Dermoscopy / methods
  • Female
  • Genetic Predisposition to Disease
  • Germ-Line Mutation
  • Humans
  • Male
  • Melanoma / genetics*
  • Melanoma / pathology*
  • Melanoma, Cutaneous Malignant
  • Microscopy, Confocal / methods
  • Middle Aged
  • Neoplastic Syndromes, Hereditary / genetics*
  • Pedigree
  • Prognosis
  • Skin Neoplasms / genetics*
  • Skin Neoplasms / pathology*
  • Tumor Suppressor Proteins / genetics*
  • Ubiquitin Thiolesterase / genetics*

Substances

  • BAP1 protein, human
  • Tumor Suppressor Proteins
  • Ubiquitin Thiolesterase