Successful treatment of trichilemmal carcinoma with distant metastasis using pembrolizumab: a case report and review

Immunotherapy. 2024;16(10):659-667. doi: 10.1080/1750743X.2024.2353535. Epub 2024 May 23.

Abstract

Trichilemmal carcinoma (TC) is a rare, malignant cutaneous adnexal tumor. TC often has nonspecific clinical manifestations and its aggressive nature is frequently overlooked. Metastasis of TC is rarely reported and there is no standard treatment for recurrent or metastatic TC. We report a complicated case of TC arising from the parotid gland with metastasis to cervical lymph nodes. The tumor progressed after multiple surgeries, radiation and chemotherapy. Finally, the patient achieved good response and disease control with pembrolizumab, an immune checkpoint inhibitor targeting programmed cell death protein-1. Currently, the patient has received 19 cycles of pembrolizumab and the disease remains well controlled. This represents the first reported use of immune checkpoint blockade to treat TC.

Keywords: PD-1/PD-L1 checkpoint blockade; cutaneous oncology; pembrolizumab; trichilemmal carcinoma.

Plain language summary

This paper discusses a rare form of skin cancer called trichilemmal carcinoma (TC) and presents a distant metastasis TC case. The patient was treated with an immunotherapy called pembrolizumab and after 19 courses of treatment, the tumor was significantly reduced and the symptoms were relieved. This case report is the first recorded case study of pembrolizumab for the treatment of TC and provides a new approach to the treatment of challenging malignancies.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Antibodies, Monoclonal, Humanized* / therapeutic use
  • Antineoplastic Agents, Immunological / therapeutic use
  • Female
  • Humans
  • Immune Checkpoint Inhibitors / therapeutic use
  • Lymphatic Metastasis
  • Male
  • Middle Aged
  • Neoplasm Metastasis
  • Parotid Neoplasms / drug therapy
  • Parotid Neoplasms / pathology
  • Skin Neoplasms* / drug therapy
  • Skin Neoplasms* / pathology

Substances

  • Antibodies, Monoclonal, Humanized
  • pembrolizumab
  • Immune Checkpoint Inhibitors
  • Antineoplastic Agents, Immunological