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December 16, 2020

Association of Programmed Cell Death 1 Inhibitor With Circumorificial Plasmacytosis

Author Affiliations
  • 1Division of Dermatology, Department of Internal Medicine, The Ohio State University, Columbus, Ohio
  • 2Central Ohio Skin and Cancer, Inc, Columbus, Ohio
  • 3Division of Hematology, Department of Internal Medicine, The Ohio State University Comprehensive Cancer Center, Columbus, Ohio
  • 4Division of Oral and Maxillofacial Pathology and Radiology, College of Dentistry, The Ohio State University, Columbus, Ohio
JAMA Dermatol. 2021;157(2):237-238. doi:10.1001/jamadermatol.2020.4273

There are myriad mucocutaneous manifestations of programmed cell death 1 (PD-1) inhibitors. We report a novel manifestation of PD-1–associated circumorificial plasmacytosis (CP).

An elderly man with undifferentiated pleomorphic sarcoma enrolled in a clinical trial of neoadjuvant treatment with pembrolizumab and radiation therapy, followed by surgical resection. Six months after starting to take pembrolizumab, he developed gum pain and bleeding. On examination, the maxillary and mandibular buccal gingivae appeared diffusely enlarged, erythematous, and cobblestoned (Figure 1A). In the right maxillary quadrant, the gingival erythema and enlargement involved the buccal and labial mucosa. Crateriform and fissured ulcerations were identified in the right maxillary buccal vestibule that extended onto the right superior aspect of the buccal mucosa. The lesional tissue had a multinodular appearance and was rubbery firm (Figure 1B). Biopsy results showed sheets of uniform CD138+ plasmacytoid cells with rare acute and chronic inflammatory cells (Figure 2, A and B). Because of the predominance of plasma cells, immunohistochemical studies using antibodies directed against κ and λ light chains were performed and confirmed a polyclonal plasma cell infiltrate, ruling out extramedullary plasmacytoma. The histopathologic and immunopathologic findings, with the clinical presentation, were consistent with CP.

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