Melkersson-Rosenthal syndrome in the context of sarcoidosis: a case report

J Med Case Rep. 2021 Oct 4;15(1):488. doi: 10.1186/s13256-021-03044-5.

Abstract

Background: Melkersson-Rosenthal syndrome is a rare disease characterized by the triad of recurrent orofacial swelling with facial paralysis and fissured dorsal tongue. Histologically, noncaseating granulomatous inflammation occurs that confirms the diagnosis. Overlaps between granulomatous diseases such as sarcoidosis and Crohn's disease are described. Systemic corticosteroid therapy is the treatment of choice for acute attacks.

Case presentation: We here present a case of a 59-year-old White woman suffering from Melkersson-Rosenthal syndrome with a past history of sarcoidosis on therapy with leflunomide in combination with low-dose tacrolimus successfully treated with the anti-leprosy drug clofazimine after failure of systemic steroid therapy.

Conclusions: We propose clofazimine as an alternative treatment in steroid-refractory cases.

Keywords: Case report; Clofazimine; Melkersson–Rosenthal syndrome.

Publication types

  • Case Reports

MeSH terms

  • Behavior Therapy
  • Crohn Disease*
  • Facial Paralysis*
  • Female
  • Humans
  • Melkersson-Rosenthal Syndrome* / complications
  • Melkersson-Rosenthal Syndrome* / diagnosis
  • Melkersson-Rosenthal Syndrome* / drug therapy
  • Middle Aged
  • Sarcoidosis* / complications
  • Sarcoidosis* / diagnosis
  • Sarcoidosis* / drug therapy