Lymphedema secondary to limited cutaneous systemic sclerosis

BMJ Case Rep. 2018 Jun 15:2018:bcr2017224148. doi: 10.1136/bcr-2017-224148.

Abstract

Systemic sclerosis (SSc) is an autoimmune connective tissue disease characterised by vascular abnormalities, immune system activation and fibrosis. Lymphatic involvement in SSc was described more recently and starts in early stages. This report describes a 46-year-old patient who developed over the last 2 years asymmetric lymphedema in lower extremities. Compromise in lymphatic drainage was confirmed by lymphoscintigraphy. She also presented Raynaud's phenomenon, a scleroderma pattern in nailfold capillaroscopy, cutaneous thickening and anticentromere antibodies, which together resulted in a new diagnosis of limited cutaneous SSc. Treatment with methotrexate, prednisolone and lymphatic drainage resulted in lymphedema improvement. To our knowledge, this is the first case of grade 2 lymphedema in the setting of anticentromere-positive limited cutaneous SSc. We highlight the importance of considering rheumatic diseases in the differential diagnosis of lymphedema.

Keywords: connective tissue disease; dermatology.

Publication types

  • Case Reports

MeSH terms

  • Female
  • Humans
  • Lymphedema / diagnostic imaging*
  • Lymphedema / drug therapy
  • Lymphedema / etiology
  • Lymphoscintigraphy
  • Methotrexate / therapeutic use*
  • Microscopic Angioscopy
  • Middle Aged
  • Prednisolone / therapeutic use*
  • Raynaud Disease / diagnostic imaging
  • Raynaud Disease / drug therapy
  • Raynaud Disease / etiology
  • Scleroderma, Limited / diagnosis*
  • Scleroderma, Limited / drug therapy
  • Treatment Outcome

Substances

  • Prednisolone
  • Methotrexate