Refractory pretibial myxoedema with response to intralesional insulin-like growth factor 1 antagonist (octreotide): downregulation of hyaluronic acid production by the lesional fibroblasts

Br J Dermatol. 2000 Nov;143(5):1083-6. doi: 10.1046/j.1365-2133.2000.03850.x.

Abstract

We report a case of refractory pretibial myxoedema (PTM) with Graves' disease in which there was a good clinical response to intralesional injection of the insulin-like growth factor 1 (IGF-1) antagonist octreotide. Intralesional octreotide (200 microg once daily) dramatically improved the tumorous lesions of PTM after 4 weeks, and the lesions remained stable even after reducing the dose to 200 microg once weekly. The amount of hyaluronic acid (HA) in the lesional skin decreased to 5.8 microg mg-1 dry weight from 16.3 microg mg-1 dry weight after 4 weeks of octreotide treatment. IGF-1 showed a dose-dependent stimulatory effect on HA secretion by both normal and patient's fibroblasts at higher concentrations in vitro. Octreotide significantly suppressed IGF-1 induced-HA secretion by the patient's fibroblasts, but not by normal fibroblasts, which suggests that expression of IGF-1 receptor on fibroblasts, or its affinity for IGF-1, are upregulated in PTM, resulting in the oversecretion of HA. These results might suggest that octreotide improves PTM through downregulation of HA production by lesional fibroblasts.

Publication types

  • Case Reports

MeSH terms

  • Down-Regulation / drug effects
  • Fibroblasts / metabolism
  • Hormones / therapeutic use*
  • Humans
  • Hyaluronic Acid / biosynthesis
  • Insulin-Like Growth Factor I / antagonists & inhibitors*
  • Leg Dermatoses / drug therapy*
  • Male
  • Middle Aged
  • Myxedema / drug therapy*
  • Octreotide / therapeutic use*

Substances

  • Hormones
  • Insulin-Like Growth Factor I
  • Hyaluronic Acid
  • Octreotide