[Barraquer and Simons lipodystrophy. Complement anomalies and cutaneous leukocytoclasic vasculitis]

Ann Dermatol Venereol. 1987;114(9):1083-91.
[Article in French]

Abstract

In a 56-year old woman progressive partial lipodystrophy began at the age of 6 years on the face, thereafter extending slowly down to mid-thigh level (fig. 1 and 2), with moderate hypertrophy of the subjacent fatty tissue and a fatty macroglossia (fig. 3). Histological examination of the lipodystrophic skin not only showed an absence of fatty tissue, but also abnormalities at the dermis-epidermis junction with hyaline bodies (fig. 4). At the age of 23 she developed purpura, predominantly on the legs, which rapidly became chronic (fig. 5); histological examination showed leucocytoclasic vasculitis of dermal vessels (fig. 6) with granular deposits of C3 on the vessels and of IgM at the dermis-epidermis junction. Episodes of polyarthralgia and headaches were frequent. Regressive neuritis of the external popliteal nerve occurred when she was 53-year old. Renal function tests proved normal, but renal biopsy was not performed. There was no diabetes mellitus, but an oral glucose tolerance test and a somatostatin insulin glucose test elicited definite resistance to insulin. A search for a serum factor inhibiting insulin receptors was negative. Permanent abnormalities in serum were a very deep fall in C3, a pronounced fall in CH50 and a low C4 level. Besides, a C3 nephritic factor (NeF) at a high level and circulating immune complexes were present (table I); a mixed IgM-IgG cryoglobulin was found intermittently (fig. 7). Clearance of the immune complexes by splenic macrophages was extremely slow. During a series of plasma exchanges, serum C3 increased transiently, whereas serum C4 remained unchanged (fig. 8).(ABSTRACT TRUNCATED AT 250 WORDS)

Publication types

  • Case Reports
  • English Abstract

MeSH terms

  • Antigen-Antibody Complex / metabolism
  • Biopsy
  • Complement C3 / deficiency*
  • Complement C3 Nephritic Factor / blood*
  • Complement C4 / deficiency*
  • Complement C4 / genetics
  • Complement Inactivator Proteins / blood*
  • Female
  • Humans
  • Hyperinsulinism / complications
  • Lipodystrophy / complications*
  • Lipodystrophy / immunology
  • Lipodystrophy / pathology
  • Middle Aged
  • Purpura / complications*
  • Purpura / immunology
  • Purpura / pathology
  • Vasculitis / complications*
  • Vasculitis / immunology
  • Vasculitis / pathology

Substances

  • Antigen-Antibody Complex
  • Complement C3
  • Complement C3 Nephritic Factor
  • Complement C4
  • Complement Inactivator Proteins