Dysferlinopathy: spectrum of pathological changes in skeletal muscle tissue

Indian J Pathol Microbiol. 2011 Apr-Jun;54(2):350-4. doi: 10.4103/0377-4929.81636.

Abstract

Background: Dysferlinopathy is an autosomal recessive-limb girdle muscular dystrophy (AR-LGMD) caused due to the defect in gene encoding dysferlin, a sarcolemmal protein. Awareness of the variants and their relative frequency is essential for accurate diagnosis.

Aim: To study the spectrum of morphologic changes in immunohistochemically proven cases of dysferlinopathies, to correlate the findings with clinical phenotype and durations of illness and determine the frequency.

Materials and methods: Dysferlinopathies seen over a period of 2 years at a tertiary neurological center were analyzed.

Results: Clinically, majority had Miyoshi phenotype (46.6%) with distal involvement and LGMD phenotype (40%) with proximal muscle involvement. In addition, a proximo-distal and tibial muscle phenotype was encountered. Morphologically, rimmed vacuoles were noted in the Miyoshi phenotype. The presence of ragged red fibers, lobulated fibers and inflammation had no preference to a particular phenotype. Significant atrophy and lobulated fibers were noted in patients with longer duration of illness.

Conclusions: Dysferlinopathy was the second most common identifiable cause (21%) of LGMD next to sarcoglycanopathies (27%).

MeSH terms

  • Adolescent
  • Adult
  • Dysferlin
  • Female
  • Humans
  • Immunohistochemistry
  • Male
  • Membrane Proteins / analysis
  • Microscopy
  • Middle Aged
  • Muscle Cells / ultrastructure
  • Muscle Fibers, Slow-Twitch / ultrastructure
  • Muscle Proteins / analysis
  • Muscle, Skeletal / pathology*
  • Muscular Dystrophies, Limb-Girdle / pathology
  • Vacuoles / ultrastructure
  • Young Adult

Substances

  • DYSF protein, human
  • Dysferlin
  • Membrane Proteins
  • Muscle Proteins

Supplementary concepts

  • Dysferlinopathy