Clinical and Electrophysiological Findings in Hereditary Inclusion Body Myopathy Compared With Sporadic Inclusion Body Myositis

J Clin Neuromuscul Dis. 2016 Jun;17(4):190-6. doi: 10.1097/CND.0000000000000113.

Abstract

Objective: To compare the clinical and electrophysiological findings in hereditary inclusion body myopathy (hIBM) and sporadic inclusion body myositis (sIBM) patients.

Methods: We retrospectively identified 8 genetically proven hIBM patients and 1 DNAJB6 myopathy with pathological features of hIBM, and compared their clinical, electromyographic, and serological data with a group of 51 pathologically proven sIBM patients.

Results: hIBM patients had a younger mean age of onset (36 vs. 60 years, P = 0.0001). Diagnostic delay was shorter in sIBM (6 vs. 15 years, P = 0.0003). Wrist flexors (P = 0.02), digit flexors (P = 0.01), digit extensors (P = 0.02), and quadriceps (P = 0.008) muscles were more frequently affected in sIBM. Fibrillation potentials were more common in sIBM patients (P = 0.03). Electrical myotonia was found in 4 hIBM patients, not significantly different from sIBM patients (P = 0.45). Creatinine kinase was higher in sIBM patients (799 vs 232, P = 0.03).

Conclusions: sIBM and hIBM seem to have similar electromyographic changes. The combination of clinical, serological, and histopathological findings can guide genetic testing to the final diagnosis.

Publication types

  • Comparative Study

MeSH terms

  • Adult
  • Age of Onset
  • Aged
  • Delayed Diagnosis
  • Distal Myopathies / diagnosis*
  • Distal Myopathies / pathology
  • Distal Myopathies / physiopathology
  • Electrodiagnosis
  • Electromyography
  • Female
  • Humans
  • Male
  • Middle Aged
  • Muscle, Skeletal / pathology
  • Muscle, Skeletal / physiopathology*
  • Myositis, Inclusion Body / diagnosis*
  • Myositis, Inclusion Body / pathology
  • Myositis, Inclusion Body / physiopathology
  • Myotonia / pathology
  • Myotonia / physiopathology
  • Retrospective Studies
  • Symptom Assessment

Supplementary concepts

  • Distal myopathy, Nonaka type