Spinal Muscular Atrophy Report summary

Spinal Muscular Atrophy Report summary

Author or authors of report : W. David Arnold, Darine Kassar, John T. Kissel,
Date of report : 2014-12-16
  • Introduction to SMA:
    • SMA is a group of disorders associated with spinal motor neuron loss.
    • The most common form of SMA is proximal or 5q SMA.
    • Historically, diagnosis was based on electromyography and muscle biopsy features of denervation. Now, molecular testing for homozygous deletion or mutation of the SMN1 gene is used for diagnosis.
  • Cause and Pathogenesis:
    • Humans have two nearly identical SMN genes on chromosome 5q13.
    • Homozygous deletion of the SMN1 gene was identified as the cause of SMA in 1995.
    • SMN1 and SMN2 genes differ by only a few base pairs. A single nucleotide difference in SMN2 affects splicing, leading to ~90% of its transcripts lacking exon7.
    • As a result, SMN2 produces a shortened, unstable protein, whereas SMN1 produces the full-length SMN protein.
    • The combined effects of homozygous loss of SMN1 and retention of SMN2 result in low levels of the full-length SMN protein.
    • Full-length SMN protein is essential for various cellular functions, including RNA metabolism.
    • While SMN plays a key role in RNA splicing in all cells, its exact role in motor neurons is still under investigation.
  • Variability of Clinical Features and Severity:
    • The main clinical features of SMA are muscle weakness and atrophy due to motor neuron dysfunction and loss.
    • The severity spectrum ranges from mild proximal limb weakness in adulthood to severe generalized weakness with respiratory failure in neonates.
    • The onset and progression of weakness in SMA are distinct from many other motor neuron disorders. There's usually a presymptomatic period, followed by rapid progression and then a relatively static phase.
    • SMA has been traditionally classified into types 1–3, but some experts suggest more subtypes. The type and severity correlate with the SMN2 copy number.
  • Clinical Features of Type 1 SMA:
    • Type 1 SMA is the most common and severe form, representing 45% of cases.
    • Infants may appear normal before developing symptoms like limb weakness, respiratory distress, weak cry, and poor feeding.
    • Affected babies often have a "frog-leg" lower limb posture due to severe hypotonic weakness.
    • Paradoxical breathing is a characteristic feature.
    • Tongue fasciculations are common, but cognition is spared.
    • By definition, these infants never achieve the ability to sit independently. Most cases have a natural history of death before age 2, but aggressive supportive care can improve survival.