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Skeletal muscular changes in Pena-Shokeir sequence

P Reiser1, J Briner, A Schinzel

  • 1Institute of Pathology, University of Zurich, Switzerland.

Insights

Pena-Shokeir sequence in fetuses and newborns showed minor muscle changes, not specific myopathies. Fetal hypokinesia from skeletal muscle issues may cause pulmonary hypoplasia and limb deformities.

Area of Science:

  • Medical science
  • Pathology
  • Developmental biology

Background:

  • Pena-Shokeir sequence is a congenital disorder characterized by joint contractures, facial anomalies, and pulmonary hypoplasia.
  • The underlying cause of Pena-Shokeir sequence, particularly the role of skeletal muscle, remains unclear.

Observation:

  • Histologic examination of skeletal muscles from 8 fetuses and newborns with Pena-Shokeir sequence was performed.
  • Minor, nonspecific changes were noted, insufficient to diagnose specific myopathies.
  • Increased muscle fiber diameters were observed in 2 of 5 examined patients.

Findings:

  • Skeletal muscle lesions in Pena-Shokeir sequence are subtle and not indicative of known myopathies.
  • No significant internal organ malformations were detected via autopsy.
  • Increased muscle fiber diameter suggests potential muscular involvement.

Implications:

  • Fetal hypokinesia, stemming from skeletal muscle abnormalities, is proposed as the cause of pulmonary hypoplasia.
  • This fetal hypokinesia may also contribute to the characteristic facial and extremity deformities seen in Pena-Shokeir sequence.
  • Further research into fetal movement disorders and skeletal muscle development is warranted.

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