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Cranial reconstruction in osteogenesis imperfecta

B L Eppley1, J E Kalsbeck, A M Sadove

  • 1Division of Plastic Surgery, James Whitcomb Riley Hospital for Children, Indiana University Medical Center, Indianapolis 46202.

The Journal of Craniofacial Surgery
|July 1, 1994
PubMed
Summary
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Osteogenesis imperfecta (OI) can cause craniosynostosis. This study reports successful surgical and non-surgical management of craniosynostosis in an infant with type 3 OI, adapting techniques for fragile bones.

Area of Science:

  • Pediatric Neurosurgery
  • Craniofacial Surgery
  • Medical Genetics

Background:

  • Osteogenesis imperfecta (OI) is a genetic disorder affecting collagen, leading to bone fragility.
  • Craniosynostosis, the premature fusion of skull sutures, can occur in OI patients.
  • Limited therapeutic strategies exist for craniosynostosis associated with OI.

Observation:

  • An 8-month-old female infant diagnosed with Osteogenesis Imperfecta type 3 presented with craniosynostosis.
  • The infant exhibited specific skull deformities: bilateral occipital flattening, biparietal widening, and frontal narrowing.
  • Conventional surgical techniques for craniosynostosis correction were considered unsuitable due to the poor bone quality in OI.

Findings:

  • A combined approach involving surgical release and reconstruction of the skull was performed.

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  • Post-operative management included the use of a molding cap therapy.
  • This combined therapeutic strategy effectively corrected the observed craniosynostosis deformities in the infant.

    • Implications:

    • This case demonstrates the feasibility of managing craniosynostosis in patients with Osteogenesis Imperfecta.
    • Modified surgical and non-surgical techniques are crucial for addressing the unique challenges posed by OI bone quality.
    • Further research into tailored interventions for OI-related craniosynostosis is warranted.