A Case of Spontaneous Intestinal Perforation in Osteogenesis Imperfecta
2010

Spontaneous Intestinal Perforation in Osteogenesis Imperfecta

publication Evidence: low

Author Information

Author(s): Katherine Wheatley, Ee Ling Cheng, Mary Sheppard, Hank Schneider, Neil Moat, Jeremy Cordingley, Sundeep Kaul

Primary Institution: Royal Brompton Hospital, London, UK

Hypothesis

Can osteogenesis imperfecta lead to spontaneous non-ischemic bowel perforation?

Conclusion

This case highlights the occurrence of non-ischemic bowel perforation in a patient with osteogenesis imperfecta.

Supporting Evidence

  • The patient had a history of multiple childhood fractures due to osteogenesis imperfecta.
  • Histology showed absent musculature with no evidence of ischemia.
  • This case is the first report of spontaneous non-ischemic bowel perforation in osteogenesis imperfecta.

Takeaway

A patient with a bone condition called osteogenesis imperfecta had a hole in his intestine that wasn't caused by a lack of blood flow, which is unusual.

Methodology

Case report detailing the clinical presentation, surgical intervention, and histological findings.

Limitations

Limited literature on gastrointestinal complications in osteogenesis imperfecta.

Participant Demographics

51-year-old male with osteogenesis imperfecta type 1.

Digital Object Identifier (DOI)

10.4021/jocmr369w

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