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Case Reports
. 2025 Feb:127:110971.
doi: 10.1016/j.ijscr.2025.110971. Epub 2025 Feb 1.

Anesthetic management in pregnancy with osteogenesis imperfecta type XI: A comprehensive case report

Korakod Punnaniti  1 , Lisa Sangkum  1 , Sivaporn Termpornlert  1 , Rojnarin Komonhirun  1 , Sasima Dusitkasem  2
Affiliations
Case Reports

Anesthetic management in pregnancy with osteogenesis imperfecta type XI: A comprehensive case report

Korakod Punnaniti et al. Int J Surg Case Rep. 2025 Feb.

Abstract

Introduction: Osteogenesis imperfecta (OI) type XI or Bruck syndrome is an extremely rare genetic disorder characterized by congenital joint contractures and bone fragility. OI presents unique and considerable challenges in the perioperative and anesthetic management of affected patients.

Presentation of case: A 29-year-old primigravida with OI type XI (100 cm, 26 kg) and severe kyphoscoliosis underwent urgent Caesarean delivery at 32 weeks under general anesthesia (sevoflurane/nitrous oxide). A female infant (1470 g) required resuscitation. Postoperative recovery was uneventful.

Discussion: The rarity of this syndrome, along with the physiological changes associated with pregnancy, creates an unprecedented clinical scenario that demands a thorough and cautious approach to patient care.

Conclusion: Osteogenesis imperfecta (OI) type XI necessitates careful anesthetic management in pregnancy. This case highlights the anesthetic management challenges and the use of a multidisciplinary approach to enhance clinical understanding and improve patient outcomes.

Keywords: Bruck syndrome; Caesarean delivery; FKBP10 mutation; General anesthesia; Osteogenesis imperfecta (OI) type XI.

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Conflict of interest statement

Declaration of competing interest All of the authors have no conflicts of interest to declare.

Figures

Fig. 1
Fig. 1
Characteristics of the patient showing multiple joint contractures and deformities.
Fig. 2
Fig. 2
Whole spine X-ray showing diffuse osteopenia and severe scoliosis with right thoracic and left lumbar convexities, causing relatively decreased volume of the right hemithorax and abnormal thoracic cage.
Fig. 3
Fig. 3
A timeline illustrating the sequence of events from patient admission to discharge.
See this image and copyright information in PMC

References

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