We describe the anesthesia management of a 12-year-old girl, diagnosed with fibrodysplasia ossificans progressiva (FOP), who presented with a submandibular abscess. FOP is a rare, inherited disorder with heterotopic bone formation and progressive musculoskeletal disability. This disability ultimately confines patients to a wheelchair. Minor trauma following dental treatment may lead to ankylosis of the jaw. Subsequent to this disability, which resulted in poor dental hygiene, our patient developed a dental abscess. This spread along the mandibular margin and under the tongue. She presented with an impending airway compromise in an already difficult situation. The options for airway management in a child with limited mouth opening are discussed.

Summary Background The local anesthetic, levobupivacaine, is the safer enantiomer of racemic bupivacaine. Present protocols for levobupivacaine are based on studies and pharmacokinetic modeling with racemic bupivacaine. Aims The aim is to investigate total serum levobupivacaine concentrations after a caudalepidural loading dose followed by a maintenance infusion over 48 hours in infants aged 3‐6 months. Methods The clinical trial was conducted in eight infants aged 3‐6 months, undergoing bladder exstrophy repair. Pharmacokinetic modeling allowed optimization of clinical sampling to measure total levobupivacaine and α1‐acid glycoprotein and prediction of the effect of α1‐acid glycoprotein on levobupivacaine plasma protein binding. Results The observed median total levobupivacaine serum concentration was 0.30 mg/L (range: 0.20‐0.70 mg/L) at 1 hour after the loading dose of 2 mg/kg. The median total levobupivacaine concentration after 47 hours of infusion, at 0.2 mg/kg/h, was 1.21 mg/L (0.07‐1.85 mg/L). Concentrations of α1‐acid glycoprotein were found to rise throughout the study period. Pharmacokinetic modeling suggested that unbound levobupivacaine quickly reached steady state at a concentration of approximately 0.03 mg/L. Conclusion The study allows the development of a pharmacokinetic model, combining levobupivacaine and α1‐acid glycoprotein data. Modeling indicates that unbound levobupivacaine quickly reaches steady state once the infusion is started. Simulations suggest that it may be possible to continue the infusion beyond 48 hours.

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