Clinical Improvements Following Posterior Cranial Vault Remodeling in a Patient with Bronchopulmonary Dysplasia: A Case Report

Bronchopulmonary dysplasia (BPD) is a chronic lung disease and known sequelae of prolonged mechanical ventilation in premature infants. The use of positive-end expiratory pressure (PEEP) can increase intracranial pressure (ICP), which can have devastating effects on neurological function and development (1). Intracranial hypertension is also associated with craniosynostosis: the premature fusion of cranial sutures which can limit appropriate expansion of an infant's developing brain (2). We present the case of an 18-month-old male born prematurely at twenty-six weeks and two days gestation with a complicated medical history including severe BPD, tracheobronchomalacia with air trapping, tracheostomy dependence on a ventilator, and significant hemodynamic instability associated with BPD spells. After five months of increasing PEEP support with minimal improvement, significant numbers of BPD spells, and the need for a morphine drip for comfort, the patient's prognosis was not promising. A computed tomography of his head demonstrated craniosynostosis of the coronal and sagittal sutures. Given the significant PEEP support, compounded with craniosynostosis, the craniofacial team recommended cranial vault expansion to increase intracranial volume and reduce suspected ICP. Over the five months following posterior cranial vault distraction, his required PEEP was successfully weaned from eighteen to nine cm H2O. Additionally, he was able to wean off his morphine drip and was placed on a methadone taper. Ultimately, the patient was discharged home with a home ventilator. This case study helps further increase our understanding of effects of ICP in the BPD patient population and the potential benefits of cranial expansion in recalcitrant BPD patients. To our knowledge, there have been no studies published on the outcomes associated with cranial vault repair in the bronchopulmonary dysplasia patient population. Only one report in the literature describes differences in calvarial bone quality in BPD patients, including calvarial thickening and 1000x increased rates of premature suture closure (4). In this case study, the posterior cranial vault remodeling appeared to dramatically improve this patient's respiratory outcome, reduced his need for pain medication, and ultimately allowed him to be discharged from the hospital, a notion that seemed impossible presurgically. Further work needs to be done to elucidate the craniofacial dysmorphism in BPD patients and to elucidate the clinical impact it may have in this patient population.