Incomplete Cantrell syndrome with respiratory disorder in an early neonate: A case report

<p> A male neonate was admitted to our hospital on the day of birth with a respiratory disorder. He had familial history of incomplete Cantrell syndrome with his half sibling. He was intubated soon after birth due to suprasternal notch retraction and groaning. During intubation, his retraction continued despite ventilator management. His breathing consequently improved, and extubating was performed on day 1. Subsequently, we started nasal continuous positive airway pressure（CPAP）therapy for him. His retraction continued, and he presented remarkable tachypnea accompanying CPAP withdrawal. Computed tomography（CT）scan revealed hypoplasia of the lower sternum and suspicion of raise in forward diaphragm. Based on the CT findings and familial history, a diagnosis of incomplete Cantrell syndrome was made. We performed thoracoscopic checking on day 56 and detected a partial defect in the muscular layer of the diaphragm. Simultaneously, thoracoscopic plication of diaphragm was performed. We introduced CPAP therapy for the neonate at his home, and he was discharged on day 81. Cantrell syndrome is a rare congenital anomalous syndrome; most cases are diagnosed with a thoracoabdominal wall defect. In our case, familial history was an important indicator, with which we could make a diagnosis. Clinicians should be aware that some patients with incomplete Cantrell syndrome show neonatal respiratory disorders with multiple abnormalities of the sternum, diaphragm or heart.</p>