Abstract
Abstract Objective Bohring-Opitz syndrome (BOS) is a rare congenital disorder with approximately 60 documented cases worldwide. Distinguishing characteristics include a complex phenotype with associated feeding intolerance, emesis, sleep difficulties, respiratory complications, seizures, and failure to thrive. Infant massage is a supportive intervention with documented positive effects in medically-fragile infants, suggesting its potential as a non-pharmacological treatment in BOS. Limited data exists regarding the clinical management of patients with BOS. To initiate this effort, we report the effects of infant massage on a male with BOS. Method Born at 32 weeks gestation, the patient presented with chronic respiratory failure, tracheostomy, and ventilator dependence. Development was complicated by chronic infections, autonomic instability, feeding intolerance, seizures, and prolonged sedation wean. Clinicians provided infant massage (~15 minutes/week) to the patient’s hands and feet from the ages of 12-20 months, with the goal of decreasing heart rate. Heart rate was measured after each massage session over a 3-month period (14 sessions total) during hospitalization. Results Decreased heart rate was recorded following all massage sessions, with an average decline of 10.4 beats per minute. Decreased non-purposeful body movements, increased tactile orientation, increased feeding tolerance, and timely discharge from the hospital were also noted. Conclusion This case study offers initial support for the use of infant massage for hospitalized patients with BOS. Infant massage was associated with lowered heart rate and improved hospitalization course. While the researchers propose integration of infant massage within the clinical management of patients with BOS, further research is warranted to elucidate its implications for improved health outcomes.
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