Related Experiment Video
Updated: Jan 20, 2026
P50 Sensory Gating in Infants
Published on: December 26, 2013
Face-mask CPAP as palliative support for diffuse tracheobronchomalacia from ring agenesis in an infant: When sleep
Abir Bouhamdi1,2, Yassin Chefchaou1, Nahla En-Nejjari3
1Sleep Medicine Center, Hassan II University Hospital, Epidemiology and Health Sciences Research Laboratory, Sidi Mohamed Ben Abdellah University, PO Box: 1835, Route Sidi Harazem, 30050, Fez, Morocco.
Insights
Congenital tracheal ring agenesis causes severe airway collapse. In this case, continuous positive airway pressure (CPAP) provided temporary support for diffuse tracheobronchomalacia (TBM) in an infant, highlighting its palliative role.
Area of Science:
- Pediatric Pulmonology
- Rare Diseases
- Airway Management
Background:
- Congenital absence or deficiency of tracheal rings is exceptionally rare.
- It can lead to diffuse tracheobronchomalacia (TBM) and life-threatening airway collapse.
- Infants may present with stridor, cyanotic spells, and nocturnal desaturation.
Abstract:
Congenital absence or deficiency of tracheal rings is exceptionally rare and may cause diffuse tracheobronchomalacia (TBM) with life-threatening airway collapse. A 6-month-old girl had persistent stridor from birth, cyanotic spells during feeding, and nocturnal desaturation. Cervicothoracic CT suggested severe airway collapse, and rigid bronchoscopy showed a flattened trachea and main bronchi without visible cartilaginous rings, consistent with ring agenesis and diffuse TBM. Curative surgery was not feasible. After in-hospital titration during a nap, face-mask CPAP with heated humidification was prescribed during sleep and postprandial windows. A minimal effective pressure of 6 cmH2O stabilized oxygenation and reduced cyanotic episodes, with good tolerance and device-recorded adherence. Despite transient clinical benefit, the infant died at one and a half years of age (18 months) due to disease progression. In extensive congenital TBM from ring agenesis not amenable to reconstruction, targeted face-mask CPAP can provide palliative/bridging support by counteracting dynamic airway collapse at times of highest vulnerability (sleep and after feeding). This case highlights practical aspects of titration, interface choice, and monitoring in infants.
Related Concept Videos
12:55P50 Sensory Gating in Infants
11:39Non-invasive Optical Measurement of Cerebral Metabolism and Hemodynamics in Infants
12:16Quantitative Measurement of the Immune Response and Sleep in Drosophila
07:40Multi-Modal Home Sleep Monitoring in Older Adults
20:06The Comprehensive Infant Examination
The newborn exam is important in establishing a baby's baseline health status as they enter the world. Newborn providers have the unique opportunity to evaluate a patient for the very first time. They are also tasked with identifying congenital anomalies that require early intervention to promote a long and healthy life for the child. Many newborn anomalies, such as heart defects, are noted on prenatal ultrasound....
08:45Polygraphic Recording Procedure for Measuring Sleep in Mice