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Unusual Mandibular Distraction Complication in Two Neonatal Pierre Robin Sequence Patients
Carmen Gloria Morovic1, Javier Torres2, Claudia Jorquera1
1Plastic and Reconstructive Surgery Unit, Luis Calvo Mackenna Children's Hospital.
Insights
This study explores sublingual salivary gland hypertrophy in Pierre Robin sequence (PRS) patients. Mandibular distraction surgery, used for airway obstruction, may be complicated by this condition.
Area of Science:
- Craniofacial Surgery
- Pediatric Otolaryngology
- Genetics and Rare Diseases
Background:
- Pierre Robin sequence (PRS) involves micrognathia, glossoptosis, and airway obstruction.
- Conservative management and surgical interventions like mandibular distraction are used for PRS.
- Severe airway obstruction in PRS necessitates prompt intervention.
Purpose of the Study:
- To report two cases of sublingual salivary gland hypertrophy in PRS patients.
- To investigate the implications of this hypertrophy in the context of mandibular distraction surgery.
- To highlight a potential complication associated with PRS airway management.
Main Methods:
- Case report of two patients diagnosed with Pierre Robin sequence.
- Review of clinical presentation and management strategies.
- Analysis of sublingual salivary gland hypertrophy in relation to mandibular distraction.
Main Results:
- Two PRS patients presented with severe airway obstruction requiring intervention.
- Sublingual salivary gland hypertrophy was identified as a complicating factor.
- Mandibular distraction was performed in both cases, with specific considerations for the hypertrophy.
Conclusions:
- Sublingual salivary gland hypertrophy can occur in Pierre Robin sequence patients with severe airway obstruction.
- This hypertrophy may present challenges during mandibular distraction surgery.
- Further investigation into the management of this specific complication is warranted.
Abstract:
The Pierre Robin sequence (PRS) is characterized by the triad of micrognathia, glossoptosis, and airway obstruction. Conservative management in Pierre Robin sequence consists of nasogastric tube feeding and positioning of the neonate (prone or lateral position) that facilitates the anterior position of the tongue or the application of continuous positive nasal pressure. In case of failure of this treatment, emergency tracheostomy and/or mandibular distraction must be performed.Mandibular distraction is a standard technique used by craniofacial surgeons to achieve an anteroposterior horizontal lengthening of the mandibular body, correcting the posterior position of the base of the tongue and thus retropharyngeal enlargement of the airway.The authors present 2 clinical cases of hypertrophy of the sublingual salivary glands with the use of mandibular distractors in SPR patients with severe airway obstruction.
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