Patterns of Oropharyngeal Dysphagia Associated With Craniofacial Microsomia

Craniofacial microsomia (CFM) is a congenital condition characterized by mandibular hypoplasia, facial asymmetry, upper airway obstruction, and potential cleft lip/palate that can impact swallowing function resulting in dysphagia. This study aims to characterize the prevalence of oropharyngeal dysphagia at all levels of CFM severity by analyzing Video Fluoroscopic Swallow Studies (VFSS) of patients with CFM, and identify patterns of oral and pharyngeal dysphagia to guide assessment and treatment.

A retrospective chart review of patients aged 0-18 years at a single institution from January 2015 to February 2021 with a diagnosis of CFM was performed. Patients who did not receive a VFSS or had a primary diagnosis other than CFM were excluded. Severity of CFM was determined using the Pruzansky-Kaban classification (PKC), while all VFSSs were scored using the Penetration Aspiration Scale (PAS). Oral and pharyngeal phase impairments were scored by level of severity. The Functional Oral Intake Scale (FOIS) was used to categorize the functional oral intake of foods and liquids.

Eighteen total patients met the inclusion criteria for this study. Eight (44%) patients were male and the median age at the time of first VFSS was 8.1 months (interquartile range: 2.1-30.3). On the basis of the Pruzansky-Kaban classification, 8 patients were grade 1, 3 were 2a, 3 were 2b, and 4 were grade 3. The majority of the patients, 14/18 (78%) were impacted on the right side. Twelve patients underwent polysomnography and 10/12 (83%) were diagnosed with obstructive sleep apnea. Laryngomalacia was present in 7 of the 18 patients (39%). The most frequently occurring oral phase swallowing impairments were anterior labial escape of less than half the bolus (12, 67%) and impaired bolus control resulting in spillage to the level of the pyriform sinuses (15, 83%). Pharyngeal residue was present in 17 of the 18 patients (94%) patients with the base of tongue (14, 78%) and valleculae (11, 61%) being the most common anatomic location. In total, 9 (50%) of the patients exhibited laryngeal penetration [Penetration Aspiration Scale (PAS) 2, 3, and 4] on at least one consistency, while 8 (44%) presented with aspiration [PAS 7, 8] on at least one consistency. Retrograde movement of the bolus into the nasopharynx/nasopharyngeal regurgitation was present in 4 out of 18 (22%). The upper esophageal phase was unremarkable in all patients. Changes to oral feeding status according to the Functional Oral Intake Scale occurred in half of the patients following VFSS evaluation.

Patients with CFM of all severities demonstrated oral and pharyngeal dysphagia on VFSS, including impaired labial closure, impaired bolus containment, pharyngeal residue, and penetration/aspiration. Instrumental assessment of patients with CFM is recommended to assess anatomy and physiology of the oropharyngeal phases of swallowing, and to identify the specific interventions and compensatory strategies to improve feeding outcomes.