Evaluation of Swallow Function in Patients with Craniofacial Microsomia: A Retrospective Study

Lara S Van De Lande,Cornelia J J M Caron,Marloes Streppel,David J Dunaway,Bonnie L Padwa,Maarten J Koudstaal,Koen F M Joosten,Britt I Pluijmers

doi:10.1007/s00455-017-9851-x

Abstract

Craniofacial microsomia (CFM) is characterized by underdevelopment of the structures derived from the first and second pharyngeal arches resulting in aesthetic, psychological, and functional problems including feeding and swallowing difficulties. The aim of this study is to gain more insight into swallowing difficulties in patients with CFM. A retrospective study was conducted in the population of patients diagnosed with CFM at three major craniofacial units. Patients with feeding difficulties and those who underwent video fluoroscopic swallow (VFS) studies were included for further analyses. The outcome of the VFS-studies was reviewed with regard to the four phases of swallowing. In our cohort, 13.5% of the 755 patients were diagnosed with swallowing difficulties. The outcome of the VFS-studies of 42 patients showed difficulties in the oral and pharyngeal phases with both thin and thick liquids. Patients with more severe mandibular hypoplasia showed more difficulties to form an appropriate bolus compared to patients who were less severely affected. This is the first study to document swallowing problems in patients with CFM. Difficulties were seen in both the oral and pharyngeal phases. We recommend routine screening for swallowing issues by a speech and language therapist in all patients with CFM and to obtain a VFS-study in patients with a type III mandible.

Highlights

Craniofacial microsomia (CFM) is a complex and heterogeneous condition characterized by underdevelopment of structures derived from the first and second pharyngeal arches including the orbit, mandible, ear, facial nerves, facial soft tissues, and muscles [1, 2]
Following IRB approval (Rotterdam: MEC-2013-575; London: 14DS25; Boston: X05-08-058), medical charts were reviewed for information on sex, affected side, severity of the deformity according to the Pruzansky–Kaban classification [4, 25], presence of feeding difficulties (FD) and type of FD, presence of cleft lip and/or palate, cleft repair, presence of tracheostomy, reports of performed video fluoroscopic swallow (VFS)-studies, and available clinical pictures and/or radiographic images
13.5% of the patients were diagnosed with a swallowing disorder, necessitating a VFS-study in 50.9% of these patients

Summary

Introduction

Craniofacial microsomia (CFM) is a complex and heterogeneous condition characterized by underdevelopment of structures derived from the first and second pharyngeal arches including the orbit, mandible, ear, facial nerves, facial soft tissues, and muscles [1, 2]. With an incidence of 1:3000 to 1:5000 live births, CFM is believed to be the second most common craniofacial anomaly following cleft lip and palate [2,3,4]. The facial anomalies seen in CFM may lead to aesthetic and psychological problems, and to functional issues such as breathing and feeding difficulties (FD) [5, 6]. FD are seen in 42–83% of the patients with CFM and include problems with suckling, chewing, failure to thrive, and swallowing [5, 7,8,9]. S. van de Lande et al.: Evaluation of Swallow Function in Patients with Craniofacial Microsomia; a Retrospective Study

Objectives

Methods

Results

Conclusion