Prospective cohort study on facial profile changes in infants with Robin sequence and healthy controls.

Abstract

Various conservative and surgical approaches exist to treat Robin sequence (RS), but their effects on facial profile and mandibular catch-up growth are unclear. A functional treatment concept, used in our centre for 25years, includes an individualized palatal plate with a velo-pharyngeal extension and intensive feeding training. We performed a prospective study to objectively describe facial profiles in infants with RS treated with the above concept. Infants with isolated RS were admitted to our tertiary perinatal and national referral centre for craniofacial malformations between May 2018 and Nov 2019. Infants with RS received 3D-photographs during clinically indicated visits. Healthy controls were recruited from Dec 2018 to Sep 2019 and received 3D-photographs every 3months. The digitally measured jaw index (JI), defined as alveolar overjet (O) x maxillary arch (U)/mandibular arch (L), and the soft tissue reference points A'-point, Nasion', B'-point angle (ANB'), describing the relative position of maxilla to mandible, were evaluated. Linear mixed models were used to examine time trajectories in JI and ANB'. A total of 207 3D images, obtained in 19 infants with RS and 32 controls, were analysed. JI and ANB' decreased over time in both groups [for JI -0.18 (95% CI -0.25 to -0.10); for ANB': -0.40° per month [(95% CI -0.48 to -0.32)]] but remained lower in controls [for JI -2.5 (95% CI -3.2 to -1.8); for ANB'-1.7° (95% CI -2.4 to -1.0)]. Also, the ANB' model showed a significant effect of the interaction term diagnosis x age. Based on longitudinal 3D images, we describe changes in objective parameters of facial profile in infants with and without RS during the first year of life. Our findings indicate catch-up growth in infants treated for RS. Video Abstract.