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Growth of the hard palate in infants with Down syndrome compared with healthy infants-A retrospective case control study.

ABSTRACT: To investigate morphological differences of the hard palate in infants with Down syndrome (DS) compared with a volumetric-matched control group (CG).Trial design: retrospective case control study. Based on inclusion and exclusion criteria, plaster casts of edentulous maxillae of 40 DS infants (20 females and 20 males, aged 221.3 ± 132.4 days) and 40 CG infants (20 females and 20 males, aged 53.9 ± 87.2 days) were digitized and converted into 3-dimensional stereolithography data. An automated landmark- and investigator-independent method for assessing two-dimensional measurements such as width, depth, and length of palate, as well as palatal index and the 3-dimensional volume, were used.Matching DS and healthy CG infants by age, we found reduced sizes in all linear and volumetric measurements in the DS group. Matching both groups by palatal volume, we found no differences between the groups according to palatal width (p = .93), palatal depth (p = .32), and palatal index (p = .31). Control infants with the same palatal volume compared with the DS infants were about 151 days younger, 95%-CI = [102, 200] (Hodges-Lehmann estimator). Except for palatal length and palatal volume, the growth pattern of DS palates decreased irregularly at age 6 to 9 months.The palate of DS infants in the first 6 to 9 month of life is of normal shape but considerably smaller compared with healthy normals. From 6 to 9 months onward, the growth pattern of the hard palate in DS infants decreases irregularly. High-arch-constricted palates could, therefore, be interpreted as secondarily acquired in later life. We therefore speculate that it could be advantageous to begin oral muscular stimulating therapy between 6 and 9 months of age which may prevent palatal shape alterations and enhance oral function which also contributes to maxillary development.

SUBMITTER: Klingel D

PROVIDER: S-EPMC5552113 | biostudies-literature | 2017

REPOSITORIES: biostudies-literature

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Growth of the hard palate in infants with Down syndrome compared with healthy infants-A retrospective case control study.

Klingel Daniel D Hohoff Ariane A Kwiecien Robert R Wiechmann Dirk D Stamm Thomas T

PloS one 20170810 8

<h4>Objective</h4>To investigate morphological differences of the hard palate in infants with Down syndrome (DS) compared with a volumetric-matched control group (CG).<h4>Methods</h4>Trial design: retrospective case control study. Based on inclusion and exclusion criteria, plaster casts of edentulous maxillae of 40 DS infants (20 females and 20 males, aged 221.3 ± 132.4 days) and 40 CG infants (20 females and 20 males, aged 53.9 ± 87.2 days) were digitized and converted into 3-dimensional stereo ...[more]

PMID: 28796822

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Project description:Oral cavity wound healing occurs in an environment that sustains ongoing physical trauma and is rich in bacteria. Despite this, injuries to the mucosal surface often heal faster than cutaneous wounds and leave less noticeable scars. Patients undergoing cleft palate repair have a high degree of wound healing complications with up to 60% experiencing oronasal fistula (ONF) formation. In this study, we developed a mouse model of hard palate mucosal injury, to study the endogenous injury response during oral cavity wound healing and ONF formation. Immunophenotyping of the inflammatory infiltrate following hard palate injury showed delayed recruitment of non-classical LY6Clo monocytes and failure to resolve inflammation. To induce a pro-regenerative inflammatory response, delivery of FTY720 nanofiber scaffolds following hard palate mucosal injury promoted complete ONF healing and was associated with increased LY6Clo monocytes and pro-regenerative M2 macrophages. Alteration in gene expression with FTY720 delivery included increased Sox2 expression, reduction in pro-inflammatory IL-1, IL-4 and IL-6 and increased pro-regenerative IL-10 expression. Increased keratinocyte proliferation during ONF healing was observed at day 5 following FTY720 delivery. Our results show that local delivery of FTY720 from nanofiber scaffolds in the oral cavity enhances healing of ONF, occurring through multiple immunomodulatory mechanisms. STATEMENT OF SIGNIFICANCE: Wound healing complications occur in up to 60% of patients undergoing cleft palate repair where an oronasal fistula (ONF) develops, allowing food and air to escape from the nose. Using a mouse model of palate mucosal injury, we explored the role of immune cell infiltration during ONF formation. Delivery of FTY720, an immunomodulatory drug, using a nanofiber scaffold into the ONF was able to attract anti-inflammatory immune cells following injury that enhanced the reepithelization process. ONF healing at day 5 following FTY720 delivery was associated with altered inflammatory and epithelial transcriptional gene expression, increased anti-inflammatory immune cell infiltration, and increased proliferation. These findings demonstrate the potential efficacy of immunoregenerative therapies to improve oral cavity wound healing.

Dataset Information

Growth of the hard palate in infants with Down syndrome compared with healthy infants-A retrospective case control study.

Publications

Growth of the hard palate in infants with Down syndrome compared with healthy infants-A retrospective case control study.

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