Complete Digital Workflow for Manufacturing Presurgical Orthodontic Palatal Plates in Newborns and Infants with Cleft Lip and/or Palate.

Deciding on the implementation or modification of steps in daily clinical care is a nuanced process that demands careful evaluation. This is crucial not only for selecting the most appropriate solution but also for achieving the best treatment outcome. Thus, implementing a workflow for treating cleft lip and/or palate patients with a presurgical orthodontic cleft-covering plate needs to consider objective factors, prioritized from most to least important: safety and quality level, user-friendliness, feasibility, and, finally, efficiency and cost. The goal of this workflow is to integrate CAD/CAM technologies into daily clinical routine to enhance technical and clinical efficiency, reduce the burden of cleft care, and simplify the implementation of these technologies in other facilities. To achieve this, a methodology based on intraoral scanning and additive manufacturing is employed to produce patient-specific passive palatal plates. The approach describes possible pitfalls and their resolution within the routine of a cleft centre, along with an exemplary case scenario. Comparative analysis between the digital workflow and the conventional process demonstrated the digital approach to be safer, higher in quality, more user-friendly, feasible, and cost- and time-effective than the conventional process.

Impact of the adenosine receptor A2BR expressed on myeloid cells on immune regulation during pregnancy.

During pregnancy, the maternal immune system must carefully balance protection against pathogens with tolerance toward the semiallogeneic fetus. Dysfunctions of the immune system can lead to severe complications such as preeclampsia, fetal growth restriction, or pregnancy loss. Adenosine plays a role in physiological processes and plasma-level increase during pregnancy. The adenosine receptor A2B (A2BR), which is expressed on both, immune and nonimmune cells, is activated by high adenosine concentrations, achieved during pregnancy. We investigated the impact of A2BR expressed on myeloid cells on immune regulation during pregnancy using a mouse model with myeloid deficiency for A2BR. We demonstrate systemic changes in myeloid and lymphoid cell populations during pregnancy in A2BR-KO (Adora2B923f/f-LysMCre) mice with increased monocytes, neutrophils, and T cells but decreased B cells as well as altered T-cell subpopulations with decreased Th1 cells and Tregs and increased Th17 cells. Lack of A2BR on myeloid cells caused an increased systemic expression of IL-6 but decreased systemic accumulation and function of MDSC and reduced numbers of uterine natural killer cells. The pregnancy outcome was only marginally affected. Our results demonstrate that A2BR on myeloid cells plays a role in immune regulation during pregnancy, but the clinical impact on pregnancy remains unclear.

Influence of catheter thickness on respiratory physiology during less invasive surfactant administration in extremely preterm infants.

Introduction: Delivering surfactant via thin catheters (minimal-invasive surfactant therapy (MIST); less invasive surfactant administration (LISA)) has become a common procedure. However, the effect of tracheal obstruction caused by catheters of different sizes on tracheal resistance in extremely low gestational age newborns (ELGANs) is unknown. Methods: To investigate the effect of catheters size 3.5, 5 and 6 French on airway resistance in ELGANs of 23-28 weeks gestational age during LISA, we performed calculations based on Hagen-Poiseuille's law and compared these with a clinically and physically more accurate method: computational fluid dynamics (CFD) simulations of respiratory airflow, performed in 3D virtual airway models derived from MRI. Results: The presence of the above catheters decreased the cross-sectional area of the infants' tracheal entrance (the cricoid ring) by 13-53%. Hagen-Poiseuille's law predicted an increase in resistance by 1.5-4.5 times and 1.3-2.6 times in ELGANs born at 23 and 28 weeks, respectively. However, CFD simulations demonstrated an even higher increase in resistance of 3.4-85.1 and 1.1-3.5 times, respectively. The higher calculated resistances were due to the extremely narrow remaining lumen at the glottis and cricoid with the catheter inserted, resulting in a stronger glottal jet and turbulent airflow, which was not predicted by Hagen-Poiseuille. Conclusion: Catheter thickness can greatly increase tracheal resistance during LISA-procedures in ELGANs. Based on these models, it is recommended to use the thinnest catheter possible during LISA in ELGANs to avoid unnecessary increases in airway resistance in infants already experiencing dyspnea due to respiratory distress syndrome.

Averaging Times for Pulse Oximeter Measurements - A Review of Manuscripts Published in the Top Five Sleep Medicine Journals.

Purpose: Clinical management decisions often rely on a patient's SpO2 level and desaturation rate. Limitations include that measurements depend on the averaging time (AVT) used, which is particularly relevant to sleep medicine, but has yet received little attention. Methods: Cross-sectional review of studies reporting pulse oximeter saturation (SpO2) measurements published in 5 leading sleep medicine journals. All papers published between 2017 and 2023 reporting SpO2 measurements were screened regarding the AVT used. Results: Of 193 papers identified, 151 were included; of these, only 9 studies mentioned the AVT, 4 of these were published in one journal. The AVT ranged from zero (beat-to-beat-mode) to 10s, with 3s being used most often (33.3%), followed by 2s (22.2%). Conclusion: The AVT is only rarely mentioned in sleep medicine papers, despite its influence on sleep study results. Reported AVTs were heterogenous. Further research is warranted to set up guidelines for using or reporting the AVT.

Lung ultrasound to predict the duration of respiratory support in newborn infants with respiratory distress.

AIM: Lung ultrasound (LU) and clinical parameters evaluated during the first postnatal hour potentially predict the length of CPAP therapy in newborns with respiratory distress. METHODS: In a single-centre, prospective observational pilot study, 130 newborns ≥36 weeks gestational age were assessed using standardised LU at 30 and 60 min postnatally. Various clinical parameters were evaluated influencing CPAP duration (<1 vs. ≥1 h) using univariate and multivariate analyses. RESULTS: Lung ultrasound score >5, FiO2 > 0.21 and respiratory acidosis 30 min postnatally were associated with CPAP ≥1 h. Our model showed good diagnostic quality (ROC AUC = 0.87) and was confirmed by classification and regression tree (CART) analysis. Additional LU findings like double lung point and pleural line abnormalities were frequently observed, with good interrater reliability for LU interpretation (ICC = 0.76-0.77). CONCLUSION: Newborns with postnatal respiratory distress at risk for prolonged CPAP therapy can be identified based on specific LU and clinical parameters assessed 30 min postnatally. Despite the need for validation in an independent sample, these findings may lay the groundwork for a prediction tool. LU proved feasible and reliable for assessing respiratory status in this population, highlighting potential utility in clinical practice.

Facial growth dynamics dictate optimal facemask fitting during the first year of life.

AIM: The aim of this study is to prevent mask leak during ventilation in infant emergencies, appropriate facemask fitting is essential. Therefore, we investigated facial profiles during the first year of life and their correlation with the correct sizing of masks. METHODS: This is a post hoc subgroup analysis of 32 healthy term infants, based on a prospective observational study performed from September 2018 to December 2019 in Tuebingen, Germany. In 3-monthly intervals, facial aspects were measured based on anatomical landmarks in three-dimensional frontal photographs to describe their changes across the first year of life. All infants were awake and breathing spontaneously; none required any anaesthesia. RESULTS: In 130 3D images, mean distance between nasion and gnathion was 54 mm (3.3) measured at birth and 70 mm (3.5) at age 12 months. Gompertz models showed relevant growth-related changes in the facial profile in vertical but not horizontal direction. Vertical growth occurred mainly in the first 6 months. Boys and girls differed by an average of about 2 mm (boys >girls). CONCLUSION: Based on our findings, it should now be verified whether the 50 mm facemasks are suitable for infants from birth to 2 months of age, respectively, the 60 mm version for infants aged three to 12 months.

Less Invasive Surfactant Administration in Preterm Infants in Tertiary Neonatal Intensive Care Units in Germany: A Survey.

INTRODUCTION: The European guideline for treatment of respiratory distress syndrome recommends less invasive surfactant administration (LISA) as the preferred method of surfactant administration in spontaneously breathing preterm infants. However, there is limited evidence on practical aspects such as sedation and catheter types, leading to considerable variability between centers. METHODS: An anonymous online survey (www.soscisurvey.de) was sent to 164 tertiary neonatal intensive care units (NICUs) in Germany including 43 questions on practical aspects of LISA. RESULTS: Of 122 (74%) participating NICUs, 117 (96%) reported experience with LISA with 82% of those reporting LISA as their preferred method of surfactant administration. Indications for surfactant administration differed widely between NICUs. Most (89%) used FiO2-thresholds only or in combination with other criteria, such as Silverman score/signs of dyspnea (41%) or lung ultrasound findings (3%). Prophylactic surfactant was administered by 42%. Differences in use of LISA in extremely immature infants were reported (e.g., 36% did not perform LISA in infants below 24-26 weeks). Preferred drugs for sedation were (Es-)Ketamine, followed by Propofol and Midazolam. Minimum time interval between subsequent LISA procedures was 4 (2-6) h. Catheters specifically designed for LISA were used by most NICUs (69%). CONCLUSION: This survey shows that LISA is common practice in German NICUs, but with considerable variability in practical aspects. These data may serve as a guidance for NICUs that have not yet implemented LISA and might be helpful design clinical trials with the aim to standardize and/or optimize LISA.

Evidence and Perspectives for Choline Supplementation during Parenteral Nutrition-A Narrative Review.

Choline is an essential nutrient, with high requirements during fetal and postnatal growth. Tissue concentrations of total choline are tightly regulated, requiring an increase in its pool size proportional to growth. Phosphatidylcholine and sphingomyelin, containing a choline headgroup, are constitutive membrane phospholipids, accounting for >85% of total choline, indicating that choline requirements are particularly high during growth. Daily phosphatidylcholine secretion via bile for lipid digestion and very low-density lipoproteins for plasma transport of arachidonic and docosahexaenoic acid to other organs exceed 50% of its hepatic pool. Moreover, phosphatidylcholine is required for converting pro-apoptotic ceramides to sphingomyelin, while choline is the source of betaine as a methyl donor for creatine synthesis, DNA methylation/repair and kidney function. Interrupted choline supply, as during current total parenteral nutrition (TPN), causes a rapid drop in plasma choline concentration and accumulating deficit. The American Society for Parenteral and Enteral Nutrition (A.S.P.E.N.) defined choline as critical to all infants requiring TPN, claiming its inclusion in parenteral feeding regimes. We performed a systematic literature search in Pubmed with the terms "choline" and "parenteral nutrition", resulting in 47 relevant publications. Their results, together with cross-references, are discussed. While studies on parenteral choline administration in neonates and older children are lacking, preclinical and observational studies, as well as small randomized controlled trials in adults, suggest choline deficiency as a major contributor to acute and chronic TPN-associated liver disease, and the safety and efficacy of parenteral choline administration for its prevention. Hence, we call for choline formulations suitable to be added to TPN solutions and clinical trials to study their efficacy, particularly in growing children including preterm infants.

Failure of an electric rocking device to improve neonatal sleep.

AIM: We wanted to investigate whether an electric rocking device (Swing2Sleep, Neumünster, Germany), sold with the claim to promote infant sleep, would increase total sleep time or reduce sleep latency once infants are put therein. METHODS: In a randomised crossover design, 20 infants (median gestational age at birth 31.7 weeks, range 25-39) were placed to sleep either first with the device rocking, then not rocking (or vice versa) for 5-7 h each. The device consisted of a hammock with three spiral springs that performed vertical swings at a rate of 100/min and an amplitude of 2.5 cm. RESULTS: There was no significant difference in %time spent asleep (83 (22-97) vs. 85% (49-96)), sleep latency (7.7 (2-45) vs. 12.3 (4-42) min), sleep fragmentation (1.3 (0.5-2.3) vs. 1.1 (0.2-5.5)) or efficiency (0.8 (0.2-1.0) vs. 0.9 (0.5-1.0)) between both conditions. CONCLUSION: At its recommended settings, the device did not achieve its intended effect in these infants.

Delineating morbidity patterns in preterm infants at near-term age using a data-driven approach.

BACKGROUND: Long-term survival after premature birth is significantly determined by development of morbidities, primarily affecting the cardio-respiratory or central nervous system. Existing studies are limited to pairwise morbidity associations, thereby lacking a holistic understanding of morbidity co-occurrence and respective risk profiles. METHODS: Our study, for the first time, aimed at delineating and characterizing morbidity profiles at near-term age and investigated the most prevalent morbidities in preterm infants: bronchopulmonary dysplasia (BPD), pulmonary hypertension (PH), mild cardiac defects, perinatal brain pathology and retinopathy of prematurity (ROP). For analysis, we employed two independent, prospective cohorts, comprising a total of 530 very preterm infants: AIRR ("Attention to Infants at Respiratory Risks") and NEuroSIS ("Neonatal European Study of Inhaled Steroids"). Using a data-driven strategy, we successfully characterized morbidity profiles of preterm infants in a stepwise approach and (1) quantified pairwise morbidity correlations, (2) assessed the discriminatory power of BPD (complemented by imaging-based structural and functional lung phenotyping) in relation to these morbidities, (3) investigated collective co-occurrence patterns, and (4) identified infant subgroups who share similar morbidity profiles using machine learning techniques. RESULTS: First, we showed that, in line with pathophysiologic understanding, BPD and ROP have the highest pairwise correlation, followed by BPD and PH as well as BPD and mild cardiac defects. Second, we revealed that BPD exhibits only limited capacity in discriminating morbidity occurrence, despite its prevalence and clinical indication as a driver of comorbidities. Further, we demonstrated that structural and functional lung phenotyping did not exhibit higher association with morbidity severity than BPD. Lastly, we identified patient clusters that share similar morbidity patterns using machine learning in AIRR (n=6 clusters) and NEuroSIS (n=8 clusters). CONCLUSIONS: By capturing correlations as well as more complex morbidity relations, we provided a comprehensive characterization of morbidity profiles at discharge, linked to shared disease pathophysiology. Future studies could benefit from identifying risk profiles to thereby develop personalized monitoring strategies. TRIAL REGISTRATION: AIRR: DRKS.de, DRKS00004600, 28/01/2013. NEuroSIS: ClinicalTrials.gov, NCT01035190, 18/12/2009.

Pulse oximetry signal loss during hypoxic episodes in preterm infants receiving automated oxygen control.

The aim of this study was to analyze signal loss (SL) resulting from low signal quality of pulse oximetry-derived hemoglobin oxygen saturation (SpO2) measurements during prolonged hypoxemic episodes (pHE) in very preterm infants receiving automatic oxygen control (AOC). We did a post hoc analysis of a randomized crossover study of AOC, programmed to set FiO2 to "back-up FiO2" during SL. In 24 preterm infants (median (interquartile range)) gestational age 25.3 (24.6 to 25.6) weeks, recording time 12.7 h (12.2 to 13.6 h) per infant, we identified 76 pHEs (median duration 119 s (86 to 180 s)). In 50 (66%) pHEs, SL occurred for a median duration of 51 s (33 to 85 s) and at a median frequency of 2 (1 to 2) SL-periods per pHE. SpO2 before and after SL was similar (82% (76 to 88%) vs 82% (76 to 87%), p = 0.3)).  Conclusion: SL is common during pHE and must hence be considered in AOC-algorithm designs. Administering a "backup FiO2" (which reflects FiO2-requirements during normoxemia) during SL may prolong pHE with SL.  Trial registration: The study was registered at www. CLINICALTRIALS: gov under the registration no. NCT03785899. WHAT IS KNOWN: • Previous studies examined SpO2 signal loss (SL) during routine manual oxygen control being rare, but pronounced in lower SpO2 states. • Oxygen titration during SL is unlikely to be beneficial as SpO2 may recover to a normoxic range. WHAT IS NEW: • Periods of low signal quality of SpO2 are common during pHEs and while supported with automated oxygen control (SPOC), FiO2 is set to a back-up value reflecting FiO2 requirements during normoxemia in response to SL, although SpO2 remained below target until signal recovery. • FiO2 overshoots following pHEs were rare during AOC and occurred with a delayed onset; therefore, increased FiO2 during SL does not necessarily lead to overshoots.

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

BACKGROUND: 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 25 years, includes an individualized palatal plate with a velo-pharyngeal extension and intensive feeding training. METHODS: 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 3 months. 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'. RESULTS: 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. CONCLUSIONS: 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.

Influence of malocclusion and orthodontic treatment in the masticatory efficiency of patients with craniofacial disorders.

This study evaluates masticatory efficiency in orthodontic patients with craniofacial disorders compared to controls without, considering the effect of an orthodontic appliance and malocclusion. A total of 119 participants (7-21 years), divided into a craniofacial disorder and control group (n = 42 and n = 77; mean age 13.5 ± 5.2 and 14.2 ± 3.3 years) were included. Masticatory efficiency was evaluated using a standard food model test, where masticated test food bodies were analyzed, and parameters like particle number (n) and area (mm2) were recorded. This study newly introduced the masticatory efficiency index (MEI), which encompasses the above terms (number and area), with a high MEI being an indicator of high masticatory ability. Younger orthodontic patients with a craniofacial disorder had a significantly decreased MEI (0.50 ± 0.25 n/mm2) compared to patients without (1.10 ± 0.48 n/mm2; p = 0.02). The presence of a crossbite significantly decreased masticatory efficiency, particularly in craniofacial disorder patients (0.69 ± 1.44 n/mm2) versus controls (0.89 ± 1.00 n/mm2, p = 0.04). As treatment progressed with age and fixed appliances, mastication group differences became non-significant, suggesting that patients with a craniofacial disorder were catching up to healthy controls in the rehabilitation of their masticatory function. Considering an early diagnosis of malocclusion during orthodontic therapy in combination with speech therapy can avoid negative malocclusion effects with growth, caused by muscle imbalances.

Impact of perinatal administration of probiotics on immune cell composition in neonatal mice.

BACKGROUND: Newborns and especially preterm infants are much more susceptible to infections than adults. The pathogens causing infections in newborns are often detectable in the intestinal flora of affected children even before disease onset. Therefore, it seems reasonable to prevent dysbiosis in newborns and preterm infants. An approach followed in many neonatal intensive care units (NICUs) is to prevent infections in preterm infants with probiotics however their mechanisms of action of probiotics are incompletely understood. Here, we investigated the effect of perinatal probiotic exposure on immune cells in newborn mice. METHODS: Pregnant mice were orally treated with a combination of Lactobacillus acidophilus and Bifidobacterium bifidum (Infloran®) from mid-pregnancy until the offspring were harvested. Immune cell composition in organs of the offspring were analyzed by flow cytometry. RESULTS: Perinatal probiotic exposure had profound effects on immune cell composition in the intestine, liver and lungs of newborn mice with reduction of myeloid and B cells and induction of T cells in the probiotic treated animals' organs at weaning. Furthermore, probiotic exposure had an effect on T cell development in the thymus. CONCLUSION: Our results contribute to a better understanding of the interaction of probiotics with the developing immune system. IMPACT: probiotics have profound effects on immune cell composition in intestines, livers and lungs of newborn mice. probiotics modulate T cell development in thymus of newborn mice. effects of probiotics on neonatal immune cells are particularly relevant in transition phases of the microbiome. our results contribute to a better understanding of the mechanisms of action of probiotics in newborns.

Randomised crossover study on pulse oximeter readings from different sensors in very preterm infants.

OBJECTIVE: In extremely preterm infants, different target ranges for pulse oximeter saturation (SpO2) may affect mortality and morbidity. Thus, the impact of technical changes potentially affecting measurements should be assessed. We studied SpO2 readings from different sensors for systematic deviations. DESIGN: Single-centre, randomised, triple crossover study. SETTING: Tertiary neonatal intensive care unit. PATIENTS: 24 infants, born at <32 weeks' gestation, with current weight <1500 g and without right-to-left shunt via a patent ductus arteriosus. INTERVENTIONS: Simultaneous readings from three SpO2 sensors (Red Diamond (RD), Photoplethysmography (PPG), Low Noise Cabled Sensors (LNCS)) were logged at 0.5 Hz over 6 hour/infant and compared with LNCS as control using analysis of variance. Sensor position was randomly allocated and rotated every 2 hours. Seven different batches each were used. OUTCOMES: Primary outcome was the difference in SpO2 readings. Secondary outcomes were differences between sensors in the proportion of time within the SpO2-target range (90-95 (100)%). RESULTS: Mean gestational age at birth (±SD) was 274/7 (±23/7) weeks, postnatal age 20 (±20) days. 134 hours of recording were analysed. Mean SpO2 (±SD) was 94.0% (±3.8; LNCS) versus 92.2% (±4.0; RD; p<0.0001) and 94.5% (±3.9; PPG; p<0.0001), respectively. Mean SpO2 difference (95% CI) was -1.8% (-1.9 to -1.8; RD) and 0.5% (0.4 to 0.5; PPG). Proportion of time in target was significantly lower with RD sensors (84.8% vs 91.7%; p=0.0001) and similar with PPG sensors (91.1% vs 91.7%; p=0.63). CONCLUSION: There were systematic differences in SpO2 readings between RD sensors versus LNCS. These findings may impact mortality and morbidity of preterm infants, particularly when aiming for higher SpO2-target ranges (eg, 90-95%). TRIAL REGISTRATION NUMBER: DRKS00027285.

Choline and Betaine Levels in Plasma Mirror Choline Intake in Very Preterm Infants.

Choline is essential for cell membrane formation and methyl transfer reactions, impacting parenchymal and neurological development. It is therefore enriched via placental transfer, and fetal plasma concentrations are high. In spite of the greater needs of very low birth weight infants (VLBWI), choline content of breast milk after preterm delivery is lower (median (p25-75): 158 mg/L (61-360 mg/L) compared to term delivery (258 mg/L (142-343 mg/L)). Even preterm formula or fortified breast milk currently provide insufficient choline to achieve physiological plasma concentrations. This secondary analysis of a randomized controlled trial comparing growth of VLBWI with different levels of enteral protein supply aimed to investigate whether increased enteral choline intake results in increased plasma choline, betaine and phosphatidylcholine concentrations. We measured total choline content of breast milk from 33 mothers of 34 VLBWI. Enteral choline intake from administered breast milk, formula and fortifier was related to the respective plasma choline, betaine and phosphatidylcholine concentrations. Plasma choline and betaine levels in VLBWI correlated directly with enteral choline intake, but administered choline was insufficient to achieve physiological (fetus-like) concentrations. Hence, optimizing maternal choline status, and the choline content of milk and fortifiers, is suggested to increase plasma concentrations of choline, ameliorate the choline deficit and improve growth and long-term development of VLBWI.

Caring for Infants with Robin Sequence Treated with the Tübingen Palatal Plate: A Review of Personal Practice.

The Tübingen Palatal Plate (TPP) is a minimally invasive yet highly effective functional orthodontic treatment for upper airway obstruction in infants with Robin Sequence (RS). It consists of a palatal plate to cover the cleft and a velar extension that shifts the root of the tongue forward. We review our practical experience with this approach. First, upon admission, our local orthodontists perform an (3-D) intraoral scan of the maxilla. Based on the scan data, the TPP is manufactured in a semi-digital workflow. The length and angulation of its extension is checked via awake laryngoscopy and the effectiveness confirmed by a sleep study. Plates are kept in place by adhesive cream. When inserting the TPP, the tip of the tongue must be visible. Next, metal fixation bows should be secured to the forehead using tape and elastic bands. Plates are removed daily for cleaning, and the oral mucosa is then checked for pressure marks. Feeding training (initially only via finger feeding) may even start before plate insertion. Breathing often normalizes immediately once the plate is inserted. For isolated RS, we have never had to perform a tracheostomy. This has largely been possible through our highly dedicated and competent team, particularly the nursing staff, and the early involvement of parents.