Respiratory outcomes after cleft palate closure in Robin sequence: a retrospective study.

OBJECTIVES: There is a paucity of information about the possible risk factors that could identify patients with Robin sequence (RS) who are more prone to developing obstructive airway complications after palate closure. This study aimed to compare the respiratory complication rates in patients with RS and isolated cleft palate (ICP). MATERIALS AND METHODS: In this retrospective study, we reviewed the medical records of 243 consecutive patients with RS and ICP who were treated at Amsterdam University Medical Centers over the past 25 years. We collected preoperative data on previous treatment, diagnostic findings, surgical technique, weight, and presence of congenital anomalies. RESULTS: During cleft palate closure, patients with RS were older (11.9 versus 10.1 months; p = 0.001) and had a lower gestational age than those with ICP (37.7 versus 38.5 weeks; p = 0.002). Patients with RS had more respiratory complications (17 versus 5%; p = 0.005), were more often non-electively admitted to the pediatric intensive care unit (PICU) (13 versus 4.1%; p = 0.022), and had a longer hospital stay duration (3.7 versus 2.7 days; p = 0.011) than those with ICP. The identified risk factors for respiratory problems were a history of tongue-lip-adhesion (TLA) (p = 0.007) and a preoperative weight of < 8 kg (p = 0.015). Similar risk factors were identified for PICU admission (p = 0.015 and 0.004, respectively). CONCLUSIONS: The possible risk factors for these outcomes were a low preoperative weight and history of TLA. Closer postoperative surveillance should be considered for patients with these risk factors. CLINICAL RELEVANCE: Identifying risk factors for respiratory complications could provide clinicians better insight into their patients and allows them to provide optimal care for their patients.

Mental Health of School-Aged Children Treated with Propranolol or Atenolol for Infantile Hemangioma and Their Parents.

BACKGROUND: Infants with infantile hemangioma (IH) have been effectively treated with propranolol or atenolol. Concerns were raised about the mental health of these children at school age, due to central nervous system effects of propranolol and visible nature of IH. OBJECTIVE: This study aimed to compare the mental health at school age of children treated with propranolol to children treated with atenolol for IHs and their parents. METHODS: This two-centered cross-sectional study included children aged ≥6 years and treated with either propranolol or atenolol for IH during infancy. Children's outcomes were performance-based affect recognition (Dutch version of the Developmental Neuropsychological Assessment-II [NEPSY-II-NL]), parent-reported emotional and behavioral functioning (Child Behavioral Checklist [CBCL]), and health-related quality of life (KIDSCREEN-27). Parents' outcome was parenting stress (Parenting Stress Questionnaire [OBVL]). RESULTS: Data of 105 children (36 propranolol, 69 atenolol; 6.0-11.8 years) were analyzed. Mental health outcomes did not differ between both ß-blocker groups. Although overall functioning was in line with norms, children presented specific problems concerning affect recognition, parent-reported attention, and social quality of life. Parents showed increased physical symptoms, depressive symptoms, and parent-child relationship problems. CONCLUSION: No difference in mental health at school age was found between children treated with propranolol or atenolol for IH. Although few overall mental health problems were found, specific problems require follow-up. Follow-up of children should be directed toward affect recognition, attention, and social functioning in daily life. Problems reported by parents could be ameliorated by mental health support during and after their infant's ß-blocker treatment.

What Parents of Children Born with a Cleft Lip and/or Palate Want to Know About the Care for their Child.

OBJECTIVE: Adequate health information that matches the needs of care recipients is a prerequisite for patient-centered care. To facilitate the provision of tailored and timely information, it isimportant to understand the information needs of parents of children and adolescents with cleft lip and/or palate (CL/P) themselves, and in addition they were asked how they experienced the provided care-related information. DESIGN: A cross-sectional study employing questionnaires and semi-structured interviews. SETTING: Participants were recruited from a cleft palate-craniofacial care unit in a major tertiary hospital in the Netherlands. PARTICIPANTS: Participants were parents or guardians of children with CL/P, and two adolescents with CLP. They were recruited through the outpatient clinic during multidisciplinary consultation or after clinical admission. RESULTS: In total, fifty-five questionnaires were completed by parents or guardians and eleven interviews were conducted with nine parents of children with CL/P and two adolescents with CL/P. In general, participants reported to be satisfied with provided information during hospital admission or multidisciplinary cleft team consultations (mean 8.0, scale 0-10). In addition, 25.5% (n = 14) indicated that information to prepare for hospital admission was lacking (eg, practical information). Thematic qualitative analysis yielded five main information needs: 1) Clear communication during the care process, 2) Overview of the care trajectory, 3) Specific care plan information, 4) Presentation of information and 5) Guidance and support. CONCLUSIONS: Our findings emphasize the importance of gaining insights into wishes and information needs from care recipients who can provide insights in their information needs. With these findings, information provision should be redesigned to improve and to foster the further transition to family-centered care.

Smart surgical glasses for orofacial cleft surgery training in high- and low-income countries: A proof of concept.

INTRODUCTION: The coronavirus 2019 pandemic urged us to find alternatives for education through remote proctoring and international surgical collaborations among high-, middle-, and low-income countries. Smart surgical glasses are promising for remote surgical education and international surgical collaborations. AIMS: This study aimed to assess the usability of smart surgical glasses during cleft surgery and explore their potential in remote surgical education and collaboration. METHODS: Six plastic surgical cases were randomly selected and recorded using the RODS&CONES glasses in 4K (3840 × 2160p). A 23-point questionnaire was sent to one plastic surgeon, one plastic surgery resident, and eight doctors who were not trained to critically appraise the video and audio quality of the smart surgical glasses and their applicability for remote surgical education. RESULTS: The participants indicated that the smart glasses had several significant advantages over conventional on-site education, such as facilitating a better view of the surgical field and providing possibilities for remote interaction. The audio quality was considered excellent. The main limitations were image stabilization issues and loss of video connection due to weak wireless fidelity. CONCLUSIONS: All participants appreciated the use of smart glasses for remote education and considered them a promising tool for enhancing the quality of surgical education. The glasses can enable remote assistance and education of local surgical residents and may facilitate sustainable surgical collaborations among high-, middle-, and low-income countries.

Improving patient care by virtual case discussion between plastic surgeons and residents of Uganda and the Netherlands.

INTRODUCTION: Traditional on-site missions of plastic surgeons from "high-income countries" in "low- and middle-income countries" are often limited in time and lack proper follow-up. Regular digital collaboration could lead to a more impactful and durable exchange of knowledge for plastic surgeons and residents in both settings. AIMS: The aim of this study was to evaluate the satisfaction of the first twelve months of weekly digital meetings, explore advantages/disadvantages, and to provide tools for similar initiatives. METHODS: Weekly meetings started from August 2021. An encrypted digital connection allowed residents and plastic surgeons from Uganda and the Netherlands to discuss cases for educational purposes, where treatment options were considered. After twelve months, a survey was sent to participants from both countries to indicate the meetings' strengths, weaknesses, and possible improvements. RESULTS: A total of 18 participants responded to the questionnaire (ten plastic surgeons, six residents, and two researchers). The strengths of the meetings were the accessibility of the meetings, knowledge exchange and practice for residents' final exams. Possible improvements included having a clear format for patient discussion, a session moderator and better internet connectivity. Moreover, a database to assess the impact of the given intervention on the patient cases by evaluating postoperatively (e.g. three months), could further improve clinical care. CONCLUSIONS: Virtual patient discussions subjectively contributed to medical education at both locations. Improved digital infrastructure and a collaborative database could further maximize learning capacity. Furthermore, digital proctoring is a promising way to establish sustainable collaborations between high- and low-resource countries.

Unraveling a Major Burden of Orofacial Clefts Analyses: Classification of Cleft Palate Fistulas by Cleft Surgeons.

OBJECTIVE: The objective of this study was to investigate how cleft surgeons classify palatal fistulas. We focused on three different anatomical locations (ie, hard palate, soft palate, junction hard/soft palate) to analyze agreement/disagreement at various anatomical locations. DESIGN: Cross-sectional survey study. PARTICIPANTS: Participants in an international webinar that focused on palatal fistula treatment were included. INTERVENTION: Participants were presented with a survey pre- and post-webinar. MAIN OUTCOMES: Frequency of used classification systems for classifying oronasal fistulas and the inter-rater reliability of the Pittsburgh classification system. RESULTS: A total of 141 participants completed the questionnaires prior to the webinar and 109 participants completed the survey after the webinar. In total, four classification systems were used (ie, Pittsburgh, Pakistan Comprehensive Fistula Classification [PCFC], anatomical and 'other'). The Pittsburgh classification was the most commonly used system in all cases. However, Pittsburgh inter-rater reliability was low (κ = 0.136 pre-webinar, and κ = 0.174 post-webinar). Surprisingly, a substantial shift was observed from the anatomical to Pittsburgh classification after the webinar, indicating increased awareness of the usability of the Pittsburgh classification system. CONCLUSIONS: This study demonstrates a large heterogeneity with regards to the classification of cleft palate fistulas. Interestingly, a shift was observed from the anatomical to Pittsburgh classification after the webinar. However, the inter-rater reliability for using the Pittsburgh classification was low. Classifying palatal fistulas in a homogenous fashion could enhance comparison of primary palate repair and could improve treatment of palatal fistulas.

Interrater Reliability for Classifying Craniofacial Microsomia Severity: A Call for Objective Evaluation.

OBJECTIVE: The severity of craniofacial microsomia (CFM) is generally classified using the Orbit, Mandible, Ear, Soft tissue, Nerve (OMENS) classification score. The global assessment of the Phenotypic Assessment Tool for Craniofacial Microsomia (PAT-CFM), is a pictorial modification of the OMENS classification. The aim of this study was to assess the interrater reliability of the PAT-CFM global assessment score. DESIGN: In this prospective cohort study, three clinicians completed the global assessment form of the PAT-CFM. The mandible was classified based on orthopantomogram- and/or computed tomography images. PARTICIPANTS: Consecutive patients with CFM or microtia.Interrater agreement was calculated using the weighted Krippendorff alpha (α), with 95% confidence intervals (CI). RESULTS: In total, 53 patients were included (106 hemifaces). The reliabilities of the main classification components ranged from high for the mandible (α = 0.904 [95% CI 0.860-0.948]) and ear (α = 0.958 [95% CI 0.934-0.983]) subscales, to tentative for the orbital summary score (α = 0.682 [0.542-0.821]), and nerve summary score (α = 0.782 [0.666-0.900]) subscales. CONCLUSIONS: The reliability of the ear and radiographic mandible scales of the PAT-CFM global classification were high, while the orbit, facial nerve and soft tissue subscales may have limited reliability. Research focusing on radiographic severity scores for hypoplasia of the orbits and soft tissues, as well as objective measures for overall facial hypoplasia using non-ionizing forms of imaging for early classification, are warranted.

Objective quantitative methods to evaluate microtia reconstruction: A scoping review.

Background: Commonly used methods to evaluate auricles are subjective and are therefore not specific, comprehensive, and precise nor effective in the assessment of microtia reconstruction outcomes. This scoping review aimed to summarize the objective methods for the accurate evaluation of microtia reconstruction. Methods: We performed a scoping review of publications that used objective measurement methods to evaluate outcomes of microtia reconstruction according to the PRISMA-ScR guidelines. A systematic literature search was conducted in the Embase, PubMed, Cochrane, CNKI, and VIP databases, and literature references were screened for additional records. Studies that evaluated auricles after microtia reconstruction using quantitative anthropometric methods were included, and data on these methods were collected. Results: Twenty-five publications reported on quantitative objective outcome measurements. Thirteen studies evaluated auricular protrusion, three articles assessed the position or symmetry, and twelve studies reported on auricle size. The quantitative measurements of fine structures, such as the tragus and concha, were described in three studies. All described measurements used manual landmarking, where fifteen studies described well-defined landmarks, fifteen studies described poorly defined landmarks, and four studies used a combination of well and poorly defined landmarks. Conclusion: The objective evaluation of microtia reconstruction outcomes is hindered by significant heterogeneity of measurement methods. The measurement methods used for general auricular measurements (auricular protrusion, auriculocephalic angle, and size) used in microtia reconstruction were abundant, while measurements of auricular position and the fine structures of the auricle were limited. Three-dimensional imaging combined with computer analyses poses promising future alternatives.

Objective evaluation of nonsurgical treatment of prominent ears: A systematic review.

Background: The prominent ear is a type of congenital ear deformity that can be corrected by a variety of nonsurgical treatments, such as splinting and the taping method. However, there is no objective evaluation method that is universally accepted. The aim of this review is to evaluate objective measurement methods that are used in the available literature to analyze nonsurgical treatment of prominent ears. Methods: A systematic review was performed in the MEDLINE and Embase databases in December 2022 and updated on April 2023 according to Preferred Reporting Items for Systematics and Meta-Analyses (PRISMA) guideline. Any study using objective measurements (continuous variables such as distance and angle) to evaluate the effect of nonsurgical treatment of prominent ears was included. The Joanna Briggs Institute (JBI) critical appraisal for case series was used for quality assessment. Results: A total of 286 studies were screened for eligibility, of which five articles were eligible for inclusion. All of the included studies were case series. The helix mastoid distance (HMD) is the most commonly used parameter to measure treatment outcome. Pinna and cartilage stiffness, length, and width were also used, but without clear statistical relevance. HMD was classified into grading groups (i.e. good, moderate, and poor) to evaluate the treatment's effect. Conclusion: Based on the included studies, objective measurements are rarely used, and when used, they are largely heterogeneous. Although HMD was the most frequent measurement used, all studies used different definitions for the measurement and grouped subsequent outcomes differently. Automated algorithms, based on three-dimensional imaging, could be used for object measurements in the nonsurgical treatment of prominent ears.

Global Prevalence and Burden of Orofacial Clefts: A Systematic Analysis for the Global Burden of Disease Study 2019.

Orofacial clefts, in particular cleft lip and cleft palate, are among the most common congenital anomalies. Despite guidelines recommending early surgical correction, a global backlog of untreated patients persists. This has made orofacial clefts an attractive target for global cleft care initiatives. The most recent global burden of orofacial clefts was estimated to be 529,758.92 disability-adjusted life years (95% uncertainty interval: 362,492.88-798,419.69 disability-adjusted life years), whereas the global prevalence of orofacial clefts was estimated to be 4.6 million (95% uncertainty interval: 3.8-5.7 million). An inverse relationship exists between the Sociodemographic Index and the burden of orofacial clefts. Sub-Saharan Africa, Middle East/North Africa, and South Asia are the regions carrying the most significant burden of orofacial clefts. This manuscript provides updated estimates of the global burden and prevalence of orofacial clefts, acting as a guide to direct future investments, resources, and initiatives from individuals and organizations engaged in global cleft care delivery with the goal of building sustainable cleft care capacity where it is needed the most.

Prognostic Factors for Long-term Aesthetic Outcome of Infantile Haemangioma Treated with Beta-blockers.

Parents of infants treated with beta-blockers for infantile haemangioma are often concerned about the long-term aesthetic outcome. This cross-sectional study assessed the influence on the long-term aesthetic outcome of characteristics of the infantile haemangioma, the beta-blocker treatment, and the infant. The study included 103 children aged 6-12 years, treated with beta-blockers (propranolol or atenolol) for infantile haemangioma during infancy (age at treatment initiation ≤1 year) for ≥6 months. Dermatologists and parents scored the Patient Observer Scar Assessment Scale, and the child scored a visual analogue scale. Dermatologists identified whether telangiectasia, fibrofatty tissue, and atrophic scar tissue were present. The long-term aesthetic outcome of infantile haemangioma was judged more negatively by dermatologists and parents in case of a superficial component, ulceration, older age at treatment initiation, higher cumulative dose, and/or shorter follow-up time. According to children, infantile haemangioma located on the head had better aesthetic outcome than infantile haemangioma located elsewhere. Close monitoring, particularly of infantile haemangioma with a superficial component, is essential for early initiation of treatment, and to prevent or treat ulceration. These outcome data can support parental counselling and guide treatment strategy.

Healthcare professionals' views on shared decision-making in plastic surgery in the Netherlands.

BACKGROUND: In shared decision-making (SDM), patients and healthcare professionals (HCPs) reach a joint clinical decision based on the best available evidence and the patient's preferences. SDM seems particularly valuable in plastic surgery, as often multiple treatment options are available. This cross-sectional online survey study aimed to assess HCPs' views and knowledge about SDM, identify facilitators and barriers of SDM, and determine specific requirements for SDM within plastic surgery. METHODS: Participants were HCPs working in plastic surgery in the Netherlands. Participant characteristics, SDM knowledge, perceived facilitators and barriers, and requirements were assessed using a custom-made online survey. Two researchers thematically analyzed qualitative data. RESULTS: We received 124 responses (with a response rate of 23%). Most respondents were attending plastic surgeons (79%), and 60% had more than 10 years of experience. Almost all respondents considered SDM important (91%), and most (78%) indicated that they applied SDM during consultations. However, only 15% of the HCPs showed a comprehensive understanding of the principle of SDM. Sufficient time, available sources of information (on treatment options and SDM), and decision support tools were identified as important requirements for SDM. CONCLUSIONS: Despite the positive attitudes toward SDM, there is a clear need for SDM training of HCPs, uniform sources of information and guidelines, and improved awareness and availability of decision support tools. National plastic surgery societies can play a crucial role in improving SDM-related knowledge, the availability of information and decision support tools, and the implementation of SDM in the field of plastic surgery.

Oncological status is not a determinant of refraining from breast reconstruction among 490 candidates for mastectomy and post-mastectomy radiotherapy.

BACKGROUND: Although breast reconstruction in the setting of post-mastectomy radiotherapy (PMRT) is controversial, we offer nipple-sparing mastectomy and immediate implant-based breast reconstruction ([N]SSM/IIBR) to women needing primary mastectomy regardless of PMRT. Nevertheless, some of these women have no reconstruction. PURPOSE: To assess the uptake of breast reconstruction in women who undergo PMRT and the patient characteristics associated with such uptake. Additionally, we assessed the determinants of forgoing breast reconstruction. METHODOLOGY: Demographic, physical and oncological characteristics of women who underwent mastectomy, PMRT and breast reconstruction were compared to the characteristics of those who did not undergo breast reconstruction from 2013 through 2018. As determinants of delaying or refraining from breast reconstruction, we distinguished between an oncological reason, patient's preference, patient's co-morbidity, combined tobacco abuse and obesity and the need for PMRT. RESULTS: 490 women received PMRT. Of these, 396 women (81%) underwent combined [N]SSM/IIBR and PMRT or mastectomy and PMRT with delayed breast reconstruction. Ninety-four additional women (19%) did not undergo breast reconstruction. The latter group differed significantly from those who did in demographic and physical characteristics but not in terms of oncological diagnosis and history. Patient's preference was the single most frequent determinant of not performing either immediate or delayed breast reconstruction among these 94 women. Oncological status was not a major determinant in refraining from reconstruction. CONCLUSION: The significant difference in non-oncological characteristics between the reconstructed and non-reconstructed women confirms the importance of these characteristics in the preference for either reconstruction or non-reconstruction.

Long-term speech outcome in patients with Robin sequence after cleft palate repair and tongue-lip adhesion: A 21-year retrospective analysis.

The purpose of this study was to assess the effect of tongue-lip adhesion (TLA) on the long-term speech and articulation outcomes of patients with Robin sequence (RS) after cleft palate repair. Outcomes were compared to those in patients with RS who required positioning alone and to patients with isolated cleft palate (ICP). All consecutive patients with RS (with or without TLA) versus isolated cleft palate (ICP) who underwent cleft palate repair were retrospectively reviewed. Speech and articulation included all assessments between the age of 3-6 years. Secondary speech operations, velopharyngeal insufficiency (VPI), hypernasality, and articulation errors by cleft-type characteristics (CTC), including 4 categories (passive), non-oral, anterior-oral, and posterior-oral. A total of 41 RS patients and 61 ICP patients underwent repair with sufficient follow-up. Of them, 23 patients underwent a TLA at median age of 12 days. Rates of hypernasality (p = 0.004), secondary speech operations (p = 0.004), and posterior oral CTC (p = 0.042) were higher in RS compared to ICP. Isolated RS had speech outcomes similar to those of ICP; however, syndromic RS patients needed more secondary speech operations compared to isolated RS (p = 0.043). TLA-RS patients did not demonstrate differences in speech outcomes or any CTCs (all p > 0.05) compared to non-TLA-RS patients, except for the anterior oral CTC (74% TLA-RS vs 28% non-TLA-RS, p = 0.005). Within the limitations of the study, it seem that TLA does not affect long-term velopharyngeal function in patients with RS. However, TLA-RS patients demonstrated higher rates of anterior-oral CTC, which might be related to a different positioning of the tongue after TLA. Every effort should be taken to treat patients with RS conservatively instead of with TLA because of this demonstrated a negative effect on one type of articulation error. However, if conservative therapy fails, a TLA is still a valuable adjunct in the treatment of RS, and cleft speech pathologists who treat such patients should be more aware of this phenomenon in order to improve long-term articulation outcomes.

The relation between clinically diagnosed and parent-reported feeding difficulties in children with and without clefts.

A cleft lip and/or palate (CL/P) is one of the most common craniofacial malformations, occurring worldwide in about one in 600-1000 newborn infants. CL/P is known to influence the feeding process negatively, causing feeding difficulties in 25-73% of all children with CL/P. Because there is a risk for serious complications in these children regarding feeding difficulties, there is often a need for intensive medical counseling and treatment. At this moment, adequate diagnosis and measurement remain a challenge and often lead to a delayed referral for professional help. Since parents play a big part in reporting feeding difficulties, it is important to help objectify parents' experiences, as well as the use of a frontline screening instrument for routine check-ups during medical appointments. The aim of this study is to investigate the relationship between parent perspective and standardized observation by medical professionals on feeding difficulties in 60 children with and without clefts at the age of 17 months. We focus on the information from parents and health professionals by comparing the Observation List Spoon Feeding and the Schedule for Oral Motor Assessment with the validated Dutch translation of the Montreal Children's Hospital Feeding Scale.  Conclusion: There is a need for timely and adequate diagnosis and referral when it comes to feeding difficulties in children with CL/P. This study underscores the importance of combining both parental observations and measurements of oral motor skills by healthcare professionals to enable this. What is Known: • Early identification of feeding difficulties can prevent adversely affected growth and development. • Clefts increase the probability of feeding difficulties; however, the diagnostic trajectory is unclear. • The Observation List Spoon Feeding (OSF) and Schedule for Oral Motor Assessment (SOMA) are validated to measure oral motor skills. The Montreal Children's Hospital Feeding Scale Dutch version (MCH-FSD) has been validated for the parental perception of infant feeding difficulties. What is New: • Parents of children with CL/P experience relatively few feeding problems in their child on average. • Oral motor skills for spoon feeding are associated with oral motor skills for solid foods in children with CL/P. • The extent of the cleft is associated with experiencing more feeding difficulties in children with CL/P.

Stakeholders' Views on Information Needed in a Patient Decision Aid for Microtia Reconstruction.

OBJECTIVE: To assess which information about microtia and the possible reconstructive options health care providers (HCPs), patients and parents believe should be included in a patient decision aid (PtDA). DESIGN: A mixed-methods study comprised of an online survey of HCPs and focus group discussions with patients and parents. PARTICIPANTS: Survey respondents were members of the International Society for Auricular Reconstruction (ISAR). Focus group participants were patients with microtia and their parents, recruited through the microtia outpatient clinic at Amsterdam UMC, and through a Dutch patient organization for cleft and craniofacial conditions. METHODS: An online, investigator-made survey was sent to ISAR members in December 2021. Semi-structured focus group discussions were held in February 2022. Quantitative results were summarized, and qualitative results were thematically grouped. RESULTS: Thirty-two HCPs responded to the survey (response rate 41%). Most respondents (n = 24) were plastic surgeons, who had a median of 15 years of experience (IQR: 7-23 years). Two focus groups were held with a total of five patients and two parents. HCPs, patients and parents generally agreed on the information needed in a PtDA, emphasizing the importance of realistic expectation management. Patients and parents also considered psychosocial and functional outcomes, patient experiences, as well as patients' involvement in decision-making important. CONCLUSIONS: A PtDA for microtia reconstruction should target all patients with microtia, and include information on at least technique-related information, expected esthetic results, possible adverse effects, psychosocial and functional outcomes and patient experiences. Preference eliciting questions should be developed for both pediatric patients and their parents.

Decrease in Prevalence of Cleft lip, Alveolus and Palate After Nationwide Introduction of the Second-Trimester Anomaly Scan in the Netherlands.

OBJECTIVE: Some studies have suggested that introducing a second-trimester anomaly scan (SAS) leads to increased rates of termination of pregnancy (TOP) in fetuses with orofacial clefts (OFCs). The aim of this study was to evaluate the impact of a nationwide introduction of SAS on the prevalence of live births with OFCs in the Netherlands. DESIGN: Retrospective cohort study. SETTING: Tertiary setting. POPULATION: Included in the study were all patients diagnosed with OFCs as recorded in the "Dutch Association for Cleft Palate Anomalies" database between 1997 and 2019. INTERVENTIONS: Patients were divided into three categories: cleft lip with or without alveolus (CL/A), cleft lip, alveolus and palate (CLAP) and cleft palate (CP) based on anatomical landmarks at the first consultation. MAIN OUTCOME MEASURES: Prevalence rates of OFCs before and after the nationwide introduction of the SAS on January 1, 2007 were compared. RESULTS: Overall, 1899 patients were diagnosed with CL/A, 2586 with CLAP and 2927 with CP. The prevalence of clefts before and after introduction of the SAS did not differ (P = 0.85). The prevalence of CL/A decreased (P = 0.04), and that of CLAP decreased (P = 0.01) and that of CP increased (P = 0.02). CONCLUSIONS: This study demonstrates a significant decrease in the prevalence of CL/A and CLAP after introduction of the SAS. However, due to an increase in CP, the prevalence of all patients born with OFCs has not changed in the Netherlands between 1997 and 2019.

Differences in analysis and treatment of upper airway obstruction in Robin sequence across different countries in Europe.

The goal of this study was to explore the availability of diagnostic and treatment options for managing upper airway obstruction (UAO) in infants with Robin Sequence (RS) in Europe. Countries were divided in lower- (LHECs, i.e., PPP per capita < $4000) and higher-health expenditure countries (HHECs, i.e., PPP per capita ≥ $4000). An online survey was sent to European healthcare professionals who treat RS. The survey was designed to determine the availability of diagnostic tools such as arterial blood gas analysis (ABG), pulse oximetry, CO2 analysis, polysomnography (PSG), and sleep questionnaires, as well as to identify the used treatment options in a specific center. Responses were received from professionals of 85 centers, originating from 31 different countries. It was equally challenging to provide care for infants with RS in both LHECs and HHECs (3.67/10 versus 2.65/10, p = 0.45). Furthermore, in the LHECs, there was less access to ABG (85% versus 98%, p = 0.03), CO2 analysis (45% versus 70%, p = 0.03), and PSG (54% versus 93%, p < 0.01). There were no significant differences in the accessibility concerning pulse oximetry, sleep questionnaires, home saturation monitoring, nasopharyngeal tubes, Tuebingen plates, and mandibular distraction.    Conclusion: This study demonstrates a large difference in available care for infants with RS throughout Europe. LHECs have less access to diagnostic tools in RS when compared to HHECs. There is, however, no difference in the availability of treatment modalities between LHECs and HHECs. What is Known: • Patients with Robin sequence (RS) require complex and multidisciplinary care. They can present with moderate to severe upper airway obstruction (UAO). There exists a large variety in the use of diagnostics for both UAO treatment indications and evaluations. In most cases, conservative management of UAO in RS is sufficient. Patients with UAO that persist despite conservative management ultimately need surgical intervention. To determine which intervention is best suitable for the individual RS patient, the level of UAO needs to be determined through diagnostic testing. • There is a substantial variation among institutions across Europe for both diagnostics and treatment options in UAO. A standardized, internationally accepted protocol for the assessment and management of UAO in RS could guide healthcare professionals in the timing of assessment and indications to prevent escalation of UAO. Creating such a protocol might be a challenge, as there are large financial differences between countries in Europe (e.g., health expenditure per capita in purchasing power parity in international dollars ranges from $600 to over $8500). What is New: • There is a substantial variation in the availability of objective diagnostic tools between European countries. Arterial blood gas analysis, CO2 analysis and polysomnography are not equally accessible for lower-healthcare expenditure countries (LHECs) compared to higher-healthcare expenditure countries (HHECs). These differences are not only limited to availability; there is also a difference in quality of these diagnostic tools. Surprisingly, there is no difference in access to treatment tools between LHECs and HHECs. • There is national heterogeneity in access to tools for diagnosis and treatment of RS, which suggests centralization of health care, showing that specialized care is only available in tertiary centers. By centralization of care for RS infants, diagnostics and treatment can be optimized in the best possible way to create a uniform European protocol and ultimately equal care across Europe. Learning what is necessary for adequate monitoring could lead to better allocation of resources, which is especially important in a low-resource setting.