A Systematic Review of the Efficacy of Haemostatic Interventions in Primary Cleft Palate Repair.

OBJECTIVE: To evaluate the effectiveness of adrenaline infiltration, topical adrenaline, systemic tranexamic acid, fibrin tissue sealants and alginate-based topical coagulants at reducing blood loss and post-operative bleeding in primary cleft palate repair. DESIGN: Systematic review according to PRISMA-P guidelines, using Covidence systematic review software to facilitate 3-stage screening and data extraction by two reviewers. SETTING: Academic cleft surgery center. INTERVENTIONS: Any peri-operative intervention to reduce intra-operative and post-operative bleeding. MAIN OUTCOME MEASURES: Estimated blood loss, rate of post-operative bleeding, rate of return to theatre for haemostasis. RESULTS: Sixteen relevant studies were identified, with a total of 1469 study participants. Nine studies examined efficacy of infiltrating vasoconstrictors and all concluded that 1:100,000-1:400,000 adrenaline infiltration reduced intra-operative blood loss, to the range of 12-60 ml. Secondary bleeding and re-operation for haemostasis were uncommon. Tranexamic acid was studied in five randomised controlled trials, two of which demonstrated a significant reduction in blood loss compared to a control group. Use of fibrin and gelatin sponge products was examined in 3 studies, all of which reported no or minimal bleeding, but did not have quantifiable outcome measures. CONCLUSIONS: Infiltration with vasoconstricting agents, administration of systemic tranexamic acid and application of fibrin sealants have a well-studied and favorable safety profile in pediatric cases, and likely contribute to the relatively low incidence of post-operative bleeding and intra-operative blood loss in primary cleft palate repair.

Unusual Stroke: Left Ventricular Non-Compaction Causing an Embolic Cerebrovascular Accident

Background Left Ventricular Non-Compaction Cardiomyopathy (LVNC) is a rare genetic, developmental disorder when the left apical chamber of the heart contains bundles or pieces of muscle that extend into the chamber called trabeculations. These trabeculations are a sponge-like network of muscle fibers that typically become compacted to transform heart muscle to become smooth and solid during a normal development process. Those who have LVNC most commonly are asymptomatic. Those who are symptomatic present with syncope, palpitations, dizziness, dyspnea, fatigue and/or unexplained weight gain or swelling. LVNC has also been suggested as a rare cause of embolic stroke, in our patient's case, "due to sluggish blood flow in deep intertrabecular recesses." Case We present a 29 year old African American female, G2P0011, with a history of cleft palate repair, and recent pregnancy complicated by COVID-19 who reported to ED after having a fall the day before, leg weakness and numbness, unable to walk, headache and a left facial droop on day of admission. No family history of SCD or other cardiac disease was noted. On assessment, was found to have NIHSS of 7 with rate lateral gaze palsy, left facial palsy, and decreased strength and sensation of LUE and LLE. TPA was not given due to being outside the therapeutic window. CT head and MRI brain were consistent with acute right MCA stroke. Secondary stroke workup with TTE revealed reduced LVEF 15-20%, loosely arranged myocardium with suspected LVNC and RV apical thrombus. Cardiac MRI showed increased trabeculations consistent with LVNC. Decision-making Currently, there are no ACC/AHA guidelines on anticoagulation in the setting of LVNC. Cardiology and Neurology had an extensive multidisciplinary discussion on the need for anticoagulation specifically with Warfarin. The patient was educated extensively on the need for medical adherence with anticoagulation and guideline directed medical therapy. Conclusion The patient was started on guideline directed medical therapy for cardiomyopathy and was started on Warfarin after bridging from Lovenox. She continued with physical therapy and was noted to have improvement in residual deficits at her outpatient follow up.Copyright © 2023 American College of Cardiology Foundation

Left Ventricular Non-Compaction: An Uncommon Cause of Cardoembolic Stroke

Introduction: Left ventricular non-compaction (LVNC) is a rare, usually congenital or hereditary cardiomyopathy. The formation of deep trabecular recesses increases the likelihood of a thrombus entering the systemic vasculature which may cause cardioembolic stroke. Clinical manifestations of LVNC range from asymptomatic to severe heart failure, thromboembolic events, and sudden death. In adults, individuals with LVNC have a 21 - 38% chance of developing a cardioembolic stroke. Case Presentation: We present a case of a 29-year-old African American female, six weeks postpartum with a history of cleft palate, right sixth nerve palsy, and recent Covid-19 infection who developed stroke-like symptoms of left side numbness and weakness, left side facial droop and slurred speech upon awakening. On initial examination, the patient displayed 4/5 strength and decreased sensation to pinprick in left upper and lower extremities as well as left lower facial palsy and dysarthria. CT head without contrast revealed a large infarct in the right middle cerebral artery (MCA) territory. A brain MRI confirmed the right MCA infarct but did not reveal acute infarct in other vascular territory. CTA head/neck reported right MCA proximal M2 segment occlusion. During admission, stroke work-up included a normal EKG. A transthoracic echocardiography (TTE) with bubble study was obtained which reported no patent foramen ovale but with severely reduced systolic function with a LVEF estimated 15-20%;questionable right ventricle apical thrombus was also suspected. Cardiology was consulted for acute systolic heart failure with plans to initiate goal directed medical therapy. A cardiac MRI was recommended which revealed left ventricle increased trabeculations meeting MRI criteria for LVNC, but no cardiac thrombus was detected. Patient was started on warfarin with enoxaparin bridging for secondary stroke prevention. Discussion(s): Given our case's young age of onset of stroke and the history of postpartum and recent COVID-19 infection, cardioembolic stroke due to other more common etiologies such as hypercoagulable disorders, postpartum cardiomyopathy or COVID-19 associated cardiomyopathy were initially suspected based on the initial TTE findings. Subsequent cardiac MRI revealed LVNC as an unusual cause of her cardioembolic stroke. This case illustrates the importance of thoroughly investigating the potential managementchanging causes of stroke, including congenital structural cardiac abnormalities, in patients with young age of onset.

Anesthetic Approach and Perioperative Complications in Cleft Lip/Palate Surgery: A Single Center Retrospective Study

Objective: The objective of this study was to determine factors that may affect anesthesia and surgical complications, difficult airway, and the need for intensive care unit (ICU) care in cleft lip and cleft palate (CLCP) surgeries. Materials and Methods: The study was a retrospective review of the records of 617 patients who underwent CLCP surgery between 2015–2019. Results: The number of anesthesia complications was higher in patients with difficult mask ventilation. Surgical complications were more common in patients >1 year of age. Isolated cleft palate (CP) surgery;presence of a concomitant disease, syndrome, or micrognathia;age >1 year;and the CP subtype were associated with a higher rate of difficult intubation. Isolated cleft palate, concomitant disease, syndrome, micrognathia, difficult intubation, difficult mask ventilation, and anesthesia complications were associated with ICU admission. Conclusion: The CP subtype was associated with a higher rate of difficult intubation and ICU hospitalization even in patients who were nonsyndromic and/or >1 year of age. Therefore, special attention should be paid to the anesthesia and surgical management of these patients.

Evaluating Efficacy, Global Impact, & Trainee Perception of International Speech Therapy Trainings

Background/Purpose: This presentation highlights the benefits and challenges of implementing speech therapy trainings utilizing a train-the-trainer model. Children born with CL+/-P need comprehensive care from a cleft team to ensure that they can meet their highest potential and engage in all aspects of life in their communities. However, in these areas it can be difficult to locate health professionals with the capacity to address cleft palate speech characteristics. As a result, many children speak with these characteristic speech patterns post-palate repair. Research has shown that in low- and middle-income countries (LMICs) a partner hospital model improves the quantity and quality of cleft care services (Purnell, McGrath, & Gosain, 2015). However, there are varying models as to how to structure and build these programs once a partner hospital is established. Limited research has been conducted focusing on the efficacy of speech therapy trainings intended to provide a sustainable impact on these communities. Methods/Description: This poster shares comprehensive data from a survey distributed to training attendees from the past 5 years. Trainings were organized by Smile Train and facilitated by local health professionals who went on to become future trainers. The survey was distributed in August 2020 and consisted of three sections: Personal & Demographic information, Global Impact & Outreach, and Speech Training Feedback. The survey elicited a group of respondents (n=55) who each attended one or more of 14 trainings over a five year span (2015-2020). Authors collected demographic data of respondents, measured respondents' self perception of competency in this area before and after speech therapy trainings, and gathered trainee feedback for future trainings. Results: Surveys were analyzed via SurveyMonkey platform. Researchers analyzed trainee's multiple choice selections, scaled scores, and open-ended responses. Results indicated that trainee confidence in treating those with speech conditions resulting from a CL +/-P significantly increased after the training (on average, trainee confidence was scored a 4/10 before training and improved to 9/10 after training). Further, 56% of trainees have now begun to train other health professionals in their communities. Constructive feedback included the need for continuing education after trainings concluded and the need to address the lack of resources/funding in home countries. Finally, trainees report difficulties in implementing tools acquired from the trainings as a result of the COVID-19 pandemic. Conclusions: Utilization of a train-the-trainer model for speech trainings can provide a lasting, sustainable impact for LIMCs based on respondent feedback. Continued survey distribution is imperative to continuously evaluate the efficacy of these trainings. Further discussion is also needed to determine how trainings can effectively provide tools and education within the constraints of the COVID-19 pandemic.

COVID Cleft: A national evaluation of cleft care operating through the COVID-19 Pandemic in the United Kingdom

Background/Purpose: COVID-19 fundamentally changed cleft teams' ability to care for their patients. This study aims to study;1) the effect of COVID-19 on elective surgery timings and outcomes;2) preoperative screening and isolation protocols;3) the impact of operating with personal protective equipment (PPE). Methods/Description: Between the start of the first UK lockdown in March 2020 and April 2021 operative details from 651 cleft procedures performed in eight UK centres were entered into a secure REDCap database. Results: 651 records were entered (59% male, 41% female). 9% patients had a known syndrome. Operations were as follows: cleft palate repair (40%), unilateral cleft lip repair +/- vomer flap (23%), alveolar bone grafting (16%), secondary speech surgery (10%), fistula repair (3.7%), lip revision (1%) and rhinoplasty (1%). 39% of surgical cases were deemed delayed compared to normal protocol timings, with 80% of the delays attributable to COVID. Mean age at initial cleft lip repair was 230 days exceeding a previous representative mean of 137 days as well as breaching the UK national standards for upper age limit of 183 days. Mean age at cleft palate repair was 387 days compared to the UK national standard for upper age limit of 396 days, and previous representative mean of 320 days. 81% of patients undertook some form of pre-operative isolation;47% isolated for two weeks. COVID screening was performed in the 72 hrs prior to surgery in 89% of patients and 13% of parents/carers. Only one patient had a positive test. 69% surgeons wore an FFP3 (N99) mask to operate, and 64% of cases involved difficulty during the operation as a result of the PPE;most commonly communication difficulties (45%). No patients developed COVID in the early post-operative period. Conclusions: This data demonstrates that initial cleft lip and palate repair in the UK has been delayed as a direct result of the COVID-19 pandemic. Secondary surgery has been significantly affected and efforts will need to be made at national level to provide capacity to catch up. Isolation and testing protocols for COVID-19 vary from unit to unit, but appear safe. Routine cleft surgery can safely continuing through the pandemic, as long as appropriate infection control measures are followed and resources allow.

Health-related Quality of Life in Pediatrics with Craniofacial Conditions during COVID-19: Comparison to a Matched Pre-pandemic Sample

Background/Purpose: Since the outbreak of COVID-19 was declared a pandemic, there has been concern regarding the social isolation born from safety mandates. Literature suggests that social distancing guidelines provoke anxiety and uncertainty among children and adolescents. This study aims to evaluate the effects of pandemic guidelines on pediatric patients with craniofacial conditions (CFCs), by studying the Health-Related Quality of Life (HRQoL) in a matched sample of pediatric patients with CFCs prior to and during the COVID-19 pandemic. Methods/Description: This matched cohort study (N=88) utilized the Craniofacial Quality of Life Scale (CFC-QoL), a bilingual patient- and parent-reported outcome measure, to assess social and psychological HRQoL domains. CFC-QoL surveys were collected from CFC patients and parents prior to March 11, 2020 (pre-pandemic) and compared to a separate cohort that completed the survey after July 1, 2020, during the pandemic. The patients in each sample (n = 44) were matched by diagnosis (bilateral or unilateral cleft lip and palate, craniosynostosis, microtia or dermatologic condition), age range (7-12, 13-17, 18+) and gender. Mean scores for both patient and parent responses to each survey item within the social and psychological impact subscales were computed and compared between the matched cohorts. Results: Comparison of mean responses found significant differences between the pre-pandemic and pandemic cohorts. On the social impact subscale, patients in the pandemic sample reported that 'People tease me', 'People ask me what is wrong with my face', and 'People notice that my face is different' less often than those in the pre-pandemic sample (p = .021;p = .041;p = .038). Parents in the pandemic sample reported 'People ask my child what is wrong with their face' and 'People notice my child's face is different' less often compared to the pre-pandemic sample (p = .040;p = .020). Within the psychological subscale, patients in the pandemic sample reported feeling less worried about their future (p = .030), less anxious or scared (p = .024), and more likely to try new things (p = .039) compared to those who answered the survey pre-pandemic. The parents in the pandemic sample reported 'My child feels bad about himself/herself' less often compared to parents in the prepandemic sample (p = .038). Conclusions: This study found that a sample of patients with CFCs and their parents reported better indications of social and psychological HRQoL during the COVID-19 pandemic compared to a matched pre-pandemic cohort. Factors that might influence these findings include decreased negative social impact due to containment measures such as school closures. Additionally, enforced mask wearing in social settings may serve as a protective barrier to decrease negative attention drawn toward a child's facial differences.

NUTRITIONAL MONITORING THROUGH THE WHATSAPP® APPLICATION: A POST-COVID-19 PANDEMIC INNOVATION

Background/Purpose: The Covid-19 pandemic has restricted in person care at our cleft treatment center, delayed patient turnout, and led to financial losses and an increased number of missed scheduled appointments. Thus, we have sought new ways to monitor the nutritional status of infant patients with clefts, by using the WhatsApp® application, so as to conduct timely nutritional interventions and to prevent malnutrition and anemia. The purpose was to monitor the nutritional status of newborn babies with cleft, using the WhatsApp® application, and to validate its use. Methods/Description: 71% of internet users in Brazil use WhatsApp® to send and receive messages. This application is easy to use, with privacy ensured by cryptography. This research project was approved by the Institutional Review Board. The protocol established: 1) new cases of infants up to 30 days old, with cleft lip and/or cleft palate;2) consent form signed by parent or guardian;3) the first appointment was in person;with mothers receiving training and guidance to send information about their children's height and weight every month;4) data comparisons were conducted against WHO benchmarks (WAZ score between 0 and - 1 = healthy weight;between - 1 and - 2 = nutritional risk;< - 2 = underweight);6) nutritional interventions considering RDI's. Results: 18 patients were followed up by regular data submission through the WhatsApp® application;8 had cleft lips, 3 cleft palates, and 7 cleft lip and palates. Among these patients, 1 had a gastrostomy tube, 2 had cow milk protein allergy, and 1 had cardiac disease. Thus far, 8 have already been submitted to lip surgery, and 10 are waiting to reach the appropriate age for surgery. Twelve were classified as coming from low-income families, and 6 from medium income families. Most of them (no. = 17) travelled between 100-500 km to receive care, and 1 more than 100 km. Eight received transportation and food subsidized by public funds, and 10 used their own funds. Thirteen children were fed breast milk for 120 days, 8 were breast fed directly and 5 were fed pumped breast milk;5 were fed formula. Nine began eating additional foods at 6 months, the remaining patients were not old enough for that yet. Eleven children had normal weight (eutrophic) since the onset, 3 were malnourished when the study began and recovered after about 54 days, 4 were at risk when the study began and recovered after about 26.5 days. No patient had to delay surgery because of anemia (mean Hb = 11.45g/ dL). Nutritional interventions included encouraging breast feeding and different breastfeeding positions, guidance regarding the appropriate concentration of infant formula and how /when to introduce of additional foods. Milk formula, bottles, scales, and measuring tapes were donated. Conclusions: The use of the WhatsApp® application for nutritional follow up proved to be a valid and effective way tomonitor the patients' growth and development, yielding efficient nutritional recovery.

Application of Safety Protocols for International Cleft Outreach Programs During the COVID-19 Pandemic: Global Smile Foundation Precaution Guidelines and Implications

Background/Purpose: Since COVID-19 was declared a worldwide pandemic by the World Health Organization (WHO) in March of 2020, foundation-based cleft outreach programs to Low- and Middle-Income Countries (LMICs) were halted considering global public health challenges, scarcity of capacity and resources, and travel restrictions. This led to an increase in the backlog of untreated patients with cleft lip and/or palate, with new challenges to providing comprehensive care in those regions. Resumption of international outreach programs requires an updated course of action to incorporate necessary safety measures in the face of the ongoing pandemic. In this manuscript, we outline safety protocols, guidelines, and recommendations implemented in Global Smile Foundation's (GSF) most recent outreach trip to Beirut, Lebanon. Methods/Description: COVID-19 safety protocols for outreach cleft care and an Action Response Plan were developed by the GSF team based on the published literature and recommendations from leading international organizations. Results: GSF conducted a 1-week surgical outreach program in Beirut, Lebanon, performing 13 primary cleft lip repairs, 7 cleft palate repairs, and 1 alveolar bone grafting procedure. Safety protocols were implemented at all stages of the outreach program, including patient pre-selection and education, hospital admission and screening, intraoperative care, and post-operative monitoring and follow-up. Conclusions: Organizing outreach programs in the setting of infectious diseases outbreaks should prioritize the safety and welfare of patients and team members within the program's local community. The COVID-19 protocols and guidelines described may represent a reproducible framework for planning future similar outreach initiatives in high risk conditions.

eP455: Fetal ultrasound presentation and neonatal diagnosis of Freeman-Sheldon syndrome in son of previously undiagnosed adult male

Background: Freeman-Sheldon syndrome [distal arthrogryposis type 2A (OMIM #193700), DA2A, Freeman-Burian syndrome] is a rare autosomal dominant multiple pterygium syndrome caused by alterations in MYH3. The phenotypic features, particularly of the face, are distinct and easily recognizable, and the diagnosis can be confirmed with molecular gene analysis. Fetal ultrasound imaging may provide important diagnostic clues to facilitate the diagnostic process. Informed consent and parental permission were provided by the parents. Case presentation: The infant’s mother presented for a Maternal Fetal Medicine genetic counseling telehealth appointment (due to COVID-19 pandemic restrictions) as a G7P2132, 32-year old female who had insulin-dependent diabetes and thrombocytosis. Her partner was a 24-year old male with a history of hearing loss, a V-shaped palate, and a lower lip cleft. Gestational age was 14 4/7 weeks and the indications were: increased nuchal translucency, paternal complex medical history, maternal G6PD heterozygote, and recurrent pregnancy loss. During the genetic counseling session, the following were addressed: 1) Maternal heterozygote status for G6PD indicated that if the fetus was male, there was a 50% chance he would be affected with G6PD-deficiency;2) Increased nuchal translucency on fetal ultrasound (US) with measurement at 98th percentile is associated with an increased risk of chromosomal abnormalities, microdeletion/duplications, and Noonan syndrome. The patient reportedly had low risk cell-free DNA but results were not available to the counselor at the time of consult. The option for additional genetic screening and diagnostic testing was declined;3) Three first trimester pregnancy losses with the father of this baby (FOB) were addressed, and parents deferred chromosome analyses at the time;4) Mother shared FOB’s complex history of bilateral sensorineural hearing loss, V-shaped cleft palate, lower lip cleft, and micrognathia. However, father was not present during the telehealth encounter. Mother was counseled regarding the possibility of an autosomal dominant condition with the potential risk to the pregnancy of up to 50%. It was recommended that the FOB have a clinical genetics evaluation, which could potentially provide a specific diagnosis and inform recurrence risk and management guidance. Follow-up MFM genetic counseling telephone visit occurred with the mother at 31 6/7 weeks gestation due to multiple congenital anomalies evident on fetal ultrasound. A 25 week fetal ultrasound revealed hypotelorism and a thickened nuchal translucency. A repeat study at 29 weeks revealed a V-shaped palate with a possible cleft, micrognathia, and midline mandibular cleft. FOB’s history was revisited. It was determined that he had 3 previous “no shows” to Genetics clinic appointments and did not pursue evaluation after the last counseling appointment. Again, it was emphasized that in order to best make a diagnosis for the family, an affected person would need to undergo a thorough evaluation, including medical and family history review, physical examination, and any indicated genetic testing. The parents were comfortable with the likelihood that the baby had the same condition as the father, but variable expressivity and broad range pf phenotypic presentation were explained. Recommendations for postnatal evaluation of the infant and pertinent genetic testing were provided. Consultative Genetics evaluation of the infant at 2 days of age revealed a short, broad forehead with supraorbital fullness leading to a horizontal brow indentation;mask-like facial appearance;hypotelorism;very deep set eyes with blepharophimosis;deep, creased nasal bridge;small, upturned nose with hypoplastic alae and narrow nares;microstomia with pursed lips;glossoptosis;micrognathia;2 deep vertical chin creases;short neck with excess nuchal skin;inverted and wide spaced nipples;clenched hands with 5th digits overlying 4th and 2nd overlying 3rd, bilaterally;bilateral vertical talus;2nd toes longer and overlying rd toes;clinodactyly of 4th and 5th toes bilaterally;and deep gluteal crease with no visible sinus. There were no evident contractures. The father has a complex history with no medical assessments prior to age 18. He reported that he did “not look like anyone else” in his family. He has a diagnosis of autistic spectrum disorder, a submucous cleft, vision issues, hearing loss necessitating a hearing aid on the left, and a history of cholesteatomas and of mastoidectomy. On brief examination, he had a mask-like face, blepharophimosis, left microphthalmia, left esotropia, narrowing of his midface, deep vertical crease on the mandibular region, microstomia, broad great toes, single flexor creases on the thumbs, and contracture of right thumb. Maxillofacial CT of the infant revealed hypoplastic mandibular body, ramus, and condyles bilaterally with micrognathia and retrognathia;hypoplastic maxilla bilaterally;and enophthalmos with retracted appearance of globes in the bony orbits bilaterally. Multiple facial bone abnormalities were seen, including microsomia, micrognathia, retrognathia, orbital hypotelorism and enophthalmos Genetic testing was performed via a custom Whole Exome Slice at GeneDx laboratories and included the MYH3 and TNNI2 genes. Results revealed a heterozygous pathogenic change in MYH3 (c.2015 G>A;p. R6724) consistent with the diagnosis of Freeman-Sheldon syndrome. Conclusion: The presentation of “midline mandibular cleft” on fetal ultrasound was the most specific prenatal finding. This is a very rare fetal finding. Thus, it should prompt further evaluation to assess for true clefting versus ridging or creasing. Additionally, targeted assessment for other findings or clinical clues for Freeman-Sheldon syndrome, such as contractures, “windmill vane” hand, and mouth size, could aid in the differential diagnosis considerations and the diagnostic process. Admittedly, these are position and quality dependent, and are challenging to assess even in ideal situations. The phenotype of the father was immediately recognizable. However, due to COVID-19 pandemic restrictions, prior to the infant’s birth, only telehealth visits were conducted and the father’s participation was by telephone. This limited the ability to narrow the differential diagnosis without visualization of his distinct phenotypic features. Finally, missed opportunities to diagnose the father prior to this pregnancy occurred. Many clinics send “no show” letters to referring providers and patients, as we do. Emphasizing the importance of diagnosis prior to pregnancy for individuals concerned about having a genetic disorder should be considered as part of the information shared in these letters.

6th International Congress of Medical Genetics

The proceedings contain 108 papers. The topics discussed include: toll-like receptor 3 c.1377C/T and -7C/A polymorphisms in COVID-19 infection;a cleft palate with 49, XXXXY karyotype: A case report;a case with atypical autism and hereditary motor sensory neuropathy;investigation of genetic etiology in gastrointestinal cancer patients with next generation sequencing method;a rare disease associated with the CDK13 gene: CHHDFIDD;a case report of pericentric inversion, inv (21) (p12;q22) in repeated pregnancy loss;a case of rare CYP26B1-related craniosynostosis in a Turkish female patient;a novel homozygous mutation in CYP11A1 gene in 46, XX patient with P450scc deficiency;and a novel homozygous variant in SUOX gene causes classic isolated sulfite oxidase deficiency: a case report.

Speech-Language Pathology Provision During the COVID-19 Pandemic for Children Born With Cleft Palate in the United Kingdom--Parent/Caregiver Perspectives and Experiences

Purpose: The purpose of this study was to examine parent/caregiver perspectives and experiences of speech-language pathology provision during the COVID-19 pandemic for children born with cleft palate. Method: An online questionnaire to parents of children born with cleft palate asked about delays and changes to speech-language pathology provision during the first U.K. national lockdown. Parents were also asked their views on the effectiveness of online speech-language pathology provision. Analysis considered variation in speech-language pathology provision by region. Chi-square and Mann-- Whitney U tests examined associations between speech-language pathology provision and socioeconomic status and child age. Free text responses were analyzed using qualitative content analysis. Results: Three hundred fifty-six (39.3%) children were receiving speechlanguage pathology intervention before the first national lockdown. A further 49 (9.0%) were due to start speech-language pathology intervention during the lockdown. Speech-language pathology provision varied both nationally and within smaller geographical regions. Overall, 146 (42.6%) children continued to receive speech-language pathology and 197 (57.4%) had intervention delayed. There was no association between delayed speech-language pathology and socioeconomic status. Older children were more likely to experience delayed speech-language pathology provision (p = .004). Qualitative analysis revealed concerns about access to speech-language pathology, challenges with adequate devices to access online provision, technological problems, and child engagement in online provision. Parents reported online provision as being "better than nothing." Conclusions: Parents/caregivers reported delays to speech-language pathology provision during the first lockdown, but this varied geographically and was more prevalent for older children. Concerns about access to speech-language pathology provision were raised, including challenges regarding online provision. Follow-on work will consider the impact of the delays experienced on longer term outcomes.

The impact of COVID-19 on cleft services in Great Britain & Northern Ireland.

INTRODUCTION: Cleft lip and/or palate is the most common craniofacial anomaly and occurs in 1 in 650 to 700 live births in the United Kingdom (UK). The majority of cleft surgery is elective, and as a result, almost all cleft surgery was suspended across the UK in March 2020 during the first national lockdown. The UK has centralised regional Cleft Services which all use the same agreed target-age standards for primary surgery including lip and palate repairs. The coronavirus disease-2019 (COVID-19) response has caused a delay in carrying out procedures. The severity of this delay depends on the impact of COVID-19 on local trusts and R-value within that region. As the country goes through the second and third wave, the impact could be long lasting, and we aimed to quantify it so that the data could be used to guide service prioritisation in the NHS and help future workforce planning. METHODS: An online survey was designed based on the cleft quality dashboard indicators and circulated nationally to all nine cleft regions in the UK. The survey was divided into three main headings: • Duration of suspended cleft services • Quantification of the impact on delayed in surgery/services • Changes needed to restart surgery/services RESULTS: We obtained a 60% response rate with five completed surveys from five out of nine regions. All regions reported that they suspended their cleft services in March 2020 around the time of the first wave and the first national lockdown. There has been an impact on delayed surgical and clinical interventions for cleft patients. Regions were affected differently with some on an exponential waiting list growth projection, whereas other teams are on track to recover from the backlog within 7-22 weeks. There has been an impact on the allied health professionals' services within the cleft multidisciplinary team. The cleft nurses' 24-h reviews, Speech And Language Therapy (SALT), and psychology maintained service delivery in some format. Patient-facing services such as audiology and dentistry were significantly disrupted and continue to experience delays due to reduced capacity. CONCLUSIONS: Various regions have seen a varied impact from COVID-19 on their services, from all cleft regions there seems to be an impact on achieving surgery within the national target age. The adverse effect of the COVID-19 impact is unlikely to be known for a few years to come; however, the data are a useful guide when supporting the allocation of resources within the healthcare setting. A prospective long-term study is required to assess the impact of COVID-19 on cleft surgery, follow-up, assess access to allied health professional MDT clinics, and long-term complications.

Telemedicine in International Cleft Care: A Systematic Review.

BACKGROUND: Cleft lip and cleft palate (CLP) are among the world's most common congenital malformation and has a higher prevalence in developing nations due to environmental and genetic factors. Global efforts have been developed in order to prevent and treat the malformation. Telemedicine has been implemented in various humanitarian global missions with success and is currently the primary means of care due to COVID-19. OBJECTIVE: To assess the benefits and barriers of telehealth in the care of patients with CLP through a global approach. METHODS: Systematic review of the PubMed and Cochrane Review databases with relevant terms related to telemedicine in cleft lip and palate surgery. RESULTS: Eight articles fit the inclusion criteria and suggested benefits with the use of telemedicine in regard to education, preoperative, and postoperative care as well as increased access to underserved populations. Barriers included connectivity and accessibility concerns. CONCLUSION: Telehealth is a beneficial way to evaluate patients with CLP in developing countries with proper care and follow-up to reduce complications and to improve health outcomes.

Running a safe and effective Cleft Network Service in response to the COVID-19 pandemic.

Maintaining an excellent level of service in a network Cleft Lip and Palate service in the UK has been an added challenge for both clinicians and patients throughout the COVID-19 pandemic. We describe the changes to our service, and report a high level of patient satisfaction with the changes. Some of the enforced changes may last beyond the duration of this pandemic.

Speech pathology telepractice intervention during the COVID-19 pandemic for Spanish-speaking children with cleft palate: A systematic review.

BACKGROUND: Due to the lockdown and quarantines caused by the COVID-19 pandemic, the need to study and use telepractice for providing speech pathology interventions for children with cleft palate has arisen. OBJECTIVE: To carry out a systematic review of the use of telepractice during the COVID-19 pandemic for providing speech pathology interventions for Spanish-speaking children with cleft palate. METHODS: In July and August 2020, the authors searched the electronic databases Medline, LILACS, SciELO, and the Cochrane Library using the following keywords in English (MeSH): Cleft palate combined with Early intervention, Speech therapy, Rehabilitation of speech and language disorders, Speech production measurement, Speech articulation tests and Telemedicine. Original articles were selected and analyzed, complemented by an analysis of flowcharts and recommendations by the GES Clinical Guide of Cleft Lip and Palate of Chile's Government and the authors' expert opinions. RESULTS: A total of 2680 articles were retrieved, of which 23 were critically analyzed and used to adapt the early stimulation, evaluation, and treatment of children with CP to speech therapy telepractice at the Gantz Foundation, a Hospital in Santiago de Chile. LIMITATIONS: Only three researchers carried out a quick review, which limited the depth of individual analysis of the studies included. Also, the suggestions and material presented should be evaluated in future investigations. CONCLUSION: This systematic review provides useful guidelines for providing speech pathology interventions through telepractice for children with cleft palate. Audiovisual materials seem to be extremely useful for families receiving the interventions. The use of interactive videos for Spanish-speaking children and educational videos for parents is manifest.

The society for craniofacial genetics and developmental biology 43rd annual meeting.

The Society for Craniofacial Genetics and Developmental Biology (SCGDB) held its 43rd annual meeting in a virtual format on October 19-20, 2020. The SCGDB meeting included the presentation of the SCGDB Distinguished Scientists in Craniofacial Research Awards to Marilyn Jones and Kerstin Ludwig and four scientific sessions on the molecular regulation of craniofacial development, craniofacial morphogenesis, translational craniofacial biology, and signaling during craniofacial development. The meeting also included workshops on career development, NIH/NIDCR funding, and the utility of the FaceBase database, as well as two poster sessions. Over 190 attendees from 21 states, representing over 50 different scientific institutions, participated. This diverse group of scientists included cell biologists, developmental biologists, and clinical geneticists. While in-person interactions were missed due to the virtual meeting format imposed by the COVID-19 pandemic, the meeting platform provided ample opportunities for participant interactions and discussions, thus strengthening the community.

Speech pathology telepractice for children with cleft palate in the times of COVID-19 pandemic.

OBJECTIVE: To study whether providing Speech and Language Pathology (SLP) interventions by telepractice (TP) could effectively improve speech performance in children with cleft palate (CCP). METHODS: Forty-three CCP were treated with TP intervention in 45 min sessions, 2 times per week for a period of one month. Children ages ranged 4-12 years (X = 7.04; SD = 2.59). All children presented with velopharyngeal insufficiency (VPI) and compensatory articulation (CA) after palatal repair. TP was provided in small groups (5-6 children) following the principles of the Whole Language Model (WLM). Severity of CA was evaluated by a standardized scale at the onset and at the end of the TP period. RESULTS: At the onset of the TP intervention period, 84% of the patients demonstrated severe CA. At the end of the TP period there was a significant improvement in severity of CA (p < 0.001). CONCLUSION: The results of this study suggests that TP can be a safe and reliable tool for improving CA. Considering that the COVID-19 pandemic will radically modify the delivery of Health Care services in the long term, alternate modes of service delivery should be studied and implemented.