Number and timing of primary cleft lip and palate repair surgeries in England: whole nation study of electronic health records before and during the COVID-19 pandemic.

OBJECTIVE: To quantify differences in number and timing of first primary cleft lip and palate (CLP) repair procedures during the first year of the COVID-19 pandemic (1 April 2020 to 31 March 2021; 2020/2021) compared with the preceding year (1 April 2019 to 31 March 2020; 2019/2021). DESIGN: National observational study of administrative hospital data. SETTING: National Health Service hospitals in England. STUDY POPULATION: Children <5 years undergoing primary repair for an orofacial cleft Population Consensus and Surveys Classification of Interventions and Procedures-fourth revisions (OPCS-4) codes F031, F291). MAIN EXPOSURE: Procedure date (2020/2021 vs 2019/2020). MAIN OUTCOMES: Numbers and timing (age in months) of first primary CLP procedures. RESULTS: 1716 CLP primary repair procedures were included in the analysis. In 2020/2021, 774 CLP procedures were carried out compared with 942 in 2019/2020, a reduction of 17.8% (95% CI 9.5% to 25.4%). The reduction varied over time in 2020/2021, with no surgeries at all during the first 2 months (April and May 2020). Compared with 2019/2020, first primary lip repair procedures performed in 2020/2021 were delayed by 1.6 months on average (95% CI 0.9 to 2.2 months). Delays in primary palate repairs were smaller on average but varied across the nine geographical regions. CONCLUSION: There were significant reductions in the number and delays in timing of first primary CLP repair procedures in England during the first year of the pandemic, which may affect long-term outcomes.

COVID-19 related risk factors and their association with non-syndromic orofacial clefts in five Arab countries: a case-control study.

BACKGROUND: The environmental etiology of non-syndromic orofacial clefts (NSOFCs) is still under research. The aim of this case-control study is to assess COVID-19 associated factors that may be related to the risk of NSOFC in five Arab countries. These factors include COVID-19 infection, COVID-19 symptoms, family member or friends infected with COVID-19, stress, smoking, socioeconomic status and fear of COVID-19. METHODS: The study took place in governmental hospitals in five Arab countries from November 2020 to November 2021. Controls are matched in the month of delivery and site of recruitment. A clinical examination was carried out using LASHAL classification. Maternal exposure to medication, illnesses, supplementation, COVID-19 infection during their pregestation and 1st trimester periods were evaluated using a validated questionnaire. Maternal exposure to stress was assessed using the Life Events scale, fear of covid-19 scale, family member or friend affected with covid-19 infection, pregnancy planning and threatened abortion. RESULTS: The study recruited 1135 infants (386 NSOFC and 749 controls). Living in urban areas, maternal exposure to medications 3-months pregestation, maternal exposure to any of the prenatal life events and maternal fear of COVID-19 significantly increased the risk of having a child with NSOFC. On the other hand, mothers exposed to supplementation 3-months pregestation, mothers suspected of having COVID-19 infection, family members or friends testing positive with COVID-19 significantly decreased the risk of having a child with NSOFC. CONCLUSIONS: This study suggests that NSOFC may be associated with maternal exposure to lifetime stress and COVID-19 fear in particular, with no direct effect of the COVID-19 infection itself. This highlights the importance of providing psychological support for expecting mothers during stressful events that affect populations such as the COVID-19 pandemic, in addition to the usual antenatal care.

Epidemiological pattern of births from the largest surveillance database of live births in Brazil "SINASC" before and during the COVID-19 pandemic in the Brazilian Amazon (preprint)

The surveillance of live births in Brazil has been carried out since 1990 by the Information System on Live Births (SINASC), which was implemented by the Ministry of Health aiming at standardized registration on a national level. The state of Para is part of the Brazilian Amazon, northern Brazil, which has several unique characteristics. Thus, the purpose of this study was to identify the epidemiological pattern of live births before and during the pandemic of COVID-19 in the state of Para, 2016 to 2020. This is an ecological epidemiological time-series study, using epidemiological surveillance data from DATASUS, referring to the Live Births Information System (SINASC). These are data that have been treated by surveillance and are in aggregate format. The study population is the live births residing in the state of Para, in the period from 2016 to 2020. The data collection instrument was the Declaration of Live Births (DLB). There were 689,454 live births, and the highest rates of births were and continued to remain in the Marajo II, Baixo Amazonas, Xingu, and Tapajos regions. The Metropolitan I and Araguaia regions were and continue to be the lowest rates in the state. Age of the mother 15 to 19 years old 22.29%, 20 to 24 years old 30.05% and 25 to 29 years old 22.58%, most of the single pregnancy type 98.32%, prenatal consultations, performed 7 or more 48.10%, followed by 4 to 6 consultations 33.98%, most presented 7 or more years of the study 48.10%, followed by 3 to 6 years 33.98%. Represented 51.21% male and 48.77% female. The occurrence of congenital anomalies represented 0.52% of live births. Another congenital malformation and deformity were the most prevalent at 25.53%, followed by Congenital deformities of the feet 14.90%, Other congenital malformations of the nervous system 14.84%, and Other congenital malformations 10.77%, Cleft lip, and cleft palate 8.88%, Other congenital malformations digestive tract 8.10%. The demographic transition has already occurred for several decades, including the reduction of fertility and birth rate, so our study showed that the reduction in the number of live births was already a reality in the country, but we emphasize that this reduction was enhanced by the pandemic. We observed greater adherence to prenatal care and a lower prevalence of low birth weight compared to other studies, but the limitation was the absence of studies in the same place of the research. Regarding data incompleteness, we emphasize the ignored fields that reflect the fragility in the surveillance of live births, which was reinforced by the literature.

Prologue to the Forum: Speech and Language Tele-Intervention: The Future Is Now.

PURPOSE: This prologue introduces the LSHSS Forum: Speech and Language Tele-Intervention: The Future Is Now. The aims of the forum are (a) to report on the process of moving established in-person interventions to virtual delivery and (b) to provide speech-language pathologists (SLPs) with empirically based guidance on designing and implementing tele-interventions, in order to support academic success for school-age children with speech and language needs. The included articles explore the process, benefits, and challenges of providing intervention in speech, language, and literacy domains in a virtual environment with children. CONCLUSION: Although there are some logistical challenges, established treatments such as those targeting morphosyntax as well as communication partner training can be adapted for successful delivery online. In addition, the forum supports the delivery of an app-based articulation-focused intervention. SLPs report a number of challenges in relation to adapting and delivering interventions through telepractice, and parents express concerns in relation to SLPs' online evaluation of complex speech sound difficulties for children born with cleft lip and palate. Finally, using the principles of Minimal Intervention Needed for Change, a systematic approach to the adaptation of evidence-based interventions for online delivery, is outlined.

Smile Train: A Sustainable Approach to Global Cleft Care.

ABSTRACT: Smile Train, the largest global cleft nonprofit organization, employs a model of establishing partnerships with treatment centers and medical professionals in low- and middle-income countries (LMICs). Having a presence in over 90 countries throughout its history, the organization provides support for cleft lip and palate repair as well as comprehensive cleft care (CCC) for patients with clefts. With the goal of reducing disparities in access to quality surgical, anesthesia, and medical care, Smile Train strengthens partners with training, education, equipment, and patient support grants. Furthermore, safety and quality protocols have been put in place as guidelines for each partner center, and partnerships with other nongovernmental agencies were created to increase safety in the operating rooms. The founder of Smile Train desired to apply technology wherever possible to build sustainability within the treatment centers and surgeons supporting their own community. Smile Train's model, aimed at increased sustainability, is supplemented by technological advancements to assist in the safety and quality of cleft care services provided in LMIC treatment centers. Examples include centralized online data record keeping for every patient, virtual simulations and training, and mobile applications to enhance care. Recently, Smile Train's focus is expanding CCC with nutrition, oral health, speech, and nursing care programs to improve functional and psychosocial outcomes for patients following their procedure. Despite the challenges imposed by the COVID-19 pandemic, Smile Train continues to provide safe, efficacious, and CCC alongside their partners in LMICs further investing tremendous efforts towards the livelihood of children with clefts globally.

Remote clinical consultations in restorative dentistry-A survey.

OBJECTIVES: To assess the patient experience of Remote Clinical Consultations (RCCs) for head and neck cancer and cleft lip and palate patients undergoing specialist restorative dentistry treatment in the context of a secondary care service. METHODS: An online survey was designed; all ninety three patients were invited to participate in its completion following a video or telephone remote clinical consultation with one restorative consultant in Addenbrooke's Hospital between August 2020 and April 2021. The video consultations were carried out via NHS England's Attend Anywhere platform. RESULTS: Sixty three responses were received. Over 70% of patients valued a video or telephone remote clinical consultation rather than an in-person attendance. Patients reported a good experience and would recommend a remote clinical consultation instead of an in-person attendance if no treatment would likely be required. Those who were less likely to recommend a remote clinical consultation were those who had difficulties logging-in to the remote platform or had technical issues. CONCLUSION: While a number of patients still would have preferred a face-to-face attendance, both telephone and video remote consultations were highly valued by patients, and the overall patient experience was that of acceptance. Benefits of remote consultations perceived by patients included having set time to discuss concerns and queries, becoming familiar with a clinician prior to any treatment, saving time, minimising travel and reducing expenses. Barriers to a positive experience included technical difficulties. This survey contributes to the evidence supporting remote clinical consultations as a practical way of delivering specialist consultations in restorative dentistry, it highlights this virtual process largely relates to the practicalities of service provision and highlights the use of remote consultations for specific appointments. CLINICAL SIGNIFICANCE: This study demonstrates patient feedback to a strategy implemented due to adaptations in working methods required as result of the Covid-19 pandemic. Patients were found to have a positive, accepting experience of both telephone and video remote clinical consultations. the study identifies a high patient acceptability and ability to overcome some of the disadvantages of an in-person attendance including saving time, travel and associated expenses while also contributing to a reduction in CO2 emissions and potentially improving clinical efficiency and capacity. These findings add to existing evidence and are indicative that remote consultations provide a practical way of delivering specialist communication in restorative dentistry as part of a unique patient pathway.

Isolation, Uncertainty and Treatment Delays: Parents' Experiences of Having a Baby with Cleft Lip/Palate During the Covid-19 Pandemic.

OBJECTIVES: Previous literature finds that having a child with a cleft lip and/or palate (CL/P) may pose social and emotional challenges for parents. For parents of children born during the Covid-19 pandemic, such challenges may be heightened. Further, novel demands brought about by the pandemic could have caused additional hardships. The aim of this study was to describe the impact of the pandemic on new parents through qualitative exploration of their experiences. DESIGN: Semi-structured interviews were conducted with 14 parents of children born in the United Kingdom with CL/P between January and June 2020, around the start of the pandemic. Data were analysed using inductive thematic analysis. RESULTS: Three themes, with sub-themes, were identified. The first theme, "Changes to Healthcare: The Impact of Restrictions and Reduced Contact", discussed the impact of the pandemic on perinatal care, the care received from the specialist CL/P teams, and parents' experiences of virtual consultations. The second theme, "Family Functioning During the Pandemic", covered parental anxiety, fathers' experiences, and social support. The third theme, "Surgical Prioritisation: Delays and Uncertainty", addressed changes to surgical protocols, coping with uncertainty, complications associated with delayed surgery, and how parents created positive meaning from this period. CONCLUSIONS: A range of increased and additional psychosocial impacts for parents were identified, along with several coping strategies, utilization of social support, and the positive aspects of their experiences. As the pandemic continues, close monitoring of families affected by CL/P remains imperative, particularly for those at risk of emotional distress.

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.

Adapting cleft care protocols in low- and middle-income countries during and after COVID-19: a process-driven review with recommendations (preprint)

Objective: A consortium of global cleft professionals, predominantly from low- and middle-income countries, identified adaptions to cleft care protocols during and after COVID as a priority learning area of need. Design: A multidisciplinary international working group met on a videoconferencing platform in a multi-staged process to make consensus recommendations for adaptions to cleft protocols within resource-constrained settings. Feedback was sought from a roundtable discussion forum and global organisations involved in comprehensive cleft care. Results: Foundational principles were agreed to enable recommendations to be globally relevant and two areas of focus within the specified topic were identified. First the safety aspects of cleft surgery protocols were scrutinised and COVID adaptions, specifically in the pre and peri-operative periods, were highlighted. Second, surgical operations and access to services were prioritized according to their relationship to functional outcomes and time-sensitivity. The operations assigned the highest priority were emergent interventions for breathing and nutritional requirements and primary palatoplasty. The cleft services assigned the highest priority were new-born assessments, paediatric support for children with syndromes, management of acute dental or auditory infections and speech pathology intervention. Conclusions: A collaborative, interdisciplinary and international working group delivered consensus recommendations to assist with the provision of cleft care in low- and middle-income countries. At a time of global cleft care delays due to COVID-19, a united approach amongst global cleft care providers will be advantageous to advocate for children born with cleft lip and palate in resource-constrained settings.

Evaluating Psychosocial Problems in School-Age Children with Cleft Lip and Palate in Bandung, Indonesia Using CBCL/6-18.

BACKGROUND: Cleft lip with or without palate (CL ± P) may impact children's eating, drinking, speaking, breathing, and hearing. We aim to evaluate psychosocial problems in Indonesian cleft center school-age patients identified after one or more surgical interventions. METHODS: This is a cross-sectional study of parent report of patients with unilateral CL ± P who had cleft surgery from 2011 to 2016 in the Bandung Cleft Center using the Bahasa Indonesia version of CBCL/6-18 questionnaire. Descriptive statistics were completed based on measure norms and score ranges. RESULTS: There were 104 participants (56.7% male) with a median age of 8 years old and 73.0% had unilateral cleft of lip, gum, and palate. We found that speech and appearance problems were not perceived by parents for 34.6% of participants after undergoing surgery. The majority of parents reported normal range scores for the Social scale (93.3%) and the School scale (92.3%). In contrast, largely due to the restrictions in the covid-19 pandemic, 78.8% of the patients had below normal range scores for the Activities scale. Borderline or Clinical range scores were reported for 6.7% of children on the Problem Items section and 15.4% of parents endorsed one or more Critical Items about their children, which indicate significant behavioral concerns. CONCLUSION: In this study, we found 6.7% of the school-age children population with CL/P had psychosocial problems. The result of this study hopefully can shed some light in the long-term psychosocial conditions of the CL/P children post-operatively.

Utilizing Teledentistry to Manage Cleft Lip and Palate Patients in an Outpatient Setting.

BACKGROUND: As a result of the current global pandemic, the dental profession has utilized teledentistry to reduce footfall in the hospitals and clinics where possible. Pediatric dental consultants form a vital part of a multidisciplinary team and regularly monitor the dental growth and development of patients with cleft lip and palate. OBJECTIVE: To assess the effectiveness of the service provided by pediatric dental consultants in the South Thames Cleft Service at Evelina Children's Hospital during the COVID-19 pandemic through virtual clinics. DESIGN: Data were collected retrospectively and include all cleft patients contacted via the virtual clinic during May to July 2020. Patients were prioritized by the Red, Amber, Green (RAG) scale to highlight the urgency of their next face-to-face appointment. RESULTS: A total of 215 patients were contacted during this period with a 97% response rate. Patients given a RAG score of GREEN (86%) meant no urgent requirement for a face-to-face consultation and AMBER (8%) patients required treatment that was deemed nonurgent. However, 3% of patients received a RED rating as they required urgent input. CONCLUSION: Through these virtual clinics, the pediatric team was able to reach 208 patients and provided advice and reassurance. The need for face-to-face appointment was eliminated for 11% of patients who were discharged to their local dental practitioners, thereby reducing the risk of spreading COVID-19.

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.

The Preventive and Control Recommendations on Patient With Cleft Lip and Palate During COVID-19 Pandemic in Shanghai.

ABSTRACT: As Corona Virus Disease 2019 (COVID-19) has been gradually controlled domestically, various industries began to resume production in an orderly way. Attention should be paid to the disease and population characteristics of patients with cleft lip with/without palate during diagnosis and treatment. This article summarized and provided prevention and control recommendations on management strategies during hospitalization and protective measures of patients and healthcare workers, hoping to minimize the spread of disease and create a relatively safe environment for medical work.

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.

Interdisciplinary Considerations for Nasolabial Repair During a Global Pandemic.

BACKGROUND: At the declaration of the global pandemic on March 11, 2020, many hospitals and institutions developed a tiered framework for the stratification and prioritization of elective surgery. Cleft lip and palate repair was classified as low acuity, and nasoalveolar molding (NAM) clinics were closed. Anticipating the consequences of delayed cleft care and the additional burden this would cause families, we reassessed our risk-stratification and perioperative algorithms. We hypothesized we could safely optimize nasolabial repair without burdening our care systems and without increasing COVID-19-related morbidity/mortality. METHODS: Our multidisciplinary cleft team reevaluated patient selection to maximize surgical impact. Perioperative protocols were adjusted, and COVID-19 preoperative testing was utilized before nasolabial repair and prior to suture removal under anesthesia. RESULTS: Early in the pandemic, unilateral cleft repair was prioritized and successfully completed on 9 patients. There were no complications related to COVID-19. Nasoalveolar molding clinic was reopened after total patient volume was significantly decreased. CONCLUSIONS: We offer an approach for surgical management of nasolabial clefts during a global pandemic. Although guidelines have suggested postponing all cleft care, we found that at our dedicated pediatric hospital with low burden of COVID-19 and adequate resources, we could follow a strategy to safely resume cleft care while decreasing burden on our patients' families and care delivery systems.

Cerebrospinal Fluid Rhinorrhea Post COVID-19 Nasopharyngeal Swab: a Case Report (preprint)

Background: Cerebrospinal fluid rhinorrhea is a potentially life threatening condition and it is a rare emergency presentation. Most cases has been related to head trauma. However, CSF leak post COVID swab has been rarely reported.Case presentation: We report a 10 year old Emirati male, who presented to emergency department complaining of unilateral clear watery nasal discharge for 14 days, the discharge started after COVID-19 nasopharyngeal swab. The patient is a known case of 3MC syndrome and he underwent surgical repair for cleft lip and cleft palate. From the history and physical examination CSF rhinorrhea was highly suspected and nasal endoscopy confirmed the diagnosis of cribriform defect on the left side. The patient underwent surgical repair and his symptoms resolved after the surgery.Conclusion: We can conclude that COVID-19 nasopharyngeal swab has potential life threatening complications and one should consider the contraindications and the alternative methods available to detect COVID-19 to minimize the potential risks.

Folate intake, markers of folate status and oral clefts: An updated set of systematic reviews and meta-analyses.

BACKGROUND: There has been a longstanding debate about the role of folate in the etiology of orofacial clefts (OFCs). Studies of different measures of nutritional intake or folate status have been done to investigate the possible role of folate in the prevention of OFC. Only one knowledge synthesis has attempted to bring together different types of evidence. The aim of the present work was to update it. METHODS: Evidence for associations between OFC and dietary folate, supplement use, folic acid fortification, biomarkers of folate status, and variants of MTHFR (C677T and A1298C) were included. Potentially eligible articles were systematically identified from PubMed, Medline, Embase, and Web of Science (2007-2020) and combined using random-effects meta-analysis when appropriate. Quality assessments were conducted using the Newcastle-Ottawa scale and Cochrane's risk of bias tool. RESULTS: Sixty-four studies published since the previous knowledge synthesis were identified, with eight of these identified through a supplementary search from October, 2018 to August, 2020. There was an inverse association between folic acid-containing supplement use before or during pregnancy and cleft lip with or without cleft palate (CL/P) (OR 0.60, 95% CI 0.51-0.69), with considerable between-study heterogeneity. The prevalence of CL/P showed a small decline post-folic acid fortification in seven studies (OR 0.94, 95% CI 0.86-1.02). No association was found between OFC and genetic markers of folate status. The coronavirus-19 pandemic has threatened food availability globally and therefore there is a need to maintain and even enhance surveillance concerning maternal intake of folate and related vitamins. CONCLUSIONS: The risk of non-syndromic OFC was reduced among pregnant women with folic acid-containing supplements during the etiologically relevant period. However, high heterogeneity between included studies, incomplete reporting of population characteristics and variation in timing of exposure and supplement types mean that conclusions should be drawn with caution.