Adapting Elements of Cleft Care Protocols in Low- and Middle-income Countries During and After COVID-19: A Process-driven Review With Recommendations.

A consortium of global cleft professionals, predominantly from low- and middle-income countries, identified adaptations to cleft care protocols during and after COVID-19 as a priority learning area of need.A multidisciplinary international working group met on a videoconferencing platform in a multi-staged process to make consensus recommendations for adaptations to cleft protocols within resource-constrained settings. Feedback was sought from a roundtable discussion forum and global organizations involved in comprehensive cleft care.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 scrutinized and COVID-19 adaptations, specifically in the pre- and perioperative periods, were highlighted. Second, surgical procedures and cleft care services were prioritized according to their relationship to functional outcomes and time-sensitivity. The surgical procedures assigned the highest priority were emergent interventions for breathing and nutritional requirements and primary palatoplasty. The cleft care services assigned the highest priority were new-born assessments, pediatric support for children with syndromes, management of acute dental or auditory infections and speech pathology intervention.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.

Sociodemographics and Quality of Life in Dutch-Speaking Adolescents and Adults With and Without a Cleft Lip and/or Palate.

OBJECTIVE: To investigate the impact of living with a cleft lip and/or cleft palate (CL/P) on sociodemographic variables, quality of life, aesthetics, life satisfaction, and social distress in Dutch-speaking adolescents and adults. DESIGN: Cross-sectional study. PARTICIPANTS: Thirty Dutch-speaking participants with a CL/P with a mean age of 26.93 years (SD = 11.69) and an age- and gender-matched control group of 30 participants (19 men and 11 women) without a CL/P with a mean age of 26.87 years (SD = 11.73). MAIN OUTCOME MEASURES: Self-reported outcomes of sociodemographics, quality of life, aesthetics, life satisfaction, social distress, and impact of cleft on well-being and functioning. RESULTS: No significant differences in educational level, employment, monthly net income, marital status, and having children were found between participants with and without a CL/P. In addition, quality of life, overall aesthetics, life satisfaction, and social distress did not differ between the 2 groups. Among participants with CL/P, there were no gender differences in the influence of their CL/P on daily functioning, well-being, social contacts, family life, applying for a job, work, education, or leisure time. CONCLUSION: The findings revealed no differences between participants with and without a CL/P with regard to sociodemographics, quality of life, aesthetics, life satisfaction, or social distress. There were no gender differences in the influence of cleft on well-being and functioning. Longitudinal research can help determine possible fluctuations in the impact of living with a CL/P across the life span.

Perceptual Speech Outcomes After Early Primary Palatal Repair in Ugandan Patients With Cleft Palate.

OBJECTIVE: To provide speech outcomes of English-speaking Ugandan patients with a cleft palate with or without cleft lip (CP±L). DESIGN: Prospective case-control study. SETTING: Referral hospital for patients with cleft lip and palate in Uganda. PARTICIPANTS: Twenty-four English-speaking Ugandan children with a CP±L (15 boys, 9 girls, mean 8.4 years) who received palatal closure prior to 6 months of age and an age- and gender-matched control group of Ugandan children without cleft palate. INTERVENTIONS: Comparison of speech outcomes of the patient and control group. MAIN OUTCOME MEASURES: Perceptual speech outcomes including articulation, resonance, speech understandability and acceptability, and velopharyngeal composite score (VPC-sum). Information regarding speech therapy, fistula rate, and secondary surgery. RESULTS: Normal speech understandability was observed in 42% of the patients, and 38% were judged with normal speech acceptability. Only 16% showed compensatory articulation. Acceptable resonance was found in 71%, and 75% of the patients were judged perceptually to present with competent velopharyngeal function based on the VPC-sum. Additional speech intervention was recommended in 25% of the patients. Statistically significant differences for all these variables were still observed with the control children (P < .05). CONCLUSIONS: Overall, acceptable speech outcomes were found after early primary palatal closure. Comparable or even better results were found in comparison with international benchmarks, especially regarding the presence of compensatory articulation. Whether this approach is transferable to Western countries is the subject for further research.

Unifying Children's Surgery and Anesthesia Stakeholders Across Institutions and Clinical Disciplines: Challenges and Solutions from Uganda.

BACKGROUND: There is a significant unmet need for children's surgical care in low- and middle-income countries (LMICs). Multidisciplinary collaboration is required to advance the surgical and anesthesia care of children's surgical conditions such as congenital conditions, cancer and injuries. Nonetheless, there are limited examples of this process from LMICs. We describe the development and 3-year outcomes following a 2015 stakeholders' meeting in Uganda to catalyze multidisciplinary and multi-institutional collaboration. METHODS: The stakeholders' meeting was a daylong conference held in Kampala with local, regional and international collaborators in attendance. Multiple clinical specialties including surgical subspecialists, pediatric anesthesia, perioperative nursing, pediatric oncology and neonatology were represented. Key thematic areas including infrastructure, training and workforce retention, service delivery, and research and advocacy were addressed, and short-term objectives were agreed upon. We reported the 3-year outcomes following the meeting by thematic area. RESULTS: The Pediatric Surgical Foundation was developed following the meeting to formalize coordination between institutions. Through international collaborations, operating room capacity has increased. A pediatric general surgery fellowship has expanded at Mulago and Mbarara hospitals supplemented by an international fellowship in multiple disciplines. Coordinated outreach camps have continued to assist with training and service delivery in rural regional hospitals. CONCLUSION: Collaborations between disciplines, both within LMICs and with international partners, are required to advance children's surgery. The unification of stakeholders across clinical disciplines and institutional partnerships can facilitate increased children's surgical capacity. Such a process may prove useful in other LMICs with a wide range of children's surgery stakeholders.

Recurrence of post burn contractures of the elbow and shoulder joints: experience from a Ugandan hospital.

BACKGROUND: Recurrence of post-burn contractures, following inadequate management of post-burn contractures (PBC), is under reported. It is associated with multiple operations and an increased cost to patients and their families. The purpose of this study was to determine the frequency of recurrence of PBC of the shoulder and the elbow joint three months after surgical intervention and the associated risk- factors. METHODS: This was a prospective cohort study conducted at CoRSU hospital from March 2012 to November 2014. All patients with PBC of the elbow and/or shoulder joint who consented to be in the study and met the inclusion criteria were enrolled. Data was collected using a pretested, coded questionnaire. A goniometer was used to measure the active range of motion of the involved joint. The measurements were recorded in degrees. The data was analysed with STATA version 12.1. RESULTS: 58 patients were enrolled consecutively in the study. There were 36 females and 22 males, with a female to male ratio of 1.6:1. The age range was 0.75-45 years, with a median age of 5 years. The average age at the time of injury was 3.4 years. The most common cause of initial burn injury was scalding. The average number of joints involved per patient was two. There was a high incidence of recurrence of PBC (52 %) among the participants. The shoulder had the highest frequency of recurrence at 67 %. The elbow joint had a frequency of recurrence of 27 %. All participants with both elbow and shoulder joint involvement had PBC recur. The risk factors for recurrence were flame burn (p = 0.007), duration of PBC of more than 1 year (p = 0.018), and incomplete release of the contracture (p = 0.002). The presence of keloids, hypertrophic scars, ulcers and the occurrence of complications at the contracture site were not associated with recurrence of PBC. CONCLUSION: Recurrence of PBC of the elbow and shoulder joint is a common problem. The risk factors should be kept in mind during management of PBC to reduce the recurrence rate.

Impact of early synchronous lip and palatal repair on speech.

AIMS: The purpose of this study was to describe articulation and resonance characteristics of Ugandan English-speaking children with cleft lip and palate (CLP) after synchronous lip and palatal closure (Sommerlad technique) prior to the age of 6 months in comparison with an age- and gender-matched control group. METHODS: Eleven Ugandan patients with CLP (mean age 4;9 years), repaired during a synchronous lip and (soft and hard) palatal closure at a mean age of 3.4 months, were included as well as a control group (n = 22) consisting of 2 Ugandan age- and gender-matched noncleft children for each patient (mean age 4;10 years). Objective and perceptual speech assessment techniques were applied. RESULTS: Consonant inventories were significantly smaller in the CLP group. Moreover, phonetic disorders and phonological processes occurred in 91 and 100% of the CLP group, respectively. Perceptual consensus evaluation revealed absence of hyponasality and cul-de-sac resonance in all patients. Hypernasality and nasal emission/turbulence occurred in 18 and 27% of the patients, respectively. No significant group differences were observed regarding the mean nasalance values of oral speech samples. CONCLUSION: Various deviations from normal speech development seem to occur in patients with CLP following synchronous lip and palatal closure before 6 months of age.

Pediatric speech-language pathology in East Africa: educational opportunities and availability of speech-language services.

OBJECTIVE: This paper describes the current situation of the provision of speech-language services in East Africa. An overview of the educational opportunities, workforce density, communication rehabilitation planning, caseload, and ethical and/or cultural challenges is provided. METHOD: A narrative review was performed. All papers referring to communication disorders in East African countries were identified from EMBASE, PubMed, ISI Web of Science: Science Citation Index Expanded, and ISI Web of Science: Conference Proceedings Citation Index-Science using the following search terms: speech therapy, speech pathology, speech-language pathology, speech-language rehabilitation, speech-language therapy, logopedics, East Africa, communication disorders, communication difficulties. Two concepts were combined with a Boolean operator 'AND'. RESULTS: At present, there is an alarming shortage of speech-language pathologists in East Africa. This issue is mainly related to the limited availability of educational programs in speech-language pathology. Although more and more universities are beginning to offer such educational programs, the number of available speech-language pathologists is still too low to meet the needs of all children with communication disorders. In addition, speech-language pathologists are often faced with different cultural and ethical obstacles when they provide intervention to East African children with communication disorders. These speech-language pathologists are therefore challenged to take a broader view of their role as a therapist. These alternative roles often include training of other individuals in the context of community-based rehabilitation. CONCLUSION: In the future, it will be important to carefully consider the development of sustainable learning opportunities in East African contexts. Initiatives, for example, "train-the-trainer" workshops, are needed to support the speechlanguage pathologists. Existing models like the communication disability model can aid to create holistic frameworks for the development of sustainable speech-language services in the East African countries.

Better speech outcomes after very early palatal repair?-A longitudinal case-control study in Ugandan children with cleft palate.

INTRODUCTION: Children born with cleft palate with or without cleft lip (CP±L) tend to use less oral pressure consonants and more glottal sounds in their babbling. The purpose of very early palatal repair (i.e., one-stage palatal closure prior to 6 months of age) is to make the palate functional before the onset of speech acquisition to reduce the anchoring of wrong patterns in the child's developing phonological system. As a result, less compensatory articulation errors are expected to be present. Currently, no detailed longitudinal speech outcomes after very early palatal closure are available. This study aimed to provide longitudinal speech outcomes in Ugandan children with CP±L who received palatal closure prior to the age of 6 months. METHODS: Ten children with CP±L were assessed at a mean age of 5 and 10 years old. Speech understandability, speech acceptability, resonance, nasal airflow and articulation were perceptually rated by two experienced speech-language pathologists. Velopharyngeal function was estimated using the velopharyngeal composite score (VPC-sum). Information regarding speech therapy, fistula rate, and secondary (speech) surgery was collected. The outcomes were compared with the longitudinal outcomes of an age- and gender-matched control group of 10 Ugandan children without CP±L. RESULTS: Speech understandability and acceptability improved significantly over time in the group with CP±L (all p's ≤ 0.05, all Z's > -2.43). At both test dates, significantly worse judgments were found for the group with CP±L compared to the control group for these variables and variables related to passive speech errors (all p's ≤ 0.05, all Z's > 2.49). A statistically significant difference with the control group was found for the presence of compensatory articulation errors at the age of 5 years but not at the age of 10 years, indicating a catch up by the children with CP±L. CONCLUSION: Whether a one-stage palatal closure prior to the age of 6 months is more favorable for speech outcomes compared to one-stage palatal closure at 12 months is still not clear. Speech of the children with CP±L improved over time, but significantly differed from the control group at the age of 5 and 10 years old. Limited access to health care facilities and possible influence of malnutrition on wound healing need to be considered when interpreting the results. Whether palatal closure prior to the age of 6 months is transferable to other countries is subject for further research, including both longitudinal and prospective designs with larger samples.

Incidence of cleft lip and palate in Uganda.

OBJECTIVE: The purpose of the study was to estimate the need for resources for cleft repairs in Uganda by determining the overall incidence of oral-facial clefts and the ratio of isolated cleft lip to isolated cleft palate to cleft lip and palate. DESIGN: A 1-year prospective study was implemented in seven hospitals and health centers with maternity units in and around Kampala. All live babies were examined for cleft lip and/or palate at birth. The birth of a baby with a cleft was communicated to the research coordinator by maternity staff, and the babies were assessed within 24 hours of delivery. Maternity data (live births) for each center were collected monthly to determine the denominator. RESULTS: Over the course of 1 year (February 1, 2008 to January 31, 2009), 26,186 babies were delivered. Nineteen babies had a cleft lip and/or palate, giving an incidence of 0.73 in 1000; 12 of the 19 babies (63.2%) had a cleft lip and palate, six (31.2%) had an isolated cleft lip, and only one (5.3%) had an isolated cleft palate. The ratio of boys to girls was 1.1∶1. CONCLUSIONS: The incidence of clefts in this study was 0.73 in 1000. With an estimated 1100 babies born with a cleft per year, this would result in the need for 1800 cleft repairs for babies in Uganda (695 babies needing two repairs each). These data will be helpful to advocate for and plan the necessary resources to effectively treat this condition.

Vascularized fibula flap in the management of segmental bone loss following osteomyelitis in children at a Ugandan hospital.

Background: Hematogenous osteomyelitis is commonly observed in the pediatric population across sub-Saharan Africa. This retrospective case series was designed to evaluate the complications and outcomes of treatment using a vascularized fibula flap (VFF) to fill segmental bone defects secondary to osteomyelitis in children in a low-resource setting in CoRSU Rehabilitation Hospital, Uganda. Methods: Clinical notes and radiographs of children with a diagnosis of osteomyelitis that subsequently underwent a VFF procedure between October 2013 and December 2017 were reviewed. All patients were clinically and radiographically evaluated in 2019. Results: Forty-four children, with an average bone defect of 10.5 cm, were included. Eighty-four percent of children had successful VFF limb reconstruction. Integration of the graft was radiologically sound in 20.8 weeks on average. The postoperative phase was uneventful in 29 % of patients. Complications were observed in the remaining patients, including flap failure (6), donor leg neurapraxia (3), cutaneous paddle necrosis (11), graft fracture (2), skin graft loss (6), fixator failure (1) and non-union (2). Functional outcomes were rated as excellent in 13 patients, good in 14, fair in 9 and poor in 8. There was no recurrence of the bone infection in any of the enrolled children. Conclusion: Despite being a complex and demanding procedure, VFF is a good option for reconstructing post-osteomyelitis bone defects, particularly when associated with loss of soft tissue envelope. Considering the more than satisfactory functional and clinical outcomes, this procedure should be kept in mind for these complex pediatric cases of bone and soft tissue loss, even in a low-resource setting.

Challenges to achieving low palatal fistula rates following primary cleft palate repair: experience of an institution in Uganda.

OBJECTIVE: To determine frequency of palatal fistula following primary cleft palate repair and the associated factors as a measure of cleft palate repair outcome and its challenges at a cleft centre in Uganda. RESULTS: Between May and December 2016, 54 children with cleft palate were followed up at Comprehensive Rehabilitation services of Uganda (CoRSU) hospital, from time of primary cleft palate repair until at least 3 months postoperative to determine whether they developed palatal fistula or not. Frequency of palatal fistula was 35%. Factors associated with increased fistula formation were cleft width wider than 12 mm (p = 0.006), palatal index greater than 0.4 (p = 0.046), presence of malnutrition at initial outpatient assessment (p = 0.0057) and at time of surgery (p = 0.008), two-stage palate repair (p = 0.005) and postoperative infection (p = 0.003). Severe clefting (palatal index greater than 0.4) was seen in 74% of patients and malnutrition (Low weight for age) seen in 48% of patients. Palatal fistula rates at our institution were high compared to reports in literature. The high proportions of severe clefting and malnutrition observed in our population that was also poor and unable to afford feeding supplements increased likelihood of fistula formation and posed challenges to achieving low fistula rates in our setting.

Delayed primary palatal closure in resource-poor countries: Speech results in Ugandan older children and young adults with cleft (lip and) palate.

AIMS: Unrepaired clefts still regularly occur in resource-poor countries as a result of limited health-care access. The purpose of the present study was to report resonance, airflow and articulation characteristics following delayed (≥8years) primary palatal closure. METHODS: Fifteen Ugandan participants with cleft (lip and) palate (CP±L) were included as well as 15 age- and gender-matched Ugandan subjects without clefts. Palatal closure was performed at a mean age of 15;10 years using the Sommerlad technique. Speech evaluations were carried out on a single occasion postoperatively (mean age: 18;10 years). Resonance and nasal airflow were perceptually evaluated and detailed phonetic and phonological assessments were carried out. Additionally, nasalance values were determined. RESULTS: Nasal emission occurred postoperatively in only 27% (4/15) of the patients, whereas resonance disorders and articulation errors were prevalent in 87% (13/15) of the patient group. Compared with the control group, a significantly higher prevalence of hypernasality and significantly higher nasalance values for all oral and oronasal speech samples were obtained in the CP±L group. Moreover, significantly smaller consonant inventories and significantly more phonetic and phonological disorders were observed. CONCLUSIONS: Delayed palatal repair (≥8years) seems to be insufficient to eliminate nasal airflow errors, resonance abnormalities, and articulation disorders. In order to prevent patients' late presentation at specialized centers, the availability of high quality surgical cleft palate treatment should increase as well as people's awareness of the possibility and importance of early surgical intervention. Moreover, speech therapy following delayed palatal closure would be beneficial. Furthermore, a standardized and validated protocol for speech assessment in future studies is advocated.

Challenges in global microsurgery: A six year review of outcomes at an East African hospital.

BACKGROUND: Free tissue transfer is an invaluable resource for reconstruction of complex defects. There is very little evidence as to the feasibility and outcomes of microsurgery performed in East Africa. This study will analyse outcomes of 114 consecutive free flaps, performed over 6 years at a single plastic surgery unit in Uganda. It aims to demonstrate that despite its challenges, successful microsurgical practice can be set up in East Africa. METHODS AND PATIENTS: The notes of 100 consecutive patients who underwent 114 free flaps between 01/06/2009 and 01/07/2015 at CoRSU Hospital, Uganda were analysed. RESULTS: One hundred and fourteen free flaps were performed on 100 patients. The types of free flaps used included free fibula (n = 41), ALT (n = 30), gracillis (n = 8), radial forearm (n = 7), latissimus dorsi (n = 9) and rectus (n = 7) amongst others (n = 12). The most common indications for surgery were head and neck cancer (n = 50), trauma (n = 19), osteomyelitis (n = 18), burns (n = 13), head and neck infection (n = 6). Over the six year period there was an overall 76% survival of the flaps. However in the last two years of the series there was a flap survival rate of over 93% (n = 50). There were 40 non-microsurgical complications including wound infection (n = 10) and graft loss (n = 8). CONCLUSION: This is one of the first studies to report on the outcomes of free flaps performed at an East African centre. There is a steep but surmountable learning curve to improve microsurgery delivery in East Africa. This study identifies challenges in patient demographics, surgical experience and resources that have been overcome to improve outcomes.

The impact of palatal repair before and after 6 months of age on speech characteristics.

OBJECTIVE: Optimal timing of palatal repair is still subject of discussion. Although literature provides some evidence that palatal closure prior to 6 months positively influence speech outcome in children with clefts, only few studies verified this hypothesis. The purpose of this study was to describe and compare articulation and resonance characteristics following early (≤6 months) and later (>6 months) palatal repair, performed using the Sommerlad technique. METHODS: Comparison was made between 12 Ugandan children with isolated cleft (lip and) palate following early palatal repair (mean age: 3.3 m) and 12 Belgian patients with later palatal repair (mean age: 11.1 m), matched for cleft type, age and gender. A Ugandan and Belgian age- and gender-matched control group without clefts was included to control for language, culture and other environmental factors. Articulation assessments consisted of consonant inventories and phonetic and phonological analyses that were based on consensus transcriptions. In addition, resonance was evaluated by perceptual consensus ratings and objective mean nasalance values. RESULTS: The Belgian and Ugandan control groups were comparable for the majority of the variables. Comparison of cleft palate groups revealed no clinically relevant significant group differences for consonant inventory or phonological processes. Phonetic analysis showed significantly more distortions in the Belgian cleft palate group due to higher occurrence frequencies for (inter)dental productions of apico-alveolar consonants. Neither perceptual consensus ratings of hypernasality, hyponasality, cul-de-sac resonance and nasal emission/turbulence, nor objective mean nasalance values for oral speech samples revealed significant group differences (p>0.05). CONCLUSION: Articulation and resonance characteristics of young children following palatal repair before and after 6 months of age seem to be at least comparable.

Parental satisfaction in Ugandan children with cleft lip and palate following synchronous lip and palatal repair.

UNLABELLED: The purpose of the present case control study was to assess parental satisfaction with speech and facial appearance in Ugandan children with complete unilateral or bilateral cleft lip and palate (CLP), who underwent a synchronous lip and palatal closure. The results are compared with an age- and gender-matched control group. The experimental group consisted of the parents or guardians of 44 Ugandan patients (21 males, 23 females) with complete unilateral or bilateral CLP (mean age: 3;1 years). The control group included the foster mothers of 44 orphan children matched by age and gender (mean age: 3;7 years). A survey based on the Cleft Evaluation Profile was used to assess the perceived satisfaction for individual features related to cleft care. Overall high levels of satisfaction were observed in the experimental group for all features (range: 56-100%). No significant differences could be established regarding age, gender, age of lip and palatal closure, cleft type or maternal vs. paternal judgments. In participants who were dissatisfied with the appearance of the lip, the time period between the cleft closure and the survey was significantly larger compared with satisfied participants. Furthermore, significantly lower levels of satisfaction were observed in the cleft group for speech and the appearance of the teeth and the nose compared with the control group. Satisfaction with speech and facial appearance in Ugandan children with cleft lip and/or palate is important since normal esthetics and speech predominantly determine the children's social acceptance in the Ugandan society. LEARNING OUTCOMES: As a result of reading this manuscript, the reader will be able to explain the attitudes of parents toward the surgical repair of their children's cleft lip and palate. As a result of reading this manuscript, the reader will be able to identify differences in parental attitudes toward synchronous lip and palate repair.