Prospective Outcomes of Noma Facial Reconstructive Surgery in Sokoto, Nigeria.

Background: Noma is a gangrenous infection of the face that results in severe facial deformity, occurring primarily in malnourished and impoverished populations. Objective: To assess clinician- and patient-reported outcomes (PROs) before and after reconstructive surgery for patients with noma in northwest Nigeria. Methods: Objective outcomes were recorded using the noma-specific NOITULP (nose, outer cheek, inner cheek, trismus, upper/lower lip, particularities) classification system. PROs were recorded using a locally developed tool. Postsurgical changes were assessed by Wilcoxon signed-rank testing. Linear regression was used to look for associated risk factors. The inter-rater reliability (IRR) of the NOITULP score was assessed using the weighted kappa statistic. Results: Forty-nine patients (median age 25 years, 71% male) underwent local/regional flap reconstruction and/or trismus release. Twelve complications were reported. Univariate analysis showed a 3.20 change in PRO score (95% confidence interval 0.59 to 5.81, p = 0.018) per kilogram the patient underwent at time of surgery. The NOITULP score improved from a presurgery median of 3.5 to 2.3 (p < 0.0001), however, the IRR was poor (kappa = 0.0894, p < 0.0001). The PRO score also improved from a median of 7.0 to 12.0 (p < 0.0001). Conclusions: Facial reconstructive surgery improves the NOITULP score and PROs in patients with noma in northwest Nigeria. Clinical Trial Registration: OCA017-63.

WHO's Investigations and Studies, Unity Studies: A global initiative creating equitable opportunities for enhanced surveillance, operational research, capacity building, and global knowledge sharing.

The World Health Organization's Unity Studies global initiative provides a generic preparedness and readiness framework for conducting detailed investigations and epidemiological studies critical for the early and ongoing assessment of emerging respiratory pathogens of pandemic potential. During the COVID-19 pandemic, the initiative produced standardized investigation protocols and supported Member States to generate robust and comparable data to inform public health decision making. The subsequent iteration of the initiative is being implemented to develop revised and new investigation protocols, implementation toolkits and work to build a sustainable global network of sites, enabling the global community to be better prepared for the next emerging respiratory pathogen with epidemic or pandemic potential.

Transmissibility and severity of COVID-19 in a humanitarian setting: First few X investigation of cases and contacts in Juba, South Sudan, 2020.

BACKGROUND: The first few 'X' (FFX) studies provide evidence to guide public health decision-making and resource allocation. The adapted WHO Unity FFX protocol for COVID-19 was implemented to gain an understanding of the clinical, epidemiological, virological and household transmission dynamics of the first cases of COVID-19 infection detected in Juba, South Sudan. METHODS: Laboratory-confirmed COVID-19 cases were identified through the national surveillance system, and an initial visit was conducted with eligible cases to identify all close contacts. Consenting cases and close contacts were enrolled between June 2020 and December 2020. Demographic, clinical information and biological samples were taken at enrollment and 14-21 days post-enrollment for all participants. RESULTS: Twenty-nine primary cases and 82 contacts were included in the analyses. Most primary cases (n = 23/29, 79.3%) and contacts (n = 61/82, 74.4%) were male. Many primary cases (n = 18/29, 62.1%) and contacts (n = 51/82, 62.2%) were seropositive for SARS-CoV-2 at baseline. The secondary attack rate among susceptible contacts was 12.9% (4/31; 95% CI: 4.9%-29.7%). All secondary cases and most (72%) primary cases were asymptomatic. Reported symptoms included coughing (n = 6/29, 20.7%), fever or history of fever (n = 4/29, 13.8%), headache (n = 3/29, 10.3%) and shortness of breath (n = 3/29, 10.3%). Of 38 cases, two were hospitalised (5.3%) and one died (2.6%). CONCLUSIONS: These findings were used to develop the South Sudanese Ministry of Health surveillance and contract tracing protocols, informing local COVID-19 case definitions, follow-up protocols and data management systems. This investigation demonstrates that rapid FFX implementation is critical in understanding the emerging disease and informing response priorities.

Proportion of paediatric admissions with any stage of noma at the Anka General Hospital, northwest Nigeria.

INTRODUCTION: Noma is a rapidly spreading infection of the oral cavity which mainly affects young children. Without early treatment, it can have a high mortality rate. Simple gingivitis is a warning sign for noma, and acute necrotizing gingivitis is the first stage of noma. The epidemiology of noma is not well understood. We aimed to understand the prevalence of all stages of noma in hospitalised children. METHODS: We conducted a prospective observational study from 1st June to 24th October 2021, enrolling patients aged 0 to 12 years who were admitted to the Anka General Hospital, Zamfara, northwest Nigeria. Consenting parents/ guardians of participants were interviewed at admission. Participants had anthropometric and oral examinations at admission and discharge. FINDINGS: Of the 2346 patients, 58 (2.5%) were diagnosed with simple gingivitis and six (n = 0.3%) with acute necrotizing gingivitis upon admission. Of those admitted to the Inpatient Therapeutic Feeding Centre (ITFC), 3.4% (n = 37, CI 2.5-4.7%) were diagnosed with simple gingivitis upon admission compared to 1.7% of those not admitted to the ITFC (n = 21, CI 1.1-2.6%) (p = 0.008). Risk factors identified for having simple gingivitis included being aged over two years (2 to 6 yrs old, odds ratio (OR) 3.4, CI 1.77-6.5; 7 to 12 yrs OR 5.0, CI 1.7-14.6; p = <0.001), being admitted to the ITFC (OR 2.1; CI 1.22-3.62) and having oral health issues in the three months prior to the assessment (OR 18.75; CI 10.65, 33.01). All (n = 4/4) those aged six months to five years acute necrotizing gingivitis had chronic malnutrition. CONCLUSION: Our study showed a small proportion of children admitted to the Anka General Hospital had simple or acute necrotizing gingivitis. Hospital admission with malnutrition was a risk factor for both simple and acute necrotizing gingivitis. The lack of access to and uptake of oral health care indicates a strong need for oral examinations to be included in routine health services. This provision could improve the oral status of the population and decrease the chance of patients developing noma.

Short communication-Lessons learnt during the implementation of Unity-aligned SARS-CoV-2 seroprevalence studies in Africa.

The WHO Unity Studies initiative engaged low- and middle-income countries in the implementation of standardised SARS-CoV-2 sero-epidemiological investigation protocols and timely sharing of comparable results for evidence-based action. To gain a deeper understanding of the methodological challenges faced when conducting seroprevalence studies in the African region, we conducted unstructured interviews with key study teams in five countries. We discuss the challenges identified: participant recruitment and retention, sampling, sample and data management, data analysis and presentation. Potential solutions to aid future implementation include preparedness actions such as the development of new tools, robust planning and practice.

Noma, a neglected disease: prevention is better than cure.

PURPOSE OF REVIEW: There is a need for concerted effort to increase Global awareness about noma (cancrum oris). This paper aims to summarize the recent literature on noma and provide suggestions that could be implemented to raise awareness about this neglected disease. RECENT FINDINGS: Noma has been recognized, diagnosed and reported for centuries. Despite significant progress in scientific methods over time, the published literature on noma has predominantly been of low level clinical and scientific evidence. Recent studies have reported on noma's global distribution and its predisposing risk factors, its treatment, its knowledge and beliefs and has included a number of literature reviews. Noma cases are being reported from an increasingly diverse set of geographical locations. SUMMARY: Noma has largely been neglected in the research sphere. Noma is a preventable disease and its progression can be halted if patients are recognized and treated in the early stages of disease. Treatment for late stage noma survivors remains complex and time consuming, requiring substantial human and financial resources most commonly not achieving functional and cosmetic anatomy. The ultimate aim is therefore prevention, initiatives should be integrated into existing health programs.

Lao Noma Survivors: A Case Series, 2002-2020.

Noma is a rapidly progressing infection of the oral cavity, mostly affecting children aged between 2 and 5 years. If untreated, mortality can reach 90% within a few weeks after the onset of symptoms. Most of the published literature on noma are case reports or case series from Africa. Studies including noma survivors in Asia are limited. We present a case series of noma survivors in Laos. A retrospective analysis of data collected to monitor the care provided to Lao noma survivors who presented for treatment from 2002 to 2020 was conducted. The review assessed data including sociodemographic characteristics, diagnosis, mouth opening, self-reported quality of life at admission and after surgery, and the names used for the disease. Of the 50 patients included, 25 (50%) were female. The median age of self-reported onset of acute noma was 4 years (interquartile range [IQR] 2-7 years). The noma survivors came from 14/17 (82%) of Lao provinces. There were 64 surgeries conducted on 45 of these survivors. There was a median of 25 years (IQR 16-33 years) between the time of acute infection and the provision of surgical care. Improvements in nutritional status and quality of life were evident after surgery. Patients referred to the disease as "Pak Phuey," which means diseased mouth. Noma survivors frequently live for years with disabling sequelae. Surgical rehabilitation improves the quality of life for noma survivors.

More than 100 years of neglect: a bibliometric analysis of global research on noma (cancrum oris).

BACKGROUND: Research plays a significant role in quantifying and addressing the burden of disease, improving healthcare delivery and developing evidence-based policy for neglected tropical diseases (NTDs). Since noma is not a recognised NTD by the World Health Organization (WHO), research activity is hypothesised to be low. This study aims to understand patterns and trends of research conducted on noma, to understand the patterns of citations, highlight networks of collaboration and analyse stakeholders in noma research. METHODS: The SCOPUS database was selected and searched. Data analysis was conducted using the bibliometrix package for R in RStudio. Areas of interest included the distribution of research output on noma by year and location, the academic impact of publication output, mapping the major academic community and conducting a thematic analysis of high-frequency keywords. RESULTS: A total of 208 documents were identified. The annual growth rate of publications is 4.3%. The average number of citations per publication was 11.0 (rounded from 10.98) and the average citations per year per publication was 0.57. The USA was the country with the highest percentage 10.6% (n=22) of corresponding authors, followed by Nigeria (6.7% [n=14]), the UK (6.3% [n=13]) and India (5.8% [n=12]). For those papers with multiple authors, there was an average of 3.22 co-authors per document. The collaboration index was 3.3. Upon plotting collaboration networks between authors, only 11 clusters were identified (frequency and intensity of collaboration). Mapping of trending topics showed a focus on skin grafting and transplantation within the literature. CONCLUSIONS: In order to progress towards eventual elimination of noma it is key that more research should be undertaken and more interest and funding should be dedicated to work on noma. Recognition of noma as an NTD by the WHO would be a first step in moving towards increased awareness.

Lockdown babies: Birth and new parenting experiences during the 2020 Covid-19 lockdown in South Africa, a cross-sectional study.

BACKGROUND: Perceived birth experiences of parents can have a lasting impact on children. We explored the birth and new parenting experiences of South African parents in 2020 during the Covid-19 lockdown. METHODS: We conducted a cross-sectional online survey with consenting parents of babies born in South Africa during 2020. Factors associated with negative birth emotions and probable depression were estimated using logistic regression. RESULTS: Most of the 520 respondents were females (n = 496, 95%) who gave birth at private hospitals (n = 426, 86%). Mothers reported having overall positive birth emotions (n = 399, 80%). Multivariable analysis showed that having a preterm baby (aOR 2.89; CI 1.51-5.53) and the mother self-reporting that Covid-19 affected her birth experience (aOR 4.25; CI 2.08-8.68) increased the odds of mothers reporting predominantly negative emotions about their birth. The mother having her preferred delivery method reduced the odds of having negative birth emotions (aOR 0.41; CI 0.25-0.66). Multivariable analysis showed that having predominantly negative emotions about the birth increased the odds of probable minor depression (aOR 3.60; CI 1.93-6.70). Being older reduced the odds of having probable minor depression (25-34 years aOR 0.36; CI 0.10-1.32; 35 years or older aOR 0.25; CI 0.06-0.91). CONCLUSIONS: Lockdown exacerbated many birth and parenting challenges including mental health and health care access. However, overall experiences were positive and there was a strong sense of resilience amongst parents.

Noma (cancrum oris): A scoping literature review of a neglected disease (1843 to 2021).

BACKGROUND: Noma (cancrum oris) is an ancient but neglected and poorly understood preventable disease, afflicting the most disenfranchised populations in the world. It is a devastating and often fatal condition that requires urgent and intensive clinical and surgical care, often difficult to access as most cases of noma occur in resource-limited settings. We conducted a scoping review of the literature published on noma to understand the size and scope of available research on the disease and identify research gaps that need to be addressed to evolve our understanding of how to address this disease. METHODS: We searched 11 databases and collected primary peer reviewed articles on noma in all languages, the final search was conducted on 24th August 2021. The oldest manuscript identified was from 28th March 1843 and the most recently published manuscript was from 3rd June 2021. Search terms included cancrum oris and noma. Data was extracted using a standardised data extraction tool and key areas of interest were identified. The Preferred Reporting Items for Systemic review and Meta-Analyses requirements were followed. RESULTS: The review included 147 articles, the majority of the studies (n = 94, 64%) were case reports. Most manuscripts (n = 81, 55%) were published in the 2000s, 49 (33%) were from the 1900s and 17 (12%) from the 1800s. The main areas of interest identified were the history and epidemiology of the disease, noma's clinical progression and aetiology, treatment regimens, mortality rates and the risk factors for the development of noma. CONCLUSIONS: Noma has been reported in the literature for hundreds of years; however important gaps in our understanding of the disease remain. Future research should focus on determining the burden and distribution of disease; the true mortality rate, pathogenic cause(s) and the factors that influence prognosis and outcomes after treatment.

Model of care, Noma Children's Hospital, northwest Nigeria.

The Nigerian Ministry of Health has been offering care for noma patients for many years at the Noma Children's Hospital (NCH) in Sokoto, northwest Nigeria, and Médecins Sans Frontières has supported these initiatives since 2014. The comprehensive model of care consists of four main components: acute care, care for noma sequelae, integrated hospital-based services and community-based services. The model of care is based on the limited evidence available for prevention and treatment of noma and follows WHO's protocols for acute patients and best practice guidelines for the surgical treatment of noma survivors. The model is updated continually as new evidence becomes available, including evidence generated through the operational research studies performed at NCH. By describing the model of care, we wish to share the lessons learned with other actors working in the noma and neglected tropical disease sphere in the hope of guiding programme development.

Filtering the Junk: Assigning Function to the Mosquito Non-Coding Genome.

The portion of the mosquito genome that does not code for proteins contains regulatory elements that likely underlie variation for important phenotypes including resistance and susceptibility to infection with arboviruses and Apicomplexan parasites. Filtering the non-coding genome to uncover these functional elements is an expanding area of research, though identification of non-coding regulatory elements is challenging due to the lack of an amino acid-like code for the non-coding genome and a lack of sequence conservation across species. This review focuses on three types of non-coding regulatory elements: (1) microRNAs (miRNAs), (2) long non-coding RNAs (lncRNAs), and (3) enhancers, and summarizes current advances in technical and analytical approaches for measurement of each of these elements on a genome-wide scale. The review also summarizes and highlights novel findings following application of these techniques in mosquito-borne disease research. Looking beyond the protein-coding genome is essential for understanding the complexities that underlie differential gene expression in response to arboviral or parasite infection in mosquito disease vectors. A comprehensive understanding of the regulation of gene and protein expression will inform transgenic and other vector control methods rooted in naturally segregating genetic variation.

Anopheles ecology, genetics and malaria transmission in northern Cambodia.

In the Greater Mekong Subregion, malaria cases have significantly decreased but little is known about the vectors or mechanisms responsible for residual malaria transmission. We analysed a total of 3920 Anopheles mosquitoes collected during the rainy and dry seasons from four ecological settings in Cambodia (villages, forested areas near villages, rubber tree plantations and forest sites). Using odor-baited traps, 81% of the total samples across all sites were collected in cow baited traps, although 67% of the samples attracted by human baited traps were collected in forest sites. Overall, 20% of collected Anopheles were active during the day, with increased day biting during the dry season. 3131 samples were identified morphologically as 14 different species, and a subset was also identified by DNA amplicon sequencing allowing determination of 29 Anopheles species. The investigation of well characterized insecticide mutations (ace-1, kdr, and rdl genes) indicated that individuals carried mutations associated with response to all the different classes of insecticides. There also appeared to be a non-random association between mosquito species and insecticide resistance with Anopheles peditaeniatus exhibiting nearly fixed mutations. Molecular screening for Plasmodium sp. presence indicated that 3.6% of collected Anopheles were positive, most for P. vivax followed by P. falciparum. These results highlight some of the key mechanisms driving residual human malaria transmission in Cambodia, and illustrate the importance of diverse collection methods, sites and seasons to avoid bias and better characterize Anopheles mosquito ecology in Southeast Asia.

Outcomes at 18 mo of 37 noma (cancrum oris) cases surgically treated at the Noma Children's Hospital, Sokoto, Nigeria.

BACKGROUND: Noma is a rapidly progressing infection of the oral cavity frequently resulting in severe facial disfigurement. We present a case series of noma patients surgically treated in northwest Nigeria. METHODS: A retrospective analysis of routinely collected data (demographics, diagnosis and surgical procedures undergone) and in-person follow-up assessments (anthropometry, mouth opening and quality of life measurements) were conducted with patients who had surgery >6 mo prior to data collection. RESULTS: Of the 37 patients included, 21 (56.8%) were male and 22 (62.9%) were aged >6 y. The median number of months between last surgery and follow-up was 18 (IQR 13, 25) mo. At admission, the most severely affected anatomical area was the outer cheek (n = 9; 36.0% of patients had lost between 26% and 50%). The most frequent surgical procedures were the deltopectoral flap (n = 16; 43.2%) and trismus release (n = 12; 32.4%). For the eight trismus-release patients where mouth opening was documented at admission, all had a mouth opening of 0-20 mm at follow-up. All patients reported that the surgery had improved their quality of life. CONCLUSIONS: Following their last surgical intervention, noma patients do experience some improvements in their quality of life, but debilitating long-term sequelae persist.

The prevalence of noma in northwest Nigeria.

Background: Noma, a rapidly progressing infection of the oral cavity, mainly affects children. The true burden is unknown. This study reports estimated noma prevalence in children in northwest Nigeria. Methods: Oral screening was performed on all ≤15 year olds, with caretaker consent, in selected households during this cross-sectional survey. Noma stages were classified using WHO criteria and caretakers answered survey questions. The prevalence of noma was estimated stratified by age group (0-5 and 6-15 years). Factors associated with noma were estimated using logistic regression. Results: A total of 177 clusters, 3499 households and 7122 children were included. In this sample, 4239 (59.8%) were 0-5 years and 3692 (52.1%) were female. Simple gingivitis was identified in 3.1% (n=181; 95% CI 2.6 to 3.8), acute necrotising gingivitis in 0.1% (n=10; CI 0.1 to 0.3) and oedema in 0.05% (n=3; CI 0.02 to 0.2). No cases of late-stage noma were detected. Multivariable analysis in the group aged 0-5 years showed having a well as the drinking water source (adjusted odds ratio (aOR) 2.1; CI 1.2 to 3.6) and being aged 3-5 years (aOR 3.9; CI 2.1 to 7.8) was associated with being a noma case. In 6-15 year olds, being male (aOR 1.5; CI 1.0 to 2.2) was associated with being a noma case and preparing pap once or more per week (aOR 0.4; CI 0.2 to 0.8) was associated with not having noma. We estimated that 129120 (CI 105294 to 1 52 947) individuals <15 years of age would have any stage of noma at the time of the survey within the two states. Most of these cases (93%; n=120 082) would be children with simple gingivitis. Conclusions: Our study identified a high prevalence of children at risk of developing advanced noma. This disease is important but neglected and therefore merits inclusion in the WHO neglected tropical diseases list.

Language and beliefs in relation to noma: a qualitative study, northwest Nigeria.

BACKGROUND: Noma is an orofacial gangrene that rapidly disintegrates the tissues of the face. Little is known about noma, as most patients live in underserved and inaccessible regions. We aimed to assess the descriptive language used and beliefs around noma, at the Noma Children's Hospital in Sokoto, Nigeria. Findings will be used to inform prevention programs. METHODS: Five focus group discussions (FGD) were held with caretakers of patients with noma who were admitted to the hospital at the time of interview, and 12 in-depth interviews (IDI) were held with staff at the hospital. Topic guides used for interviews were adapted to encourage the natural flow of conversation. Emergent codes, patterns and themes were deciphered from the data derived from IDI's and FGDs. RESULTS: Our study uncovered two main themes: names, descriptions and explanations for the disease, and risks and consequences of noma. Naming of the disease differed between caretakers and heath care workers. The general names used for noma illustrate the beliefs and social system used to explain the disease. Beliefs were varied; participant responses demonstrate a wide range of understanding of the disease and its causes. Difficulty in accessing care for patients with noma was evident and the findings suggest a variety of actions taking place before reaching a health center or health worker. Patient caretakers mentioned that barriers to care included a lack of knowledge regarding this medical condition, as well as a lack of trust in seeking medical care. Participants in our study spoke of the mental health strain the disease placed on them, particularly due to the stigma that is associated with noma. CONCLUSIONS: Caretaker and practitioner perspectives enhance our understanding of the disease in this context and can be used to improve treatment and prevention programs, and to better understand barriers to accessing health care. Differences in disease naming illustrate the difference in beliefs about the disease. This has an impact on health seeking behaviours, which for noma cases has important ramifications on outcomes, due to the rapid progression of the disease.

'I treat it but I don't know what this disease is': a qualitative study on noma (cancrum oris) and traditional healing in northwest Nigeria.

BACKGROUND: Noma, a neglected disease mostly affecting children, with a 90% mortality rate if untreated, is an orofacial gangrene that disintegrates the tissues of the face in <1 wk. Noma can become inactive with early stage antibiotic treatment. Traditional healers, known as mai maganin gargajiya in Hausa, play an important role in the health system and provide care to noma patients. METHODS: We conducted 12 in-depth interviews with caretakers who were looking after noma patients admitted at the Noma Children's Hospital and 15 traditional healers in their home villages in Sokoto state, northwest Nigeria. We explored perceptions of noma, relationship dynamics, healthcare practices and intervention opportunities. Interviews were audiorecorded, transcribed and translated. Manual coding and thematic analysis were utilised. RESULTS: Traditional healers offered specialised forms of care for specific conditions and referral guidance. They viewed the stages of noma as different conditions with individualised remedies and were willing to refer noma patients. Caretakers trusted traditional healers. CONCLUSIONS: Traditional healers could play a crucial role in the early detection of noma and the health-seeking decision-making process of patients. Intervention programmes should include traditional healers through training and referral partnerships. This collaboration could save lives and reduce the severity of noma complications.