Maguire Brittany J, Shrestha Rujan, Dahal Prabin, Ngu Roland, Nizigama Lionel, Rashan Sumayyah, Shrestha Poojan, Harriss Elinor, Newton Paul, Makino Yuka, Varenne Benoit, Guerin Philippe J
Infectious Diseases Data Observatory (IDDO), University of Oxford, Oxford, Oxfordshire, UK.
Centre for Tropical Medicine and Global Health, Nuffield Department of Medicine, University of Oxford, Oxford, Oxfordshire, UK.
BMJ Glob Health. 2025 Jul 31;10(7):e018023. doi: 10.1136/bmjgh-2024-018023.
Noma (cancrum oris) is a severe gangrenous disease of the mouth and oro-facial structures. Noma often affects young children living in extreme poverty, malnutrition and poor sanitation. Gaps remain in understanding its aetiology, pathogenesis, prevention and treatment.
We systematically searched databases for all primary research studies (clinical trials, cohort studies, case-control, cross-sectional, other observational studies, case studies/series) reporting noma patients of any age up to 7 December 2022. The 366 publications (published between 1839 and 2022) included in our scoping review describe 15 082 patients. Although 53 cohort and 29 cross-sectional studies were identified, enrolling 13 489 patients, interventional research remains extremely limited, with only six studies identified (101 patients, range: 7-26) and only one in the past decade, highlighting a critical gap in treatment evaluation. A total of 380 different treatment modalities were described, which underscores lack of a standardised practice. Disease aetiology remains unclear, with 117 microorganisms reported across 113 studies, yet none more consistently linked to noma development. Since 2000, 91.2% of cases have been reported in Sub-Saharan Africa, though occurrences outside the 'noma belt' and into Asia and the Americas suggest a broader risk. The 212 potential risk factors identified in 269 (73.5%) publications reflect substantial heterogeneity, complicating efforts to determine definitive causative factors. Additionally, the inconsistent definition and reporting of noma staging significantly hinder comparability across studies, with wide adoption of the WHO staging classification needed.
This comprehensive review of the literature underscores the urgent need for robust, policy-driven research to address the vast knowledge gaps in the physiopathology of noma and the limited evidence currently available to guide therapeutic and preventive policies. Collective action and increased research investment are crucial, especially now that noma is officially recognised as a neglected tropical disease by the WHO.
坏疽性口炎（走马疳）是一种发生于口腔和口面部结构的严重坏疽性疾病。坏疽性口炎常影响生活在极度贫困、营养不良和卫生条件差环境中的幼儿。在其病因、发病机制、预防和治疗方面仍存在认知空白。
我们系统检索了数据库中截至2022年12月7日报告的所有年龄段坏疽性口炎患者的原发性研究（临床试验、队列研究、病例对照研究、横断面研究、其他观察性研究、病例研究/系列）。我们的范围综述纳入的366篇出版物（发表于1839年至2022年之间）描述了15082例患者。虽然确定了53项队列研究和29项横断面研究，纳入了13489例患者，但干预性研究仍然极为有限，仅确定了6项研究（101例患者，范围：7 - 26例），且在过去十年中仅有1项，这凸显了治疗评估方面的关键差距。共描述了380种不同的治疗方式，这突出表明缺乏标准化的治疗方法。疾病病因仍不明确，113项研究报告了117种微生物，但没有一种与坏疽性口炎的发生有更一致的关联。自2000年以来，91.2%的病例报告来自撒哈拉以南非洲，不过在“坏疽性口炎带”之外以及亚洲和美洲出现的病例表明风险范围更广。在269篇（73.5%）出版物中确定的212个潜在风险因素反映出很大的异质性，使得确定明确的致病因素变得复杂。此外，坏疽性口炎分期的定义和报告不一致，严重阻碍了各研究之间的可比性，因此需要广泛采用世界卫生组织的分期分类。
这项全面的文献综述强调迫切需要开展强有力的、政策驱动的研究，以填补坏疽性口炎生理病理学方面巨大的知识空白，以及目前指导治疗和预防政策的证据有限的问题。集体行动和增加研究投入至关重要，特别是现在坏疽性口炎已被世界卫生组织正式确认为一种被忽视的热带病。
