|Year : 2017 | Volume
| Issue : 2 | Page : 101-105
Pattern of tissue destruction among patients diagnosed with cancrum oris (Noma) at a Northwestern Nigerian Hospital, Sokoto
Semiu Adetunji Adeniyi1, Abdurazzaq Olanrewaju Taiwo2, Adebayo Aremu Ibikunle2, Ramat Oyebunmi Braimah2, Olalekan Micah Gbotolorun2, Mike Eghosa Ogbeide2, Lateef Alani Yekini2, F Moshood Adeyemi3
1 Department of Clinical Services, Noma Children Hospital, Sokoto, Nigeria
2 Department of Surgery, Dental and Maxillofacial Surgery, Usmanu Danfodiyo University Teaching Hospital, Usmanu Danfodiyo University, Sokoto, Nigeria
3 Department of Surgery, University of Ilorin Teaching Hospital, Ilorin, Kwara, Nigeria
|Date of Web Publication||25-Jul-2017|
Adebayo Aremu Ibikunle
Department of Surgery, Dental and Maxillofacial Surgery, Usmanu Danfodiyo University Teaching Hospital, Usmanu Danfodiyo University, Sokoto
Source of Support: None, Conflict of Interest: None
Background: Cancrum oris is a debilitating and rapidly progressive gangrenous disease. It is life-threatening and results in severe devastation. Tissue loss constitutes a formidable cause of morbidity in patients and also poses an enormous challenge to a reconstructive surgeon. This study aims to describe the pattern of tissue loss among patients with cancrum oris at our center.
Methods: A review of the data of patients treated for cancrum oris at Noma Children Hospital, Sokoto, Nigeria, from December 1999 to 2011, was done. Data on the age, gender, and pattern of tissue loss were retrieved. The pattern of tissue loss was assessed anatomically and clinically. Analysis of data was done using SPSS (IBM SPSS Statistics for Windows Version 20 [Armonk, NY: IBM Corp]).
Results: A total of 159 cases of cancrum oris were included in this study. The age range was 1–33 years with a mean (± standard deviation) of 3.5 (3.23) years. A male/female ratio of 1:1.2 was observed. A total of 425 sites were affected in 159 patients. In 109 (68.6%) patients, only soft tissue involvement was seen, while a combination of soft and hard tissue destructions was observed in 50 (31.4%) patients. The most frequently affected site was the cheek, 268 (63.1%), followed by the lips, 106 (66.7%). The nose was affected in 40 (25.2%) patients. Osseous involvement was observed in 50 (7.6%) patients.
Conclusion: Cancrum oris, though considered to be a rare disease, continues to constitute a grievous cause of orofacial tissue loss and consequent morbidity in our environment.
Keywords: Cancrum oris, Noma, pattern of tissue destruction, tissue loss
|How to cite this article:|
Adeniyi SA, Taiwo AO, Ibikunle AA, Braimah RO, Gbotolorun OM, Ogbeide ME, Yekini LA, Adeyemi F M. Pattern of tissue destruction among patients diagnosed with cancrum oris (Noma) at a Northwestern Nigerian Hospital, Sokoto. Saudi J Oral Sci 2017;4:101-5
|How to cite this URL:|
Adeniyi SA, Taiwo AO, Ibikunle AA, Braimah RO, Gbotolorun OM, Ogbeide ME, Yekini LA, Adeyemi F M. Pattern of tissue destruction among patients diagnosed with cancrum oris (Noma) at a Northwestern Nigerian Hospital, Sokoto. Saudi J Oral Sci [serial online] 2017 [cited 2021 Apr 22];4:101-5. Available from: https://www.saudijos.org/text.asp?2017/4/2/101/211567
| Introduction|| |
Cancrum oris is a rapidly spreading gangrenous disease that typically runs a fulminant course, destroying both soft and hard tissues in its path, thereby resulting in crippling esthetic, functional limitations and diminished quality of life where the patient survives.,, Its etiology is multifactorial, with socioeconomic, immunological, and microbial factors implicated. However, specific bacterial organisms such as Erysipelothrix rhusiopathiae, Prevotella spp., Veinonella spp., and Fusobacterium spp. among others have been linked with its etiology., It is a disease of poverty; hence, pockets of it exist in Sub-Saharan Africa where food security is a formidable challenge.,, The disease is commonly seen among children aged between 2 and 6 years, who are typically malnourished and are from indigent families.,
The associated tissue loss is known to transcend anatomical barriers, i.e., involves hard and soft tissues [Figure 1] and [Figure 2]., Studies have shown that anatomic extent of tissue loss correlates with the type of reconstruction that will be done.,, Hence, the purpose of this study is to describe the pattern of tissue loss in patients who presented with cancrum oris at our center.
|Figure 1: A female with soft and hard tissue loss involving the lips, left cheek, nose, and maxillary alveolus|
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|Figure 2: A male child with extensive tissue loss involving the right cheek|
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| Methods|| |
A review of the data of patients treated for cancrum oris at Noma Children Hospital, Sokoto, Nigeria, from December 1999 to 2011, was done. Data on the age, gender, and pattern of tissue loss were retrieved. The pattern of tissue loss was assessed anatomically and clinically. Soft tissue loss was classified as mild (<25% of anatomical site tissue loss); moderate (25%–50% of anatomical site tissue loss); severe (51%–75% of anatomical site tissue loss); and very severe (76%–100% of anatomical site tissue loss). The side affected was also documented and labeled as right- or left-sided soft tissue/osseous involvement. Analysis of data was done using SPSS (IBM SPSS Statistics for Windows Version 20 [Armonk, NY: IBM Corp]).
| Results|| |
A total of 159 patients with acute cancrum oris were seen over a period of 12 years, giving an average of 14 patients per annum. The age range was 1–33 years with a mean (± standard deviation [SD]) of 3.5 (3.23) years. There was a slight female preponderance with a male/female ratio of 1:1.2. A total of 425 sites were affected in 159 patients, giving an average of 2.7 sites per patient. In 109 (68.6%) patients, only soft tissue involvement was seen, while a combination of soft and hard tissue destruction was observed in 50 (31.4%) patients [Figure 3]. Overall, right-sided tissue destruction, i.e., 83 (52.2%), was more commonly encountered though it was less frequently affected in isolation compared with left-sided tissue destruction [Figure 4]. Isolated left-sided tissue destruction was seen in 57 (35.8%) of patients, while isolated right-sided tissue involvement was observed in 47 (29.6%) patients. Bilateral tissue involvement was observed in 55 (34.6%) patients.
The most frequently affected site was the cheek, which accounted for 268 (57.1%) of all the sites affected [Figure 5]. The lips were affected in 106 (22.6%) patients. The upper lip was more frequently involved than the lower lip, being affected in 63 (13.4%) and 53 (11.3%) patients, respectively [Figure 5].
Osseous involvement was observed in 50 (7.6%) patients affecting only one osseous site per patient in 32 (20.1%) cases. Multiple osseous sites were affected in 18 (11.3%) patients. The maxilla was the most commonly involved bone and it was affected in 38 (23.9%) patients [Figure 5]. The nasal bone was affected in 7 (4.4%) patients, both mandible and maxilla were involved in 6 (3.7%) patients, while the palate was affected in 4 (2.5%) patients and the nasal bone was affected in 7 (4.4%) patients. The vomer bone was affected in 3 (1.9%) patients who also had total loss of the premaxilla bilaterally.
While upper lip involvement was observed, majority of the patients presented with either mild 25 (39.7%) or moderate 26 (41.3%) tissue loss [Figure 6]. Total loss of the upper and lower lips was observed in 4 (0.9%) and 2 (0.5%) patients, respectively [Figure 6]. The nose was the least frequently affected site in 40 (25.2%) patients. Majority of the patients who presented with nasal tissue destruction experienced moderate nasal tissue loss, 20 (50%) [Figure 6]. Total loss of the nasal tissues was encountered in 4 (0.9%) patients. The cartilaginous nasal septum was affected in 32 (20.1%) patients, with its total loss in 26 (16.4%) patients.
| Discussion|| |
The average number of patients seen per annum in this study is much lower than figures reported about two decades ago by Enwonwu et al., from a study in the same locale where this present study was conducted, where an average of 55–75 cases of cancrum oris cases was seen annually., This significant decline in the number of cancrum oris cases as revealed by this study may be an expression of improved economic indices and immunization coverage in this part of Nigeria.,,,
However, the average number of cancrum oris recorded in this study is higher than the figures reported by Obiechina et al., who encountered 173 cases over a period of 16 years, thus seeing an average of 11 patients per annum. Notably, their study was done in the more economically buoyant southwestern part of Nigeria. In addition, their study was done at a time of great economic distress for Nigerians, which was mainly in the 1980s and 1990s., Adeola et al. in a review of cancrum oris among Nigerian children observed a progressive decline in the number of patients diagnosed with cancrum oris from 1991 to 2000. The worldwide incidence of this disabling disease is still obscure because a significant percentage of patients do not seek orthodox care or do not seek care at all. Indeed, it is estimated that over 85% of cases do not seek orthodox health care.
The age range of patients seen in this study was 1–33 years with majority of them <5 years old, with a mean (± SD) of 3.5 (3.23) years. This corresponds to reports from previous studies where most of the patients seen are <6 years old and the modal age group is 3–5 years. Only two patients were older than 18 years. Although this disease is said to be infrequent among adults, it may occur nonetheless. When seen in adults, retroviral tests are advocated to rule out human immunodeficiency virus infection., This observation may be attributable to the susceptibility of young children to adverse effects of poverty and malnutrition, especially in societies with scarce or trapped resources.,, Atypically, two cases were seen in adults in this study, indicating that this disease is not limited to childhood. This is in agreement with the assertion that 90% of cancrum oris cases develop in the first decade of life., A slight female preponderance was observed in this study. This is dissimilar to the report by Adeola et al., who reported a male to female ratio of 1.5:1. However, some other studies have reported equal sex predilection or a slight male predominance.,,, We propose that the female predominance observed in this study may be because more females tend to survive cancrum oris and not necessarily because females are more often affected than males by this disease.
An average of 2.7 sites per patient was observed in this study. This is a reflection of the aggressive nature of this disease and perhaps the general inclination of patients for late presentation in this environment.,, Tissues on the right facial region were slightly more frequently affected than those on the left. Bilateral extensive tissue destruction was observed in a significant number of patients in contrast with reports from the literature.
The most frequently affected site was the cheek, with the outer and inner cheeks affected with equal frequency. This is in agreement with other reports by Bourgeois et al. in a Senegalese study. This is probably due to the nature of spread of this disease, which often commences as gingivitis in the premolar/molar region; thereafter, it widens to involve the buccal vestibule and thence to the labial/cheek mucosa. Hence, the cheek is commonly involved and this corresponds with the findings of this study.
In this study, the lips were affected in a significant proportion of patients though it was not the modal site of involvement. This may be related to the fact that the lips are closely related to the cheeks and contiguous spread of the disease can easily involve the lips. The nose was infrequently affected, which agrees with other reports in the literature.,
The frequency of osseous involvement observed in this study was much lower than that reported by Lazarus and Hudson, who in a review of 26 cases reported osseous involvement in 69.2% of the cases. The maxilla was the most frequently affected bone in this series, which is consistent with other reports in the literature.,,
Reconstruction of orofacial defects secondary to cancrum oris is quite challenging, especially where composite tissue or complex tissue architecture is lost. Reconstruction may be single-staged or multi-staged depending on a number of factors. Some authors have recommended the one-staged reconstruction model in resource-limited settings. Options for reconstruction include local flaps, regional flaps, distant flaps, and microvascular surgery., Prosthetic devices have also been used with varying degrees of success.
| Conclusion|| |
Cancrum oris, though considered to be a rare disease, continues to constitute a grievous cause of orofacial tissue loss and consequent morbidity in our environment. Rehabilitation of patients is quite challenging, especially where there is extensive composite tissue loss. There is an urgent need to address predisposing factors as this is a highly preventable disease. In addition, early treatment and initiation of full rehabilitation of survivors are paramount.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Reddy BP, Reddy BS, Kiran G, Chembolu N. Cancrum oris: A devastating orofacial gangrene. J Dr NTR Univ Health Sci 2012;1:192.
Obiechina AE, Arotiba JT, Fasola AO. Cancrum oris (noma): Level of education and occupation of parents of affected children in Nigeria. Odontostomatol Trop 2000;23:11-4.
Vaidya S, Agrawal A, Sharma VK. Cancrum oris: A case report. Indian J Otolaryngol Head Neck Surg 2006;58:411-3.
Whiteson KL, Lazarevic V, Tangomo-Bento M, Girard M, Maughan H, Pittet D, et al.
Noma affected children from Niger have distinct oral microbial communities based on high-throughput sequencing of 16S rRNA gene fragments. PLoS Negl Trop Dis 2014;8:e3240.
Falkler WA Jr., Enwonwu CO, Idigbe EO. Microbiological understandings and mysteries of noma (Cancrum oris). Oral Dis 1999;5:150-5.
Enwonwu CO. Infectious oral necrosis (Cancrum oris) in Nigerian children: A review. Community Dent Oral Epidemiol 1985;13:190-4.
Enwonwu CO. Noma – The ulcer of extreme poverty. N Engl J Med 2006;354:221-4.
Maley A, Desai M, Parker S. Noma: A disease of poverty presenting at an urban hospital in the United States. JAAD Case Rep 2014;1:18-20.
Denloye OO, Aderinokun GA, Lawoyin JO, Bankole OO. Reviewing trends in the incidence of cancrum oris in Ibadan, Nigeria. West Afr J Med 2003;22:26-9.
Evrard L, Laroque G, Glineur R, Daelemans P. Noma: Clinical and evolutive aspect. Acta Stomatol Belg 1996;93:17-20.
Dean JA, Magee W. One-stage reconstruction for defects caused by cancrum oris (noma). Ann Plast Surg 1997;38:29-35.
McGurk M, Marck R. Treatment of noma: Medical missions in Ethiopia. Br Dent J 2010;208:179-82.
Adekeye EO, Ord RA. Cancrum oris: Principles of management and reconstructive surgery. J Maxillofac Surg 1983;11:160-70.
Enwonwu CO, Phillips RS, Ferrell CD. Temporal relationship between the occurrence of fresh noma and the timing of linear growth retardation in Nigerian children. Trop Med Int Health 2005;10:65-73.
Enwonwu CO, Falkler WA Jr., Phillips RS. Noma (cancrum oris). Lancet 2006;368:147-56.
Girei AA, Dire B, Bello BH. Economics of cattle marketing on the socio-economic characteristics of cattle marketers in central zone of Adamawa State, Nigeria. Int J Adv Agric Res 2014;2:1-7.
Weisbrot, M. and Ray, R. The Scorecard on Development, 1960-2010: Closing the Gap? Washington DC: Center for Economic and Policy Research; 2011.
Ophori EA, Tula MY, Azih AV, Okojie R, Ikpo PE. Current trends of immunization in Nigeria: Prospect and challenges. Trop Med Health 2014;42:67-75.
Oche MO, Umar AS, Ibrahim MT, Sabitu K. An assessment of the impact of health education on maternal knowledge and practice of childhood immunization in Kware, Sokoto State. J Public Health Epidemiol 2011;3:440-7.
Lewis P. From prebendalism to predation: The political economy of decline in Nigeria. J Mod Afr Stud 1996;34:79-103.
Adeola DS, Eguma SA, Ononiwu CN. Cancrum oris among Nigerian children. Niger J Surg Res 2004;6:1-2.
Tonna JE, Lewin MR, Mensh B. A case and review of noma. PLoS Negl Trop Dis 2010;4:e869.
Chidzonga MM, Mahomva L. Noma (cancrum oris) in human immunodeficiency virus infection and acquired immunodeficiency syndrome (HIV and AIDS): Clinical experience in Zimbabwe. J Oral Maxillofac Surg 2008;66:475-85.
Jordans MJ, Tol WA, Komproe IH, Susanty D, Vallipuram A, Ntamatumba P, et al.
Development of a multi-layered psychosocial care system for children in areas of political violence. Int J Ment Health Syst 2010;4:15.
Betancourt TS, Meyers-Ohki SE, Charrow AP, Tol WA. Interventions for children affected by war: An ecological perspective on psychosocial support and mental health care. Harv Rev Psychiatry 2013;21:70-91.
Bain LE, Awah PK, Geraldine N, Kindong NP, Sigal Y, Bernard N, et al.
Malnutrition in Sub-Saharan Africa: Burden, causes and prospects. Pan Afr Med J 2013;15:120.
Auluck A, Pai KM. Noma: Life cycle of a devastating sore – Case report and literature review. J Can Dent Assoc 2005;71:757.
Adolph HP, Yugueros P, Woods JE. Noma: A review. Ann Plast Surg 1996;37:657-68.
Adeola DS, Obiadazie AC. Protocol for managing acute cancrum oris in children: An experience in five cases. Afr J Paediatr Surg 2009;6:77-81.
] [Full text]
Oginni FO, Oginni AO, Ugboko VI, Otuyemi OD. A survey of cases of cancrum oris seen in Ile-Ife, Nigeria. Int J Paediatr Dent 1999;9:75-80.
Shambe IH. Review of article palliative care in Nigeria: Challenges and prospects. Jos J Med2014;8:53-5.
Ibikunle AA, Taiwo AO, Gbotolorun OM, Braimah RO. Challenges in the management of cervicofacial necrotizing fasciitis in Sokoto, Northwest Nigeria. J Clin Sci2016;13:143. [Full text]
Bourgeois DM, Diallo B, Frieh C, Leclercq MH. Epidemiology of the incidence of oro-facial noma: A study of cases in Dakar, Senegal, 1981-1993. Am J Trop Med Hyg 1999;61:909-13.
Ashok N, Tarakji B, Darwish S, Rodrigues JC, Altamimi MA. A review on noma: A recent update. Glob J Health Sci 2015;8:53-9.
Lazarus D, Hudson DA. Cancrum oris – A 35-year retrospective study. S Afr Med J 1997;87:1379-82.
Agbara R, Obiadazie AC, Fomete B, Omeje KU. Orofacial soft tissue reconstruction with locoregional flaps in a health resource-depleted environment: Experiences from Nigeria. Arch Plast Surg 2016;43:265-71.
Ayoub A. Innovation in the reconstruction of orofacial region: Challenges and opportunities. Natl J Maxillofac Surg 2012;3:1.
] [Full text]
Guttal KS, Naikmasur VG, Rao CB, Nadiger RK, Guttal SS. Orofacial rehabilitation of patients with post-cancer treatment – An overview and report of three cases. Indian J Cancer 2010;47:59-64.
] [Full text]
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]