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 Table of Contents  
ORIGINAL ARTICLE
Year : 2017  |  Volume : 5  |  Issue : 2  |  Page : 172-176

Management of 159 cases of acute cancrum oris: Our experience at the noma children hospital, Sokoto


1 Department of Surgery/Dental and Maxillofacial Surgery, Usmanu Danfodiyo University/Usmanu Danfodiyo University Teaching Hospital, Sokoto, Nigeria
2 Department of Clinical Services, NOMA Children Hospital, Sokoto, Nigeria
3 Department of Oral and Maxillofacial Surgery, Lagos University Teaching Hospital, Lagos, Nigeria
4 Department of Surgery, Obafemi Awolowo University Teaching Hospital, Ile-ife, Nigeria
5 Department of Dental And Maxillofacial Surgery, Usmanu Danfodiyo University Teaching Hospital, Sokoto, Nigeria

Date of Web Publication15-Dec-2017

Correspondence Address:
Adebayo Aremu Ibikunle
Department of Surgery/Dental and Maxillofacial Surgery, Usmanu Danfodiyo University/Usmanu Danfodiyo University Teaching Hospital, Sokoto
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/amhs.amhs_23_17

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  Abstract 

Background: Cancrum oris (Noma) is a rapidly progressive gangrenous infection of the oral cavity and its surrounding structures, which typically results in destruction of both soft and hard tissues. It requires urgent intervention, especially while still in the acute phase. We present an overview of our experience in the management of acute cancrum oris at the Noma children hospital, which is a regional referral center for such cases in Nigeria. Materials and Methods: This was a retrospective study of 159 patients managed for acute cancrum oris at the Noma children hospital, Sokoto, Nigeria. Early recognition of the disease, baseline investigations, identification of underlying disease/diseases, resuscitation of the patient, and institution of specific treatment were done. Results: The age range was 1–33 years with a mean (±standard deviation [SD]) of 3.5 (3.23) years. The male:female ratio was 1:1.2. Comorbidities were observed in 148 (93.1%) patients. The most commonly seen comorbidities observed were measles and protein–energy malnutrition, which were seen in 75 (47.2%) and 67 (42.1%) cases, respectively. The mean (±SD) hemoglobin of 8.59 (±2.9) g/dl and 11.38 (±1.5) g/dl were recorded on admission and at discharge, respectively. Leukocytosis of >12,000 cells/mm3 was observed in all patients. The electrolyte urea/creatine results were generally within normal range, while serum urea was raised in 52 (32.7%) patients. The duration of admission was for an average of 13 days. A mortality rate of 18.2% was observed. Conclusion: Acute cancrum oris requires urgent management. For successful management, patients should be evaluated for underlying diseases and systemic causes of immunosuppression.

Keywords: Acute Cancrum Oris, children, management


How to cite this article:
Ibikunle AA, Adeniyi SA, Taiwo AO, Braimah RO, Gbotolorun OM, Soyele OO, Adeyemi M, Amoo A, Jaffar R, Bashir M. Management of 159 cases of acute cancrum oris: Our experience at the noma children hospital, Sokoto. Arch Med Health Sci 2017;5:172-6

How to cite this URL:
Ibikunle AA, Adeniyi SA, Taiwo AO, Braimah RO, Gbotolorun OM, Soyele OO, Adeyemi M, Amoo A, Jaffar R, Bashir M. Management of 159 cases of acute cancrum oris: Our experience at the noma children hospital, Sokoto. Arch Med Health Sci [serial online] 2017 [cited 2018 Sep 22];5:172-6. Available from: http://www.amhsjournal.org/text.asp?2017/5/2/172/220823




  Introduction Top


Cancrum oris (Noma) is a relentless, gangrenous and aggressive tissue infection of the orofacial region, which often results in severe impairment of patients' quality of life and high mortality rates.[1],[2] It has been heavily linked with poverty, malnutrition and exanthematous diseases.[2],[3] Children between the ages of 2-9 years are commonly affected, although some cases among adults have been reported.[1],[2],[3] Varying mortality rates of 70-90% have been reported, especially when the disease is allowed to run its natural course natural course.[2],[3] Where the patient survives, they are often left with disfigurement and debilitating complications.[4],[5] This disease has a rapid acute phase, characterized by intense inflammation and ulceration.[1],[4],[5] This is then followed by the chronic phase, where the ulcer and tissue destruction begins to heal.[1] The promptness and mode of management especially in the acute phase is an important determinant of patients' survival and postillness quality of life.[1],[6]

To reduce the mortality and morbidity associated with this devastating disease, early and aggressive management of patients in acute phase of the disease is of utmost importance.[1],[6] We describe our experience with the management of acute cancrum oris at our center.


  Materials and Methods Top


This was a retrospective study in which a review of the management of patients diagnosed with acute cancrum oris at the Noma Children Hospital, Sokoto, Nigeria, over a period of 12 years was done. Age, gender, comorbidity, extent of tissue destruction, mode of intervention, complications, and number of deaths were recorded.

Full blood count, electrolyte and urea/creatine (EU/Cr), microscopy culture and sensitivity (MCS) were done. Diagnosis was made clinically in all cases. Ethical approval was obtained through the management of Noma Children Hospital, Sokoto, Nigeria.


  Results Top


Patients' characteristics

One hundred and fifty-nine patients diagnosed with acute cancrum oris were included in this study. The age range was 1–33 years with a mean (±standard deviation [SD]) of 3.5 (3.23) years. Nearly 87% of them were below 6 years of age [Table 1]. The male:female ratio was 1:1.2. At the time of presentation, comorbidities were observed in 148 (93.1%) patients. The most commonly seen comorbidities observed were measles and protein–energy malnutrition, which were seen in 75 (47.2%) and 67 (42.1%) cases, respectively. Others were gastroenteritis, whooping cough, and retroviral infection, which were observed in 3 (1.9%), 2 (1.3%), and 1 (0.6%) patients, respectively. Importantly, 11 (6.9%) cases had no identified comorbidities [Figure 1].
Table 1: The distribution of patients by age and sex

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Figure 1: Proportion of patients with identified comorbidities

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The mean (±SD) hemoglobin levels of 8.59 (±2.9) g/dl and 11.38 (±1.5) g/dl were recorded on admission and at discharge, respectively. Hemoglobin level was <10 g/dl in 103 patients. Leukocytosis of >12,000 cells/mm 3 was observed in all patients. MCS reported no growth for all specimens sent. The EU/Cr results were generally within normal range, while serum urea was raised in 52 (32.7%) patients. The duration of admission was for an average of 13 days. The observed mortality rate in this study was 29 (18.2%).

Presentation

In general, the patients presented with severe pain and orofacial ulcerations of short duration, ranging from 3 to 7 days. The children were frequently irritable and drooled saliva. They often had sparse brownish hair, protuberant abdomen, and dry wrinkled skin and lips [Figure 2]. In addition, they were typically small for age and irritable. A purplish-black roughly oval skin discoloration or ulceration was often present on the extraorally corresponding with the intraoral site of the lesion. Classically, it exhibits what might be referred to as three concentric circles; the central circle was the darkest, often black/dark brown in color, nonresponsive to pinprick, but still tender to palpation [Figure 3]. The intermediate circle was typically pale with overlying slough, while the outermost circle was a ring of normal cutaneous tissue. It was invariably tender to palpation although nonresponsive to the pinprick test. Submandibular lymph nodes were typically palpable and tender.
Figure 2: A male child with sparse hair, thin extremities, and protruding abdomen

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Figure 3: A skin ulceration with skin sloughing off (arrow)

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Intraorally, the affected site was classically the premolar/molar region [Figure 4]. The gingivae were erythematous and ulcerated sometimes with exposure of necrotic underlying bone [Figure 4]. The ulcerations had grayish white slough and exhibited irregular shape and edges. Frank pus was not encountered, but necrotic tissues which sloughed off with little effort were evident [Figure 4].
Figure 4: Denuded necrotic bone intraorally (arrow)

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Patient management

The patients were promptly admitted and treatment commenced. In patients with poor appetite and clinically established dehydration, intravenous (IV) lines were established and 4.3% dextrose saline which was alternated with 0.9% saline was given at the rate of 7 ml/kg/h. This was continued until the patient could either tolerate feeding per oral or a nasogastric tube was passed. A urinary catheter was also passed in lethargic patients with poor appetite.

If a patient is hydrated, clinically stable, and has stable vital signs, debridement of the lesion under local anesthesia (2% lignocaine with 1:80,000 adrenaline) and sedation with IV ketamine (4 mg/kg) and IV diazepam (0.1 mg/kg) was commenced. Empirical antibiotics and analgesics were commenced 45 min before initiation of debridement: IV metronidazole 15 mg/kg stat, IV ceftriaxone 30 mg/kg stat, and intramuscular paracetamol 11 mg/kg stat. Debridement was done with 3% hydrogen peroxide (1 in 10 dilution with normal saline), following which copious amounts of normal saline were used to irrigate the wound. Sharp debridement of soft tissue with scalpel blade and necrotic bone removal with bone rongeurs were done, when necessary until fresh bleeding tissues were encountered. After achieving hemostasis, povidone iodine-soaked gauze dressing was applied. Twice-daily debridement was done until infection was controlled. The use of hydrogen peroxide was stopped once necrotic tissue was completely removed from the wound and irrigation continued with only normal saline.

The patients/parents/guardians had nutritional counseling. In addition, patients were advised to commence conventional high-protein diet and fruits. Furthermore, nutritional supplements such as Vitamin B complex tablets (1 tablet 8 hourly × 2/52), Vitamin C (1.5 mg/kg 8 hourly × 2/52), and eeZeePaste™ NUT manufactured by Compact India Pvt. Ltd., were advised. Patients were encouraged to consume a minimum of 3 sachets of the eeZeePaste™ NUT per day [Figure 5]. The patients' weights were measured daily to monitor progress or otherwise. Furthermore, physiotherapy majorly in the form of jaw exercise with the aid of stacks of wooden spatula was performed. Instructions on oral hygiene and self-administered physiotherapy were delivered. The patients were then discharged home and scheduled for further treatment at later dates.
Figure 5: eeZeePaste™ NUT paste satchet

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  Discussion Top


Cancrum oris is a devastating disease that frequently starts as an innocuous ulceration which rapidly progresses and destroys both soft and hard tissues in a relentless manner, especially when allowed to fester.

Very often, patients in our environment present late. This is evident in this study, as the earliest presenters reported to the hospital 3 days into the infection. For a lesion with such rapidity as cancrum oris, extensive damage was often done at the time of patient presentation at our hospital. Reasons for late presentation may be related to poor access to health care which has been variously linked with the out-of-pocket payment system that predominates in our climes and patronage of unorthodox practitioners. Other proposed reasons for late hospital presentation include poor educational level, spirituality/religiosity, and inadequate/lack of social support.

In this study, diagnoses were made clinically. In managing this disease, the clinician should have a high index of suspicion. Classically, an orofacial ulceration of short duration with or without bone exposure and teeth mobility, severe halitosis, drooling of saliva, and history of debilitating disease in a child that appears malnourished should raise suspicion. Several differential diagnoses of this lesion have been described, which includes Buruli ulcer, mucocutaneous leishmaniasis, midline granuloma, and ecthyma gangrenosum.[4],[7],[8] Most are infrequently seen in children <6 years of age, while ecthyma gangrenosum rarely affects the orofacial region.[4],[7],[8]

MCS was not done in all cases seen because of the resource-limited environment of ours, although, when done, no growth of microorganisms was observed. This observation may be because of microorganisms associated with cancrum oris are majorly anaerobes, which were not cultured in this study due to the nonavailability of anaerobic transport media.[4],[9]

Ceftriaxone and metronidazole combination was observed to be quite effective in this study, despite the late presentation by patients often encountered. This may be because of the wide coverage of both aerobes and anaerobes that this antibiotic combination gives.[10],[11],[12] Hydrogen peroxide may have improved the efficacy of these drugs because of its potent local action against anaerobes.[11],[13] Indeed, hydrogen peroxide has been said to exert catastrophic effects on any microorganism that lacks the enzyme, catalase.[11],[13] Some authors have reported the use of Edinburgh University solution of Lime (EUSOL).[14],[15] EUSOL also possesses oxidizing and antibacterial properties; however, it requires frequent preparations because of its short shelf life.[14],[15],[16]

Researchers have expressed concerns about possible detrimental effects of these solutions on wound healing, including impediment of fibroblast proliferation and migration.[15],[17] Moreover, peroxide has been reported to cause soft tissue emphysema in certain cases.[13],[17],[18] In this study, the use of diluted 3% hydrogen peroxide was only in the initial phase of wound debridement. As the wound became cleaner, it was discontinued. In addition, to avoid the detrimental effects of indiscriminate hydrogen peroxide use, the wound was irrigated liberally with normal saline after each use.

The average hemoglobin levels observed in this study are similar that observed by Adeola et al., who reported an average of 7.5 mg/dl.[19] Some authors advocate for transfusion of patients with hemoglobin of <10 g/dl.[19],[20],[21] Indeed, Adeola and Obiadazie reported transfusion with packed cells as part of their treatment regimen in their case series.[20] Blood transfusion was not done despite the less than optimal hemoglobin of some patients. Hitherto, the critical hemoglobin level that was judged clinically acceptable was hemoglobin ≥10 g/dl.[21],[22] Blood transfusion was not done in this study because none of the patients had hemoglobin level of <7.0 g/dl. Hébert et al., in a multicenter randomized controlled trial to examine transfusion requirements among critically ill patients, recommended red cell transfusions when hemoglobin is <7.0 g/dl.[21]

The eeZeePaste Nut is a ready-to-use therapeutic food. The ingredients include peanut, sugar, nonhydrogenated vegetable oils, skim milk powder, minerals, emulsifier, vitamins, antioxidants, tocopherol, and citric acid esters. Each sachet weighs 92 g and gives 500 kcal of energy. It is compact, easy to distribute, easy to use, and easy to handle. Some authors have documented the use of fortified local foods such as pap and fresh crayfish (Paranephrops planifrons) mixture.[23],[24] Other preparations contain soya beans, skim milk among others.[23],[24] These preparations may be cheaper, more accessible, and more sustainable than the eeZeePaste Nut.

Jaw exercise was with the aid of stacked spatula which was inserted incrementally into the patient's mouth as carefully as possible. It is a cheap, easily available means of physiotherapy in these patients. Various options for jaw physiotherapy such as utilization of TheraBite for jaw exercise have been described by several authors.[25] While it may be more convenient to use, it is more expensive and less accessible than other options, such as the use of stacked spatula in our environment.


  Conclusion Top


Cancrum oris requires urgent intervention which should be conservative in nature initially. To give optimum results, underlying systemic conditions should be identified and adequately treated appropriately. Furthermore, surgical, psychosocial, and nutritional rehabilitation of patients should be given due attention.

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

Nil.

Conflicts of interest

There are no conflicts of interest.



 
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