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| CASE REPORT |
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| Year : 2012 | Volume
: 1
| Issue : 3 | Page : 192-194 |
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Cancrum oris: A devastating orofacial gangrene
Bal P Reddy1, B Sridhar Reddy1, G Kiran1, Neelima Chembolu2
1 Department of Oral and Maxillofacial Surgery, GDCH, Hyderabad, India
2 Department of Oral and Maxillofacial Surgery, Meghna Institute of Dental Sciences, Nizambad, Andhra Pradesh, India
| Date of Web Publication | 15-Oct-2012 |
Correspondence Address:
G Kiran
H. No: 1-5-166, New Maruthinagar, Road No. 11A, Kothapet, Hyderabad - 500 060, Andhra Pradesh
India
 Source of Support: None, Conflict of Interest: None  | Check |
Cancrum oris or noma is a rapidly spreading gangrenous stomatitis which occurs chiefly in debilitated or malnourished children, destroying the soft and hard tissue structures. Its fulminating course causes progressive and mutilating destruction of the affected tissues. As most patients with noma do not report until the disease is at an advanced stage, its onset and progression still remains a mystery. Survivors of this disease suffer severe facial deformity due to loss of facial tissues and scarring. We report a case report of noma highlighting its onset and progression, the extent of tissue necrosis and its management with an emphasis on the need for early diagnosis and prompt treatment. Keywords: Cancrum oris, gangrenous stomatitis, noma
How to cite this article:
Reddy BP, Reddy B S, Kiran G, Chembolu N. Cancrum oris: A devastating orofacial gangrene. J NTR Univ Health Sci 2012;1:192-4 |
| Introduction | |  |
Noma is a rapidly spreading mutilating gangrenous stomatitis caused by normal oral flora that becomes pathogenic during periods of compromised immune status. [1],[2] Recent debilitating disease or illness usually precede noma. It is chiefly seen in children, but also reported in adults under certain conditions like malnourishment. [3] Noma is considered as the face of poverty as factors like chronic malnutrition, poor oral hygiene, and exposure to microbial infections contribute to disease progression. [4] This case report describes an adult patient of noma with extensive involvement of the orofacial region.
| Case Report | | |
A 45-year-old male patient presented to the department of oral and maxillofacial surgery with a large defect over the middle and lower third of the left side of face since 6 months. He complained of inability to chew, swallow, non-coherent speech, and restricted mouth opening. One year back, he had tooth ache on the left lower back tooth which slowly loosened and avulsed on its own. Later the patient developed gingival swelling adjacent to the lost tooth which gradually increased in size and involved the surrounding tissues. It progressed to necrosis of intraoral tissues and extended extraorally to involve middle and lower third of the left face.
On examination, his face appeared to be deformed with extensive exposure of oral cavity and mid-face on the left side. There was extensive loss of skin, underlying muscles, and lining mucosa. The defect extended superiorly up to the zygomatic complex, medially up to left nostril and left one-third of lips, laterally up to the ear pinna and inferiorly up to the lower border of the mandible, exposing it on the left side [Figure 1] and [Figure 2]. The involved area was covered with bluish black necrotic mass. Intra-oral examination revealed Lower left first molar (36). | Figure 1: Photograph showing the extensive defect on left side of the face
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Blood investigations showed anemia, raised serum urea and creatinine, and the patient was negative to HIV. OPG and PA view skull revealed osteomyelitic changes in the mandible. Ultrasound abdomen showed bilateral grade III renal parenchymal disease.
After taking physician's consent, blood transfusion was done to raise the hemoglobin percentage to 10 g. Culture and sensitivity tests were done and appropriate antibiotics were administered. Patient was adequately hydrated, and correction of electrolytes and vitamin deficiencies were done with adequate nutritional support. Debridement of the sloughened area was carried out followed by reconstruction under general anesthesia. Deltopectoralis major region was selected as donor site and a myocutaneous flap was raised for soft tissue coverage over the defect [Figure 3] and [Figure 4]. Regular dressing was carried out every day for 1 week. The patient was followed-up after 10 days and healing was uneventful at both the donor and recipient sites except for a small area of necrosis at the recipient site, which healed by formation of scar. We planned for secondary reconstruction for necrosed area [Figure 5]. Later the patient was recalled at 15-day interval for 2 months, healing was normal, and the patient was able to take solid and liquid foods. | Figure 4: Pectoralis major myocutaneous flap raised for the inner lining
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| Discussion | | |
Cancrum oris is an infectious disease, which involves the orofacial tissues and adjacent neighboring structures in its fulminating course. It is not a primary disease, usually preceded by measles, tuberculosis, leukemia, and AIDS. [5] Noma is usually seen in children, whereas our patient was a 45-year-old male whose diet was severely deficient in proteins and vitamins and had grade III renal parenchymal disease. Malnutrition causes alteration in cell-mediated immune function resulting in early breakdown of the epithelial tissues and changes in the oral mucosa which facilitates invasion by pathogens. [5] Stress on malnourished individuals living in poor environmental conditions increases the level of circulating cortisol, which impairs the immune system and favors the growth of pathogenic bacteria. [5],[6] Therefore, malnutrition is considered as the major predisposing factor in this patient.
The exact causative organism is not clear, although spirochetes and fusiform bacillus in symbiosis have been considered to be the primary cause of the condition. [7] Fusobacterium necrophorum  produces dermatotoxins that help in rapid progression of the disease. [8] Without appropriate treatment, the mortality rate is reported to be 70-90% and surviving patients suffer with functional disturbances and disfigurement. The prognosis will be better if it is detected in early stages and antibiotics are administered. [7]
Surgical treatment for noma is possible if the surgeon carefully evaluates each patient individually choosing simple, sound, and satisfactory techniques, based on sex and age of the patients. [7] There have been reports of single-stage reconstruction of defects caused by noma, which is both cost effective and safe. In the case of bony involvement, muscle island flaps such as pectoralis major and latissimus dorsi were found to be excellent for interposition to prevent re-ankylosis. [7],[9] There have been few reports of using microvascular free tissue transfer for facial defects caused by noma. [10] In the present case, deltopectoralis major flap was raised for covering the defect.
"There is nothing like noma," a statement that certainly holds true for the advanced stages of the disease. The differential diagnosis should include physical trauma (including burns), syphilis, oral cancer, mucocutaneous leishmaniasis, lupus erythematosus, leprosy, and agranulocytic ulcerations.
Noma can be treated successfully but it leaves behind an unesthetic scar. Furthermore, infections from the oral cavity can spread to other parts of the body. Recurrence in noma is rare as only very few reports have described its recurrence. Long-term follow-up of noma patients may enable us to know its exact rate of recurrence. Local complications due to inadequate treatment of noma include ankylosis of temperomandibular joint, trismus, problems in mastication, speech, and severe facial disfigurement. Systemic complications such as toxemia, dehydration, and bronchopneumonia can occur leading to death of the patient. [5] Hence, one should possess adequate knowledge about the onset, progression, and management of noma in order to reduce its high mortality rate.
| Conclusion | | |
This case report is of clinical interest because of its extensive and rapid involvement. Literature updates on the cause and nature of progression may guide clinicians in better management of this devastating disease. A multidisciplinary team consisting of oral and maxillofacial surgeons and plastic surgeons should treat this dreadful disease.
| References | | |
| 1. | Wazir SM, Khan SU. Cancrum Oris. J Pak Assoc Dermatol 2008;18:110-2. 
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| 2. | Adolph HP, Yugueros P, Woods JE. Noma: A review. Ann Plast Surg 1996;37:657-68.
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| 3. | Falkler WA Jr, Enwonwu CO, Idigbe EO. Isolation of Fusobacterium necrophorum from cancrum oris (noma O). Am J Trop Med Hyg 1999;60:150-6.
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| 4. | Auluck A, Pai KM. Noma; Life cycle of a devastating sore: Case report and literature review. J Can Dent Assoc 2005;71:757.
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| 6. | Enwowu CO, Falkler WA Jr, Idigbe EO, Savage KO. Noma (cancrum oris): Questions and answers. Oral Dis 1999;5:144-9.
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| 7. | Devi SR, Gogoi M. Aesthetic restoration of facial defect caused by cancrum oris: A case report. Indian J Plast Surg 2003;36:131-3.
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| 8. | Enwonwu CO, Falker WA Jr, Idigbe EO, Afolabi BM, Ibrahim M, Onwujekwe D, et al. Pathogenesis of cancrum oris (noma): Confounding interactions of malnutrition with infection. Am J Trop Med Hyg 1999;60:223-32.
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| 9. | Adekeye WO, Lavery KM, Nasser NA. The versatility of pectoralis major and latissimus dorsi myocutaneous flaps in the reconstruction of cancrum oris defects of children and adolescents. J Maxillofac Surg 1986;14:99-102
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| 10. | Stassen LF, Batchelor AG, Rennie JS, Moos KF. Cancrum oris in an adult Caucasian female. Br J Oral Maxillofac Surg 1989;27:417-22.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
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