Cancrum Oris: A Single Stage Correction of Ankylosis of Jaw and Cheek Fistula Defect

Abstract

A 55-year-old lady presented with trismus and full thickness oral defect exposing upper and lower row of teeth. This was a sequel of the cancrum oris that she had had in the childhood. The operative intervention consisted of radical excision of scar tissue, release of bony block of right temporo-mandibular joint ankylosis and the cheek fistula defect was corrected in two layers by bipaddled island pectoralis major myocutaneous flap. The correction of jaw ankylosis and the flap cover was done in a single stage and is presented in details.

INTRODUCTION

Cancrum oris is an acute gangrenous disease affecting the face. The condition usually affects young, poorly nourished children and occurs as a complication of measles and other childhood illnesses. Malnutrition is a predisposing factor and the presence of erupted teeth appears necessary for the onset.¹ It is also known as Noma (from the Greek meaning, "to devour"), which aptly describes the process which usually starts as a pustule on the buccal mucosa. The mortality in the acute phase is unknown. The patient usually presents with gross scarring which prevents movement of the mandible, leading on to secondary ankylosis of the jaw and often, there is a large oro-cutaneous fistula with varying degrees of destruction of soft tissue, cartilage and bony framework of face.

CASE REPORT

A 55-year old presented with a 5x4 cm oro-cutaneous fistula in the right cheek, exposing the upper and lower row of teeth (Fig.1 & 2). She had trismus with mouth opening of less than 3 mm. There was extensive scarring around the defect which she had since childhood. An orthopantomogram revealed ankylosis between the temporal bone and the condyle and coronoid process of the mandible. The anaemia was corrected by haematinics and diet counseling. After the pre-anaesthetic checkup, she was admitted for the corrective surgery.
At surgery under general anaesthesia, the dense scar tissue around the oral defect was excised. As the soft tissue release was not sufficient for the mouth opening, the temporo-mandibular area was accessed through the check fistula. The bony block was wide and deep and extended between the ramus and the upper articular surface, completely replacing the architecture of the joint. 1.5cm x 4cm block of bone was excised beneath the zygomatic arch. Once the bony block was removed, the mouth opening achieved was 6cm.
Though the apparent defect in the cheek was of 5 cm x 4 cm, the real defect after the excision of the scar was larger and required an inner lining of 6cm x 5 cm and outer cover of 7cm x 5.5cm. This was provided by a bipaddled pectoralis major myo-cutaneous flap (Fig 3). One of the skin paddles was outlined on the medial aspect of the breast skin over the para-sternal area. The second paddle was outlined over the infra-mammary area, extending a little beyond the pectoralis major muscle but over the rectus fascia. The skin paddles were incised. The dermis of the skin paddle was sutured to the muscle fascia with several interrupted sutures. Care was taken to enter the space deep to the subpectoral fascia. Once the pectoralis major muscle was elevated, the thoraco-acromial pedicle was clearly identified. The muscle pedicle was dissected on either side parallel to the vascular pedicle diretly up to the clavicle. The flap was then tunneled under the neck skin. The bridge of skin between the two paddles was de-epithelised and the flap was folded upon itself and sutured to the margins. The lady had an uneventful post-operative period. She is on regular follow up (eleven months since surgery) and doing well (Fig. 4).

DISCUSSION

Mr. Michael N. Tempest in his paper on Cancrum oris in 1966 presented a detailed study of 250 children with Cancrum oris in Nigeria. This was followed in 1983 by a further series of 140 cases presented by Adekeye and Ord, again from Nigeria.

The basic principles of treatment of the established case of cancrum oris have remained the same, viz.: a) to release the ankylosis if present and b) to carry out reconstruction of the facial defect. To undertake the latter before achieving the former is disastrous for the patient.
A 5-cm of inter-incisor mouth opening in adults is considered to be a good result after release of ankylosis. This patient too, after surgery, has a mouth opening of 6 cm. Adekeye and Ord stressed that lower success rates were achieved in patients with a soft tissue defect.

Since the classic paper of Tempest, there have been significant advances in the knowledge of skin flaps and it is in this area that there have been the most changes. The forehead flap is no longer the first choice for either inner or outer lining on account of the cosmetic deformity of the donor site on the forehead. Other options are the cervical turnover flap, tongue flap, deltopectoral flap, pectoralis major myocutneous flap and free flaps. The cervical and tongue flaps do not provide adequate tissue in large defects. The deltopectoral flap is a versatile flap but it requires a second stage for flap detachment and complete insetting. Unfortunately, free flaps remain an unrealistic option in most of the developing countries, where the demand for reconstructive surgery is growing.

The pectoralis major myocutaneous flap has been the work-horse for head and neck reconstruction. It provides good quality chest skin for cover and / or check lining with minimal morbidity and an acceptable donor site. The greatest advantage lies in the fact that the pectoralis major island flap allows insetting in a single stage procedure. The double paddle technique provides excellent primary reconstruction for full thickness cheek defects, alleviating the need for elevating two different flaps. However, in women, the dissection of pectoralis major flaps is cumbersome because of the overlying breast, and the perforators which are sheared are likely to get damaged while traversing the breast to reach the overlying skin. The reconstructed cheek now has a pin-cushion effect due to circular shape of the pectoralis major flap. This will be corrected subsequently by z-plasty.
As in so many other tropical diseases, the ultimate cure is prevention by raising socio-economic standards. However, in established cases, it is now possible to achieve a reasonable functional and cosmetic by using modern technique of reconstructive surgery.

REFERENCES

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2. Tempest MN. Cancrum oris. Br J Surg 1966; 53: 949.
3. Juri, J. and Juri, C. Advancement and rotation of a large cervicofacial flap for cheek repairs. Plast Reconstr Surg. 1979;61:692.
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