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Introduction
The aetiology of temporomandibular joint ankylosis is
varied. It includes inflammatory diseases, neoplasia,
trauma, infection, radiation and systemic disease (Thoma
1945, Schulte 1967, El Mofty 1972). However, the cause of
loss of mandibular movement in some cases has so far not
yet been identified.
Authentic cases of congenital ankylosis of the temporomandibular joint are very rare. Many reported cases of temporomandibular joint ankylosis occurring in early infancy
were erroneously described as congenital (Topazian 1964,
Guilhem and Cadenat 1955). Topazian (1964), in a review
of the literature, found 5 cases of congenital ankylosis of
the temporomandibular joint out of 185 cases of ankylosis
reviewed. Mathis (1962) and Salleh (1965) reported two
cases of true congenital fusion of the mandible and the
maxilla.
A review of the literature appears to reveal only seven
known cases of true congenital fusion of the mandible to
the maxilla. This case report of bony union of the mandible
to maxilla, noticed at birth, represents an additional case of
true congenital fusion of the jaws to the extremely scanty
literature on this subject.
Report of Case
On November 28th, 1977 a twelve-day-old baby girl (C.N.
Luth, No. 1977) was admitted to the paediatric ward of the
Lagos University Teaching Hospital, Lagos, because of
complete immobility of the mandible and inability of the
mother to feed her. She was then referred to the Maxillofacial Surgeon in the Hospital.
Investigations
Laboratory investigations and blood chemistry were within
the normal limits. Radiographic examination of the chest
and extremities was noncontributory. Antero-posterior
views of the mandible showed a normal mandible. However, lateral views revealed that the coronoid process was
elongated, deformed and fused to the region of the maxillary tuberosity and temporal bone on both sides (Fig. 2). It
was not possible to obtain a good radiograph of the temporomandibular joints.
Treatment:
On admission, a nasogastric tube was inserted for feeding,
combined with supportive therapy of multivite, 20 drops
tds, ferrous sulphate 5 ml. tds and folic acid. By the end of
two weeks the general condition of the baby was found to
be suitable for us to contemplate an operation.
On January 6th, 1978, the baby was taken to the operating
theatre and blind naso-endotracheal intubation was performed. An intraoral incision was made on each side in the
buccal sulcus extending from the ramus upward towards
the coronoid process.
Exposure of the ramus and coronoid process confirmed the
clinical and radiological diagnosis of bony fusion between
the coronoid process and the maxillary tuberosity. The
temporalis muscle was inserted laterally. Coronoidectomy
was performed on both sides and the temporalis muscle
divided, immediate opening of the mouth was achieved, the
interalveolar ridge space being 30 ram. (Fig. 3).
The tongue, palate and oral cavity could then be examined,
and were found to be normal. Partial closure of the surgical
wound was performed using 4.0 chromic catgut sutures.
The post-operative regime prescribed consisted of 125 nag.
Ampicillin syrup 8 hourly for 7 days, valium 2.5 rag. b.i.d.
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Fig. 3 Photograph showing immediate post-operative result following bilateral intraoral coronoidectomy. The interalveoJar distance
measured 30 mm.
Fig. 4 Postoperative condition 1 year later spontaneous mouthopening is 25 mm, Note normal mandibular growth.
the interalveolar ridge distance established during hospitalisation, the temporomandibular joint is still functional on
each side. When last seen in 1982, five years after operation, the deciduous teeth had all erupted and spontaneous
mouth-opening was 22 mm. (I1 D). Even though we
strongly advised 6-monthly follow-up, the patient did not
come back for further review.
Result:
Mouth-opening exercises were commenced on the second
post-operative day using finger pressure three times daily.
In order to encourage passive exercise of the joint the
mother was advised to start breast-feeding the baby at the
same time. Assessment of mouth opening was made by
making her cry. She was discharged home on March 20th
1978 with a constant mouth opening of 25 mm. interalveolar ridge distance (Fig. 4). She has been under our
supervision on an outpatient basis since, and although there
has been a decrease in the interincisal distance, compared to
Discussion
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