REDUPLICATION OF THE MOUTH AND MANDIBLE. Groote Schuur Hospital and the Red Cross Hospital, Cape Town, South Africa

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IJHtlsh Journnl of Plaslic NurgeO, (1973), 26, 84-89 REDUPLICATION OF THE MOUTH AND MANDIBLE By D. DaviEs, M.B., Ch.B., F.R.C.S., G. MoRgIso~r, M.B., Ch.B., F.C.S. and B. H. MILLER, B.D.S., F.D.S., Dip.

1 IJHtlsh Journnl of Plaslic NurgeO, (1973), 26, REDUPLICATION OF THE MOUTH AND MANDIBLE By D. DaviEs, M.B., Ch.B., F.R.C.S., G. MoRgIso~r, M.B., Ch.B., F.C.S. and B. H. MILLER, B.D.S., F.D.S., Dip.Orth. Groote Schuur Hospital and the Red Cross Hospital, Cape Town, South Africa Tins congenital deformity, although well described in I948 by McLaughlin and in I956 by Beatty, is sufficiently rare to warrant the addition of a third case history to the literature. FIG, I. Two-year-old child showing reduplication of mouth. CASE REPORX In I97 o a-a-year-old child was referred to The Red Cross War Memorial Children's Hospital in Cape Town with a protuberance on the right cheek resembling a small mouth. Examination showed a healthy child with a swelling overlying the right side of the mandible (Fig. I). The pouting orifice pointed laterally rather than forwards, was 40 ram. in diameter and projected 3o ram. from the normal cheek line. ~lell marked upper and lower lips, which showed evidence of sphincter action, could be separated to reveal a cavity containing teeth (Fig. 2). There was a fairly copious discharge of salivary secretion. The lining of the cavity had the appearance of normal buccal mucosa, ending at a well-defined lip margin with vermilion ridge. 84

2 REDUPLICATION OF THE MOUTH AND MANDIBLE 85 On palpation a bony swelling was found to underlie the reduplicared mouth. This was firmly attached to the right side of the mandible and extended upwards towards the right temporo-mandibular joint. A cleft between the normal and the reduplicated mandible was noted at the mental end, No tongue was visible and there was no evidence of communication with the normal mouth. "'t~:~~l V -I, r,.,-..,.,,.,:" Fzc,. 2. Lips of reduplicated mouth parted to show teeth and mandible. Note saliva in this oral cavity. FIG. 3- Panorex view of mandible. X-Ray Examination. Multiple views including a Panorex (Fig. 3) demonstrated a V-shaped mandible lying to the right of the normal right ramus which itself appeared enlarged. One received the impression that an upward extension of the reduplicated mandible shared the fossa of the right temporo-mandibl~lar joint with the ascending ramus of the normal mandible on that side.

3 86 BRITISH JOURNAL OF PLASTIC SURGERY FI(3. 4. Partially dissected specimen showing probe accessory salivary duct. in F~6.5- Inferior dental nerve leaving reduplicated mandible. FIG. 6. Abnormal branch of maxillary artery passing from the right of the illustration and entering the mandibular foramen of the reduplicated mandible.

REDUPLICATION OF THE MOUTH AND MANDIBLE 87 Operation. A lenticular incision was made around the accessory mouth which was packed to define its extent.

4 REDUPLICATION OF THE MOUTH AND MANDIBLE 87 Operation. A lenticular incision was made around the accessory mouth which was packed to define its extent. The lips and mouth were freed from the surrounding tissues with great care in an attempt to define any structures that might have been attached to the reduplicated mouth and mandible, An accessory salivary duct was found to run from the reduplicated mouth towards the anterior border of the masseter (Fig. 4)- This was divided and a probe passed up the lumen in the direction of the parotid gland showed quite clearly that this was a sal~ivary duct. FIG. 7- Operative specimen showing mucous retention cyst. FIG. 8. Pathological specimen clearly showing both rami and condylar heads. Unfortunately a sialogram was not done at the time of operation. Behind the alveolar arch and the teeth was a firm bulge approximately I5 x Io ram. in size resembling a small tongue. Gradually a complete rudinlentary mandible was exposed and it was possible to demonstrate both inferior dental nerves leaving the mandible (Fig. 5), a rudimentary "left" masseter muscle and muscle fibres attached to the region of the ~enial tubercles. The left ramus of the accessory mandible was attached to the expanded normal right ramus over an area of about 3 sq. cm. A fine osteotome was introduced into the groove below this junction and the two rami parted. Further dissection revealed two small condyles lying just anterior to the right mastoid bone and t[ae right condyle appeared to have a false joint Oavit3~.

5 88 I~RI'rlSH JOURNAL OF PLASTIC SURGERY A large branch of an abnormal maxillary artery (Fig. 6) which supplied both sides of the mandible from the narrow gap between the small ascending rami was divided, enabling the specimen to be removed. The wound was closed without drainage. Post-operatively a small wound abscess was drained and this was obviously due to haematoma formation in the wound. At one stage a well-defined swelling could be t~it and we thought that a mucous-lined cyst may have formed as in McLaughlin and Beatty's cases. This was confirmed at operation (Fig. 7). The cyst was removed and thereafter healing was uneventful. A post-operative Pa~mrex showed an enlarged normal mandible and the area of attachment to the reduplicated mandible was clearly seen. A small spicule of bone projected from the site of division. Fro. 9 FIo, 9- Radiogram of reduplicated mandible showing deciduous and permanent dentitions. FIG. Io. Photograph of teeth dissected from the reduplicated mandible. FIG. I0 A right sialogram demonstrated a small right parotid gland which appeared to be only half the size of the normal parotid on the unaffected side. This suggested that the fairly large accessory duct that was exposed af the time of operation was fed by a separate parotid gland on the right side. Pathology Report. "A malformed mandible to which is attached a nodular excrescence at the mental end where there is a mucosa-lined cavity containing two incisor teeth. Posterior to this is a firm nodule which may be a rudimentary tongue. Around this is a malformed lip." (Fig. 8.) Histology. "Sections from the lip and adjoining mucosa show normal squamous epithelium with underlying muscle in the lip region and small mixed salivary glands and ducts under

REDUPLICATION OF THE MOUTH AND MANDIBLE 89 the internal mucosa. Sections from the nodule which was thought to be tongue showed acanthotic squamous epithelium without gonal papillae.

6 REDUPLICATION OF THE MOUTH AND MANDIBLE 89 the internal mucosa. Sections from the nodule which was thought to be tongue showed acanthotic squamous epithelium without gonal papillae. A fair number of keratotic pearls are, however, seen and the tissue underlying the epithelium is dense connective tissue without any identification of striated muscle fibre." Dental Report. "The radiograph of the accessory mandible showed the presence of deciduous and permanent dentitions (Fig. 9). Two deciduous incisors were present in the 'mouth cavity', while two unerupted deciduous canines and one molar were dissected out. Three unerupted permanent teeth, two incisors and a canine, were also dissected from the specimen (Fig. l o). "All the teeth showed normal crown and root formation with the exception of the deciduous molar which had 3 roots in contrast to the 2-rooted formation usually associated with these teeth. The crown of tiffs molar was also somewhat atypical, being triangular in shape with a well-developed palatal cusp. The permanent teeth were developing normally, with welldefined dentine papillae." DISCUSSION A full general examination together with intravenous pyeiogram and contrast studies of the gastro-intestinal tract failed to reveal any other anomalies in this child. Beatty's case was noted to have right anophthalmos and a polycystic left kidney in addition to the reduplicated mouth. We can add no new thoughts to those of McLaughlin concerning the origin of the reduplication and it must presumably be regarded as a true developmental anomaly arising from sequestrated totipotent cells. The same author gives a full review of the literature and we have found no subsequent relevant articles. It is interesting to note that in all three cases a mucous retention cyst formed postoperatively. This is possibly due to incomplete removal of the salivary apparatus of the reduplicated mouth and mandible. S UMMARY A rare case of reduplication of the mouth and mandible is reported and compared with two previous reports of a similar condition. We wish to thank the Superintendent of Red Cross Hospital for permission to publish this casereport and Professor ft. H. Louw, Head of tkc Department of Sm~e~y, for his encouragement. REFERENCES BEATTY, H, G. (I956). A report of a case of an-unusual embryologic defect of the face. Plastic attd Reconstructive Surgery, 1% MCLAUGHLIN, C. R. (x948). Reduplication of mouth, tongue and mandible. British Journal of Plastic Surgery, x,

Upper arch. 1Prosthodontics. Dr.Bassam Ali Al-Turaihi. Basic anatomy & & landmark of denture & mouth

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