Bilateral pseudoankylosis of the temporomandibular joint due to synostoses between the mandible and maxilla

J Oral Maxlllofac

Surg

40:590-592.1992

Bilateral Pseudoan kylosis of the Temporomandibular Joint Due to Synostoses Between the Mandible and Maxilla STUART SUPER, DMD,* AND J. STEVENS COTTEN, JR., DMDt Kazanjian’ proposed classification of ankylosis of the temporomandible joint into true and false varieties. A true ankylosis is defined as any condition that produces fibrous or bony adhesions between the articular surfaces of the temporomandibular joint. A false ankylosis, or pseudoankylosis, results from pathologic conditions outside the joint that limit mobility of the mandible. Pseudoankylosis may result from myogenic disorders, such as chronic infection of the elevator muscles of mastication, myositis ossificans, and intramuscular hematomas. Coronoid impingement is another cause of pseudoankylosis; it may result from exostosis or elongation of the coronoid process or from a depressed malunited fracture of the zygomatic arch.* A rare form of temporomandibular joint ankylosis is the congenital form, which results from prenatal maldevelopment or intrauterine injury.

A thorough radiographic examination revealed bilateral bony bridges between the right and left maxillary tuberosities and the coronoid processes of the mandible (Fig. 3). The bony bridge on the left appeared more prominent. The patient was then taken to the operating room. Following nasotracheal intubation, attention was directed intraorally. where the region of the left maxillary tuberosity was exposed through an incision in the maxillary left mucobuccal fold. Elevation of a full-thickness mucoperiostrium flap revealed the bony bridge (Fig. 4). The bony attachment of the coronoid to the maxillary tuberosity was sectioned with burs and osteotomes. A similar procedure was then performed on the right side. Following section of the bony bridge on the right, opening of the mandible was still restricted because of the impingement of both coronoid processes. The coronoids and attached bone from the maxillary tuberosities were then removed through bilateral Risdon incisions. It was now possible to open the mandible by means of Molt mouth props to a measured distance of 36 mm between the anterior teeth (Fig. 5). Postoperatively, the patient did well. She was instructed to perform exercises using tongue blades, and advised to eat a regular diet. Four months after the operation she was functioning well. She later moved back to Panama and was lost to follow-up.

Report of a Case The patient was a ‘I-year-old black girl born in Panama and living with an aunt in New York. The patient’s aunt sought help for the patient because of her inability to open her mouth. The birth history was not obtainable, but the aunt stated that the condition had existed since birth. The child had used doll spoons and utensils to eat. There were no carious teeth or functional growth defects. No history of trauma was obtained. The patient supposedly had a 5year-old brother with a similar condition. Clinical examination showed that the girl was well developed and well nourished. Frontal and profile views of the patient were normal (Fig. I). Oral examination revealed a normal occlusal relationship of the posterior teeth and a slight anterior open bite (Fig. 2). Measured opening was 5 mm between the maxillary and mandibular incisors on forced opening. No other physical abnormalities were observed.

Discussion Two similar cases have been reported in the literature, both by Converse.3 One case involved an

FIGURE 1. Profile view showing relatively normal facial development.

* Director of Oral and Maxillofacial Surgery. tFormerly Chief Resident, Oral and Maxillofacial Surgery: presently in private practice in Biloxi, Received from the Department of Surgery, Bellevue Hospital Center, Street, New York. New York 10010. Address correspondence and reprint

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SUPER

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extensive synostosis of the enlarged left ramus that extended from the left external bony auditory canal, the infratemporal surface, the zygomatic arch, and the zygoma to the mandibular coronoid process. The other case was of a synostosis from the lingula of the mandible to the base of the skull. In both cases, the left side was involved. Poswillo, in a note in the article by Converse,3 describes the possible pathogenesis as a local ne-

FIGURE

FIGURE

2.

View of patient’s

occlusion

showing

4.

Intraoperative

view showing

bilateral

bridging

bridge.

open bite.

FIGURE 5. Opening of mouth following bridge and bilateral coronoidectomy.

FIGURE 3. Radiographs showing maxilla and coronoid process.

bony

between

sectioning

of bony

erotic phenomenon taking place in the morphogenesis of this area and resulting in repair by bone rather than differentiation into a normal temporomandibular apparatus. Poswillo4 produced evidence, based on experiments with mice, supporting such a vascular origin. He also concluded that the extent of focal hemorrhage occurring during formation of the stapedial arterial stem could explain the variability that is often seen in such cases. He stated that pressor or other changes that may lead to rupture of the developing anastomosis are unlikely to be equal on both sides. The bilateral and symmetrical deformity in this case, present since birth, differs from the deformities previously reported cases. It is also significant that a Syear-old sibling, not yet examined, is reported to have a similar deformity. If that is true,

BILATERAL

it would provide support for a familial component the anomaly.

in

Summary The occurrence of pseudoankylosis of the temporomandibular joints secondary to maxillary-mandibular synostoses is rare. These authors know of only two cases, previously reported and both were unilateral. A case is presented that is bilateral and symmetrical with a possible familial component.

J Oral Maxillofac

PSEUDOANKYLOSIS

References 1. Kazanjian VH: Ankylosis of the temporomandibular joint. Am J Orthod 24:1181, 1938 2. Miller GA, Page HL, Griffith CR: Temporomandibular joint ankylosis: Review of the literature and report of two cases of bilateral involvement. J Oral Surg 33:792, 1975 3. Converse JM: Surgical release of bilateral, intractable, temporomandibular ankylosis. Plast Reconstr Surg 64:404. 1979 4. Poswillo DE: The pathogenesis of the first and second branchial arch syndrome. Oral Surg 35:301, 1973

Surg

4059%593,1982

Metastatic Carcinoma to the Submandibular Salivary Gland RAPHAEL FEINMESSER, MD, GRATZIELLA LAHOVITZKI, MD, MENAHEM R. WEXLER, MD, AND ISAAC G. PELED, MD To our knowledge, only three cases of metastasis to the submandibular salivary gland have been reported. 1,2,3In both cases the primary lesion was a carcinoma of the breast. We present a case of carcinoma of the lip with late metastasis to the submandibular salivary gland. Report of a Case In 1980 an 85-year-old man was admitted to the Department of Plastic and Maxillofacial Surgery at the Hadassah Medical Center because of a painless mass of the left submandibular region, of one month’s duration. The patient had no other complaints, nor had he experienced loss of weight. Previously, in June 1977, an incisional biopsy and frozen section of a lesion on the right side of the lower lip had been performed and had showed the presence of squamous cell carcinoma. Wedge resection of the lesion and a cross-lip flap had been performed. The postoperative period had been uneventful, and the patient had been free of symptoms for three years. At the present admission, a 2 cm, firm, nontender, immobile mass was palpable under the inferior border of the body of the left side of the mandible. Oral examination and nasopharyngoscopy did not reveal any pathologic condition. Enlarged lymph glands were not palpated. Laboratory tests, radiographs, and scan were interpreted as normal. The patient was given general anesthesia, and a 5 cm horizontal incision was made along the lower border of the mandible. The tumor was deep in the muscle plane, localized, and easy to remove. It appeared to be an enReceived from the Department of Plastic and Maxillofacial Surgery and of Pathology, Hadassah Medical Center, Jerusalem, ISlXl. Address correspondence and reprint requests to Dr. Feinmesser: Hadassah Medical Center, Ein Karem, Jerusalem, Israel.

larged submandibular salivary gland. Three associated lymph glands were removed as well. The histologic examination revealed three normal lymph nodes and a metastatic squamous cell carcinoma encapsulated in connective tissue attached to the submandibular salivary gland (Fig. 1). The submandibular gland itself showed signs of nonspecific inflammatory changes but no evidence of carcinoma in the gland itself, thus refuting a diagnosis of primary squamous cell carcinoma. The postoperative period was uneventful, and the patient was referred for radiation therapy.

Discussion Metastases to the major salivary glands are well known, most of them being located in the parotid and only rarely in the submandibular and sublingual salivary glands. Most metastases in the parotid originate from malignant melanomas of the skin of the head and neck. Squamous cell carcinomas of the oral cavity, nasal sinuses, and pharynx, as well as tumors of the lung, kidney, pancreas, stomach, and breast, are known to metastasize to the parotid.3,4 The incidence of tumor in the submandibular salivary gland is low, accounting for only 8% of primary tumors of the major salivary glands. Metastases to this gland are even rarer. The anatomy of lymphatic pathways in the submandibular area may explain the rarity. A review of the literature concerning the anatomy of the lymphatic system shows that some authors question the existence of lymphatic tissue in the submandibular salivary gland and its vicinity. In Tobias’s translation of Rouviere’s classic treatise on

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