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13. Colclasure JB, Shea MC, Graham SS: giant cell lesions of the temperal bone. Am J Otol 2:188, 1981 14. Wolfe JT, Scheithauer BW, Dahlin DC: Giant cell tumor of the sphenoid bone. J Neurosurg 59:322, 1983 15. Chuong R, Kaban LB, Kozakewich H, et al: Central giant cell lesions of the jaws: A clinicopathologic study. J Oral Maxillofac Surg 44:708, 1986 16. Monchik JM, Martin HF: Ionized calcium in the diagnosis of

primary hyperthyroidism.

Surgery 88:185, 1980

17. Morin LG: Direct calorimetric determination of serum calcium with o-cresolphthein complexion. Am J Clin Path01 61:114, 1974 18. Hawker CD, DiBella FP: Human parathyroid hormones: A review of the radioimmunoassay procedures and clinical internretation. in Kinn JS ted): Current Tonics in Clinical Chemistry: Clinical Immunochemistry, vol 3. Washington, DC, AACL 1978, p 329 19. Habener JF: Recent advances in parathyroid search. Clin Biochem 14:223. 1981

J Oral Maxillofac

hormone re-

20. Gordan GS, Roof BS: Laboratory tests for hyperparathyroidism. JAMA 206:729, 1968 21. Kao PC. Klee GG: Serum narathvroid hormone. Adv Clin Chemll:l, 1982 _ * 22. Palmer FJ, Nelson JC, Bacchus H: The chloride-phosphate ratio in hypercalcemia. AM Intern Med 80:20& 1980 23. Weldon L. Cozzi G: Muhinle aiant cell lesions of the iaws. J Oral Maxillofac Surg 40‘:526, 1982 24. Batsakis JG: Tumors of the head and neck, clinical and pathological considerations. Baltimore, MD, Williams 8t Wilkins. 1979, p 379 25. Kenan S, Kirby EJ, Buchalter J, et al: The potential role of the laser in marginal sterilization of giant cell tumor following curettage. Bull Hosp Joint Dis Orthop Institutes 48:93, 1988 26. Webb DJ, Brockbank J: Combined curettage and cryosurgical treatment of the aggressive “giant cell lesion” of the mandible. Int J Oral Maxillofac Surg 15:780, 1986 27. Thawley SE, Panje WR, Bat&is JG, et al: Comprehensive Management of Head and Neck Tumors. Philadelphia, PA, Saunders, 1987, pp 1534-1536

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48:305-3&3,1990

Squamous Cell Carcinoma Arising in a Plunging Ranula M. KARIM ALI, MD, FACS,* GIANCARLO CHIANCONE, GLENN W. KNOX, MD+-

A plunging ranula is a collection

of salivary secretion that extends from the involved salivary gland into the neck.‘,* It generally has a connective tissue capsule. There have been no previous reports of a ranula associated with a malignancy. The following report describes such a case. Report of a Case A 36-year-old white man was admitted to the hospital with progressive respiratory difficulty and inability to eat secondary to an enlarging mass that initially had started on the right side of the floor of his mouth approximately 15 years previously (Figs 1 and 2). During this period the mass had gradually enlarged to occupy most of the oral cavity and extend down both sides of the neck. Two years Received from the Section of Otolaryngology, Hunter H. McGuire Veterans Medical Center. Richmond. VA. * Chief. t In private practice, Richmond, VA. $ In private practice, Washington, DC. Address correspondence and reprint requests to Dr Ali: Chief, Otolaryngology Section, Veterans Administration Medical Center, 1201 Broad Rock Blvd, Richmond, VA 23249. 0 1990 geons

American

Association

0278-2391/90/4803-0014$3.00/O

of Oral

and Maxillofacial

Sur-

MD,t AND

before admission the mass had exhibited a more rapid growth to the extent that the patient was unable to eat. He also developed mild respiratory distress in the supine position. The patient was a heavy smoker. Physical examination showed the patient to be in no acute distress. A cystic mass occupied the entire oral cavity, pushing the tongue to the left in a superolateral manner (Fig 3). Due to the extent of the neck mass, no lymphadenopathy could be palpated on either side of the neck. There were no significant findings other than decayed teeth. The patient had camouflaged his abnormal neck appearance with a beard. Laboratory values, including complete blood cell count, electrolytes, prothrombin time, partial thromboplastin time, and urinalysis, were within normal limits. Results of the chest radiograph were normal. Computerized axial tomography of the neck revealed a large cystic mass extending from the floor of the mouth to the right side of the neck (Fig 4). A diagnosis of plunging ranula was made. On the third hospital day, a tracheostomy was performed under local anesthesia, and exploration of the neck was performed under general anesthesia. During the dissection of the right lateral portion of the cyst, it ruptured and expelled a greenish cheesy material. The plane of dissection in this area was not clear, and the cyst was adherent to the surrounding tissues. The cyst wall in this area was corrugated and glandular (Fig 5). Malignancy was suspected, and a frozen section of the cyst was ob-

306

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FIGURE 1. Clinical view of patient showing the extent of the mass intraorally.

FIGURE 2.

tained which revealed squamous cell carcinoma. Because the cancerous tissue was fixed to the surrounding tissue, a drain was placed and the wound was closed. Direct laryngoscopy was then performed without significant findings. The patient was subsequently referred for postoperative radiation therapy, receiving a total of 54 Gy to both sides of the neck. Permanent sections from the cyst wall showed nests and cords of well-differentiated squamous cell carcinoma (Fig 6). Foreign body giant cells and cholesterol clefts were also present within the fibrous tissue. The rest of the cyst wall was lined with flattened cuboidal epithelium

with areas of dense fibrosis. Electronmicrographs showed that the tumor cells had abundant tonofibrils and that the cells were connected by desmosomes, thus confirming the lesion as a squamous cell carcinoma. The patient has been followed closely in the tumor clinic and has shown no evidence of recurrent disease in a 2-year period.

Clinical view of patient showing extension of the mass into the neck.

Discussion Approximately one third of the population have a hiatus in the mylohyoid muscle that might allow a

FIGURE 3. Intra oral view showing the tongue displaced superolaterally to thle left.

ALI. CHIANCONE,

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AND KNOX

FIGURE 4. CT scan showing large cystic mass in the anterior part of the neck.

ranula to extend anteriorly into the neck.3 Others extend posteriorly behind the free margin of the mylohyoid muscle, displacing the lingual nerve and submandibular gland anteriorlye These lesions are not common. Blaele and Croft4 reported eight cases in a lo-year experience, and Mair et al’ noted four cases of plunging ranula in a 6-year period. Patients often escape medical attention due to the slow growth and initial painless nature of these lesions. Ranulas are believed to originate from extravasation of sublingual salivary secretions6 Biochemical and histochemical analyses of the contents of ranulas are consistent with a sublingual secretory origin,2.4 and sialography has repeatedly demonstrated a sublingual rather than submandibular origin.‘,” Moreover, submandibular sialograms on patients with ranulas have not revealed extravasation of contrast material.5.6 Two clinical types of ranula are recognized: the

FIGURE 5. Gross specimen showing cyst wall where squamous cell carcinoma arises from within the cyst.

._I :,. “.‘-I.

“L..

,‘_,,

FIGURE 6. Photomicrograph of cyst wall showing welldifferentiated squamous cell carcinoma within the fibrous tissue. (Original magnification, X 100.)

first type is contained entirely within the oral cavity; the second type, termed plunging or cervical ranula, has an associated cervical extension in continuity with the oral cavity lesion.6 This continuity can be demonstrated by injecting radiographic contrast material.2 It has been noted that extirpation of

308 the sublingual gland leads to cure of oral and plunging ranulas. Because treatment of both oral and cervical (plunging) ranulas is often followed by recurrence, many methods of treatment have been suggested. These have been reviewed by van den Akker, et al’ and include the following: 1) marsupialization, 2) intraoral sublingual gland excision, 3) combined marsupialization and sublingual gland excision, 4) cervical excision of ranula and sublingual gland, and 5) radiation. Total excision of the ranula with sublingual gland is the favored method. Total excision of plunging ranulas is difficult, however, because they lack an epithelial lining and are pseudocysts rather than true cysts. For this reason some authors’ advocate excision of the sublingual gland with simple drainage of the ranula. Ranulas have been classically described as having an epithelial lining.’ However, several investigators have noted that the majority of these lesions (80% to 95%) have a dense connective tissue capsule rather than a squamous epithelial lining.” Quick and Lowell” theorized that this represents two modes of pathogenesis: partial obstruction of the sublingual ductule could produce an epitheliallined cyst by dilatation, or disruption of the duct or ductules could lead to a pseudocyst lined with connective tissue. When a mylohyoid hiatus exists, this could extend to the neck; hence, the plunging ranula. In the case presented, the diagnosis of squamous cell carcinoma was not considered preoperatively due to the enormous size of the cystic neck mass. Fine-needle aspiration biopsy of such a neck mass

SQUAMOUS CELL CARCINOMA IN PLUNGING

RANULA

yielding fluid consistent with salivary secretion would have established the diagnosis of ranula, but would have missed the occult carcinoma. Contrast radiography, computed tomography (CT) sialography, or magnetic resonance imaging would delineate the ranula cavity.’ It should be noted that any irregularity in the cyst or pseudocyst lining noted radiographically should alert one to the possibility of a malignancy. Summary A case report of a 36year-old man with a plunging ranula of 15 years’ duration, in which squamous cell carcinoma arose from the cyst wall, is presented. Pathogenesis and treatment of ranulas is briefly discussed. References 1. Fund EH: Ranula. Aust Dent J 26:214, 1981 2. Roediger WEW: Mucus extravasation theory as a cause of plunging ranulas. Br J Surg 60:720, 1973 3. Gaughram GRL: Mylohyoid boutonniere and sublingual bouton. J Anat 97565, I%3 4. Blaele RJ, Crost CD: Ranula: Pathogenesis and management. Clin Otolaryngol 7:299, 1982 5. Mair IWS, Schewitsch I, Svendsen: Cervical ranula. J Laryngol Otol43:623, 1979 6. Roediger WEW, Kay J: Pathogenesis and treatment of plunging ranulas. Surg Gyn Obstet 144:862, 1977 7. van den Akker HP, et al: Plunging or cervical ranula. J Maxillofac Surg 6:286, 1978 8. Abbey FS, Cohlmia DD, Reynolds GG: Sublingual cystRenort of a case. Oral Sum 14: 1155. l%l 9. Gorlin RJ, Goldman HM: O&l Pathoiogy. St Louis, MO, Mosby, 1970, p 66 10. Quick CA, Lowell SA: Ranula and the sublingual glands. Arch Otol 103:397, 1977

Squamous cell carcinoma arising in a plunging ranula.

A case report of a 36-year-old man with a plunging ranula of 15 years' duration, in which squamous cell carcinoma arose from the cyst wall, is present...
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