Squamous cell carcinoma arising in residual odontogenic cyst Report

of a case and review

of literature

Alan M. Schwimmer, DDS,a Faruk Aydin, MD,b and S. Neil Morrison, DDS,c New York, N.Y. BETH ISRAEL MEDICAL OF NEW YORK

CENTER-MOUNT

SINAI SCHOOL OF MEDICINE,

Odontogenic squamous pathologic

cysts are common pathologic lesions found in the oral and cell carcinoma arising within the lining of a large odontogenic aspects of the previously reported cases are presented. (ORAL SURG ORAL MED ORAL PATHOL 1991;72:218-21)

THE CITY UNIVERSITY

perioral structures. cyst is described.

A case of Clinical and

quamous cell carcinoma arising from the epithelial lining of an odontogenic cyst is a rare but distinct pathologic entity. Herman’ first described malignant transformation in an odontogenic cyst in 1889. Gardner2 reviewed all documented cases from 1889 to 1967 and determined that there were 25 acceptable instances of malignant transformation within the epithelial lining of an odontogenic cyst. In 1975 Eversole et a1.3 updated Gardner’s list by increasing the number of acceptable cases to 36. Cases where alternative interpretations were considered, such as carcinoma arising from adjacent oral mucosa, cystic degeneration of a primary or metastatic carcinoma, or a primary or metastatic carcinoma of bone in close proximity to a cyst with subsequent union of the two entities, were excluded from these reports. Gardner2 stated that to establish that the malignancy was primary within the cyst, it is essential to demonstrate the transition of the epithelial lining from normal to carcinoma in situ and to invasive carcinoma. Eversole and colleagues,3 in a review of 36

cases, found that central epidermoid carcinoma was associated with a cystic lining in 75% of the reported cases. They also reported that 41% of acceptable cases occurred in residual cysts. Some authors have considered the presence of keratinization in the cyst lining to be a risk factor for malignant transformation.4q 5 Clinical symptoms include swelling, dull pain, occasionally draining fistulas, and cervical lymphadenopathy. Cases in which the mandible is affected demonstrate bony expansion that is usually nontender and without related paresthesia. The radiologic appearance is characterized by an irregular radiolucent area surrounded by a relatively well-defined radiopaque border. Because the clinical and radiologic findings are nonspecific, definitive diagnosis must be made by histologic examination. We report a histopathologically documented case demonstratingmalignanttransformationarisingwithin the wall of a large odontogenic cyst. It was managed by an en bloc resection, local lymph node dissection, and primary reconstruction.

Thief,

Oral and Maxillofacial Surgery; Associate Director, Department of Dentistry Beth Israel Medical Center-Mount Sinai School of Medicine of The City University of New York. bFormerly Chief Resident, Department of Pathology and Laboratory Medicine, Beth Israel Medical Center-Mount Sinai School of Medicine, The City University of New York; currently with the Department of Neuropathology, School of Medicine, Medical College of Virginia, Virginia Commonwealth University, Richmond. CFormerly Chief Resident, Oral and Maxillofacial Surgery, Mount Sinai School OFMedicine, The City University of New York; currently in private practice, Virginia Beach, Va.

CASEREPORT

7/14/22103

A 59-year-old black woman was first seen at Beth Medical Center in October 1988 with a chief complaint of a swelling of 48 hours’ duration of the left mandible. The patient denied any history of pain, paresthesia, dysphagia, or hemorrhage. Her medical history was unremarkable. She had no history of cysts, tumors, or malignancies. Physical examination revealed a fluctuant expansion in the left mandibular buccal vestibule. The patient was partially edentulous in the area of swelling. There were no palpable lymph nodes. The panoramic radiograph revealed a 7 to 8 cm, unilocular,

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Squamous cell carcinoma in residual odontogenic cyst

219

1. Preoperative panoramic radiograph demonstrating unilocular radiolucency extending from right parasymphyseal region to left body. Fig.

Fig. 3. Epithelial lining of cyst wall showing transition from normal (right side) to carcinoma in situ (center and left side). (Hematoxylin-eosin stain; original magnification, X51.5.)

Fig. 2. Cyst wall with benign squamous epithelium. (Hematoxylin-eosin stain; original magnification, x220.)

radiolucent lesion extending from the left midbody of the mandible to the right parasymphyseal region (Fig. 1). Apexes of teeth 22 to 26 were within the lesion but were vital on pulp testing. An incisional biopsy was performed, and this was reported as a squamous cell carcinoma in situ with questionable areas of invasion into the surrounding soft tissue structures; focal areas of surface ulceration, severe chronic inflammation, and many cholesterol clefts were present. Because the areas of invasion were questionable, the patient underwent a marginal resection intraorally with concurrent extraction of teeth 22 to 26. At the time of this resection, serial biopsy specimenswere obtained. These revealed a squamous epithelial-lined cyst with focal areas of squamous cell carcinoma in situ and frank bone invasion (Figs. 2 to 5). Periosteal biopsy specimens were also obtained and were found to be negative for tumor. Becauseof the presenceof invasivecarcinoma, an en bloc resection of the mandible extending from the left body to the right parasymphyseal region was performed. Suprahyoid lymph node dissection and primary reconstruction with a corticocancellous iliac crest bone graft were carried out at this time. All lymph nodes were negative for tumor, no residual tumor was present in the mandible, and all margins of resection were free of tumor. The postoperative course

Fig. 4. High-power view showing severely dysplastic squamous mucosa. (Hematoxylin-eosin stain; original magnification, X210.)

was unremarkable. In view of the negative margins and lymph nodes, follow-up radiation therapy was not planned. DISCUSSION

Neoplastic transformation in the epithelial lining of an odontogenic cyst is a rare but well-described phenomenon. The neoplasms associated with epithelial cyst lining include ameloblastoma, squamous cell carcinoma, and mucoepidermoid carcinoma.33 6 Various forms of squamous cell carcinoma arise from the cystic lining, ranging from very anaplastic to very well-differentiated ones, including the verrucous type.

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Schwimmer, Aydin, and Morrison

ORAL

Fig. 6. Three-month strating reconstruction

Fig. 5. Close-up view of bone and soft tissue showing infiltrating squamous cell carcinoma. (Hematoxylin-eosin stain; original magnification, X5 10.)

Table

1. Clinical data of previously reported cases No. (%) Total No. of cases Age Median (yr): 57 Range (yr): 4-90 Decade of life o-2 3-5 5-8 Sex F M Symptoms Mass Pain Painful mass Site Mandible Maxilla Nature of cyst Residual Keratinized residual Dentigerous Apical periodontal Lateral periodontal Radicular Unclassified

56

3 (5.4) 11 (25.0)

39 (69.6) 18 (33.9) 33 (66.1) 33 (58.9) 10 (19.6) 8 (21.5) 42 (80.3) 14 (19.7) 21 10 11 7 2 1 4

(37.5) (17.8) (19.7) (12.5) (3.5) (1.8) (7.2)

Gardner’ 1 reviewed all cases of epidermoid carcinoma arising in odontogenic cysts reported in the literature from 1889 to 1967 and cited 25 acceptable cases. Since Eversole’s review3 of 36 cases in 1975, an additional 20 cases, including this present instance, have been reported. The total of 56 cases has been reviewed, and the clinical, pathologic, and therapeutic results are shown in Table I.

SURC ORAL

MED

ORAL PATHOI. August 1991

postresection radiograph demonplate and bone graft in place.

In evaluating an odontogenic cyst for the occurrence of a primary malignancy, several other possibilities have to be excluded, such as invasion of the cyst wall from an adjacent primary or metastatic carcinoma and cystic degenerative change in a primary or metastatic carcinoma. The histopathologic criteria employed to document malignant transformation of the cyst lining is identification of a transition from the normal lining epithelium to dysplasia, carcinoma in situ, and, eventually, infiltrating carcinoma.*+ 7-9 Secondary squamous epithelial changes such as pseudoepitheliomatous hyperplasia, acanthosis, and hyperkeratosis may be observed but are nonspecific and nondiagnostic. The fibrous capsule of the cyst can be thickened as a result of superimposed chronic inflammation. Occasionally, mucoid metaplasia and cholesterol deposits can also be seen. Long-standing chronic inflammation has been proposed as the principle predisposing factor of the malignant transformation in the epithelial lining of the cyst yet cannot be substantiated. Several reports emphasize that keratinization of the lining epithelium is more often associated with high risk of malignant transformation 43539, IO

The mean age of patients in all reported cases is 57 years. Seventy percent of patients have been in the fifth to eighth decades of life, with a 2:l male/female ratio. The mandible has been affected four times as often as the maxilla. The most common cyst undergoing malignant change has been the residual cyst, constituting 55% of all cases (Table I). Keratinization in the epithelial lining of a cyst has been demonstrated in approximately 18% of all cases. Clinical symptoms include swelling, dull pain, draining fistulas, and cervical lymphadenopathy. The presence of paresthesia is uncommon and usually occurs as a result of local invasion of the tumor through the neurovascular bundle of the mandible. The 2-year survival rate in 27 well-documented cases has been 63%. This case fulfills the clinical, radiologic, and histopathologic criteria for malignant transformation of an

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Squamous cell carcinoma in residual odontogenic cyst 221

odontogenic cyst. 2, ’ ’ Radiographically, the lesion was typical of a large residual odontogenic cyst and appeared to be completely cystic without solid components. The patient had no previous history of smoking or alcohol abuse. Physical examination, laboratory values, and radiologic workup were within normal limits, except for the aforementioned local findings. Of major importance is that the histologic examination of the cystic lesion revealed a gradual transition of normal squamous epithelial lining to dysplasia, squamous cell carcinoma in situ, and invasive carcinoma. In a review of 35 previously reported cases, histologic demonstration of malignant transformation has been documented in very few cases. There is no difficulty in establishing a malignant diagnosis in the undifferentiated squamous cell carcinomas with anaplasia, but the difficulty usually arises in differentiating a very well-differentiated squamous cell carcinoma from pseudoepitheliomatous hyperplasia within the cyst wall. Surgical treatment for this entity has varied from enucleation to mandibulectomy with lymph node dissection to radical neck dissection, depending on the degree of invasion noted at biopsy and the presence or absence of lymph node involvement. The immediate reconstruction of large mandibular defects after ablative surgery has become an increasingly accepted mode of treatment.12* I3 In cases of anterior mandibular defects, labial incompetence and salivary incontinence are common sequelae. The ability to reestablish the mandibular continuity at the time of excision offers the patient a rapid return to function and avoids severe cosmetic and dysfunctional complications (Fig. 6).

REFERENCES 1. Hermann M. Beitrag zur Entwicklung der Kieferzystem. Erlangen: lnnag Dies, 1889. 2. Gardner AF. A survey of odontogenic cysts and their relationship to squamous cell carcinoma. J Can Dent Assoc 1975;3: 16i-7. . 3. Eversole LR, Sabes WR, Rovin S. Aggressive growth and neoplastic potential of odontogenic cysts. Cancer 1975;35:27081. 4.

5.

6. 7.

8.

9.

10.

Areen RG, McClatchey KD, Baker HL. Squamous cell carcinoma developing in an odontogenic keratocyst: report of a case. Arch Otolarvnaol 1981:107:568-9. Van Der Wall I, Rauhamaa R, Van der Kwast WA, Snow GB. Squamous cell carcinoma arising in the lining of the odontogenie cyst: report of 5 cases. Int J Oral Surg 1985;14:146-52. Shear M. Cysts of the oral regions. 2nd ed. Bristol: John Wright and Sons, 1983. Bradley N, Thomas DM, Antoniades K, Anavi Y. Squamous cell carcinoma arising in an odontogenic cyst. Int J Oral Maxillofac Surg 1988;17:260-3. Berenholz L, Gottlieb RD, Cho YS, Lowry LD. Squamous cell carcinoma arising in a dentigerous cyst. Ear Nose Throat J 1988;67:764-72. Ward TG, Cohen B. Squamous cell carcinoma in a mandible cyst. Br J Oral Surg 1963;1:8. McLeod RI, Soames JV. Squamous cell carcinoma arising in an odontogenic keratocyst. Br J Oral Maxillofac Surg 1988; 26152-7.

II. Gardner AF. The odontogenic cyst as a potential carcinoma, a clinicopathological appraisal. J Am Dent Assoc 1969;78:74655.

Taylor GI. Reconstruction of the mandible with free composite iliac bone grafts. Ann Plast Surg 1982;9:361-76. 13. Gullane GP, Holmes H. Mandible reconstruction. New Concepts Arch Otolaryngol 1986; 10 I : I 12,7 14-9.

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Squamous cell carcinoma arising in residual odontogenic cyst. Report of a case and review of literature.

Odontogenic cysts are common pathologic lesions found in the oral and perioral structures. A case of squamous cell carcinoma arising within the lining...
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