J Oral Maxillofac Surg 48:188-192,1990
Mandibular Osseous Metastasis From Esophageal Carcinoma: A Case Report and Review of the Literature RUSSELL SCOTT ANDERSON, MD,· AND WILLIAM J. PEEPLES, MDt Involvement of the jaws by metastatic disease is an unusual but well-described occurrence. Malignancies of the oral cavity account for approximately 5% of primary malignancies, whereas only 1% to 4% of oral cavity malignancies are the result of metastatic disease. 1-2 Metastases to the jaws are usually from breast, lung, kidney, prostate, or thyroid.I" The case described here appears to be the fifth case in the literature of oral cavity metastasis from the esophagus and only the second case found to involve the mandible.
scan of the abdomen and thorax showed thickening of the esophageal wall, with three visible nodes between the esophagus and the carina. No evidence' of intraabdominal nodes or evidence of abdominal metastasis was seen. The left jaw pain was evaluated by conventional and panoramic radiographs which revealed a lytic lesion in the left ramus and midbody of the mandible, with destruction of the trabecular bony pattern (Fig 4). Biopsy was subsequently performed by an enucleation procedure which removed most of the tumor bulk but left gross residual disease. The histologic diagnosis was welldifferentiated adenocarcinoma similar in appearance to the primary eosphageal lesion (Fig 5). A bone scan exhibited many areas of increased uptake in the left mandible, several ribs bilaterally, and the right ischium and femur (Fig 6). Plain radiographs confirmed the lytic lesions of the right femur and acetabulum. Significant laboratory data included an alkaline phosphatase level of 144UIL and a hemogram, urinalysis, acid phosphatase level, and carcinoembryonic antigen level within the normal ranges . Palliative radiotherapy was felt to be the most effective course of management. The esophagus was treated with 6·MV photons to 60 Gy in 32 fractions using parallel opposed AP-PA fields for the first 40 Gy and continuing with an anterior and two posterior oblique fields for the remaining 20 Gy. The dose of 60 Gy was used to achieve good long-term palliation. The patient had an excellent functional status, but was extremely concerned about esophageal recurrence. The mandibular lesion received 30 Gy in 10 fractions using 9 MeV electrons. The femoral lesion was treated with 6 MV photon to 30 Gy in 10 fractions using parallel opposed fields. The patient responded quite well to therapy with improvement of swallowing and reduction of bony pain. His long-term prognosis, however, is poor. Clausen and Poulsen" found that 70% of patients died within I year of detection of mandibular involvement.
Report of a Case A 61-year-old white man initially presented in August 1987 with a single episode of hernaternesis. A barium swallow examination at that time showed an area of irregularity in the midesophagus, but there was no further evaluation by his family physician. He again sought medical attention in January 1988 with the complaint of pain and swelling of his left mandible, a hypoesthesia of the lower lip, a 2- to 3-month history of progressive dysphagia to the point that he could tolerate only liquids and with an associated 25-lb weight loss, and pain in the right hip. His past medical history was unremarkable. The patient was a nonsmoker and denied alcohol usc. A repeated barium swallow study again showed an irregular mass in the midesophagus with a reduced luminal diameter (Fig I). Endoscopy was performed which showed a hiatal hernia with free reflux and a Barrett's esophagus involving the distal 8 to 10 em. Just distal to the squamocolumnar junction, 28 em from the upper incisors, there was a fleshy circumferential luminal mass extending for 4 em longitudinally. Multiple biopsies were obtained which were histologically interpreted as a well-differentiated adenocarcinoma (Figs 2 and 3). A computed tomography
Review of the Literature
Received from the Department of Radiation Oncology and Biophysics, Eastern Virginia Medical School, Norfolk . * Clinical Instructor. t Associate Professor. Address correspondence and reprint requests to Dr Anderson: Eastern Virginia Medical School, Department of Radiation Oncology and Biophysics , 600 Gresham Dr, Norfolk, VA 23507.
In 1954 Castigliano and Rominger" conducted a review of the medical literature from 1902 to 1953 and found 176 instances of metastatic malignancies involving the jaws; however, they included in this series metastatic sarcomas and lesions involving the gingiva as well as some cases in which histologic confirmation or identification of primary site was
© 1990 American Association of Oral and Maxillofacial Surgeons 0278-2391/90/4802-0011 $3.00/0
ANDERSON AND PEEPLES
FIGURE 1. Barium swallow radiograph showing the irregular intraluminal mass.
lacking. Clausen and Poulsen," reviewing much the same data in 1963, found 92 cases reported from 1884 to ·1961 which met their stricter criteria for inclusion. These were as follows: 1) The lesion had to represent a metastatic carcinoma localized to bone, excluding extension from adjacent soft tissue; 2) it had to be verified histologically; and 3) the site of the primary tumor had to be identified. To this review they added five cases of their own. The sites of primary carcinomas metastasizing to the jaws are presented in Table 1. Also shown are the results of 32 histologically verified cases reported by McDaniel et al" in 1971,24 of which met the above criteria, as well as 25 cases reported by Meyer and Shklar,' 20 of which met the criteria. The primary sites most frequently found were breast, lung, kidney, thyroid, prostate, and colon. No esophageal primary tumors were reported in any of these series. A slight female predominance (52% to 62%) was found. Sixty per cent of lesions were solitary in nature, and using Clausen and Poulsen's.' series, the mandible was most frequently involved with 67 cases, the maxilla was involved in 17, and both jaws in only two. Mandibular involvement was most frequent in the mid to posterior mandible, which may be due to the fact that bone metastasis occurs much more commonly in red bone marrow than in yellow bone marrow. The composition of the mandibular marrow cornpart-
FIGURE 2. A photomicrograph of the esophageal primary tumor shows typical features of an adenocarcinoma. (Hematoxylin-eosin stain. Original magnification, x40.)
ment is primarily yellow marrow, with no red marrow at all found in the mandibles of 75% of adults. When present, red marrow is most commonly located posterior to the incisors. 70S Bones other than the jaws were involved in over half of the reported cases. Both Castigliano et al'' and Clausen et al5 emphasized the importance of recognizing the symptom complex of anesthesia and paresthesia of the lower jaw secondary to mental nerve involvement. Other common presenting symptoms are pain, swelling, and loosening of teeth. The anesthesia/paresthesia symptom complex was found to be most specific for malignant involvement, with the other symptoms seen in both benign and malignant processes. Seventy per cent of the cases in the series by Meyer et al3 were adenocarcinomas, and examination of the data of others appears to confirm this finding. In 1986, Scholosky et all reported a case of a poorly differentiated squamous cell carcinoma of the esophagus metastatic to the mandible, and Tideman et af reported a case of an esophageal adeno-
MANDIBULAR METASTASIS FROM ESOPHAGEAL CARCINOMA
FIGURE 3. A photomicrograph of the primary tumor shows typical features of an adenocarcinoma. (Hematoxylin-eosin stain. Original magnification, x400.)
FIGURE 5. Photomicrograph of the mandibular lesion is consistent with metastatic adenocarcinoma. It is quite similar to the pattern seen in the esophageal primary lesion. (Hematoxylineosin stain. Original magnification, x 100.)
carcinoma metastatic to the maxilla. Both cases were histologically verified at both metastatic and primary sites; one of the primary lesions was at the junction of the middle and lower thirds of the esoph-
agus, and the other occurred in the lower third. This appears to be only the third reported case of esophageal carcinoma metastatic to the jaws, 1-2 and the second found to involve the mandible.
FIGURE 4. Panoramic radiograph showing a lytic lesion of the left midbody and ramus of the mandible and destruction of normal trabecular architecture.
ANDERSON AND PEEPLES
Table 1. Major Series Reported: Authors' Data Site
Clausen and Poulsen"
McDaniel et al 4
Meyer and Shklarl
Breast Lung Kidney Thyroid Colon/rectum Prostate Melanoma Stomach Testis Bladder Liver Cervix Ovary Total
30 17 15 6 6 6 5 5 3 1 1 1 1 97
9 5 t 4 0 2 2 0 0 0 1 0 0 24
5 2 4 1 3 2 1 1 0 0 0 0 0 20
Sex More common 59% females 60% females predilection in females Average age (yr) 55.6 51.5 57.2 Most significant Most common 70% Adenosymptom was in the carcinomas posterior paresthesia body of the jaw 67 Mandibular 17 Maxillary 2 Both
FIGURE 6. Bone scan shows multiple areas of increased uptake in addition to the mandible and right femur.
Metastatic spread of esophageal carcinoma is not an unusual phenomenon. An estimated 1.5% to 2% of metastatic disease is of esophageal origin, and metastasis to bone is estimated to occur in 7% to 9% of esophageal carcinomas.v'"!' The likelihood of metastatic spread to bone by esophageal carcinomas has also been related to the site of origin, using the convention of subdividing the esophagus into thirds. . The esophagus is a muscular tube lacking a serosa and lined by a nonkeratinized stratified squamous epithelium. The predominant method of spread of esophageal malignancy is by direct extension, although tumor embolization in the lymphatic and vascular system does occur. The most probable route for development of bony metastasis is by hematogenous dissernination.Z'F The vast majority of primary esophageal tumors are squamous cell carcinomas (95%), with only approximately 5% being adenocarcinomas. In contrast to squamous cell carcinomas, adenocarcino-
mas are more likely to develop in white individuals and are not associated with alcohol intake. They are also more likely in males and usually occur at approximately age 50, as do squamous cell carcinomas. Adenocarcinomas rarely develop in the upper third of the esophagus, occurring more commonly in the middle or lower thirds. Care must be taken, particularly in cases arising from the lower third, to exclude adenocarcinomas of gastric origin that have spread to involve the esophagus. It has been reported that up to 86% of adenocarcinomas of the esophagus are associated with a Barrett's esophagus. Acquired Barrett's esophagus is usually associated with free reflux and occurs most frequently in the distal 10 ern of the esophagus.P'l" Summary
A case of mandibular osseous metastasis from a primary esophageal adenocarcinoma is reported. Review of the literature shows this to be the third reported case of metastatic involvement of the jaws by esophageal carcinoma, and the second to involve the mandible. References 1. Scholosky M. Bouquot J, Graves R: Esophageal carcinoma metastatic to the oral cavity. J Oral Maxillofac Surg 44:825, 1986
SUBMANDIBULAR TUBERCULOUS LYMPHADENITIS
2. Tidemen H, Avier JF, Benesquet AG, et al: Esophageal adenocarcinoma metastatic to the maxilla. Oral Surg Oral Med Oral Pathoi 62:564, 1986 3. Meyer I, Shklar G: Malignant tumors metastatic to mouth and jaws. Oral Surg Oral Med Oral Pathol 20:350, 1969 4. McDaniel RK, Luna MA, Stimson PG: Metastatic tumors in the jaws. Oral Surg 31:380, 1971 5. Clausen F, Poulsen H: Metastatic carcinoma to the jaws. Acta Pathol Microbiol Scand 57:361, 1963 6. Castigliano SG, Rominger CJ: Metastatic malignancy of the jaws. Am J Surg 87:4%, 1954 7. Bigelow NH, Walsh TS: Metastatic carcinoma of the mandible. Ann Surg 137:138, 1953 8. Holland OJ: Metastatic carcinoma to the mandible. Oral Surg Oral Med Oral Pathoi 6:567, 1953
9. Buirge RE: Secondary carcinoma of the mandible. Surgery 15:553, 1944 10. Abrams HL, Spiro R, Goldstein N: Metastasis in carcinoma. Cancer 3:74, 1950 11. Chan KW, Chan EY, Chan CW: Carcinoma of the esophagus on autopsy study of231 cases. Pathology 18:400, 1986 12. Sachs RL: Metastatic carcinoma to the jaw bones. J Dent Res 43:799. 1955 13. Rosenberg JC, Roth JA, Lichter AS, et al: Cancer of the esophagus, in DeVita VT Jr, Hellman S, Rosenberg SA (eds): Cancer. Principles & Practices of Oncology (ed 2). Philadelphia, PA, Lippincott, 1985, pp 621-649 14. de Regato JA, Spjut HJ: Cancer. Diagnosis, Treatment and Prognosis (ed 5). St Louis, MO, Mosby, 1977, pp 446-462
J Oral MaxiUofac Surg 48:192-196,1990
Submandibular Tuberculous Lymphadenitis (Scrofula): Report of Two Cases L1PA BODNER, DMD,* JACK LEWIN-EPSTEIN, DDS,t AND ARIE SHTEYER, DMD:f: Submandibular swellings are common phenomena that require a precise diagnosis. The differential diagnosis involves mainly the pathologic conditions involving the regional lymph nodes and the submandibular salivary glands. Both of these can have either an inflammatory or neoplastic basis. The possibility of a developmental lesion or granulomatous disease must also be considered. Tuberculosis, an infectious granulomatous disease, is presumed to be quite a rare entity and, for that reason, might be overlooked in the differential diagnosis whenever dealing with a submandibular swelling. 1 The purpose of this report is to describe two cases of submandibular swelling consistent with tuberculous
* Section of Oral and Maxillofacial Surgery, The Maurice and Gabriela Goldschleger School of Dental Medicine, Tel-Aviv University, Israel. t Deceased; formerly Professor and Head of the Department of Oral Maxillofacial Surgery, Hadassah Faculty of Dental Medicine, Jerusalem, Israel. :j: Professor, Department of Oral and Maxillofacial Surgery, Hebrew University-Hadassah Faculty of Dental Medicine, Jerusalem, Israel. Address correspondence and reprint requests to Prof Shteyer: Department of Oral and Maxillofacial Surgery, Hebrew University, Hadassah Faculty of Dental Medicine, PO Box 12000,IL-91 120 Jerusalem, Israel. © 1990 American Association of Oral and Maxillofacial Surgeons 0278-2391/90/4802-0012$3.00/0
lymphadenitis, each with a different clinical course, and to discuss their differential diagnosis and workup. Case 1 A 5-year-old girl was admitted to the Department of Oral and Maxillofacial Surgery at the Hadassah Medical Center with a swelling of the left submandibular region. Six weeks previously, the girl's mother had observed a small swelling in this area and contacted her pediatrician, who did not advise any treatment. Two weeks later, the swelling became more extensive, and a course of ampicillin I g/d and Orebenin (cloxacillin sodium) I g1d was administered for about 2 weeks with no apparent change. There was no history of systemic disease, except for the usual childhood diseases. An extraoral examination revealed severe facial asymmetry due to a left submandibular mass extending from the mandibular angle to the midportion of the body of the mandible (Fig I). The mass was painless, firm, and mobile and did not show discoloration or abnormal warmth. Intraoral examination did not show any abnormalities. Salivary flow of all glands was normal. A clinical differential diagnosis of submandibular lymphadenitis or lymph node tumor was made. The periapical, occlusal, panoramic, and chest radiographs were within normal limits, as were the blood count and blood biochemistry tests. A moderate increase in erythrocyte sedimentation rate was found. Bacterial culture of the sputum for tuberculosis was negative. A radioisotope scan with gallium citrate Ga 67 showed a positive uptake in three submandibular lymph nodes on the left side. The anterior node measured approximately 1.5 x 2 em, and two posterior nodes measured approximately