JOURNAL OF ENDODONTICS I VOL 1, NO 2, FEBRUARY 1975
ADENOID CYSTIC CARCINOMA OF THE MANDIBLE MASQUERADING AS PERIAPICAL INFLAMMATION
E. Jefferson Burkes, Jr., DDS, C h a p e l Hill
Radiolucent defects of the jaws that are associated with pulpless teeth and that produce pain are most commonly related to inflammatory conditions. However, there are many disease conditions that may produce radiolucency of the jaw, some of these are malignant neoplasms. Because of these possibilities, the dentist performing root canal therapy must be constantly alert for neoplasia and must be thorough in his diagnosis and follow-up. Schlagel and others1 have said, "Perhaps apicoectomy should be used more routinely for its diagnostic value rather than as a treatment modality of last resort." They further emphasized that all tissues surgically removed should be submitted for microscopic examination. This report presents a case in which an adenoid cystic carcinoma developed into a radiolucent lesion that simulated periapical inflammation. Report of C a s e In February 1968, a 50-year-old white woman was originally seen by her dentist for construction of a bridge to replace the mandibular right first molar (Fig 1). In March 1969, she sought further consultation because of a slightly enlarged and ques76
Fig 1--Radiograph tion of bridge.
before prepara-
tionably tender area in the right sublingual gland. Clinically, the gland was functional and moved freely. An incisional biopsy and subsequent removal of the sublingual gland showed only obstructive sialoadenitis. From that time until July 1970, when the patient consulted an oral surgeon, she continued to have vague nonspecific sensations in the area. Radiographs of the area showed an enlarged right inferior alveolar canal and a diffuse, poorly defined radiolucent area involving the bone around the first premolar and canine (Fig 2). The pulp of the first premolar did not respond to thermal tests. Because of the pain, the radiolucent area, and the negative reaction to the pulpal test of the first premolar,
endodontic treatment with curettage and biopsy was advised. The patient was referred to her dentist for this treatment. The curettage and root canal filling were performed in September; however, the tissue that was removed was not submitted for microscopic examination. Radiographs again were obtained in January 1971 because of continued pain (Fig 3, top), but there was no treatment done at that time. By November, the pain had become more severe. The patient returned to the oral surgeon who had originally recommended the endodontic therapy. At that time, radiographs showed enlargement of the radiolucent area with involvement of the second premolar and incisors. The borders of the lesion were irregular and the bone appeared ragged in adjacent areas of the mandible (Fig 3, bottom). An incisional biopsy and exploration of the area showed an adherent, tan, glistening mass that filled a large bony crypt. The lingual cortical plate appeared to be intact except for a 2-mm ovoid perforation in the first premolar area. The histologic diagnosis was adenoid cystic carcinoma. Large cystic spaces within islatads of hyperchromatic epithelial cells were scattered throughout the specimen (Fig 4). Because of the patient's reluctance to undergo extensive surgery, therapy
q .t,
Fig 2--Radiograph before endodontic treatment.
JOURNAL OF ENDODONTICS I VOL 1, NO 2, FEBRUARY 1975
was delayed for almost three months. At the preoperative examination, a firm mass that was attached to the lingual side of the right anterior portion of the mandible could be palpated. The results of the physical examination were essentially normal; there was no sign of involvement of neoplastic disease in the regional lymph nodes, cranial nerves, or lungs. The findings from preoperative clinical laboratory procedures did not show any significant abnormal deviations. At surgery, the right half of the mandible was removed along with the right part of the floor of the mouth and the right half of the tongue. Between the mandible and the tongue, there was a 2-cm mass of neoplastic tissue as well as islands of neoplastic tissue within the bone of the mandible, The patient's recovery from surgery was uncomplicated.
Fig 4--Photomicrograph of adenoid cystic carcinoma with invasion o/ bone (orig mag X40, H & E).
Three years postoperatively, there has been no evidence of recurrent or metastatic disease. The patient has tolerated the surgical defect well; a prosthetic replacement is currently being considered.
Discussion
Fig 3--Top: Radiograph four months after endodontic treatment; bottom, panoramic radiograph a year after endodontic treatment.
This case illustrates several important principles to be considered in the practice of endodontics. First, diagnosis of a periapical lesion cannot be made from a radiograph alone. Even though this is commonly said, it is an axiom that frequently is tempting to ignore. Inflammatory lesions form the basis for endodontic practice, but each lesion should be evaluated carefully; a differential diagnosis should be formulated. Inclusions of salivary gland tissue in bone may be a source of confusion. 2 Regarding neoplastic lesions of the salivary gland, Worth a said that it may not be possible to identify the lesion as a tumor rather than as a cyst. Dodd and Jing ~ have given several helpful suggestions for radiographic differentiation. They emphasized destruction about the canal or foramen as a pertinent sign and enlargement of the mental foramen or canal as a suggestive sign. This latter observation is es-
pecially significant since adenoid cystic carcinoma has a tendency for perineural lymphatic invasion. Since the tumor follows the nerves, adenoid cystic carcinomas frequently are painful. Eby and associates ~ have questioned the frequency of pain as a symptom. They said that symptoms cannot be correlated with perineural extension; in their series of patients, perineural extension bore no relation to the clinical course or prognosis. Adenoid cystic carcinoma has been described as having a protracted clinical course that usually follows a pattern of relentless local infiltration; it has ultimate potential for regional and distant metastatic spread. ~ As a lesion of the mandible, the adenoid cystic carcinoma is rare and indistinguishable histologically from the adenoid cystic carcinoma that arises in primary sites elsewhere in the region of the head and neck. 7 It is recognized more frequently now; this is shown by the reports of Bumsted, '~ Richard and Ziskind, 9 Bradley, 1~ Verbin and Stiff, 11 and Schlagel and associates. 1 Clinically, there is no method to determine whether salivary tissue is present; only by surgical removal and examination of the tissue can a diagnosis be made. 1~ Sialography has recently been suggested as a help
77
JOURNAL OF ENDODONTICS ] VOL I, NO 2, FEBRUARY 1975
in showing certain inclusions of the salivary gland. 14 Inclusions of the salivary gland are most frequently seen as well-defined radiolucent areas below the inferior alveolar nerve and in the area of the angle of the mandible. Sublingual glands and minor salivary glands are frequently the sites of adenoid cystic carcinomas. 4,~ Since the original complaint of this patient in 1968 was swelling in the right side of the floor of the mouth, the biopsy material was reviewed carefully; however, no neoplastic tissue was found. At the time of the biopsy of the jaw in 1971, a small opening was found in the lingual cortical bone. It is possible that the tumor originated in the sublingual gland, perforated the lingual cortical plate, and caused destruction of the bone. Under these circumstances, the tumor should have been discovered in 1968 or 1969. It also is possible that tissue of the salivary gland was originally present in the mandible without sufficient radiographic evidence of it. It is possible that the tumor began as a central bony lesion and later penetrated the lingual cortex. This case emphasizes the need for adequate follow-up and evaluation. Radiographic evidence of reossification in a radiolucent periapical lesion as a result of inflammation is to be expected within six months. ~.' Further treatment must be considered if the lesion has enlarged rather than resolved during this time. In view of the current case, surgical exploration should be the foremost treatment in the armamentarium of the endodontist. The importance of periapical curettage and of the submission of any tissue removed for microscopic study also is illustrated by this case. If the curetted tissue had been submitted for microscopic diagnosis, the lesion could have been treated almost a year 78
earlier. It must be recognized that early diagnosis of malignant diseases is a major factor in determining prognosis. A biopsy at the initial endodontic surgery would have enabled earlier treatment, improved the prognosis, and eliminated the unnecessary pain and anxiety the patient experienced for almost a year. In addition to periapical granulomas and cysts, the differential diagnosis of radiolucent lesions in the mandible of an adult must include metastatic neoplasms. Waldon IG said that metastatic carcinoma is the most common malignant process involving the jaws. Some metastatic foci may be well circumscribed; they will show few of the radiographic features usually associated with malignancies. Constant suspicion of any radiolucency of the jaw must be maintained, particularly in patients who may have cancer of the breast, prostate gland, lung, or thyroid gland. Summary
A 50-year-old woman had a painful radiolucent lesion involving the bone around the right premolars. Since root canal filling and apical curettage were not successful, a biopsy was performed almost a year later. The resultant diagnosis was adenoid cystic carcinoma. Three years after hemimandibulectomy and partial glossectomy, the patient remains free of tumor. The submission of any tissue that is surgically removed from a patient is mandatory since primary and metastatic neoplastic lesions have the potential to appear as radiolucent lesions in the jaw. Close follow-up for all endodontic patients is a necessary part of endodontic practice.
Dr. Burkes is associate professor and head of the section of oral pathology at the UNC School of Dentistry. Requests
for reprints should be directed to Dr. Burkes, School of Dentistry, University of North Carolina, Chapel Hill, NC 27514. References 1. Schlagel, E.; Seltzer, R.J.; and Newman, J.l. Apicoectomy as an adjunct to diagnosis. NY State Dent J 39:156 March 1973. 2. Palladino, V.S.; Rose, S.A.; and Curran, T. Salivary gland tissue in the mandible and Stafne's mandibular 'cysts'. JADA 7{):388 Feb 1965. 3. Worth, H.M. Principles and practices of oral radiographic interpretation. Chicago, Year Book Medical Publishers, 1963, p 545. 4. Dodd, G.D., and Jing, B.S. Radiographic findings in adenoid cystic carcinoma of the head and neck. Ann Otol Rhinol Laryngol 81:591 Aug 1972. 5. Eby, L.A.; Johnson, D.S.; and Baker, H.W. Adenoid cystic carcinoma of the head and neck. Cancer 29:1160 May 1972. 6. Smith, L.C.; Lane, N.; and Rankow, R.M. Cylindroma. A report of fifty-eight cases. Am J Surg 110:519 Oct 1965. 7. Stoil, H.C.; Marchetta, F.C.; and Schobinger, R. Malignant epithelial turfiors of the mandible and maxilla. Arch Pathol 64:239 Sept 1957. 8. Bumsted, W.D. Cylindroma of the mandible. Oral Surg 8:546 May 1955. 9. Richard, E.L., and Ziskind, J. Aberrant salivary gland tissue in mandible. Oral Surg 10:1086 Oct 1957. 10. Bradley, J.C. A case of cylindroma of the mandible. Br J Oral Surg 5:186 March 1968. 11. Verbin, R.S., and Stiff, R.H. What's your diagnosis? Adenocystic carcinoma. Odontol Bull 53:12 March 1973. 12. Abramson, A.S. Ectopic submaxillary gland in the mandible: report of case. JADA 73:1114 Nov 1966. 13. Araiche, M., and Brode, H. Aberrant salivary gland tissue in mandible. Oral Surg 12:727 June 1959. 14. Seward, G.R. Salivary gland inclusions in the mandible. Br Dent J 108: 321 May 3, 1960. 15. Bender, I.B.; Seltzer, S.; and Turkenkopf, S. To culture or not to culture. Oral Surg 18:527 Oct 1964. 16. Waldron, C.A. Nonodontogenic neoplasms, cysts, and allied conditions of the jaws. Semin Roentgenol 6:414 Oct 1971.
ADENOID CYSTIC CARCINOMA OF THE MANDIBLE MASQUERADING AS PERIAPICAL INFLAMMATION
E. Jefferson Burkes, Jr., DDS, C h a p e l Hill
Radiolucent defects of the jaws that are associated with pulpless teeth and that produce pain are most commonly related to inflammatory conditions. However, there are many disease conditions that may produce radiolucency of the jaw, some of these are malignant neoplasms. Because of these possibilities, the dentist performing root canal therapy must be constantly alert for neoplasia and must be thorough in his diagnosis and follow-up. Schlagel and others1 have said, "Perhaps apicoectomy should be used more routinely for its diagnostic value rather than as a treatment modality of last resort." They further emphasized that all tissues surgically removed should be submitted for microscopic examination. This report presents a case in which an adenoid cystic carcinoma developed into a radiolucent lesion that simulated periapical inflammation. Report of C a s e In February 1968, a 50-year-old white woman was originally seen by her dentist for construction of a bridge to replace the mandibular right first molar (Fig 1). In March 1969, she sought further consultation because of a slightly enlarged and ques76
Fig 1--Radiograph tion of bridge.
before prepara-
tionably tender area in the right sublingual gland. Clinically, the gland was functional and moved freely. An incisional biopsy and subsequent removal of the sublingual gland showed only obstructive sialoadenitis. From that time until July 1970, when the patient consulted an oral surgeon, she continued to have vague nonspecific sensations in the area. Radiographs of the area showed an enlarged right inferior alveolar canal and a diffuse, poorly defined radiolucent area involving the bone around the first premolar and canine (Fig 2). The pulp of the first premolar did not respond to thermal tests. Because of the pain, the radiolucent area, and the negative reaction to the pulpal test of the first premolar,
endodontic treatment with curettage and biopsy was advised. The patient was referred to her dentist for this treatment. The curettage and root canal filling were performed in September; however, the tissue that was removed was not submitted for microscopic examination. Radiographs again were obtained in January 1971 because of continued pain (Fig 3, top), but there was no treatment done at that time. By November, the pain had become more severe. The patient returned to the oral surgeon who had originally recommended the endodontic therapy. At that time, radiographs showed enlargement of the radiolucent area with involvement of the second premolar and incisors. The borders of the lesion were irregular and the bone appeared ragged in adjacent areas of the mandible (Fig 3, bottom). An incisional biopsy and exploration of the area showed an adherent, tan, glistening mass that filled a large bony crypt. The lingual cortical plate appeared to be intact except for a 2-mm ovoid perforation in the first premolar area. The histologic diagnosis was adenoid cystic carcinoma. Large cystic spaces within islatads of hyperchromatic epithelial cells were scattered throughout the specimen (Fig 4). Because of the patient's reluctance to undergo extensive surgery, therapy
q .t,
Fig 2--Radiograph before endodontic treatment.
JOURNAL OF ENDODONTICS I VOL 1, NO 2, FEBRUARY 1975
was delayed for almost three months. At the preoperative examination, a firm mass that was attached to the lingual side of the right anterior portion of the mandible could be palpated. The results of the physical examination were essentially normal; there was no sign of involvement of neoplastic disease in the regional lymph nodes, cranial nerves, or lungs. The findings from preoperative clinical laboratory procedures did not show any significant abnormal deviations. At surgery, the right half of the mandible was removed along with the right part of the floor of the mouth and the right half of the tongue. Between the mandible and the tongue, there was a 2-cm mass of neoplastic tissue as well as islands of neoplastic tissue within the bone of the mandible, The patient's recovery from surgery was uncomplicated.
Fig 4--Photomicrograph of adenoid cystic carcinoma with invasion o/ bone (orig mag X40, H & E).
Three years postoperatively, there has been no evidence of recurrent or metastatic disease. The patient has tolerated the surgical defect well; a prosthetic replacement is currently being considered.
Discussion
Fig 3--Top: Radiograph four months after endodontic treatment; bottom, panoramic radiograph a year after endodontic treatment.
This case illustrates several important principles to be considered in the practice of endodontics. First, diagnosis of a periapical lesion cannot be made from a radiograph alone. Even though this is commonly said, it is an axiom that frequently is tempting to ignore. Inflammatory lesions form the basis for endodontic practice, but each lesion should be evaluated carefully; a differential diagnosis should be formulated. Inclusions of salivary gland tissue in bone may be a source of confusion. 2 Regarding neoplastic lesions of the salivary gland, Worth a said that it may not be possible to identify the lesion as a tumor rather than as a cyst. Dodd and Jing ~ have given several helpful suggestions for radiographic differentiation. They emphasized destruction about the canal or foramen as a pertinent sign and enlargement of the mental foramen or canal as a suggestive sign. This latter observation is es-
pecially significant since adenoid cystic carcinoma has a tendency for perineural lymphatic invasion. Since the tumor follows the nerves, adenoid cystic carcinomas frequently are painful. Eby and associates ~ have questioned the frequency of pain as a symptom. They said that symptoms cannot be correlated with perineural extension; in their series of patients, perineural extension bore no relation to the clinical course or prognosis. Adenoid cystic carcinoma has been described as having a protracted clinical course that usually follows a pattern of relentless local infiltration; it has ultimate potential for regional and distant metastatic spread. ~ As a lesion of the mandible, the adenoid cystic carcinoma is rare and indistinguishable histologically from the adenoid cystic carcinoma that arises in primary sites elsewhere in the region of the head and neck. 7 It is recognized more frequently now; this is shown by the reports of Bumsted, '~ Richard and Ziskind, 9 Bradley, 1~ Verbin and Stiff, 11 and Schlagel and associates. 1 Clinically, there is no method to determine whether salivary tissue is present; only by surgical removal and examination of the tissue can a diagnosis be made. 1~ Sialography has recently been suggested as a help
77
JOURNAL OF ENDODONTICS ] VOL I, NO 2, FEBRUARY 1975
in showing certain inclusions of the salivary gland. 14 Inclusions of the salivary gland are most frequently seen as well-defined radiolucent areas below the inferior alveolar nerve and in the area of the angle of the mandible. Sublingual glands and minor salivary glands are frequently the sites of adenoid cystic carcinomas. 4,~ Since the original complaint of this patient in 1968 was swelling in the right side of the floor of the mouth, the biopsy material was reviewed carefully; however, no neoplastic tissue was found. At the time of the biopsy of the jaw in 1971, a small opening was found in the lingual cortical bone. It is possible that the tumor originated in the sublingual gland, perforated the lingual cortical plate, and caused destruction of the bone. Under these circumstances, the tumor should have been discovered in 1968 or 1969. It also is possible that tissue of the salivary gland was originally present in the mandible without sufficient radiographic evidence of it. It is possible that the tumor began as a central bony lesion and later penetrated the lingual cortex. This case emphasizes the need for adequate follow-up and evaluation. Radiographic evidence of reossification in a radiolucent periapical lesion as a result of inflammation is to be expected within six months. ~.' Further treatment must be considered if the lesion has enlarged rather than resolved during this time. In view of the current case, surgical exploration should be the foremost treatment in the armamentarium of the endodontist. The importance of periapical curettage and of the submission of any tissue removed for microscopic study also is illustrated by this case. If the curetted tissue had been submitted for microscopic diagnosis, the lesion could have been treated almost a year 78
earlier. It must be recognized that early diagnosis of malignant diseases is a major factor in determining prognosis. A biopsy at the initial endodontic surgery would have enabled earlier treatment, improved the prognosis, and eliminated the unnecessary pain and anxiety the patient experienced for almost a year. In addition to periapical granulomas and cysts, the differential diagnosis of radiolucent lesions in the mandible of an adult must include metastatic neoplasms. Waldon IG said that metastatic carcinoma is the most common malignant process involving the jaws. Some metastatic foci may be well circumscribed; they will show few of the radiographic features usually associated with malignancies. Constant suspicion of any radiolucency of the jaw must be maintained, particularly in patients who may have cancer of the breast, prostate gland, lung, or thyroid gland. Summary
A 50-year-old woman had a painful radiolucent lesion involving the bone around the right premolars. Since root canal filling and apical curettage were not successful, a biopsy was performed almost a year later. The resultant diagnosis was adenoid cystic carcinoma. Three years after hemimandibulectomy and partial glossectomy, the patient remains free of tumor. The submission of any tissue that is surgically removed from a patient is mandatory since primary and metastatic neoplastic lesions have the potential to appear as radiolucent lesions in the jaw. Close follow-up for all endodontic patients is a necessary part of endodontic practice.
Dr. Burkes is associate professor and head of the section of oral pathology at the UNC School of Dentistry. Requests
for reprints should be directed to Dr. Burkes, School of Dentistry, University of North Carolina, Chapel Hill, NC 27514. References 1. Schlagel, E.; Seltzer, R.J.; and Newman, J.l. Apicoectomy as an adjunct to diagnosis. NY State Dent J 39:156 March 1973. 2. Palladino, V.S.; Rose, S.A.; and Curran, T. Salivary gland tissue in the mandible and Stafne's mandibular 'cysts'. JADA 7{):388 Feb 1965. 3. Worth, H.M. Principles and practices of oral radiographic interpretation. Chicago, Year Book Medical Publishers, 1963, p 545. 4. Dodd, G.D., and Jing, B.S. Radiographic findings in adenoid cystic carcinoma of the head and neck. Ann Otol Rhinol Laryngol 81:591 Aug 1972. 5. Eby, L.A.; Johnson, D.S.; and Baker, H.W. Adenoid cystic carcinoma of the head and neck. Cancer 29:1160 May 1972. 6. Smith, L.C.; Lane, N.; and Rankow, R.M. Cylindroma. A report of fifty-eight cases. Am J Surg 110:519 Oct 1965. 7. Stoil, H.C.; Marchetta, F.C.; and Schobinger, R. Malignant epithelial turfiors of the mandible and maxilla. Arch Pathol 64:239 Sept 1957. 8. Bumsted, W.D. Cylindroma of the mandible. Oral Surg 8:546 May 1955. 9. Richard, E.L., and Ziskind, J. Aberrant salivary gland tissue in mandible. Oral Surg 10:1086 Oct 1957. 10. Bradley, J.C. A case of cylindroma of the mandible. Br J Oral Surg 5:186 March 1968. 11. Verbin, R.S., and Stiff, R.H. What's your diagnosis? Adenocystic carcinoma. Odontol Bull 53:12 March 1973. 12. Abramson, A.S. Ectopic submaxillary gland in the mandible: report of case. JADA 73:1114 Nov 1966. 13. Araiche, M., and Brode, H. Aberrant salivary gland tissue in mandible. Oral Surg 12:727 June 1959. 14. Seward, G.R. Salivary gland inclusions in the mandible. Br Dent J 108: 321 May 3, 1960. 15. Bender, I.B.; Seltzer, S.; and Turkenkopf, S. To culture or not to culture. Oral Surg 18:527 Oct 1964. 16. Waldron, C.A. Nonodontogenic neoplasms, cysts, and allied conditions of the jaws. Semin Roentgenol 6:414 Oct 1971.
