Glossopyrosis due to adenoid cystic carcinoma h’amuel V. Pellegrixo,
D.D.S., Reading, Pa.
A combination of radiation therapy and surgical results in the treatment of adenoid cystic carcinoma.
intervention
produces
the
best
T
wo of the most difficult and frustrating conditions to diagnose and treat are glossopyrosis and glossodynia. Burning and neuralgia of the tongue and inner surface of the mandible may be associated with excessive smoking, drinking of alcoholic beverages, or eating of highly seasoned foods. Lipsticks, dentifrices, denture bases, or mouthwashes may be responsible. Occasionally, decreased intermaxillary space or irritation of the tongue may reflex pain to the anterior region of the tongue. Glossopharyngeal neuralgia produces severe paroxysms of pain which arise in certain areas of the posterior part of the tongue on swallowing. When the symptoms of burning or pain are associated with change of color in the tongue, the cause is usually systemic. Among the possible causes are pernicious anemia, B-complex nutritional deficiencies, gonadal deficiency, or irondeficiency anemia. There are positive findings, such as redness, atrophy of the lingual papillae, and ulceration of the lateral borders of the tongue. The more difficult diagnostic problems are related to those symptoms with no obvious lesions. There is always the possibility that these symptoms may be unrecognized diabetes, secondary anemia, or early pernicious anemia before the typical lingual changes occur. A characteristic finding is that the pain or burning does not interfere with eating or sleeping. It is usually present only in the daytime and when the patient is fatigued or the mind is unoccupied. The distribution does not follow any recognized anatomic pattern. Visual signs are not present. Blood tests result in normal findings. Mouthwashes are of no value and may cause chemical irritation. Any local irritative factors should be removed, including smoking. Large therapeutic doses of B-complex (three capsules of a therapeutic formula per day) can be given if vitamin deficiency is suspected. Hormone replacement therapy has not been effective.l No treatment should be instituted if no cause is found. Pain or distorted sensation in the tongue, however, may be referred and mediated through the lingual nerve, and not originate in the tongue at a11.2 521
522
Pellegrino
CASE
REPORT
Oral April,
Burg. 1977
D. M., a 5%year-old woman, was referred by her physician on Jan. 23, 1975, because of episodes of burning, pain, and numbness in the left side of the tongue. The burning was aggravated when she ate pretzels and spicy foods. The symptoms had been present for several months. Examination of the tongue revealed no lesions or changes. Radiographs of the mandible were negative for pathoses. A sharp lingual cusp of the mandibular left second premolar was removed. The salivary flow from the submaxillary gland was considered to be normal, and there was a questionable left submaxillary lymphadenopathy. Although the patient seemed to be a well-adjusted woman, emotional instability was considered. She had undergone menopause 9 years earlier. Because of the paucity of physical signs, the patient was given tranquilizers and asked to return. She returned on March 19, 1975, still complaining of burning of the tongue in the morning accompanied by excessive salivation. Intraoral examination was again nonproductive, except that saliva no longer flowed freely from Wharton’s duct on the left side. There was now a fixed, firm swelling at the inferior border of the left mandible. Because of the character of the swelling, neoplasm was suspected. Consequently, a biopsy was performed, and the submaxillary mass was determined to be an adenoid cystic carcinoma.
DISCUSSION
Adenoid cystic carcinoma may have a wide variation of architectural patterns, not all of which individually suggest the malignant character of the lesion. Small incisional biopsy specimensor needle biopsies may represent only a portion of the tumor and may lead to inaccuracies in diagnosis.3Some surgeons think that open biopsies of this tumor seedsthe skin with malignant cells. Needle biopsies in several areas will minimize the possibilities of error and seeding. If an open biopsy is to be performed, the surgeon should be prepared to complete the excision of the tumor and possible neck dissection at the same time. Adenoid cystic carcinoma was named cylindroma by Billroth in 1895.4 It is a malignant tumor that originates mainly in salivary glands. Because it resembles basal-cell carcinoma of the skin in its growth, it has been called basalionta. It occurs more often in women in the fifth and sixth decadesof life. Squamous-cell carcinoma makes up about 90 per cent of all oral cancers and causes99 per cent of the deaths from mouth cancer. Adenoid tumors of the mixed tumor type and adenocarcinoma make up most of the remainder, or about 8 per cent, but they contribute little to the death rate.’ Cylindroma accounts for about 4 per cent of malignant submaxillary tumors.4 Adenocarcinoma grows far more slowly than does epidermoid carcinoma and metastasizes less frequently. Delay in treatment, although serious, is not frequently tragic, as is delay in treatment of epidermoid carcinoma.1 Adenoid cystic carcinoma grows slowly and infiltrates the surrounding tissues, bone nerve sheaths, and lymph vessels. The lesion often extends farther than clinical examination reveals. For this reason, surgical cure is difficult to achieve and recurrence is common. The tumor is composed of two types of tissue, the undifferentiated secretory cells and radio-resistant myoepithelial cells, thus making it difficult to control by radiation alone. A combination of radiation and operation seemsto provide the best chance of cure. Metastasis occurs late, traveling by perineural lymphatics. This accounts for the neurologic symptoms. Cervical node involvement eventually occurs in about
Volume Number
Glossopyrosis
43 4
due to adenoid
cystic
carcinovm
523
30 per cent of the cases,but distant metastasesto lung, bone, and brain occur in a high proportion of the patients.j The patient in the present report received 5,000 rads to the tumor and lymph nodes of the neck over a period of 5 weeks. Subsequent to the irradiation, there was a scaling of the skin and a mucositis of the oral mucosa. A slight fullness was still felt in the submandibular area. The radiation therapy was followed by excision of the tumor and radical neck dissection on the left side. Tumor cells were found throughout the specimen. Because of the invasive nature of adenoid cystic adenoma, the lingual nerve was sacrificed, which produced insensitivity in the left side of the tongue. Chest x-ray films and brain scan 1 year later failed to show distant metastasis. There is no evidence of a return of the tumor, and the symptoms of burning and pain in the tongue have not returned. CONCLUSION
Glossopyrosis and glossodynia may occur from local or systemic factors or from any irritation along the course of the lingual nerve. Although cylindroma is relatively rare, it must be considered along with other malignant lesions when neurologic symptoms of burning and pain of the tongue persist. Examination of the lesion is best carried out as multiple needle biopsies rather than as open biopsy, in order to avoid seeding of the skin and lymphatics of the neck. Recurrences are frequent and, because of the slow growth, there must be a long follow-up period. Metastases to the lung and brain, although late, occur in large numbers of patients. Best results are obtained by a combination of radiation therapy and operation because the infiltration is so extensive that operation alone may not eliminate the tumor completely. REFERENCES
1. Burket, L. W.: Oral Medicine, ed. 3, Philadelphia, 1975, J. B. Lippineott Company, p. 116. 2. Shafer, W. CT., Hines, M. K., and Levy, M.: A Textbook of Oral Pathology, ed. 2, Philadelphia! 1964, W. B. Saunders Company, p. 596. 3. Adkms, K. F., and Campbell, A. F. G.: Adenocystic Carcinoma of the Palate, ORAL SURG. 30: 734-741, 1970. 4. Fuchihata, H., Takuro, W., and Toshihiko, I.: Radiotherapy of Adenoid Cystic Carcinoma of the Head and Neck, ORAL SURG.~~: 753-759,1973. 5. Forman, G. H.: Adenoid Cystic Carcinoma of the Floor of the Mouth Presenting by Metastasis, ORAL SURG. 28: l-8, 1970. Reprint
requests
to:
Dr. Samuel V. Pellegrino 234236 North 6th St. Reading, Pa. 19601
D.D.S., Reading, Pa.
A combination of radiation therapy and surgical results in the treatment of adenoid cystic carcinoma.
intervention
produces
the
best
T
wo of the most difficult and frustrating conditions to diagnose and treat are glossopyrosis and glossodynia. Burning and neuralgia of the tongue and inner surface of the mandible may be associated with excessive smoking, drinking of alcoholic beverages, or eating of highly seasoned foods. Lipsticks, dentifrices, denture bases, or mouthwashes may be responsible. Occasionally, decreased intermaxillary space or irritation of the tongue may reflex pain to the anterior region of the tongue. Glossopharyngeal neuralgia produces severe paroxysms of pain which arise in certain areas of the posterior part of the tongue on swallowing. When the symptoms of burning or pain are associated with change of color in the tongue, the cause is usually systemic. Among the possible causes are pernicious anemia, B-complex nutritional deficiencies, gonadal deficiency, or irondeficiency anemia. There are positive findings, such as redness, atrophy of the lingual papillae, and ulceration of the lateral borders of the tongue. The more difficult diagnostic problems are related to those symptoms with no obvious lesions. There is always the possibility that these symptoms may be unrecognized diabetes, secondary anemia, or early pernicious anemia before the typical lingual changes occur. A characteristic finding is that the pain or burning does not interfere with eating or sleeping. It is usually present only in the daytime and when the patient is fatigued or the mind is unoccupied. The distribution does not follow any recognized anatomic pattern. Visual signs are not present. Blood tests result in normal findings. Mouthwashes are of no value and may cause chemical irritation. Any local irritative factors should be removed, including smoking. Large therapeutic doses of B-complex (three capsules of a therapeutic formula per day) can be given if vitamin deficiency is suspected. Hormone replacement therapy has not been effective.l No treatment should be instituted if no cause is found. Pain or distorted sensation in the tongue, however, may be referred and mediated through the lingual nerve, and not originate in the tongue at a11.2 521
522
Pellegrino
CASE
REPORT
Oral April,
Burg. 1977
D. M., a 5%year-old woman, was referred by her physician on Jan. 23, 1975, because of episodes of burning, pain, and numbness in the left side of the tongue. The burning was aggravated when she ate pretzels and spicy foods. The symptoms had been present for several months. Examination of the tongue revealed no lesions or changes. Radiographs of the mandible were negative for pathoses. A sharp lingual cusp of the mandibular left second premolar was removed. The salivary flow from the submaxillary gland was considered to be normal, and there was a questionable left submaxillary lymphadenopathy. Although the patient seemed to be a well-adjusted woman, emotional instability was considered. She had undergone menopause 9 years earlier. Because of the paucity of physical signs, the patient was given tranquilizers and asked to return. She returned on March 19, 1975, still complaining of burning of the tongue in the morning accompanied by excessive salivation. Intraoral examination was again nonproductive, except that saliva no longer flowed freely from Wharton’s duct on the left side. There was now a fixed, firm swelling at the inferior border of the left mandible. Because of the character of the swelling, neoplasm was suspected. Consequently, a biopsy was performed, and the submaxillary mass was determined to be an adenoid cystic carcinoma.
DISCUSSION
Adenoid cystic carcinoma may have a wide variation of architectural patterns, not all of which individually suggest the malignant character of the lesion. Small incisional biopsy specimensor needle biopsies may represent only a portion of the tumor and may lead to inaccuracies in diagnosis.3Some surgeons think that open biopsies of this tumor seedsthe skin with malignant cells. Needle biopsies in several areas will minimize the possibilities of error and seeding. If an open biopsy is to be performed, the surgeon should be prepared to complete the excision of the tumor and possible neck dissection at the same time. Adenoid cystic carcinoma was named cylindroma by Billroth in 1895.4 It is a malignant tumor that originates mainly in salivary glands. Because it resembles basal-cell carcinoma of the skin in its growth, it has been called basalionta. It occurs more often in women in the fifth and sixth decadesof life. Squamous-cell carcinoma makes up about 90 per cent of all oral cancers and causes99 per cent of the deaths from mouth cancer. Adenoid tumors of the mixed tumor type and adenocarcinoma make up most of the remainder, or about 8 per cent, but they contribute little to the death rate.’ Cylindroma accounts for about 4 per cent of malignant submaxillary tumors.4 Adenocarcinoma grows far more slowly than does epidermoid carcinoma and metastasizes less frequently. Delay in treatment, although serious, is not frequently tragic, as is delay in treatment of epidermoid carcinoma.1 Adenoid cystic carcinoma grows slowly and infiltrates the surrounding tissues, bone nerve sheaths, and lymph vessels. The lesion often extends farther than clinical examination reveals. For this reason, surgical cure is difficult to achieve and recurrence is common. The tumor is composed of two types of tissue, the undifferentiated secretory cells and radio-resistant myoepithelial cells, thus making it difficult to control by radiation alone. A combination of radiation and operation seemsto provide the best chance of cure. Metastasis occurs late, traveling by perineural lymphatics. This accounts for the neurologic symptoms. Cervical node involvement eventually occurs in about
Volume Number
Glossopyrosis
43 4
due to adenoid
cystic
carcinovm
523
30 per cent of the cases,but distant metastasesto lung, bone, and brain occur in a high proportion of the patients.j The patient in the present report received 5,000 rads to the tumor and lymph nodes of the neck over a period of 5 weeks. Subsequent to the irradiation, there was a scaling of the skin and a mucositis of the oral mucosa. A slight fullness was still felt in the submandibular area. The radiation therapy was followed by excision of the tumor and radical neck dissection on the left side. Tumor cells were found throughout the specimen. Because of the invasive nature of adenoid cystic adenoma, the lingual nerve was sacrificed, which produced insensitivity in the left side of the tongue. Chest x-ray films and brain scan 1 year later failed to show distant metastasis. There is no evidence of a return of the tumor, and the symptoms of burning and pain in the tongue have not returned. CONCLUSION
Glossopyrosis and glossodynia may occur from local or systemic factors or from any irritation along the course of the lingual nerve. Although cylindroma is relatively rare, it must be considered along with other malignant lesions when neurologic symptoms of burning and pain of the tongue persist. Examination of the lesion is best carried out as multiple needle biopsies rather than as open biopsy, in order to avoid seeding of the skin and lymphatics of the neck. Recurrences are frequent and, because of the slow growth, there must be a long follow-up period. Metastases to the lung and brain, although late, occur in large numbers of patients. Best results are obtained by a combination of radiation therapy and operation because the infiltration is so extensive that operation alone may not eliminate the tumor completely. REFERENCES
1. Burket, L. W.: Oral Medicine, ed. 3, Philadelphia, 1975, J. B. Lippineott Company, p. 116. 2. Shafer, W. CT., Hines, M. K., and Levy, M.: A Textbook of Oral Pathology, ed. 2, Philadelphia! 1964, W. B. Saunders Company, p. 596. 3. Adkms, K. F., and Campbell, A. F. G.: Adenocystic Carcinoma of the Palate, ORAL SURG. 30: 734-741, 1970. 4. Fuchihata, H., Takuro, W., and Toshihiko, I.: Radiotherapy of Adenoid Cystic Carcinoma of the Head and Neck, ORAL SURG.~~: 753-759,1973. 5. Forman, G. H.: Adenoid Cystic Carcinoma of the Floor of the Mouth Presenting by Metastasis, ORAL SURG. 28: l-8, 1970. Reprint
requests
to:
Dr. Samuel V. Pellegrino 234236 North 6th St. Reading, Pa. 19601
