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Bronchial Adenocarcinoma Presenting as a Lingual Tonsillar Metastasis
fore, as a last resort, a 14 mm Dacron graft, 6 cm long, was utilized for construction of the pathway to the left atrium. Cobanoglu et al' have reported on surgical treatment of the scimitar syndrome involving use of a 12 mm polytetrafiuoroethylene (PTFE) graft. However, in that case, the graft was extracardiac and used concurrently with an intracardiac tunnel created with a PTFE patch. A satisfactory prosthetic replacement for the venous system in man is not yet available. Craft failures are well documented, 5 and a PTFE graft might have been better in this situation, except, unfortunately, it was unavailable in the correct size at that time. In this patient, the 21-month postoperative clinical course has been uneventful. In conclusion, we believe that the method of surgical treatment described herein may be a viable alternative in certain rare cases where, due to anatomic relationships, the usual procedures are either very difficult or impossible.
FIGURE 1. Lingual tonsil. Lymphoid tissue has been replaced by cords of epithelial neoplastic cells which are typical of poorly differentiated adenocarcinoma (original magnification x 130).
ACKNOWLEDGMENT: We appreciate the great help of Mr. Jack Arends and Mr. Hiromi Shioneri in preparation of this manuscript. REFERENCES
l Wright JS, McCredie RM, Torda TA. Total right anomalous pulmonary venous connection. Ann Thorac Surg 1973; 16:512-17 2 Murphy JW, Kerr AR, Kirklin JW. lntracardiac repair for anomalous pulmonary venous connection of right lung to inferior vena cava. Ann Thorac Surg 1971; 11:38-42 3 Neill CA. Development of the pulmonary veins, with reference to the embryology of anomalies of pulmonary venous return. Pediatrics 1956; 18:880-87 4 Cobanoglu A, Hanlon JT, Combs OT, Starr A. Combined intracardiac and extracardiac repair of scimitar syndrome with anomalous pulmonary veins to both cavae. J Thorac Cardiovasc Surg 1984; 88: 141-46 5 Dale WA, Harris J, Terry RB. Polytetrafluoroethylene reconstruction of the inferior vena cava. Surgery 1984; 95:625-30
Bronchial Adenocarclnoma Presenting as a Lingual Tonslllar Metastasis* R. Monforte, M.D.; A Ferrer, M.D.;}. M. Montserrat, M.D.;
C. Picado, M.D.; and A Palacfn, M.D. We describe a case of a lingual tonsillar metastasis as the first manifestation of a bronchial adenocarcinoma. Tonsillar metastases infrequently become manifest before the diagnosis of the primary neoplasm. A review of the literature disclosed 89 cases of carcinoma metastasizing to the pa1atine tonsil, but no one has reported the involvement of the lingua1 tonsil. Our patient is the first described case of bronchogenic carcinoma with this unusua1 form of presentation and furthermore is a1so the first carcinoma metastasizing to the lingua] tonsil.
T
onsillar metastases from nonhematologic malignant neoplasms are very uncommon, and at the present time,
*From the Servei de Neumologia and Servei de Patolog(a, Hospital Clinic i Provincial, Barcelona, Spain.
Reprint requests: Dr: Montserrat, Seroei Neumologia, Hospital Clinic, Villarroel 170, Barcelona, Spain 08036
1122
only 89 cases have been described. i.u In 12 of them, the primary neoplasm was bronchogenic carcinoma, mainly of the small-cell type, and only one case was adenocarcinoma.12 Tonsillar metastases rarely were the first evidence of neoplastic disease. In the majority of cases, these metastases become manifest after the diagnosis of the primary tumor and generally are part of widespread systemic disease. 1 We describe a young man with adenocarcinoma of the lung in whom the presenting symptoms were due to lingual tonsillar metastasis. CASE REPORT
A 25-year-old man was admitted because of fever, dyspnea, and a right tonsillar mass. He was well until 20 days before admission, when he started to complain of pain in the right side of the throat. Later, he became aware of a mass in the right tonsil and developed fever and exertional dyspnea. His temperature was 38°C. Examination revealed a right nonulcerated mass involving the lingual tonsil. There were signs of right pleural effusion. No lymphadenopathy was found. The erythrocyte sedimentation rate was 5 mm at the 6rst hour, the hematocrit reading was 37.2 percent, the white blood cell count was 14. 5 X 10"/L, with a normal differential cell count. The platelet count was 100 x 10"/L. Blood gas levels with the patient breathing room air disclosed that the arterial oxygen pressure was 48 mm Hg, arterial carbon dioxide tension was 30 mm Hg, and pH was 7.44. The chest x-ray Sim showed a right hilar mass with a massive right-sided pleural effusion. Biopsy ofthe lingual tonsillar mass revealed a poorly differentiated adenocarcinoma with poorly developed papillary structures (Fig 1). Bronchoscopic examination showed diffuse infiammatory mucosa in the carina and in the right upper lobe bronchus. Cytologic examination ofthe bronchial brushing, right thoracocentesis, and pericardiocentesis disclosed atypical cells of adenocarcinoma type with papillary structures. Bronchial biopsy disclosed lymphangitic spread. After combined chemotherapy (cyclophosphamide, vinblastine, bleomycin, and cisplatin), the tonsillar mass diminished, but later the patient developed progressive dyspnea is spite of pericardia! and pleural drainage and died 25 days after admission. In the autopsy a peripheral mass in the right upper lobe and metastases to hilar nodes, right pleura, pericardium, and lingual tonsil were round. On light microscopy, the neoplasm was mainly composed ofan adenocarcinoma with papillary and solid areas with mucinous contents. Some areas resembled bronchioloalveolar carcinoma, but others showed atypical cells of large and giant cell type (Fig 2). Bronchial Adenocarcinoma (Monforte et al)
metastasis diagnosed befure the primary tumor. 1 Our case is very unusual in a number of aspects. It is the first case ofbronchogenic carcinoma presenting as a tonsillar metastasis. Furthermore, we do not find any other reported case of metastatic carcinoma in the lingual tonsil. Finally, it is interesting to note the atypical histologic features of the primary, with mucous, papillary, bronchioloalveolar, and large-giant cell differentiation. REFERENCES
FIGURE 2A (left). Bronchogenic carcinoma with distinct papillary pattern (original magnification x 80). B (center). Neoplastic cells in alveolar linings (original magnification x 80). C (right). Tumor giant cells with pleomorphic nuclei (original magnification x 130). DISCUSSION
The tonsil is an unusual site of metastatic disease. While review of the literature discloses only 89 cases of carcinoma metastatic to the palatine tonsil, we have not fuund any cases of metastases in the lingual tonsil. 1-ll Primary tonsillar carcinomas are squamous or undifferentiated, but not adenocarcinomas. 13 This fact supports the hypothesis that the lingual mass of our patient was metastatic and not primary. The most common primary tumors fuund are melanoma, hypemephroma, breast carcinoma, and bronchogenic carcinoma, while seminoma and carcinomas of the stomach and pancreas are less common. 1 Metastatic lesions in this part of the oral cavity are generally part of widespread systemic disease. Nevertheless, in six cases ofhypemephroma and in one case of pancreatic carcinoma, the tonsillar involvement was the first evidence of neoplastic disease. 1-lo To our knowledge, only 12 cases of bronchogenic carcinomas metastasizing to the tonsil have been reported. 1 All of them were of the oat cell type except one, which was adenocarcinoma. 12 The patients were men, and their ages ranged from 44 to 71 years. In none was the tonsillar
1 Brownson RJ, Jaques WE, La Monte SE, 7.ollinger WK. Hypernephroma metastatic to the palatine tonsils. Ann Oto! Rhino! Laryngol 1979; 88:235-40 2 Draizin D, Matucci IC, Rothfeld S. Bilateral metastatic tonsillar disease due to renal-cell carcinoma. Ear Nose Throat J 1978; 57:477-80 3 Ashur H, Mizrahi S, Ben-Hur N, Dolberg L. Metastatic malignant melanoma to the palatine tonsils: report of a case. J Oral Surg 1979; 37:110-12 4 Barton TIC, Kesterson GH, Wellman D, McCarty KS. Tonsillar metastasis from carcinoma of the breast with ultrastructural and steroid receptor analyses. Laryngoscope 1980; 90:477-85 5 Feleppa AE, Ellison NM. Metastatic pancreatic adenocarcinoma of the tonsil. Ear Nose Throat J 1981; 60:44-50 6 Craig RM, Glass BJ, Rhyne RR. Malignant melanoma: metastasis to the tonsil. J Am Dent Assoc 1982; 104:89.3-94 7 Passmore AL, Hugh TB, Coleman MJ. Tonsillar metastases from adenocarcinoma of the stomach. Aust NZ J Surg 1982; 52:371-72 8 Born JW, Baarsma EA. Metastatic hypemephroma in the tonsil. J Laryngol Otol 1982; 96:1159-63 9 Myer CM, Wood MD, Donegan JO. Metastatic melanoma to the palatine tonsil. Ear Nose Throat J 1983; 62:62-4 10 Maor E, Tovi F, Sacks M. Carcinoma of the pancreas presenting with bilateral tonsillar metastases. Ann Otol Rhino! Laryngol 1983; 92:192-95 11 Bychko V, Ghosh L, Lundine M, Manaligod JR, McQuire WP, Rose C. Ovarian androblastoma metastatic to tonsil. J Surg Oncol 1984; 27:275-79 12 Ardovin P, Bavdoin J. Jobard J. Sur un cas d'epithelioma glandulaire de l'amygdale palatine accompagnant un cancer du poumon. Rev Laryngol Oto! Rhino! (Bord) 1956; 77:878-91 13 Res ta L, Ricco R, Santangelo A. The histologic typing of tonsillar carcinoma. Tumori 1984; 70:409-16
CHEST I 92 I 8 I DECEMBER, 1987
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FIGURE 1. Lingual tonsil. Lymphoid tissue has been replaced by cords of epithelial neoplastic cells which are typical of poorly differentiated adenocarcinoma (original magnification x 130).
ACKNOWLEDGMENT: We appreciate the great help of Mr. Jack Arends and Mr. Hiromi Shioneri in preparation of this manuscript. REFERENCES
l Wright JS, McCredie RM, Torda TA. Total right anomalous pulmonary venous connection. Ann Thorac Surg 1973; 16:512-17 2 Murphy JW, Kerr AR, Kirklin JW. lntracardiac repair for anomalous pulmonary venous connection of right lung to inferior vena cava. Ann Thorac Surg 1971; 11:38-42 3 Neill CA. Development of the pulmonary veins, with reference to the embryology of anomalies of pulmonary venous return. Pediatrics 1956; 18:880-87 4 Cobanoglu A, Hanlon JT, Combs OT, Starr A. Combined intracardiac and extracardiac repair of scimitar syndrome with anomalous pulmonary veins to both cavae. J Thorac Cardiovasc Surg 1984; 88: 141-46 5 Dale WA, Harris J, Terry RB. Polytetrafluoroethylene reconstruction of the inferior vena cava. Surgery 1984; 95:625-30
Bronchial Adenocarclnoma Presenting as a Lingual Tonslllar Metastasis* R. Monforte, M.D.; A Ferrer, M.D.;}. M. Montserrat, M.D.;
C. Picado, M.D.; and A Palacfn, M.D. We describe a case of a lingual tonsillar metastasis as the first manifestation of a bronchial adenocarcinoma. Tonsillar metastases infrequently become manifest before the diagnosis of the primary neoplasm. A review of the literature disclosed 89 cases of carcinoma metastasizing to the pa1atine tonsil, but no one has reported the involvement of the lingua1 tonsil. Our patient is the first described case of bronchogenic carcinoma with this unusua1 form of presentation and furthermore is a1so the first carcinoma metastasizing to the lingua] tonsil.
T
onsillar metastases from nonhematologic malignant neoplasms are very uncommon, and at the present time,
*From the Servei de Neumologia and Servei de Patolog(a, Hospital Clinic i Provincial, Barcelona, Spain.
Reprint requests: Dr: Montserrat, Seroei Neumologia, Hospital Clinic, Villarroel 170, Barcelona, Spain 08036
1122
only 89 cases have been described. i.u In 12 of them, the primary neoplasm was bronchogenic carcinoma, mainly of the small-cell type, and only one case was adenocarcinoma.12 Tonsillar metastases rarely were the first evidence of neoplastic disease. In the majority of cases, these metastases become manifest after the diagnosis of the primary tumor and generally are part of widespread systemic disease. 1 We describe a young man with adenocarcinoma of the lung in whom the presenting symptoms were due to lingual tonsillar metastasis. CASE REPORT
A 25-year-old man was admitted because of fever, dyspnea, and a right tonsillar mass. He was well until 20 days before admission, when he started to complain of pain in the right side of the throat. Later, he became aware of a mass in the right tonsil and developed fever and exertional dyspnea. His temperature was 38°C. Examination revealed a right nonulcerated mass involving the lingual tonsil. There were signs of right pleural effusion. No lymphadenopathy was found. The erythrocyte sedimentation rate was 5 mm at the 6rst hour, the hematocrit reading was 37.2 percent, the white blood cell count was 14. 5 X 10"/L, with a normal differential cell count. The platelet count was 100 x 10"/L. Blood gas levels with the patient breathing room air disclosed that the arterial oxygen pressure was 48 mm Hg, arterial carbon dioxide tension was 30 mm Hg, and pH was 7.44. The chest x-ray Sim showed a right hilar mass with a massive right-sided pleural effusion. Biopsy ofthe lingual tonsillar mass revealed a poorly differentiated adenocarcinoma with poorly developed papillary structures (Fig 1). Bronchoscopic examination showed diffuse infiammatory mucosa in the carina and in the right upper lobe bronchus. Cytologic examination ofthe bronchial brushing, right thoracocentesis, and pericardiocentesis disclosed atypical cells of adenocarcinoma type with papillary structures. Bronchial biopsy disclosed lymphangitic spread. After combined chemotherapy (cyclophosphamide, vinblastine, bleomycin, and cisplatin), the tonsillar mass diminished, but later the patient developed progressive dyspnea is spite of pericardia! and pleural drainage and died 25 days after admission. In the autopsy a peripheral mass in the right upper lobe and metastases to hilar nodes, right pleura, pericardium, and lingual tonsil were round. On light microscopy, the neoplasm was mainly composed ofan adenocarcinoma with papillary and solid areas with mucinous contents. Some areas resembled bronchioloalveolar carcinoma, but others showed atypical cells of large and giant cell type (Fig 2). Bronchial Adenocarcinoma (Monforte et al)
metastasis diagnosed befure the primary tumor. 1 Our case is very unusual in a number of aspects. It is the first case ofbronchogenic carcinoma presenting as a tonsillar metastasis. Furthermore, we do not find any other reported case of metastatic carcinoma in the lingual tonsil. Finally, it is interesting to note the atypical histologic features of the primary, with mucous, papillary, bronchioloalveolar, and large-giant cell differentiation. REFERENCES
FIGURE 2A (left). Bronchogenic carcinoma with distinct papillary pattern (original magnification x 80). B (center). Neoplastic cells in alveolar linings (original magnification x 80). C (right). Tumor giant cells with pleomorphic nuclei (original magnification x 130). DISCUSSION
The tonsil is an unusual site of metastatic disease. While review of the literature discloses only 89 cases of carcinoma metastatic to the palatine tonsil, we have not fuund any cases of metastases in the lingual tonsil. 1-ll Primary tonsillar carcinomas are squamous or undifferentiated, but not adenocarcinomas. 13 This fact supports the hypothesis that the lingual mass of our patient was metastatic and not primary. The most common primary tumors fuund are melanoma, hypemephroma, breast carcinoma, and bronchogenic carcinoma, while seminoma and carcinomas of the stomach and pancreas are less common. 1 Metastatic lesions in this part of the oral cavity are generally part of widespread systemic disease. Nevertheless, in six cases ofhypemephroma and in one case of pancreatic carcinoma, the tonsillar involvement was the first evidence of neoplastic disease. 1-lo To our knowledge, only 12 cases of bronchogenic carcinomas metastasizing to the tonsil have been reported. 1 All of them were of the oat cell type except one, which was adenocarcinoma. 12 The patients were men, and their ages ranged from 44 to 71 years. In none was the tonsillar
1 Brownson RJ, Jaques WE, La Monte SE, 7.ollinger WK. Hypernephroma metastatic to the palatine tonsils. Ann Oto! Rhino! Laryngol 1979; 88:235-40 2 Draizin D, Matucci IC, Rothfeld S. Bilateral metastatic tonsillar disease due to renal-cell carcinoma. Ear Nose Throat J 1978; 57:477-80 3 Ashur H, Mizrahi S, Ben-Hur N, Dolberg L. Metastatic malignant melanoma to the palatine tonsils: report of a case. J Oral Surg 1979; 37:110-12 4 Barton TIC, Kesterson GH, Wellman D, McCarty KS. Tonsillar metastasis from carcinoma of the breast with ultrastructural and steroid receptor analyses. Laryngoscope 1980; 90:477-85 5 Feleppa AE, Ellison NM. Metastatic pancreatic adenocarcinoma of the tonsil. Ear Nose Throat J 1981; 60:44-50 6 Craig RM, Glass BJ, Rhyne RR. Malignant melanoma: metastasis to the tonsil. J Am Dent Assoc 1982; 104:89.3-94 7 Passmore AL, Hugh TB, Coleman MJ. Tonsillar metastases from adenocarcinoma of the stomach. Aust NZ J Surg 1982; 52:371-72 8 Born JW, Baarsma EA. Metastatic hypemephroma in the tonsil. J Laryngol Otol 1982; 96:1159-63 9 Myer CM, Wood MD, Donegan JO. Metastatic melanoma to the palatine tonsil. Ear Nose Throat J 1983; 62:62-4 10 Maor E, Tovi F, Sacks M. Carcinoma of the pancreas presenting with bilateral tonsillar metastases. Ann Otol Rhino! Laryngol 1983; 92:192-95 11 Bychko V, Ghosh L, Lundine M, Manaligod JR, McQuire WP, Rose C. Ovarian androblastoma metastatic to tonsil. J Surg Oncol 1984; 27:275-79 12 Ardovin P, Bavdoin J. Jobard J. Sur un cas d'epithelioma glandulaire de l'amygdale palatine accompagnant un cancer du poumon. Rev Laryngol Oto! Rhino! (Bord) 1956; 77:878-91 13 Res ta L, Ricco R, Santangelo A. The histologic typing of tonsillar carcinoma. Tumori 1984; 70:409-16
CHEST I 92 I 8 I DECEMBER, 1987
1123