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Hodgkin's disease of the maxillary gingiva
Hodgkin's disease of the maxillary gingiva
Junichi T a n a k a 1, Kiyoshi Yoshida 1, Mitsugu S u z u k i 1, Yuh S a k a t a 2 1Department of Dentistry and Oral Surgery and 2First Department of Internal Medicine, Hirosaki University School of Medicine, Japan
A case report J. Tanaka, K. Yoshida, M. Suzuki, Y. Sakata: Hodgkin ~ disease o f the maxillary gingiva. A case report. Int. J. Oral Maxillofac. Surg. 1992; 21: 45-46. Abstract. A case of extranodal Hodgkin's disease limited to the maxillary gingiva is presented. After appropriate staging procedures, treatment consisted of chemotherapy. The patient is alive and free of disease 2.10 years after initial p r e s e n -
Accepted for publication 5 September 1991
tation.
Malignant lymphomas are often p r e s e n t in the head and neck region but intraoral presentation is rare. A case of extranodal H o d g k i n ' s disease including the maxillary gingiva is presented. STEINER6 reviewed in 1943 more than 2,000 cases of H o d g k i n ' s disease and reported only one maxillary case. Since that time STEG et al. 7, PETERS et al. 4, BATHARDSMITH et al. 1, RONCHI et al. 5, and LELLO & RAUBENHEIMER2 reported on extranodal manifestation of Hodgkin's disease in the maxilla. O f these cases, only those described by PETERS et al. 4, BATHARD-SMITH et al. i, and LELLO & RAUBENHEIMER 2 c a n be considered as primarily evolving from the gingiva. Case report A 55-year-old Japanese man had noticed a mass in the right maxillary canine region. The swelling was symptomless but became painful after approximately 2 months. Enlarged lymphnodes were found on palpation in both submandibular areas. Further examination revealed no other enlarged lymphnodes and neither were spleen and liver enlarged. The chest X-ray showed no abnormalities. On
Fig. 1. Intraoral photograph showing soft tissue lesion over right maxillary canine region.
Key words: Hodgkin's disease; extranodal presentation; gingiva.
oral examination a 40 x 35 mm lesion was visible with elevated margins at the right maxillary canine area (Fig. 1). Radiographs revealed no bone involvement. A biopsy of the tumor was performed and the specimen sent to histology. Histopathological examination revealed diffuse proliferation of small lymphoid cells. Interspersed among these cells were large, multinucleated atypical lymphoid cells, consistent with Reed-Sternberg cells (Fig. 2). Immunohistological cell-marker analysis showed the stain for leukocyte common antigen (LCA) to be invariably positive. MT-1 reaction for T cell was not positive in the majority of lymphocytes, and MB-1 reaction for B cell was negative. The lesion was histologically diagnosed as Hodgkin's disease. The patient was admitted to hospital for clinical staging of the disease, CT scans of the neck, chest and abdomen. The CT scan showed enlarged, contrast-enhanced nodes in both submandibular regions. The results of bone marrow aspiration were within normal limits. The disease was rated as stage I I E according to the Ann Arbor Classification. The patient received combination chemotherapy, i.e. cyclophosphamide, vincristine, procarbazine, and prednisolone (COPP) 3 and adriamycin, to which he responded well. The
Fig. 2. Biopsy specimen of the maxillary lesion showed the lympho-histocytic form with rare Reed-Sternberg cells. (H&E, original magnification x 400).
gingival tumor and the bilateral submandibular lymphnode swelling disappeared. Subsequently, an additional course of COP (cyclophosphamide, vincristine, prednisolone) was given the following year. The patient is still alive and has been well for 2 years and 10 months after the initial diagnosis was made (Fig. 3). Discussion Hodgkin's disease primarily involving the maxilla is rare. WOOD & COLTMAN9, in a review of the world literature, estimated the incidence to be 0.25%. The case presented is the 7th patient described with primary Hodgkin's d i s e a s e involving the maxilla. Clinically, the maxillary lesions reported looked like tumours and were often painful. The radiological features were not specific and the diagnosis was always made histologically 5. Diagnosis of Hodgkin's disease should be based on proper immunohistochemical examination by ruling out other lymphoid tumours. Several immunohistochemical examination methods have been developed for the characterization of T cell, B cell, and
Fig. 3. Intra-oral appearance after 2 years post-chemotherapy (COPP).
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Tanaka et al.
histiocytic l y m p h o m a s which c a n be used for p a r a f f i n e m b e d d e d sections 8. C o m b i n a t i o n t h e r a p y in H o d g k i n ' s disease has p r o v e n to be of greater efficacy t h a n single agent t h e r a p y 3. Different c o m b i n a t i o n s with 3 or 4 drugs with alkylating agents, vinca alkaloids, procarbazine, a n d steroids h a v e b e e n employed. A f a v o u r a b l e t h e r a p e u t i c result was obtained, with complete remission after 4 courses o f C O P P c h e m o t h e r a p y . References 1. BATHARD-SMITHPJ,, COONARHS, MARKUS A E Hodgkin's disease presenting intra-orally. Br J Oral Surg 1978: 16:64 9. 2. LELLO GE, RAUBENHEIMERE. Hodgkin's disease presenting in the maxilla. Int J Oral Maxillofac Surg 1989: 18:7 9.
3. MORGENFELD MC, PAVLOVSKYA, SUAREZ A, et al. Combined cyclophosphamide, vincristine, procarbazine, and prednisone (COPP) therapy of malignant lymphoma. Evaluation of 190 patients. Cancer 1975: 36:1241 9. 4. PETERS RA, BELTAOSE, GREENLAWRH, et al. Intraoral extranodal Hodgkin's disease. J Oral Surg 1977: 35: 311-2. 5. RONCHI P, EPIFANI C, LUNETTA PH. Hodgkin's disease of the jaw. Report of a case. J Oral Maxillofac Surg 1988: 46: 155-8. 6. STEINERPE. Hodgkin's disease. The incidence, distribution, nature and possible significance of the lymphogranulomatous lesions in the bone marrow. A review with original data. Arch Pathol 1943: 36: 627 37. 7. STEG RF, DAHLIN DC, GORES RJ. Malignant lymphoma of the mandible and
maxillary region. Oral Surg 1959: 12: 128-41. 8. TAKAHASHIH, TSUDA N, TEZUKAF, et al. Non-Hodgkin's lymphoma of the major salivary gland: a morphologic and immunohistochemical study of 15 cases. J Oral Pathol Med 1990: 19:306 12. 9. WOOD NL, COLTMAN CA. Localized primary extranodal Hodgkin's disease. Ann Intern Med 1973: 78:113 8.
Address: Dr. Junichi Tanaka Department of Dentistry and Oral Surgery Hirosaki University School o f Medicine 5 Zaifu-cho, Hirosaki-shi Aomori 036 Japan
Junichi T a n a k a 1, Kiyoshi Yoshida 1, Mitsugu S u z u k i 1, Yuh S a k a t a 2 1Department of Dentistry and Oral Surgery and 2First Department of Internal Medicine, Hirosaki University School of Medicine, Japan
A case report J. Tanaka, K. Yoshida, M. Suzuki, Y. Sakata: Hodgkin ~ disease o f the maxillary gingiva. A case report. Int. J. Oral Maxillofac. Surg. 1992; 21: 45-46. Abstract. A case of extranodal Hodgkin's disease limited to the maxillary gingiva is presented. After appropriate staging procedures, treatment consisted of chemotherapy. The patient is alive and free of disease 2.10 years after initial p r e s e n -
Accepted for publication 5 September 1991
tation.
Malignant lymphomas are often p r e s e n t in the head and neck region but intraoral presentation is rare. A case of extranodal H o d g k i n ' s disease including the maxillary gingiva is presented. STEINER6 reviewed in 1943 more than 2,000 cases of H o d g k i n ' s disease and reported only one maxillary case. Since that time STEG et al. 7, PETERS et al. 4, BATHARDSMITH et al. 1, RONCHI et al. 5, and LELLO & RAUBENHEIMER2 reported on extranodal manifestation of Hodgkin's disease in the maxilla. O f these cases, only those described by PETERS et al. 4, BATHARD-SMITH et al. i, and LELLO & RAUBENHEIMER 2 c a n be considered as primarily evolving from the gingiva. Case report A 55-year-old Japanese man had noticed a mass in the right maxillary canine region. The swelling was symptomless but became painful after approximately 2 months. Enlarged lymphnodes were found on palpation in both submandibular areas. Further examination revealed no other enlarged lymphnodes and neither were spleen and liver enlarged. The chest X-ray showed no abnormalities. On
Fig. 1. Intraoral photograph showing soft tissue lesion over right maxillary canine region.
Key words: Hodgkin's disease; extranodal presentation; gingiva.
oral examination a 40 x 35 mm lesion was visible with elevated margins at the right maxillary canine area (Fig. 1). Radiographs revealed no bone involvement. A biopsy of the tumor was performed and the specimen sent to histology. Histopathological examination revealed diffuse proliferation of small lymphoid cells. Interspersed among these cells were large, multinucleated atypical lymphoid cells, consistent with Reed-Sternberg cells (Fig. 2). Immunohistological cell-marker analysis showed the stain for leukocyte common antigen (LCA) to be invariably positive. MT-1 reaction for T cell was not positive in the majority of lymphocytes, and MB-1 reaction for B cell was negative. The lesion was histologically diagnosed as Hodgkin's disease. The patient was admitted to hospital for clinical staging of the disease, CT scans of the neck, chest and abdomen. The CT scan showed enlarged, contrast-enhanced nodes in both submandibular regions. The results of bone marrow aspiration were within normal limits. The disease was rated as stage I I E according to the Ann Arbor Classification. The patient received combination chemotherapy, i.e. cyclophosphamide, vincristine, procarbazine, and prednisolone (COPP) 3 and adriamycin, to which he responded well. The
Fig. 2. Biopsy specimen of the maxillary lesion showed the lympho-histocytic form with rare Reed-Sternberg cells. (H&E, original magnification x 400).
gingival tumor and the bilateral submandibular lymphnode swelling disappeared. Subsequently, an additional course of COP (cyclophosphamide, vincristine, prednisolone) was given the following year. The patient is still alive and has been well for 2 years and 10 months after the initial diagnosis was made (Fig. 3). Discussion Hodgkin's disease primarily involving the maxilla is rare. WOOD & COLTMAN9, in a review of the world literature, estimated the incidence to be 0.25%. The case presented is the 7th patient described with primary Hodgkin's d i s e a s e involving the maxilla. Clinically, the maxillary lesions reported looked like tumours and were often painful. The radiological features were not specific and the diagnosis was always made histologically 5. Diagnosis of Hodgkin's disease should be based on proper immunohistochemical examination by ruling out other lymphoid tumours. Several immunohistochemical examination methods have been developed for the characterization of T cell, B cell, and
Fig. 3. Intra-oral appearance after 2 years post-chemotherapy (COPP).
46
Tanaka et al.
histiocytic l y m p h o m a s which c a n be used for p a r a f f i n e m b e d d e d sections 8. C o m b i n a t i o n t h e r a p y in H o d g k i n ' s disease has p r o v e n to be of greater efficacy t h a n single agent t h e r a p y 3. Different c o m b i n a t i o n s with 3 or 4 drugs with alkylating agents, vinca alkaloids, procarbazine, a n d steroids h a v e b e e n employed. A f a v o u r a b l e t h e r a p e u t i c result was obtained, with complete remission after 4 courses o f C O P P c h e m o t h e r a p y . References 1. BATHARD-SMITHPJ,, COONARHS, MARKUS A E Hodgkin's disease presenting intra-orally. Br J Oral Surg 1978: 16:64 9. 2. LELLO GE, RAUBENHEIMERE. Hodgkin's disease presenting in the maxilla. Int J Oral Maxillofac Surg 1989: 18:7 9.
3. MORGENFELD MC, PAVLOVSKYA, SUAREZ A, et al. Combined cyclophosphamide, vincristine, procarbazine, and prednisone (COPP) therapy of malignant lymphoma. Evaluation of 190 patients. Cancer 1975: 36:1241 9. 4. PETERS RA, BELTAOSE, GREENLAWRH, et al. Intraoral extranodal Hodgkin's disease. J Oral Surg 1977: 35: 311-2. 5. RONCHI P, EPIFANI C, LUNETTA PH. Hodgkin's disease of the jaw. Report of a case. J Oral Maxillofac Surg 1988: 46: 155-8. 6. STEINERPE. Hodgkin's disease. The incidence, distribution, nature and possible significance of the lymphogranulomatous lesions in the bone marrow. A review with original data. Arch Pathol 1943: 36: 627 37. 7. STEG RF, DAHLIN DC, GORES RJ. Malignant lymphoma of the mandible and
maxillary region. Oral Surg 1959: 12: 128-41. 8. TAKAHASHIH, TSUDA N, TEZUKAF, et al. Non-Hodgkin's lymphoma of the major salivary gland: a morphologic and immunohistochemical study of 15 cases. J Oral Pathol Med 1990: 19:306 12. 9. WOOD NL, COLTMAN CA. Localized primary extranodal Hodgkin's disease. Ann Intern Med 1973: 78:113 8.
Address: Dr. Junichi Tanaka Department of Dentistry and Oral Surgery Hirosaki University School o f Medicine 5 Zaifu-cho, Hirosaki-shi Aomori 036 Japan