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An unusual oral presentation of a metastatic basal cell carcinoma
An unusual oral presentation of a metastatic basal cell carcinoma
K. C. Silvester, P. Speight Department of Oral and Maxillofaeial Surgery, University College Hospital Dental School, and Department of Oral Pathology, Institute of Dental Surgery, Eastman Dental Hospital, London, England
K. C. Silvester, P. Speight." An unusual oral presentation of a metastatic basal cell carcinoma. Int. J. Oral Maxillofae. Surg. 1991; 20: 106-107. Abstract. A case report of a metastasising basal cell carcinoma is presented. The primary lesion was situated on the right upper lip with metastasis to soft tissue on the buccal aspect of the right molar region of the mandible. The literature regarding this unusual occurrence is reviewed.
Basal cell carcinoma is the c o m m o n e s t malignant tumour of skin. A l t h o u g h it is locally invasive, metastasis is unusual in the c o m m o n course of the condition. When it occurs it may be via lymphatic, blood or embolic spread through interstitial tissues. It creates difficulties in management and the prognosis is often grave.
Case report
A 50-year-old man presented in January 1989, regarding a painless intraoral swelling over his right mandible. It had slowly increased in size over a 6-month period. In June 1984, a basal cell carcinoma was removed by curettage from his right upper lip. It recurred 1 year later and was again curetted. In January 1986 a further recurrence was noted which was treated with radical, superficial ratiotherapy. He was given a course of 4200 cGy over a 1-week period to the right upper lip only. In May 1986 the area was noted to be well healed. On examination in 1989 a firm swelling was palpable on the buccal aspect of the edentulous right, lower molar region, below the level of the vestibular reflection. The mucosa and skin appeared normal. Radiographs were normal. An examination under anaesthetic and open biopsy of the lesion were carried out. It was uncertain from the histology whether the lesion was a low grade adenocarcinoma of salivary gland origin, or a recurrent basal cell carcinoma. He was readmitted and an excision of his mandible from the right angle to the right premolar region carried out, together with a right radical neck dissection. The defect was reconstructed with a free iliac crest bone graft and a primary intra-oral closure.
to that seen in the initial oral biopsy. The epithelial elements comprised strands and islands o f epithelial cells (Fig. 1) many o f which had a palisaded peripheral layer surrounding central areas of spindle or squamous cells. The t u m o u r was invading and destroying adjacent submandibular gland, muscle and fibro fatty connective tissue, but the soft tissue excision margins appeared tumour-free. L y m p h nodes showed reactive changes, but there was no evidence of tumour. The features were consistent with a metastatic basal cell carcinoma which appeared to have been completely excised. This view was substantiated by review of the original biopsies taken in 1984 and 1985. Both were curettage specimens obtained with cautery and showed multiple fragments of skin with severe disruption of architecture. The lesions were composed of islands
Key words: basal cell carcinoma; metastasis Accepted for publication 8 November 1990
of tumour cells in a fibrous stroma with extensive ulceration of the epidermis. The islands were irregular in shape and size with only occasional evidence of a more typical rounded morphology and peripheral cell palisading. There was a moderate degree of nuclear pleomorphism but mitoses were not prominent (Fig. 2). Occasionally, and particularly in the second biopsy, there were small cords of spindle-shaped cells arranged in an infiltrative pattern and often continuous with the overlying epidermis or with larger t u m o u r islands. Neither lesion had been completely excised. Discussion
In a study of 9,050 cases of basal cell carcinoma over a 14-year period COT R A N 3 found only 9 cases of metastasis - an incidence of 0.1%. F r o m 1894 to
Histopathology
The fibro-fatty connective tissue adjacent to the submandibular gland contained an epithelial tumour mass similar
Fig. 1. Photomicrograph of the oral lesion showing cords and sheets of epithelial cells with evidence of peripheral palisading and some squamous differentiation. (H&E stain, magnification x 63).
Metastatic basal cell carcinoma
107
autotransplants of B C C survived only in the presence of associated connective tissue stroma. CRANMERet al. 4 suggested that the metastatic BCC cell may be independent of stromal support and that therein lies its propensity to metastasise. H o s t immunity may also play an as yet undefined role 1.
Acknowledgement. We thank Professor M. Harris for permission to report this case.
References 1. BRYARLY RC JR, VEACH SR, KORNBLUT
Fig. 2. Photomicrograph of the first skin biopsy taken in 1984. The appearances are similar to the metastatic lesion. There is occasional peripheral palisading and slight nuclear pleomorphism. (H&E stain, magnification x 100).
1977, 119 cases of metastatic BCC were reported in the world literature 9. In 72% of the more recent cases, the primary t u m o u r was in the head and neck region. In a typical case the primary lesion had a number of recurrences before metastatis took place 8. It has been suggested 7 that involvement of multiple embryonic planes, close proximity of the neoplasm to bone and cartilage, and inadequate excision may be responsible for the high incidence of recurrence of the mid-face region. The latter was certainly true of this case and doubt must be cast on the use of curretage as a mode of treatment, if not for primary lesions then certainly for recurrences. In a survey of 36 cases of metastasis between 1962 and 19729 , 50% involved bone, 39% lung, 31% regional lymph nodes, 11% liver, and 11% showed subcutaneous metastases. The case presented is unusual in that the metastasis presented as an intraoral, submucosal swelling. In a review of the literature only one similar case could be found 9. This was a 47-year-old man who had a similar series of 3 excisions of BCC from the upper lip region, followed by radiotherapy. The patient presented 5 years later with a mass in the left submandibular region, which was excised together with a portion of the mandible and a radical neck dissection. There was no evidence of disease in regional lymph nodes. He was alive and well 7 years post surgically. The p o o r prognosis of metastatic basal carcinoma
at other sites, (in one study of 17 patients 5 the mean survival time after metastatis was 1.6 years), and the lack of similar cases p r o m p t e d the decision to carry out a simultaneous radical neck dissection despite the absence of palpable nodes. The m o d e of metastasis of the lesion in both these cases is of interest, Scanlon 9 felt that in his case it was due to embolic spread of t u m o u r within interstitial tissue. Although WILHS 11 stated specifically that this p h e n o m e n o n does not occur, it is difficult to explain it in any other way, as in both cases there was no evidence o f nodal involvement by t u m o u r or any distant blood-borne metastasis. The possibility of X-ray induced tumorgenesis is unlikely. The radiotherapy treatment was well localised to the upper lip, with shielding of the surrounding areas, although it has been noted that a shorter duration for metastasis seemed to occur when primary lesions had been initially managed by radiotherapy 2. The reason why some basal carcin o m a become aggressive and metastasise is unclear. The histological features of the original 2 skin biopsies show features which have been shown to be associated with a more aggressive behaviour 6. These include irregular tumour islands and an infiltrative growth pattern with cords o f single cells. There is evidence o f a stromal dependence in BCC. VAN SCOTT ~¢ REINERTON 1° noted that
AD. Metastasising auricular basal cell carcinoma. Otolaryngol Head Neck Surg 1980: 88: 40-3. 2. CONWAY H, HUGO NE. Metastatic basal ceil carcinoma. Am J Surg 1965: 110: 620M. 3. COTRANR. Metastasising basal cell carcinoma. Cancer 1961: 14: 1036-40. 4. CRANMER L, REINGOLD I, WILSON J. Basal cell carcinoma of skin metastatic to bone. Arch Dermatol 1970: 102: 3379. 5. FARMER ER, ELSON B, HELWIG MD. Metastatic basal cell carcinoma. Cancer 1980: 46: 748-57. 6. JACOBSG, RIPVEV J, ALTINI M. Prediction of aggressive behaviour in basal cell carcinoma. Cancer 1982: 49: 533-7. 7. LEW~ HL, BAILINPL. Basal cell carcinoma of the head and neck: identification of the high risk patient. Laryngoscope 1980: 90: 955. 8. Liu R McGREGORD, CIHAKR, II Y, ISHIMARU T. Basal cell carcinoma with pulmonary metastases. Hiroshima J Med Sci 1971: 20:281 89. 9. SCANLON E, VOLKMER D, OVIEDO M, KHANDEKAR J, VICTOR T. Metastatic basal cell carcinoma. Surg Oncol 1980: 15: 171-80. 10. VAN SCOTT E, REINERTSONR. The modulating influence of stroma: Environment of epithelial cells studied in human autotransplants. Invest Dermatol 1961: 36: 109-31. 11. WILLIS R. The spread of tumours in the human body. 3rd ed. London: Butterworth, 1973.
Address: Mr K. C. Silvester Department of Oral and Maxillofacial Surgery The Royal London Hospital Whitechapel London E1 1BB England
K. C. Silvester, P. Speight Department of Oral and Maxillofaeial Surgery, University College Hospital Dental School, and Department of Oral Pathology, Institute of Dental Surgery, Eastman Dental Hospital, London, England
K. C. Silvester, P. Speight." An unusual oral presentation of a metastatic basal cell carcinoma. Int. J. Oral Maxillofae. Surg. 1991; 20: 106-107. Abstract. A case report of a metastasising basal cell carcinoma is presented. The primary lesion was situated on the right upper lip with metastasis to soft tissue on the buccal aspect of the right molar region of the mandible. The literature regarding this unusual occurrence is reviewed.
Basal cell carcinoma is the c o m m o n e s t malignant tumour of skin. A l t h o u g h it is locally invasive, metastasis is unusual in the c o m m o n course of the condition. When it occurs it may be via lymphatic, blood or embolic spread through interstitial tissues. It creates difficulties in management and the prognosis is often grave.
Case report
A 50-year-old man presented in January 1989, regarding a painless intraoral swelling over his right mandible. It had slowly increased in size over a 6-month period. In June 1984, a basal cell carcinoma was removed by curettage from his right upper lip. It recurred 1 year later and was again curetted. In January 1986 a further recurrence was noted which was treated with radical, superficial ratiotherapy. He was given a course of 4200 cGy over a 1-week period to the right upper lip only. In May 1986 the area was noted to be well healed. On examination in 1989 a firm swelling was palpable on the buccal aspect of the edentulous right, lower molar region, below the level of the vestibular reflection. The mucosa and skin appeared normal. Radiographs were normal. An examination under anaesthetic and open biopsy of the lesion were carried out. It was uncertain from the histology whether the lesion was a low grade adenocarcinoma of salivary gland origin, or a recurrent basal cell carcinoma. He was readmitted and an excision of his mandible from the right angle to the right premolar region carried out, together with a right radical neck dissection. The defect was reconstructed with a free iliac crest bone graft and a primary intra-oral closure.
to that seen in the initial oral biopsy. The epithelial elements comprised strands and islands o f epithelial cells (Fig. 1) many o f which had a palisaded peripheral layer surrounding central areas of spindle or squamous cells. The t u m o u r was invading and destroying adjacent submandibular gland, muscle and fibro fatty connective tissue, but the soft tissue excision margins appeared tumour-free. L y m p h nodes showed reactive changes, but there was no evidence of tumour. The features were consistent with a metastatic basal cell carcinoma which appeared to have been completely excised. This view was substantiated by review of the original biopsies taken in 1984 and 1985. Both were curettage specimens obtained with cautery and showed multiple fragments of skin with severe disruption of architecture. The lesions were composed of islands
Key words: basal cell carcinoma; metastasis Accepted for publication 8 November 1990
of tumour cells in a fibrous stroma with extensive ulceration of the epidermis. The islands were irregular in shape and size with only occasional evidence of a more typical rounded morphology and peripheral cell palisading. There was a moderate degree of nuclear pleomorphism but mitoses were not prominent (Fig. 2). Occasionally, and particularly in the second biopsy, there were small cords of spindle-shaped cells arranged in an infiltrative pattern and often continuous with the overlying epidermis or with larger t u m o u r islands. Neither lesion had been completely excised. Discussion
In a study of 9,050 cases of basal cell carcinoma over a 14-year period COT R A N 3 found only 9 cases of metastasis - an incidence of 0.1%. F r o m 1894 to
Histopathology
The fibro-fatty connective tissue adjacent to the submandibular gland contained an epithelial tumour mass similar
Fig. 1. Photomicrograph of the oral lesion showing cords and sheets of epithelial cells with evidence of peripheral palisading and some squamous differentiation. (H&E stain, magnification x 63).
Metastatic basal cell carcinoma
107
autotransplants of B C C survived only in the presence of associated connective tissue stroma. CRANMERet al. 4 suggested that the metastatic BCC cell may be independent of stromal support and that therein lies its propensity to metastasise. H o s t immunity may also play an as yet undefined role 1.
Acknowledgement. We thank Professor M. Harris for permission to report this case.
References 1. BRYARLY RC JR, VEACH SR, KORNBLUT
Fig. 2. Photomicrograph of the first skin biopsy taken in 1984. The appearances are similar to the metastatic lesion. There is occasional peripheral palisading and slight nuclear pleomorphism. (H&E stain, magnification x 100).
1977, 119 cases of metastatic BCC were reported in the world literature 9. In 72% of the more recent cases, the primary t u m o u r was in the head and neck region. In a typical case the primary lesion had a number of recurrences before metastatis took place 8. It has been suggested 7 that involvement of multiple embryonic planes, close proximity of the neoplasm to bone and cartilage, and inadequate excision may be responsible for the high incidence of recurrence of the mid-face region. The latter was certainly true of this case and doubt must be cast on the use of curretage as a mode of treatment, if not for primary lesions then certainly for recurrences. In a survey of 36 cases of metastasis between 1962 and 19729 , 50% involved bone, 39% lung, 31% regional lymph nodes, 11% liver, and 11% showed subcutaneous metastases. The case presented is unusual in that the metastasis presented as an intraoral, submucosal swelling. In a review of the literature only one similar case could be found 9. This was a 47-year-old man who had a similar series of 3 excisions of BCC from the upper lip region, followed by radiotherapy. The patient presented 5 years later with a mass in the left submandibular region, which was excised together with a portion of the mandible and a radical neck dissection. There was no evidence of disease in regional lymph nodes. He was alive and well 7 years post surgically. The p o o r prognosis of metastatic basal carcinoma
at other sites, (in one study of 17 patients 5 the mean survival time after metastatis was 1.6 years), and the lack of similar cases p r o m p t e d the decision to carry out a simultaneous radical neck dissection despite the absence of palpable nodes. The m o d e of metastasis of the lesion in both these cases is of interest, Scanlon 9 felt that in his case it was due to embolic spread of t u m o u r within interstitial tissue. Although WILHS 11 stated specifically that this p h e n o m e n o n does not occur, it is difficult to explain it in any other way, as in both cases there was no evidence o f nodal involvement by t u m o u r or any distant blood-borne metastasis. The possibility of X-ray induced tumorgenesis is unlikely. The radiotherapy treatment was well localised to the upper lip, with shielding of the surrounding areas, although it has been noted that a shorter duration for metastasis seemed to occur when primary lesions had been initially managed by radiotherapy 2. The reason why some basal carcin o m a become aggressive and metastasise is unclear. The histological features of the original 2 skin biopsies show features which have been shown to be associated with a more aggressive behaviour 6. These include irregular tumour islands and an infiltrative growth pattern with cords o f single cells. There is evidence o f a stromal dependence in BCC. VAN SCOTT ~¢ REINERTON 1° noted that
AD. Metastasising auricular basal cell carcinoma. Otolaryngol Head Neck Surg 1980: 88: 40-3. 2. CONWAY H, HUGO NE. Metastatic basal ceil carcinoma. Am J Surg 1965: 110: 620M. 3. COTRANR. Metastasising basal cell carcinoma. Cancer 1961: 14: 1036-40. 4. CRANMER L, REINGOLD I, WILSON J. Basal cell carcinoma of skin metastatic to bone. Arch Dermatol 1970: 102: 3379. 5. FARMER ER, ELSON B, HELWIG MD. Metastatic basal cell carcinoma. Cancer 1980: 46: 748-57. 6. JACOBSG, RIPVEV J, ALTINI M. Prediction of aggressive behaviour in basal cell carcinoma. Cancer 1982: 49: 533-7. 7. LEW~ HL, BAILINPL. Basal cell carcinoma of the head and neck: identification of the high risk patient. Laryngoscope 1980: 90: 955. 8. Liu R McGREGORD, CIHAKR, II Y, ISHIMARU T. Basal cell carcinoma with pulmonary metastases. Hiroshima J Med Sci 1971: 20:281 89. 9. SCANLON E, VOLKMER D, OVIEDO M, KHANDEKAR J, VICTOR T. Metastatic basal cell carcinoma. Surg Oncol 1980: 15: 171-80. 10. VAN SCOTT E, REINERTSONR. The modulating influence of stroma: Environment of epithelial cells studied in human autotransplants. Invest Dermatol 1961: 36: 109-31. 11. WILLIS R. The spread of tumours in the human body. 3rd ed. London: Butterworth, 1973.
Address: Mr K. C. Silvester Department of Oral and Maxillofacial Surgery The Royal London Hospital Whitechapel London E1 1BB England