184
Brief reports
manner analogous to the phenomenon seen in salivary tumours4. Mixed tumours in which both epithelial and myoepithelial components are malignant have been described in dogs but these components also arise within a pre-existing benign mixed tumour6. Our preference, therefore, is to classify this tumour descriptively as a breast carcinoma with ductal, myoepithelial, squamous and sebaceous elements while accepting there are some similarities to salivary gland tumours. This unusual phenotypic constellation, particularly the sebaceous component, makes this a very rare, if not unique, breast neoplasm.
Acknowledgements We are most grateful to Dr M.Harris for his advice in the preparation of this manuscript and to Mr D.E.Edmond-
son and Miss S.Clews for assistance in photography and electronmicroscopy respectively.
References 1. Raju GC, Wee A. Spindle cell carcinomaof the breast. HistoputhologH 1990: 16; 497-499. 2. Wargotz ES,Deos PH. N o d s HJ. Metaplastic carcinomas of the breast. II. Spindle cellcarcinoma.Hum. Pathol. 1989: 20;732-740. 3. Rosen PP, Emsberger D. Low-grade adenosquamous carcinoma: a variant of metaplastic mammary carcinoma. Am. J. Surg. Pathol. 1987: 11; 351-358. 4. Thackray AC, Lucas RB.Tumours of major salivary glands. In Atlas of Tumour Puthologtl, Second series, Fascicle 10. Washington, D.C.: Armed Forces Instituteof Pathology, 1 9 7 4 20-30,107-117. 5. Sheth MT. Hathway D. PetrelIi M. Pleomorphic adenoma (‘mixed’ tumour) of human female breast mimicking carcinoma clinicoradiologically.Cancer 1978: 41; 659-665. 6. Moulton JE. Mammary gland. In Tumours in Domestic Animals. 2nd edn. Berkeley: University of California Press, 1977; 365-366.
Histopathology 1992,21,184-187
BRIEF REPORT
Derrnatofibrosarcoma protuberans recurring as a giant cell fibroblastoma J.COYNE, S.M.KAFTAN & R.D.P.CRAIG* Departments of Histopathology and *Plastic Surgery, Withington Hospital, Manchester, UK Date of submission 13 February 1992 Accepted for publication 12 March 1992
Keywords: giant cell fibroblastoma, dermatofibrosarcoma protuberans
Introduction Giant cell fibroblastoma, a rare mesenchymal tumour, is thought to have a close histogenetic relationship with dermatofibrosarcomaprotuberans. It has been proposed that giant cell fibroblastoma represents the juvenile form of dermatofibrosarcoma protuberans’, a concept strengthened by a recent single report2. Two cases of dermatofibrosarcoma protuberans with giant cell fibroblastoma-like areas have also been described and illustrated in the recent literature3. We present a case which
documents the recurrence of a lesion with the features of giant cell fibroblastoma 3 years after the local excision of a dermatofibrosarcomaprotuberans and, while supporting their close relationship, suggests that their different morphologicalmanifestationsare not simply age related.
Case report A 17-year-old male was noted to have a mass present in the left groin for several months. This was excised at another hospital. At age 20, he underwent re-excision of a recurrent tumour from the same region. PATHOLOGICAL FINDINGS
Address for correspondence: Dr J.Coyne. Histopathology Department. Withington Hospital, Nell Lane, Mancheater M20 8LR. UK.
The flrst specimen was described as a skin ellipse 4 x 1.5 cm with a 0.7 cm nodule located within the dermis.
Brief reports
18 5
Figure 1. a Cellular dermatofibrosarcoma protuberaus infiltrating subcutaneous fat and showing b a stodform pattern.
Microscopically,the overall histological appearance was that of a densely cellular, spindle cell tumour displaying a fascicular and storiform pattern. Approximately three mitotic figures per 10 h.p.f. were present: all of the mitoses were of normal appearance. The tumour surrounded residual skin appendage structures and infiltrated subcutaneous fat. Superficially, there was a small protuberant nodule in which spindle shaped cells with cellular processes were arranged haphazardly in a myxoid stroma. The appearances were interpreted on review as being those of a dermatofibrosarcoma protuberans (Figure 1). The recurrent lesion consisted of a skin ellipse 9 x 4 x 1.5 cm with an underlying lobulated, pink fleshy tumour measuring 7 x 3 cm. This was h l y attached to the overlying skin. Microscopically,the tumour showed a heterogeneous appearance with features typical of giant cell fibroblastoma (Figure 2a). Paucicellular, collagenized areas alternated with cellular, spindle cell areas. Areas containing diffusely arranged spindle cells dispersed within a myxoid stroma were also present. Many
spindle cells with large, hyperchromatic nuclei were present together with numerous multinucleated giant cells (Figure 2b). Frequent sinusoid-like spaces, partially lined by giant cells, were also present dispersed throughout the tumour (Figure 2a). Adnexal structures were frequently surrounded by the proliferation which i d trated the subcutaneous fat. An occasional normal mitotic figure was noted and a very patchy lymphoplasmacytic infiltrate was present at the tumour periphery. Occasional areas, composed of spindle cells in a myxoid stroma, were focally present as were small, mainly superficial areas of compactly arranged, tightly packed spindle cells in a fascicular pattern. In places the tumour extended up to the overlying epidermis while in others a narrow zone of normal papillary dermis was present.
Discussion Although distinctive histological entities, giant cell fibroblastoma and dermatofibrosarcomaprotuberans do
186
Brief reports
Figure 2. a Sinusoidal spaces, variable stromal cellularlty and giant cells. b Splndle stromal cells and scattered @antcells (shown at a higher power in the inset).
share some similar morphological characteristics. These include spindle cells, myxoid stroma, location within the dermis, entrapment of adnexal structures and infiltration of the subcutaneous fat. Indeed a myxoid stroma, which is a focal characteristic of giant cell fibroblastoma, can occasionally be the major feature of dermatofibrosarcoma protuberans*. Based on these similar histological features and on the mean age of occurrence of these entities, it was proposed that giant cell fibroblastoma represents the juvenile form of dermatofibrosarcoma protuberans'. A recent report describing a giant cell fibroblastoma recurring as a dermatofibrosarcoma protuberans would appear to lend support to this interpretation2. Beham & Fletcher3,while further demonstrating the close relationship between both tumours, considered this theory an oversimplification as they had encountered dermatofibrosarcoma protuberans occurring in children and giant cell fibroblastomas arising in adults. The present case tends to support this latter contention
and argues against the concept that giant cell fibroblastoma is simply a juvenile variant of dermatofibrosarcoma protuberans. While both may be different morphological manifestations of a neoplastic fibroblast proliferation, factors other than age must also be involved and these two entities may represent different degrees of tumour differentiation with different biological implications.
Acknowledgements The authors wish to acknowledge Mrs L.Garstang for excellent secretarial assistance and Dr A.Curry for the photomicrographs.
References 1. ShmooklerBM. Enzinger FM,Weiss SW. Glant cell fibroblastoma.A juvenile form of dermatofibrosarcomaprotuberans. Cancer 1989: 64; 2154-2161.
Brie! reports
2. AlguacilGarciaA. Giant cell fibroblastoma recurring as dermatofibrosarcoma protuberans (Case Report). Am. I. Surg. Pathol. 1991; 15(8); 798-801. 3. Beham A, Fletcher CDM. Dermatolibrosarcoma protuberans with
18 7
areas resembling giant cell fibroblastoma. Report of two cases. HistopathdwH 1 9 9 0 17; 165-167. 4. Frierson HF. Cooper PH. Myxoid variant of dermatoEbrosarcoma protuberans. Am. 1. Surg. Pathol. 1983: 7; 445-450.
Histopathol~g~ 1992,21, 187-189
BRIEF REPORT
Focal lymphoid hyperplasia of the oesophagus: report of a case E.GERVAZ, F.POTET, R.MAHE & G.LEMASSON Service d'tinatomie et de Cytologie Pathologiques, H6pital Bichat, Paris, France Date of submission 7 November 1991 Accepted for publication 16 March 1992
Keywords: oesophagus, lymphoreticular disorder, lymphoid hyperplasia
Introduction Focal lymphoid hyperplasia of the gastrointestinal tract is an uncommon benign lesion, defined as a follicular reaction with well-defined germinal centres, in response to a B-cell hyperplasia. Follicular lymphoid hyperplasia is frequent in the stomach (associated with peptic lesions) and in the rectum. Involvement of the oesophagus remains exceptional'. This report describes a case of gastrointestinal focal lymphoid hyperplasia located in the oesophagus and exhibiting very unusual lymphoreticular aspects.
Case report
An alcoholic, heavy smoker, HIV negative, 59-year-old man, had a 3-month history of increasing dysphagia. regurgitation and abdominal pain. Endoscopy showed an exophytic and ulcerated lesion located in the middle third of the anterior oesophageal wall associated with benign peptic stenosis and a hiatus hernia. Biopsy study of the exophytic lesion displayed a lymphoid infiltrate associated with clusters of undifTerentiated large cells. Immunohistochemistry was not Address for correspondence: Professor F.Potet, Service dAnatomie Pathologique,Hapita1 Bichat, 46 Rue Henri Huchard, 75877 Paris cedex, France.
contributory in assessing their malignant nature. A resection of the lower two-thirds of the oesophagus and upper third of the stomach including the two oesophageal lesions was carried out. The outcome was good, and the patient is alive and well. PATHOLOGICAL FINDINGS
The specimen consisted of 11 cm of distal oesophagus and the upper third of the stomach. Near the proximal margin, there was an exophytic and ulcerated lesion, 2 cm in length, involving one-quarter of the oesophageal circumference and located 9 cm above a peptic stenosis of the cardia. Histologically,the lesion at the proximal margin was a submucosal process, sharply delimited (Figure l),exhibiting a follicular pattern constituted by round to oval follicles, sometimes confluent, surrounded by lymphocytes and numerous plasma cells. The follicles were composed of clusters of large cells with faint cytoplasm, and very large oval and regular nuclei intermingledwith centroblasts (Figure 2). This pattern suggested a follicular dendritic cell nature. Some follicles showed penetration of lymphocytes associated with disruption of the folliculo-dendritic network. The interfollicular zones showed a vascularized stroma with sheets of lymphocytes and plasma cells without hyalin vascular change. The histological study of the peptic stenosis showed a fibrous and thickened submucosa. Lymph nodes from the para-oesophageal tissue showed reactive changes. Immunohistochemistry was performed on par&
Brief reports
manner analogous to the phenomenon seen in salivary tumours4. Mixed tumours in which both epithelial and myoepithelial components are malignant have been described in dogs but these components also arise within a pre-existing benign mixed tumour6. Our preference, therefore, is to classify this tumour descriptively as a breast carcinoma with ductal, myoepithelial, squamous and sebaceous elements while accepting there are some similarities to salivary gland tumours. This unusual phenotypic constellation, particularly the sebaceous component, makes this a very rare, if not unique, breast neoplasm.
Acknowledgements We are most grateful to Dr M.Harris for his advice in the preparation of this manuscript and to Mr D.E.Edmond-
son and Miss S.Clews for assistance in photography and electronmicroscopy respectively.
References 1. Raju GC, Wee A. Spindle cell carcinomaof the breast. HistoputhologH 1990: 16; 497-499. 2. Wargotz ES,Deos PH. N o d s HJ. Metaplastic carcinomas of the breast. II. Spindle cellcarcinoma.Hum. Pathol. 1989: 20;732-740. 3. Rosen PP, Emsberger D. Low-grade adenosquamous carcinoma: a variant of metaplastic mammary carcinoma. Am. J. Surg. Pathol. 1987: 11; 351-358. 4. Thackray AC, Lucas RB.Tumours of major salivary glands. In Atlas of Tumour Puthologtl, Second series, Fascicle 10. Washington, D.C.: Armed Forces Instituteof Pathology, 1 9 7 4 20-30,107-117. 5. Sheth MT. Hathway D. PetrelIi M. Pleomorphic adenoma (‘mixed’ tumour) of human female breast mimicking carcinoma clinicoradiologically.Cancer 1978: 41; 659-665. 6. Moulton JE. Mammary gland. In Tumours in Domestic Animals. 2nd edn. Berkeley: University of California Press, 1977; 365-366.
Histopathology 1992,21,184-187
BRIEF REPORT
Derrnatofibrosarcoma protuberans recurring as a giant cell fibroblastoma J.COYNE, S.M.KAFTAN & R.D.P.CRAIG* Departments of Histopathology and *Plastic Surgery, Withington Hospital, Manchester, UK Date of submission 13 February 1992 Accepted for publication 12 March 1992
Keywords: giant cell fibroblastoma, dermatofibrosarcoma protuberans
Introduction Giant cell fibroblastoma, a rare mesenchymal tumour, is thought to have a close histogenetic relationship with dermatofibrosarcomaprotuberans. It has been proposed that giant cell fibroblastoma represents the juvenile form of dermatofibrosarcoma protuberans’, a concept strengthened by a recent single report2. Two cases of dermatofibrosarcoma protuberans with giant cell fibroblastoma-like areas have also been described and illustrated in the recent literature3. We present a case which
documents the recurrence of a lesion with the features of giant cell fibroblastoma 3 years after the local excision of a dermatofibrosarcomaprotuberans and, while supporting their close relationship, suggests that their different morphologicalmanifestationsare not simply age related.
Case report A 17-year-old male was noted to have a mass present in the left groin for several months. This was excised at another hospital. At age 20, he underwent re-excision of a recurrent tumour from the same region. PATHOLOGICAL FINDINGS
Address for correspondence: Dr J.Coyne. Histopathology Department. Withington Hospital, Nell Lane, Mancheater M20 8LR. UK.
The flrst specimen was described as a skin ellipse 4 x 1.5 cm with a 0.7 cm nodule located within the dermis.
Brief reports
18 5
Figure 1. a Cellular dermatofibrosarcoma protuberaus infiltrating subcutaneous fat and showing b a stodform pattern.
Microscopically,the overall histological appearance was that of a densely cellular, spindle cell tumour displaying a fascicular and storiform pattern. Approximately three mitotic figures per 10 h.p.f. were present: all of the mitoses were of normal appearance. The tumour surrounded residual skin appendage structures and infiltrated subcutaneous fat. Superficially, there was a small protuberant nodule in which spindle shaped cells with cellular processes were arranged haphazardly in a myxoid stroma. The appearances were interpreted on review as being those of a dermatofibrosarcoma protuberans (Figure 1). The recurrent lesion consisted of a skin ellipse 9 x 4 x 1.5 cm with an underlying lobulated, pink fleshy tumour measuring 7 x 3 cm. This was h l y attached to the overlying skin. Microscopically,the tumour showed a heterogeneous appearance with features typical of giant cell fibroblastoma (Figure 2a). Paucicellular, collagenized areas alternated with cellular, spindle cell areas. Areas containing diffusely arranged spindle cells dispersed within a myxoid stroma were also present. Many
spindle cells with large, hyperchromatic nuclei were present together with numerous multinucleated giant cells (Figure 2b). Frequent sinusoid-like spaces, partially lined by giant cells, were also present dispersed throughout the tumour (Figure 2a). Adnexal structures were frequently surrounded by the proliferation which i d trated the subcutaneous fat. An occasional normal mitotic figure was noted and a very patchy lymphoplasmacytic infiltrate was present at the tumour periphery. Occasional areas, composed of spindle cells in a myxoid stroma, were focally present as were small, mainly superficial areas of compactly arranged, tightly packed spindle cells in a fascicular pattern. In places the tumour extended up to the overlying epidermis while in others a narrow zone of normal papillary dermis was present.
Discussion Although distinctive histological entities, giant cell fibroblastoma and dermatofibrosarcomaprotuberans do
186
Brief reports
Figure 2. a Sinusoidal spaces, variable stromal cellularlty and giant cells. b Splndle stromal cells and scattered @antcells (shown at a higher power in the inset).
share some similar morphological characteristics. These include spindle cells, myxoid stroma, location within the dermis, entrapment of adnexal structures and infiltration of the subcutaneous fat. Indeed a myxoid stroma, which is a focal characteristic of giant cell fibroblastoma, can occasionally be the major feature of dermatofibrosarcoma protuberans*. Based on these similar histological features and on the mean age of occurrence of these entities, it was proposed that giant cell fibroblastoma represents the juvenile form of dermatofibrosarcoma protuberans'. A recent report describing a giant cell fibroblastoma recurring as a dermatofibrosarcoma protuberans would appear to lend support to this interpretation2. Beham & Fletcher3,while further demonstrating the close relationship between both tumours, considered this theory an oversimplification as they had encountered dermatofibrosarcoma protuberans occurring in children and giant cell fibroblastomas arising in adults. The present case tends to support this latter contention
and argues against the concept that giant cell fibroblastoma is simply a juvenile variant of dermatofibrosarcoma protuberans. While both may be different morphological manifestations of a neoplastic fibroblast proliferation, factors other than age must also be involved and these two entities may represent different degrees of tumour differentiation with different biological implications.
Acknowledgements The authors wish to acknowledge Mrs L.Garstang for excellent secretarial assistance and Dr A.Curry for the photomicrographs.
References 1. ShmooklerBM. Enzinger FM,Weiss SW. Glant cell fibroblastoma.A juvenile form of dermatofibrosarcomaprotuberans. Cancer 1989: 64; 2154-2161.
Brie! reports
2. AlguacilGarciaA. Giant cell fibroblastoma recurring as dermatofibrosarcoma protuberans (Case Report). Am. I. Surg. Pathol. 1991; 15(8); 798-801. 3. Beham A, Fletcher CDM. Dermatolibrosarcoma protuberans with
18 7
areas resembling giant cell fibroblastoma. Report of two cases. HistopathdwH 1 9 9 0 17; 165-167. 4. Frierson HF. Cooper PH. Myxoid variant of dermatoEbrosarcoma protuberans. Am. 1. Surg. Pathol. 1983: 7; 445-450.
Histopathol~g~ 1992,21, 187-189
BRIEF REPORT
Focal lymphoid hyperplasia of the oesophagus: report of a case E.GERVAZ, F.POTET, R.MAHE & G.LEMASSON Service d'tinatomie et de Cytologie Pathologiques, H6pital Bichat, Paris, France Date of submission 7 November 1991 Accepted for publication 16 March 1992
Keywords: oesophagus, lymphoreticular disorder, lymphoid hyperplasia
Introduction Focal lymphoid hyperplasia of the gastrointestinal tract is an uncommon benign lesion, defined as a follicular reaction with well-defined germinal centres, in response to a B-cell hyperplasia. Follicular lymphoid hyperplasia is frequent in the stomach (associated with peptic lesions) and in the rectum. Involvement of the oesophagus remains exceptional'. This report describes a case of gastrointestinal focal lymphoid hyperplasia located in the oesophagus and exhibiting very unusual lymphoreticular aspects.
Case report
An alcoholic, heavy smoker, HIV negative, 59-year-old man, had a 3-month history of increasing dysphagia. regurgitation and abdominal pain. Endoscopy showed an exophytic and ulcerated lesion located in the middle third of the anterior oesophageal wall associated with benign peptic stenosis and a hiatus hernia. Biopsy study of the exophytic lesion displayed a lymphoid infiltrate associated with clusters of undifTerentiated large cells. Immunohistochemistry was not Address for correspondence: Professor F.Potet, Service dAnatomie Pathologique,Hapita1 Bichat, 46 Rue Henri Huchard, 75877 Paris cedex, France.
contributory in assessing their malignant nature. A resection of the lower two-thirds of the oesophagus and upper third of the stomach including the two oesophageal lesions was carried out. The outcome was good, and the patient is alive and well. PATHOLOGICAL FINDINGS
The specimen consisted of 11 cm of distal oesophagus and the upper third of the stomach. Near the proximal margin, there was an exophytic and ulcerated lesion, 2 cm in length, involving one-quarter of the oesophageal circumference and located 9 cm above a peptic stenosis of the cardia. Histologically,the lesion at the proximal margin was a submucosal process, sharply delimited (Figure l),exhibiting a follicular pattern constituted by round to oval follicles, sometimes confluent, surrounded by lymphocytes and numerous plasma cells. The follicles were composed of clusters of large cells with faint cytoplasm, and very large oval and regular nuclei intermingledwith centroblasts (Figure 2). This pattern suggested a follicular dendritic cell nature. Some follicles showed penetration of lymphocytes associated with disruption of the folliculo-dendritic network. The interfollicular zones showed a vascularized stroma with sheets of lymphocytes and plasma cells without hyalin vascular change. The histological study of the peptic stenosis showed a fibrous and thickened submucosa. Lymph nodes from the para-oesophageal tissue showed reactive changes. Immunohistochemistry was performed on par&
