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Solitary fibrous tumour of the oral cavity: report of two cases
Pathology
ISSN: 0031-3025 (Print) 1465-3931 (Online) Journal homepage: http://www.tandfonline.com/loi/ipat20
SOLITARY FIBROUS TUMOUR OF THE ORAL CAVITY: REPORT OF TWO CASES Anna A. Talacko, Michael J. Aldred, William R. Sheldon, N. Richard Hing To cite this article: Anna A. Talacko, Michael J. Aldred, William R. Sheldon, N. Richard Hing (2001) SOLITARY FIBROUS TUMOUR OF THE ORAL CAVITY: REPORT OF TWO CASES, Pathology, 33:3, 315-318 To link to this article: https://doi.org/10.1080/00313020126298
Published online: 06 Jul 2009.
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Article views: 5
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Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=ipat20
Pathology (2001 ) 33, pp. 315– 318
SOLITARY FIBROUS TUMOUR OF THE ORAL CAVITY: REPORT OF TWO CASES ANNA A. TALACKO *, MICHAEL J. ALDRED *, WILLIAM R. SHELDON † AND N. RICHARD HING ‡ *Oral Pathologist, Mayne Health Dorevitch Pathology, Heidelberg, †Oral and Maxillofacial Surgeon, East Melbourne, and ‡Oral and Maxillofacial Surgeon, Mount Waverley, Victoria, Australia
Summary The solitary fibrous tumour is an uncommon, benign neoplasm of adults involving the pleura. It is now recognised to occur in extrapleural sites. Only a limited number of cases have been reported in the oral cavity. This paper reports two further cases, which presented as clinically benign masses in the palate and buccal mucosa respectively. Key words: Solitary fibrous tumour, solitary fibrous tumor, oral cavity, CD34. Received 22 February 2000; revised 21 December 2000; accepted 8 January 2001
INTRODUCTION The solitary fibrous tumour ( SFT) was first described in 1931 as a localised fibrous mesothelioma.1 It is an uncommon, benign neoplasm of adults involving the pleura. However, its rare occurrence at other sites has only become appreciated in the last decade.2,3 Sites reported in the head and neck include the major salivary glands,4 –7 nasal cavity,8,9 orbit,10– 12 infratemporal fossa,13 thyroid gland14 and deep soft tissues of the neck.15 Only 10 cases in the world literature have been reported in the oral cavity.16– 22 Eight of these have occurred in the buccal mucosa.16– 21 The remaining two reported sites include the tongue17 and retromolar region.20 This paper reports two further cases of SFT occurring in the mouth.
CASE REPORTS Case 1 A 28-year-old male presented with a painless, firm, non-ulcerated mass approximately 2´ 2 cm in diameter in the right side of the hard palate ( Fig. 1) of 4 years duration. CT scans did not show any erosion of the palatal bone. A provisional clinical diagnosis of pleomorphic adenoma was made and the swelling was excised.
Case 2 A 51-year-old female presented with an asymptomatic, sessile swelling of the right buccal mucosa approximately 0.8 cm in diameter. Clinically, this was felt to be a fibroepithelial polyp and it was excised.
Fig. 1 Clinical photograph showing the swelling of the right side of the hard palate in Case 1.
Pathological findings In Case 1, the excised specimen comprised a circumscribed spindle cell tumour featuring interlacing bundles of connective tissue fibres interspersed with cells ( Fig. 2). The fibrous component appeared to be collagenous in places but there were many areas of myxomatous change. The cells were elongated but in some areas were plump and angular. Occasional mast cells were present. It was felt that this was a benign spindle cell lesion with features suggestive of a neurofibroma. Immunohistochemical markers revealed that the tumour cells were CD34-positive ( Fig. 3) and S100-negative, thus excluding a diagnosis of neurofibroma . The final diagnosis of SFT was made on the basis of the positive CD34 immunohistochemical result. In Case 2, the histopathological features were those of a wellcircumscribed, richly cellular, spindle cell tumour ( Fig. 4). The tumour cells were ovoid and spindle-shaped with pale-staining cytoplasm; mitotic activity was absent. In areas, the spindle cells were separated by bands of collagen ( Fig. 5). There was no convincing storiform or fascicular pattern. Immunohistochemical studies included S100, smooth muscle actin, Factor XIIIa and CD34. The spindle cells were CD34-positive ( Fig. 6), while the remaining markers were negative. On the basis of the histopathologica l features and strong positive staining for CD34, a diagnosis of SFT was made.
ISSN 0031–3025 printed/ISSN 1465– 3931 online/01/030315 – 04 © 2001 Royal College of Pathologists of Australasia DOI:10.1080/00313020120062910
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TALACKO et al.
Pathology ( 2001 ), 33, August
Fig. 2 Photomicrograph of Case 1 showing spindle cells separated by bands of collagen ( H&E, original magnification, ´ 20).
Fig. 3 Photomicrograph of Case 1 showing immunoperoxidase stain positive for CD34 ( anti-CD34, original magnification, ´ 10 ).
Fig. 4 Photomicrograph of Case 2 showing rounded submucosal nodule ( H&E, original magnification, ´ 2).
SOLITARY FIBROUS TUMOUR
317
Fig. 5 Photomicrograph of Case 2 showing irregularly arranged spindle cells in a collagenous background ( H&E, original magnification, ´ 20 ).
DISCUSSION Clinically, the SFT presents as a painless, firm mass, which may grow to a significant size; other symptoms are sitespecific. Histologically, the SFT is well circumscribed and comprises spindle cells in a collagenous background. The spindle cells are cytologically bland and arranged with no obvious pattern, although focally storiform and fascicular growth patterns are seen. Prominent vascularity, showing a haemangiopericytoma-like pattern, is sometimes seen. The tumour has a strong immunoreactivity with CD34, a vascular marker which has also been found to be positive in fibroblast-like cells and various mesenchymal tumours. The cell of origin is thought to be a fibroblast.23
The differential diagnosis lies between benign spindle cell neoplasms including leiomyoma, haemangiopericytoma, fibrohistiocytic and neural tumours, as well as fibromatoses, malignant fibrous histiocytoma and sarcomas. Benign spindle cell lesions previously reported under these categories and many haemangiopericytomas may in fact have been SFTs. The correct diagnosis of SFT has been assisted by immunohistochemistry ( Table 1). The term SFT has been applied in preference to “fibroma”, which is often loosely and incorrectly used as a diagnosis for varying fibrous lesions, both reactive and neoplastic. Although some pleural tumours undergo malignant transformation or are malignant de novo, extrapleural tumours are generally benign. Surgical excision is curative for benign lesions. The two cases reported here demonstrate
Fig. 6 Photomicrograph of Case 2 showing immunoperoxidase stains positive for CD34 ( anti-CD34, original magnification, ´ 20) .
318
Pathology ( 2001 ), 33, August
TALACKO et al.
TA BLE 1
Immunohistochemical differential diagnostic features of benign spindle cell lesions
Diagnosis
SFT Leiomyoma Haemangiopericytoma Fibrous histiocytoma Neurofibroma
Immunohistochemical findings S100
Smooth muscle actin
Factor XIIIa
– – – –
–
– –
–
– –
– –
+
+
– – –
that the SFT is being increasingly recognised in the oral cavity as well as elsewhere in the head and neck region. Clinically, the oral presentation may mimic a reactive lesion such as a fibroepithelial polyp or a neoplastic mass such as a minor salivary gland tumour. Doubtless, similar cases from the oral cavity have fallen under the diagnostic category of benign spindle cell tumours in the past, but their definitive diagnosis is now possible with the identification of the characteristic histological features of SFT – the “patternless pattern” – and confirmation with immunohistochemical markers. AC KN OW LED GEM ENTS Dr Phillip Ironside and Dr Sant Khan are thanked for their assistance in the histopathological diagnosis in each of these cases.
+
Desmin
Vimentin
CD68
+
– ± – – –
+ + + + +
– – –
+
9. 10. 11. 12. 13. 14. 15. 16.
Address for correspondence: Dr A. A. Talacko, Mayne Health Dorevitch Pathology, 18 Banksia Street, Heidelberg, Vic 3084, Australia. Email: [email protected]
17.
References
19.
1. Klemperer P, Ratin CB. Primary neoplasms of the pleura: a report of five cases. Arch Pathol 1931; 11: 385–412. 2. Chan JK. Solitary fibrous tumour – everywhere, and a diagnosis in vogue. Histopathology 1997; 31: 568–76. 3. Goodlad J, Fletcher C. Solitary fibrous tumour arising at unusual sites: analysis of a series. Histopathology 1997; 19: 515–22. 4. Ferreiro JA, Nascimento AG. Solitary fibrous tumour of the major salivary glands. Histopathology 1996; 28: 261– 4. 5. Guarino M, Giordano F, Pallotti F, Ponzi S. Solitary fibrous tumour of the submandibular gland. Histopathology 1998; 32: 571– 3. 6. Gunnan O, Yildiz FR, Celasun B, et al. Solitary fibrous tumour arising from sublingual gland; report of a case. J Laryngol Otol 1994; 108: 998–1000. 7. Sato J, Asakura K, Yokoyama Y, Satoh M. Solitary fibrous tumour of the parotid gland extending to the parapharyngeal space. Eur Arch Otorhinolaryngol 1998; 255: 18– 21. 8. Mentzel T, Bainbridge TC, Katenkamp D. Solitary fibrous tumour:
CD34
18.
20.
21.
22. 23.
+
–
clinicopathological, immunohistochemical, and ultrastructural analysis of 12 cases arising in soft tissues, nasal cavity and nasopharynx , urinary bladder and prostate. Virchows Arch 1997; 430: 445– 53. Stringfellow HF, Khan IA, Sissons MC. Solitary fibrous tumour arising in the nasal cavity: report of a case. J Laryngol Otol 1996; 110: 468–70. Heathcote JG. Pathology update: solitary fibrous tumour of the orbit. Can J Ophthalmol 1997; 32: 432– 5. McElvanney AM, Noble JL, O’Donovan DG, et al. Solitary fibrous tumour: an atypical presentation within the orbit. Eye 1996; 10: 396– 9. Sciot R, Goffin J, Fossion E, et al. Solitary fibrous tumour of the orbit. Histopathology 1996; 28: 188– 91. Rayappa CS, McArthur PD, Gangopadhyay K, Antonius JI. Solitary fibrous tumour of the infratemporal fossa. J Laryngol Otol 1996; 110: 594–7. Kie JH, Kim JY, Park YN, et al. Solitary fibrous tumour of the thyroid. Histopathology 1997; 30: 365– 8. Greig AV, Spraggs PD, Mannion E, et al. Solitary fibrous tumour of the deep soft tissues of the neck. J Laryngol Otol 1998; 112: 503–5. Fornelli A, Cocchi R, Eusebi V. Solitary fibrous tumor of the cheek. Pathologica 1996; 88: 36– 9. Piattelli A, Fioroni M, Rubini C. Solitary fibrous tumour of the tongue. Oral Oncol 1998; 34: 431–4. Kurihara K, Mizuseki K, Sonobe J, Yanagihara J. Solitary fibrous tumour of the oral cavity: report of a case. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999; 87: 223– 6. Eversole L, Christensen R, Ficarra G, et al. Nodular fasciitis and solitary fibrous tumour of the oral region. Tumours of fibroblast heterogeneity. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999; 87: 471–6. Perez-Ordonez B, Koutlas IG, Strich E, et al. Solitary fibrous tumour of the oral cavity. An uncommon location for a ubiquitous neoplasm. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999; 87: 589– 93. Iwai S, Nakazawa M, Yoshikawa F, et al. Solitary fibrous tumour of the buccal mucosa: report of a case with immunhistochemical studies. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999; 88: 461– 5. Lukinmaa P, Heitanen J, Warfvinge G, et al. Solitary fibrous tumour of the oral cavity: clinicopathological and immunohistochemical characterization of three cases. J Oral Pathol Med 2000; 29: 186– 92. Ali S, Hoon V, Hoda S, et al. Solitary fibrous tumour. A cytologichistologic study with clinical, radiologic and immunohistochemica l correlations. Cancer ( Cancer Cytopathol) 1997; 81: 116– 2.
ISSN: 0031-3025 (Print) 1465-3931 (Online) Journal homepage: http://www.tandfonline.com/loi/ipat20
SOLITARY FIBROUS TUMOUR OF THE ORAL CAVITY: REPORT OF TWO CASES Anna A. Talacko, Michael J. Aldred, William R. Sheldon, N. Richard Hing To cite this article: Anna A. Talacko, Michael J. Aldred, William R. Sheldon, N. Richard Hing (2001) SOLITARY FIBROUS TUMOUR OF THE ORAL CAVITY: REPORT OF TWO CASES, Pathology, 33:3, 315-318 To link to this article: https://doi.org/10.1080/00313020126298
Published online: 06 Jul 2009.
Submit your article to this journal
Article views: 5
View related articles
Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=ipat20
Pathology (2001 ) 33, pp. 315– 318
SOLITARY FIBROUS TUMOUR OF THE ORAL CAVITY: REPORT OF TWO CASES ANNA A. TALACKO *, MICHAEL J. ALDRED *, WILLIAM R. SHELDON † AND N. RICHARD HING ‡ *Oral Pathologist, Mayne Health Dorevitch Pathology, Heidelberg, †Oral and Maxillofacial Surgeon, East Melbourne, and ‡Oral and Maxillofacial Surgeon, Mount Waverley, Victoria, Australia
Summary The solitary fibrous tumour is an uncommon, benign neoplasm of adults involving the pleura. It is now recognised to occur in extrapleural sites. Only a limited number of cases have been reported in the oral cavity. This paper reports two further cases, which presented as clinically benign masses in the palate and buccal mucosa respectively. Key words: Solitary fibrous tumour, solitary fibrous tumor, oral cavity, CD34. Received 22 February 2000; revised 21 December 2000; accepted 8 January 2001
INTRODUCTION The solitary fibrous tumour ( SFT) was first described in 1931 as a localised fibrous mesothelioma.1 It is an uncommon, benign neoplasm of adults involving the pleura. However, its rare occurrence at other sites has only become appreciated in the last decade.2,3 Sites reported in the head and neck include the major salivary glands,4 –7 nasal cavity,8,9 orbit,10– 12 infratemporal fossa,13 thyroid gland14 and deep soft tissues of the neck.15 Only 10 cases in the world literature have been reported in the oral cavity.16– 22 Eight of these have occurred in the buccal mucosa.16– 21 The remaining two reported sites include the tongue17 and retromolar region.20 This paper reports two further cases of SFT occurring in the mouth.
CASE REPORTS Case 1 A 28-year-old male presented with a painless, firm, non-ulcerated mass approximately 2´ 2 cm in diameter in the right side of the hard palate ( Fig. 1) of 4 years duration. CT scans did not show any erosion of the palatal bone. A provisional clinical diagnosis of pleomorphic adenoma was made and the swelling was excised.
Case 2 A 51-year-old female presented with an asymptomatic, sessile swelling of the right buccal mucosa approximately 0.8 cm in diameter. Clinically, this was felt to be a fibroepithelial polyp and it was excised.
Fig. 1 Clinical photograph showing the swelling of the right side of the hard palate in Case 1.
Pathological findings In Case 1, the excised specimen comprised a circumscribed spindle cell tumour featuring interlacing bundles of connective tissue fibres interspersed with cells ( Fig. 2). The fibrous component appeared to be collagenous in places but there were many areas of myxomatous change. The cells were elongated but in some areas were plump and angular. Occasional mast cells were present. It was felt that this was a benign spindle cell lesion with features suggestive of a neurofibroma. Immunohistochemical markers revealed that the tumour cells were CD34-positive ( Fig. 3) and S100-negative, thus excluding a diagnosis of neurofibroma . The final diagnosis of SFT was made on the basis of the positive CD34 immunohistochemical result. In Case 2, the histopathological features were those of a wellcircumscribed, richly cellular, spindle cell tumour ( Fig. 4). The tumour cells were ovoid and spindle-shaped with pale-staining cytoplasm; mitotic activity was absent. In areas, the spindle cells were separated by bands of collagen ( Fig. 5). There was no convincing storiform or fascicular pattern. Immunohistochemical studies included S100, smooth muscle actin, Factor XIIIa and CD34. The spindle cells were CD34-positive ( Fig. 6), while the remaining markers were negative. On the basis of the histopathologica l features and strong positive staining for CD34, a diagnosis of SFT was made.
ISSN 0031–3025 printed/ISSN 1465– 3931 online/01/030315 – 04 © 2001 Royal College of Pathologists of Australasia DOI:10.1080/00313020120062910
316
TALACKO et al.
Pathology ( 2001 ), 33, August
Fig. 2 Photomicrograph of Case 1 showing spindle cells separated by bands of collagen ( H&E, original magnification, ´ 20).
Fig. 3 Photomicrograph of Case 1 showing immunoperoxidase stain positive for CD34 ( anti-CD34, original magnification, ´ 10 ).
Fig. 4 Photomicrograph of Case 2 showing rounded submucosal nodule ( H&E, original magnification, ´ 2).
SOLITARY FIBROUS TUMOUR
317
Fig. 5 Photomicrograph of Case 2 showing irregularly arranged spindle cells in a collagenous background ( H&E, original magnification, ´ 20 ).
DISCUSSION Clinically, the SFT presents as a painless, firm mass, which may grow to a significant size; other symptoms are sitespecific. Histologically, the SFT is well circumscribed and comprises spindle cells in a collagenous background. The spindle cells are cytologically bland and arranged with no obvious pattern, although focally storiform and fascicular growth patterns are seen. Prominent vascularity, showing a haemangiopericytoma-like pattern, is sometimes seen. The tumour has a strong immunoreactivity with CD34, a vascular marker which has also been found to be positive in fibroblast-like cells and various mesenchymal tumours. The cell of origin is thought to be a fibroblast.23
The differential diagnosis lies between benign spindle cell neoplasms including leiomyoma, haemangiopericytoma, fibrohistiocytic and neural tumours, as well as fibromatoses, malignant fibrous histiocytoma and sarcomas. Benign spindle cell lesions previously reported under these categories and many haemangiopericytomas may in fact have been SFTs. The correct diagnosis of SFT has been assisted by immunohistochemistry ( Table 1). The term SFT has been applied in preference to “fibroma”, which is often loosely and incorrectly used as a diagnosis for varying fibrous lesions, both reactive and neoplastic. Although some pleural tumours undergo malignant transformation or are malignant de novo, extrapleural tumours are generally benign. Surgical excision is curative for benign lesions. The two cases reported here demonstrate
Fig. 6 Photomicrograph of Case 2 showing immunoperoxidase stains positive for CD34 ( anti-CD34, original magnification, ´ 20) .
318
Pathology ( 2001 ), 33, August
TALACKO et al.
TA BLE 1
Immunohistochemical differential diagnostic features of benign spindle cell lesions
Diagnosis
SFT Leiomyoma Haemangiopericytoma Fibrous histiocytoma Neurofibroma
Immunohistochemical findings S100
Smooth muscle actin
Factor XIIIa
– – – –
–
– –
–
– –
– –
+
+
– – –
that the SFT is being increasingly recognised in the oral cavity as well as elsewhere in the head and neck region. Clinically, the oral presentation may mimic a reactive lesion such as a fibroepithelial polyp or a neoplastic mass such as a minor salivary gland tumour. Doubtless, similar cases from the oral cavity have fallen under the diagnostic category of benign spindle cell tumours in the past, but their definitive diagnosis is now possible with the identification of the characteristic histological features of SFT – the “patternless pattern” – and confirmation with immunohistochemical markers. AC KN OW LED GEM ENTS Dr Phillip Ironside and Dr Sant Khan are thanked for their assistance in the histopathological diagnosis in each of these cases.
+
Desmin
Vimentin
CD68
+
– ± – – –
+ + + + +
– – –
+
9. 10. 11. 12. 13. 14. 15. 16.
Address for correspondence: Dr A. A. Talacko, Mayne Health Dorevitch Pathology, 18 Banksia Street, Heidelberg, Vic 3084, Australia. Email: [email protected]
17.
References
19.
1. Klemperer P, Ratin CB. Primary neoplasms of the pleura: a report of five cases. Arch Pathol 1931; 11: 385–412. 2. Chan JK. Solitary fibrous tumour – everywhere, and a diagnosis in vogue. Histopathology 1997; 31: 568–76. 3. Goodlad J, Fletcher C. Solitary fibrous tumour arising at unusual sites: analysis of a series. Histopathology 1997; 19: 515–22. 4. Ferreiro JA, Nascimento AG. Solitary fibrous tumour of the major salivary glands. Histopathology 1996; 28: 261– 4. 5. Guarino M, Giordano F, Pallotti F, Ponzi S. Solitary fibrous tumour of the submandibular gland. Histopathology 1998; 32: 571– 3. 6. Gunnan O, Yildiz FR, Celasun B, et al. Solitary fibrous tumour arising from sublingual gland; report of a case. J Laryngol Otol 1994; 108: 998–1000. 7. Sato J, Asakura K, Yokoyama Y, Satoh M. Solitary fibrous tumour of the parotid gland extending to the parapharyngeal space. Eur Arch Otorhinolaryngol 1998; 255: 18– 21. 8. Mentzel T, Bainbridge TC, Katenkamp D. Solitary fibrous tumour:
CD34
18.
20.
21.
22. 23.
+
–
clinicopathological, immunohistochemical, and ultrastructural analysis of 12 cases arising in soft tissues, nasal cavity and nasopharynx , urinary bladder and prostate. Virchows Arch 1997; 430: 445– 53. Stringfellow HF, Khan IA, Sissons MC. Solitary fibrous tumour arising in the nasal cavity: report of a case. J Laryngol Otol 1996; 110: 468–70. Heathcote JG. Pathology update: solitary fibrous tumour of the orbit. Can J Ophthalmol 1997; 32: 432– 5. McElvanney AM, Noble JL, O’Donovan DG, et al. Solitary fibrous tumour: an atypical presentation within the orbit. Eye 1996; 10: 396– 9. Sciot R, Goffin J, Fossion E, et al. Solitary fibrous tumour of the orbit. Histopathology 1996; 28: 188– 91. Rayappa CS, McArthur PD, Gangopadhyay K, Antonius JI. Solitary fibrous tumour of the infratemporal fossa. J Laryngol Otol 1996; 110: 594–7. Kie JH, Kim JY, Park YN, et al. Solitary fibrous tumour of the thyroid. Histopathology 1997; 30: 365– 8. Greig AV, Spraggs PD, Mannion E, et al. Solitary fibrous tumour of the deep soft tissues of the neck. J Laryngol Otol 1998; 112: 503–5. Fornelli A, Cocchi R, Eusebi V. Solitary fibrous tumor of the cheek. Pathologica 1996; 88: 36– 9. Piattelli A, Fioroni M, Rubini C. Solitary fibrous tumour of the tongue. Oral Oncol 1998; 34: 431–4. Kurihara K, Mizuseki K, Sonobe J, Yanagihara J. Solitary fibrous tumour of the oral cavity: report of a case. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999; 87: 223– 6. Eversole L, Christensen R, Ficarra G, et al. Nodular fasciitis and solitary fibrous tumour of the oral region. Tumours of fibroblast heterogeneity. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999; 87: 471–6. Perez-Ordonez B, Koutlas IG, Strich E, et al. Solitary fibrous tumour of the oral cavity. An uncommon location for a ubiquitous neoplasm. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999; 87: 589– 93. Iwai S, Nakazawa M, Yoshikawa F, et al. Solitary fibrous tumour of the buccal mucosa: report of a case with immunhistochemical studies. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999; 88: 461– 5. Lukinmaa P, Heitanen J, Warfvinge G, et al. Solitary fibrous tumour of the oral cavity: clinicopathological and immunohistochemical characterization of three cases. J Oral Pathol Med 2000; 29: 186– 92. Ali S, Hoon V, Hoda S, et al. Solitary fibrous tumour. A cytologichistologic study with clinical, radiologic and immunohistochemica l correlations. Cancer ( Cancer Cytopathol) 1997; 81: 116– 2.