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Angioleiomyoma: An unusual diagnosis for a lump in the cheek
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British Journal of Oral and Maxillofacial Surgery 47 (2009) 641–642
Short communication
Angioleiomyoma: An unusual diagnosis for a lump in the cheek Helen McParland, Saman Warnakulasuriya, Richard J. Cook ∗ Department Oral Medicine, King’s College London Dental Institute, Floor 22 Tower Wing, Guy’s Campus, Great Maze Pond, London SE1 9RT, United Kingdom Accepted 11 December 2008 Available online 4 February 2009
Abstract Buccal swellings such as fibroepithelial polyps and granulomas are commonly seen in any oral diagnostic clinic, but occasionally an unusual mass presents. We describe a rare case of a man with a four-year history of a slowly enlarging, painless buccal mass that was confirmed histologically as an angioleiomyoma. Deep dissection to excise the mass completely caused no damage to nerves and there has been no recurrence. © 2009 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Angioleiomyoma; Buccal; Resection; Curative
Introduction Exophytic growths are often seen in oral diagnostic clinics; fibroepithelial polyps and granulomas being the most common that are benign, although occasionally minor salivary gland adenomas or squamous cell malignancies should be considered. We describe a rare case that was confirmed histologically as an angioleiomyoma.
Case report A 42-year-old man presented with a four-year history of a slowly enlarging, painless, left buccal mucosal swelling. He had no other dentoalveolar symptoms and no signs of local infection, but his teeth were worn with bruxing. He was fit and was not taking any drugs, did not smoke tobacco, and drank alcohol modestly. Examination showed a solitary, firm, non-pulsatile, sessile swelling 3 cm × 3 cm, arising from the left buccal submu∗
Corresponding author. Tel.: +44 2071 885399; fax: +44 7778 1709882. E-mail address: Richard [email protected] (R.J. Cook).
cosa (Fig. 1). The outer aspect was flattened with an intact epithelium although he had a central ulcer before biopsy examination. There was no locoregional lymphadenopathy, and we made provisional diagnoses of fibroma, fibroepithelial polyp as a result of trauma, or minor salivary gland adenoma. There were no clinical features of malignancy. We took an excisional biopsy specimen through an intraoral approach under local anaesthesia, mindful of the proximity of the parotid duct and neurovascular structures, particularly VII nerve in the depths of the excision site. Histopathological analysis (Fig. 2) showed a wellcircumscribed angioleiomyoma with all margins free of tumour. There were no nuclear atypia or signs of mitosis. Reviews at both three weeks and six months after excision, showed that VII nerve function was intact and there was no evidence of recurrence.
Discussion Leiomyomas are usually derived from uterine, gastrointestinal tract, and smooth muscle in skin.1,2 They are identified by their smooth muscle cell lineage and are histologically and
0266-4356/$ – see front matter © 2009 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2008.12.008
642
H. McParland et al. / British Journal of Oral and Maxillofacial Surgery 47 (2009) 641–642
Fig. 1. Photograph showing the lesion in the left buccal mucosa.
Leiomyomas present as well-defined, typically painless, slow growing, firm mucosal lumps, often pale in colour, or with normal overlying mucosa; they are typically less than 2 cm in diameter.1,2,4,5 Angioleiomyomas can present in bone as unilocular or multilocular radiolucencies with well or poorly defined borders and cortical involvement. They can cause mobility of teeth and occasionally dysphagia and dysgeusia.2,5,7 The widely accepted treatment is excision; recurrence is rare if excised completely.1,2,4 Because of its non-specific clinical appearance, diagnosis is principally histological, showing a proliferation of smooth muscle cells with few mitoses and no necrosis.1,3,4 Our case had an intact overlying mucosa at presentation, which suggested a potential neoplastic aetiology rather than a reactionary change, although occasional damage from chewing is inevitable in such an exophytic mass. We know of only one case reported with a granular cell change.8 Common histological differential diagnoses include myofibroma, palisaded encapsulated neuroma, and solitary fibrous tumour.9 Leiomyomas are typically fully encapsulated (as in our case), and there is no invasion; differentiation from malignant leiomyosarcoma is essential.2,5
References
Fig. 2. Photomicrographs showing sections of the well circumscribed angioleiomyoma. All margins were free from tumour. Smooth muscle bands (S) are surrounded by slit like vascular spaces (V) of variable calibres. A part of the fibrous capsule (E) that encapsulates the tumour is shown (field width = 1.5 mm, haematoxylin and eosin stain).
morphologically classified as either solid, angioleiomyoma, or epithelioid types.3 Leiomyomas are rare in the oral cavity because of the paucity of smooth muscle. When detected they are typically found on the tongue, lip, or palate, with few buccal examples.1,2,4,5 Their peak incidence is the fifth decade and they are unusual in children or older patients where myosarcoma is more likely.1,2 In the head and neck, vascular leiomyomas are the most common benign smooth muscle tumours in the turbinates and larynx.6
1. Luaces Rey R, Lorenzo Franco F, Gómez Oliveira G, Pati˜no Seijas B, Guitián D, López-Cedrún Cembranos JL. Oral leiomyoma in retromolar trigone. A case report. Med Oral Patol Oral Cir Bucal 2007;12:E53–5. 2. Wertheimer-Hatch L, Hatch 3rd GF, Hatch BSKF, Davis GB, Blanchard DK, Foster Jr RS, Skandalakis JE. Tumors of the oral cavity and pharynx. World J Surg 2000;24:395–400. 3. Damm DD, Neville BW. Oral leiomyomas. Oral Surg Oral Med Oral Pathol 1979;47:343–8. 4. Lloria-Benet M, Bagán JV, Lloria de Miguel E, Borja-Morant A, Alonso S. Oral leiomyoma: a case report. Med Oral 2003;8:215–9. 5. Brooks JK, Nikitakis NG, Goodman NJ, Levy BA. Clinicopathologic characterization of oral angioleiomyomas. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:221–7. 6. Pathology and genetics of head and neck tumours.Barnes L, Eveson JW, Reichart P, Sidransky D, editors. World Health Organization Classification of Tumours.. Lyon: IARC Press; 2005. 7. Liang H, Frederiksen NL, Binnie WH, Cheng YS. Intraosseous oral leiomyoma: systematic review and report of one case. Dentomaxillofac Radiol 2003;32:285–90. 8. Bhattacharyya I, Summerlin DJ, Cohen DM, Ellis GL, Bavitz JB, Gillham LL. Granular cell leiomyoma of the oral cavity. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:353–9. 9. Jordan RC, Regezi JA. Oral spindle cell neoplasms: a review of 307 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;95:717–24.
British Journal of Oral and Maxillofacial Surgery 47 (2009) 641–642
Short communication
Angioleiomyoma: An unusual diagnosis for a lump in the cheek Helen McParland, Saman Warnakulasuriya, Richard J. Cook ∗ Department Oral Medicine, King’s College London Dental Institute, Floor 22 Tower Wing, Guy’s Campus, Great Maze Pond, London SE1 9RT, United Kingdom Accepted 11 December 2008 Available online 4 February 2009
Abstract Buccal swellings such as fibroepithelial polyps and granulomas are commonly seen in any oral diagnostic clinic, but occasionally an unusual mass presents. We describe a rare case of a man with a four-year history of a slowly enlarging, painless buccal mass that was confirmed histologically as an angioleiomyoma. Deep dissection to excise the mass completely caused no damage to nerves and there has been no recurrence. © 2009 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Angioleiomyoma; Buccal; Resection; Curative
Introduction Exophytic growths are often seen in oral diagnostic clinics; fibroepithelial polyps and granulomas being the most common that are benign, although occasionally minor salivary gland adenomas or squamous cell malignancies should be considered. We describe a rare case that was confirmed histologically as an angioleiomyoma.
Case report A 42-year-old man presented with a four-year history of a slowly enlarging, painless, left buccal mucosal swelling. He had no other dentoalveolar symptoms and no signs of local infection, but his teeth were worn with bruxing. He was fit and was not taking any drugs, did not smoke tobacco, and drank alcohol modestly. Examination showed a solitary, firm, non-pulsatile, sessile swelling 3 cm × 3 cm, arising from the left buccal submu∗
Corresponding author. Tel.: +44 2071 885399; fax: +44 7778 1709882. E-mail address: Richard [email protected] (R.J. Cook).
cosa (Fig. 1). The outer aspect was flattened with an intact epithelium although he had a central ulcer before biopsy examination. There was no locoregional lymphadenopathy, and we made provisional diagnoses of fibroma, fibroepithelial polyp as a result of trauma, or minor salivary gland adenoma. There were no clinical features of malignancy. We took an excisional biopsy specimen through an intraoral approach under local anaesthesia, mindful of the proximity of the parotid duct and neurovascular structures, particularly VII nerve in the depths of the excision site. Histopathological analysis (Fig. 2) showed a wellcircumscribed angioleiomyoma with all margins free of tumour. There were no nuclear atypia or signs of mitosis. Reviews at both three weeks and six months after excision, showed that VII nerve function was intact and there was no evidence of recurrence.
Discussion Leiomyomas are usually derived from uterine, gastrointestinal tract, and smooth muscle in skin.1,2 They are identified by their smooth muscle cell lineage and are histologically and
0266-4356/$ – see front matter © 2009 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2008.12.008
642
H. McParland et al. / British Journal of Oral and Maxillofacial Surgery 47 (2009) 641–642
Fig. 1. Photograph showing the lesion in the left buccal mucosa.
Leiomyomas present as well-defined, typically painless, slow growing, firm mucosal lumps, often pale in colour, or with normal overlying mucosa; they are typically less than 2 cm in diameter.1,2,4,5 Angioleiomyomas can present in bone as unilocular or multilocular radiolucencies with well or poorly defined borders and cortical involvement. They can cause mobility of teeth and occasionally dysphagia and dysgeusia.2,5,7 The widely accepted treatment is excision; recurrence is rare if excised completely.1,2,4 Because of its non-specific clinical appearance, diagnosis is principally histological, showing a proliferation of smooth muscle cells with few mitoses and no necrosis.1,3,4 Our case had an intact overlying mucosa at presentation, which suggested a potential neoplastic aetiology rather than a reactionary change, although occasional damage from chewing is inevitable in such an exophytic mass. We know of only one case reported with a granular cell change.8 Common histological differential diagnoses include myofibroma, palisaded encapsulated neuroma, and solitary fibrous tumour.9 Leiomyomas are typically fully encapsulated (as in our case), and there is no invasion; differentiation from malignant leiomyosarcoma is essential.2,5
References
Fig. 2. Photomicrographs showing sections of the well circumscribed angioleiomyoma. All margins were free from tumour. Smooth muscle bands (S) are surrounded by slit like vascular spaces (V) of variable calibres. A part of the fibrous capsule (E) that encapsulates the tumour is shown (field width = 1.5 mm, haematoxylin and eosin stain).
morphologically classified as either solid, angioleiomyoma, or epithelioid types.3 Leiomyomas are rare in the oral cavity because of the paucity of smooth muscle. When detected they are typically found on the tongue, lip, or palate, with few buccal examples.1,2,4,5 Their peak incidence is the fifth decade and they are unusual in children or older patients where myosarcoma is more likely.1,2 In the head and neck, vascular leiomyomas are the most common benign smooth muscle tumours in the turbinates and larynx.6
1. Luaces Rey R, Lorenzo Franco F, Gómez Oliveira G, Pati˜no Seijas B, Guitián D, López-Cedrún Cembranos JL. Oral leiomyoma in retromolar trigone. A case report. Med Oral Patol Oral Cir Bucal 2007;12:E53–5. 2. Wertheimer-Hatch L, Hatch 3rd GF, Hatch BSKF, Davis GB, Blanchard DK, Foster Jr RS, Skandalakis JE. Tumors of the oral cavity and pharynx. World J Surg 2000;24:395–400. 3. Damm DD, Neville BW. Oral leiomyomas. Oral Surg Oral Med Oral Pathol 1979;47:343–8. 4. Lloria-Benet M, Bagán JV, Lloria de Miguel E, Borja-Morant A, Alonso S. Oral leiomyoma: a case report. Med Oral 2003;8:215–9. 5. Brooks JK, Nikitakis NG, Goodman NJ, Levy BA. Clinicopathologic characterization of oral angioleiomyomas. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:221–7. 6. Pathology and genetics of head and neck tumours.Barnes L, Eveson JW, Reichart P, Sidransky D, editors. World Health Organization Classification of Tumours.. Lyon: IARC Press; 2005. 7. Liang H, Frederiksen NL, Binnie WH, Cheng YS. Intraosseous oral leiomyoma: systematic review and report of one case. Dentomaxillofac Radiol 2003;32:285–90. 8. Bhattacharyya I, Summerlin DJ, Cohen DM, Ellis GL, Bavitz JB, Gillham LL. Granular cell leiomyoma of the oral cavity. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:353–9. 9. Jordan RC, Regezi JA. Oral spindle cell neoplasms: a review of 307 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2003;95:717–24.