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Cellular angiofibroma of oral mucosa: report of two cases.
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MedLine Citation:
PMID:  19644547     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Cellular angiofibroma is a benign vascular neoplasm that typically arises in the vulva, perineal, and paratesticular region. Microscopically the lesions exhibit multiple small, non-dilated capillary channels, many of which contain erythrocytes. The endothelial lining cells are prominent, with monomorphic oval nuclei. Interposed among the vessels are both delicate and mature collagen fibers with fibroblastic hypercellularity that is variable in older lesions where sclerosis is prominent. The lesions usually do not recur following simple excision. Recent evidence indicates that cellular angiofibromas may be cytogenetically related to spindle cell lipoma. This represents the first reported instances of cellular angiofibroma in the oral cavity.
Authors:
Lewis R Eversole
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Publication Detail:
Type:  Case Reports; Journal Article     Date:  2009-05-31
Journal Detail:
Title:  Head and neck pathology     Volume:  3     ISSN:  1936-0568     ISO Abbreviation:  Head Neck Pathol     Publication Date:  2009 Jun 
Date Detail:
Created Date:  2010-07-02     Completed Date:  2010-10-05     Revised Date:  2013-05-23    
Medline Journal Info:
Nlm Unique ID:  101304010     Medline TA:  Head Neck Pathol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  136-9     Citation Subset:  IM    
Affiliation:
Oral Pathology Diagnostic Services, San Diego, CA 92111, USA. lrebge@aol.com
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MeSH Terms
Descriptor/Qualifier:
Adult
Aged
Angiofibroma / pathology*,  surgery
Female
Humans
Mouth Mucosa / pathology*
Mouth Neoplasms / pathology*,  surgery
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From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine

Full Text
Journal Information
Journal ID (nlm-ta): Head Neck Pathol
ISSN: 1936-055X
ISSN: 1936-0568
Publisher: Humana Press Inc, New York
Article Information
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© Humana 2009
Received Day: 10 Month: 1 Year: 2009
Accepted Day: 3 Month: 3 Year: 2009
Electronic publication date: Day: 31 Month: 5 Year: 2009
Print publication date: Month: 6 Year: 2009
Volume: 3 Issue: 2
First Page: 136 Last Page: 139
ID: 2715461
PubMed Id: 19644547
Publisher Id: 112
DOI: 10.1007/s12105-009-0112-7
issue-copyright-statement: © Humana 2009

Cellular Angiofibroma of Oral Mucosa: Report of Two Cases
Lewis R. EversoleAff1 Address: +800-055-4765 +928-282-8447 lrebge@aol.com
Oral Pathology Diagnostic Services, San Diego, CA 92111 USA

Introduction

Vascular and fibrous lesions are common in the head and neck area including both benign and malignant subtypes. Angiofibromas are typically encountered in the nasopharynx among teenage males, accounting for the appellation “juvenile nasopharyngeal angiofibroma”. Recently another fibrovascular lesion has been described in the genital/perineal region among females that is distinctly different from juvenile nasopharyngeal angiofibroma both histologically and behaviorally [18]. The term cellular angiofibroma has been applied to this lesion which has also been found in the paratesticular region among males and in skin [3, 912]. These benign fibrovascular tumors do not have a tendency to recur after simple excision. No instances of cellular angiofibroma have been reported in the head and neck area. Two instances are reported herein. One case appears to represent an early cellular stage while the second case is hypocellular with sclerosis.


Case Reports

Case 1: A 38 year old female presented with a submucosal nodule in the soft palate thought to represent a salivary neoplasm. The patient’s past medical history was non-contributory. The lesion was removed by surgical biopsy. The excised lesion measured 1.4 × 1.0 mm. Microscopically, surface stratified squamous epithelium was present with an underlying well demarcated and partially encapsulated mass of fibrovascular connective tissue (Fig. 1a, b). The fibrous tissue is cellular with adipocytes scattered in small clusters. Coursing throughout are prominent vascular channels with plump endothelial cells.

Case 2: A solitary submucosal mass was detected on the dorsal tongue of a 74 year old female. The lesion was stated to be painful, appearing as a dome shaped nodule with surface ulceration. The clinical impression was traumatic fibroma versus granular cell tumor. The lesion was excised and submitted for microscopic examination. The excised specimen measured 1 × 7 × 4 cm and on hemisection a well defined nodule was seen in the submucosa. Microscopically, a single lobule of fibrovascular tissue was well delineated and partially encapsulated. The endothelial element was found to be prominent with plump conspicuous endothelial cells. Adipocytes were present as isolated cells for small conglomerates. Unlike case 1, the fibrous element was hypocellular with mature sclerotic collagen fibers. (Fig. 1c, d).

Immunostaining demonstrated reactivity for factor VIII in endothelial cells (Fig. 1e) and CD 34 was found in both endothelial cells and fibroblasts (Fig. 1f). S-100 protein and smooth muscle actin were negative in the fibroblastic cell population (IHC/DAB).


Discussion

The microscopic differential diagnosis for CAF includes angiomyolipoma, angiomyofibroblastoma, juvenile nasopharyngeal angiofibroma, solitary fibrous tumor, spindle cell lipoma, and superficial angiomyxoma [1319]. All of these lesions are vessel rich with a fibroblastic or myofibroblastic element, variable sclerosis and harbor focal aggregates of adipocytes. Angiomyolipoma is a fatty tumor with prominent muscular arterioles while angiomyofibroblastoma is a fibrovascular neoplasm with a loose, somewhat myxoid fibroblastic element. Juvenile nasopharyngeal angiofibroma is uniquely confined to the nasopharynx and has a very distinct histology with a homogeneous fibroplasia and delicate immature collagen with interposed sinusoidal-like vascular channels that lack prominent endothelium. Solitary fibrous tumors, comprised of CD34 fibroblasts are typically hypercellular, fasiculated and do not exhibit prominent endothelial elements, although some variants do in fact manifest a hemangiopericytoma-like pattern. Angiomyxomas of deep tissues are aggressive neoplasms while superficial lesions are non-aggressive, may arise in the head and neck region as subcutaneous nodules comprised of myxoid tissue with prominent vascularity. These lesions may occur as a component of the Carney complex. Whereas lipocytes are commonly encountered in CAF, and were prominent in case 2 reported here, they are compartmentalized and not intimately interspersed with spindle cells as seen in spindle cell lipoma. CAF shows a spectrum of histologic patterns which may represent progressive stages of development with more highly vascular and fibrocellular lesions representing an early stage and less vascular more sclerotic lesions being older. CAFs are well demarcated or partially encapsulated, show hyperplastic vessels with prominent, plump endothelial cells, and fibroblastic hypercellularity. The fibrous element may also show mitotic activity with typical mitotic figures.

Differentiation from reactive lesions such as pyogenic granuloma or sclerosing pyogenic granuloma is usually not problematical because these reactive lesions are diffuse or multilobular, lacking the demarcation and encapsulation of cellular angiofibroma. Furthermore, CAF is not infiltrated by inflammatory cells unless superficially ulcerated yet the inflammation does not pervade the tumor stroma; rather leukocytes are zonal, in continuity with the area of ulceration. CAF represents a unique lesion, common in the gentital regions yet may occur in other sites including skin and the head and neck area. In the series reported by Iwasa and Fletcher [3], the fibroblastic component of CAF may be immunoreactive with antibodies to smooth muscle actin and/or CD34. In the two cases reported here, CD34, yet not SMA was positive in the fibroblasts. Genetic studies disclose a relationship to spindle cell lipoma [1721]. Simple excision is the treatment of choice.


References
1.. Nucci MR,Granter SR,Fletcher CD. Cellular angiofibroma: a benign neoplasm distinct from angiomyofibroblastoma and spindle cell lipomaAm J Surg Pathol 1997;21:636–644. [doi: 10.1097/00000478-199706000-00002]
1.. Nucci MR, Granter SR, Fletcher CD. Cellular angiofibroma: a benign neoplasm distinct from angiomyofibroblastoma and spindle cell lipoma. Am J Surg Pathol. 1997;21:636–44. doi:10.1097/00000478-199706000-00002. [pmid: 9199640]
2.. Nucci MR,Fletcher CD. Vulvovaginal soft tissue tumours: update and reviewHistopathology 2000;36:97–108. [doi: 10.1046/j.1365-2559.2000.00865.x]
2.. Nucci MR, Fletcher CD. Vulvovaginal soft tissue tumours: update and review. Histopathology. 2000;36:97–108. doi:10.1046/j.1365-2559.2000.00865.x. [pmid: 10672053]
3.. Iwasa Y,Fletcher CD. Cellular angiofibroma: clinicopathologic and immunohistochemical analysis of 51 casesAm J Surg Pathol 2004;28:1426–1435. [doi: 10.1097/01.pas.0000138002.46650.95]
3.. Iwasa Y, Fletcher CD. Cellular angiofibroma: clinicopathologic and immunohistochemical analysis of 51 cases. Am J Surg Pathol. 2004;28:1426–35. doi:10.1097/01.pas.0000138002.46650.95. [pmid: 15489646]
4.. Lane JE,Walker AN,Mullis EN Jr,Etheridge JG. Cellular angiofibroma of the vulvaGynecol Oncol 2001;81:326–329. [doi: 10.1006/gyno.2001.6167]
4.. Lane JE, Walker AN, Mullis EN Jr, Etheridge JG. Cellular angiofibroma of the vulva. Gynecol Oncol. 2001;81:326–9. doi:10.1006/gyno.2001.6167. [pmid: 11330972]
5.. Dargent JL,Saint Aubain N,Galdón MG,Valaeys V,Cornut P,Noël JC. Cellular angiofibroma of the vulva: a clinicopathological study of two cases with documentation of some unusual features and review of the literatureJ Cutan Pathol 2003;30:405–411. [doi: 10.1034/j.1600-0560.2003.00085.x]
5.. Dargent JL, de Saint Aubain N, Galdón MG, Valaeys V, Cornut P, Noël JC. Cellular angiofibroma of the vulva: a clinicopathological study of two cases with documentation of some unusual features and review of the literature. J Cutan Pathol. 2003;30:405–11. doi:10.1034/j.1600-0560.2003.00085.x. [pmid: 12834491]
6.. Dikmen Y,Yucebilgin MS,Kazandi M,Zekioglu O,Akalin T,Ozdemir N. Cellular angiofibroma of the vulva: report of a caseEur J Gynaecol Oncol 2004;25:242–244.
6.. Dikmen Y, Yucebilgin MS, Kazandi M, Zekioglu O, Akalin T, Ozdemir N. Cellular angiofibroma of the vulva: report of a case. Eur J Gynaecol Oncol. 2004;25:242–4. [pmid: 15032293]
7.. Kerkuta R,Kennedy CM,Benda JA,Galask RP. Vulvar cellular angiofibroma: a case reportAm J Obstet Gynecol 2005;193:1750–1752. [doi: 10.1016/j.ajog.2005.08.021]
7.. Kerkuta R, Kennedy CM, Benda JA, Galask RP. Vulvar cellular angiofibroma: a case report. Am J Obstet Gynecol. 2005;193:1750–2. doi:10.1016/j.ajog.2005.08.021. [pmid: 16260223]
8.. Nielsen GP,Young RH. Mesenchymal tumors and tumor-like lesions of the female genital tract: a selective review with emphasis on recently described entitiesInt J Gynecol Pathol 2001;20:105–127. [doi: 10.1097/00004347-200104000-00002]
8.. Nielsen GP, Young RH. Mesenchymal tumors and tumor-like lesions of the female genital tract: a selective review with emphasis on recently described entities. Int J Gynecol Pathol. 2001;20:105–27. doi:10.1097/00004347-200104000-00002. [pmid: 11293156]
9.. Samaratunga H,Fitzpatrick P. Cellular angiofibroma of the scrotumPathology 2008;40:330–333. [doi: 10.1080/00313020701716409]
9.. Samaratunga H, Fitzpatrick P. Cellular angiofibroma of the scrotum. Pathology. 2008;40:330–3. doi:10.1080/00313020701716409. [pmid: 18428064]
10.. Sabah M,Mohan P,Kay E. Para-testicular cellular angiofibroma: a rare tumour in a male renal transplant patientVirchows Arch 2006;449:489–499. [doi: 10.1007/s00428-006-0289-z]
10.. Sabah M, Mohan P, Kay E. Para-testicular cellular angiofibroma: a rare tumour in a male renal transplant patient. Virchows Arch. 2006;449:489–99. doi:10.1007/s00428-006-0289-z. [pmid: 16957933]
11.. Val-Bernal JF,Rubio S,Garijo MF,González-Vela MC. Extragenital subcutaneous cellular angiofibroma. Case reportAPMIS 2007;115:254–258. [doi: 10.1111/j.1600-0463.2007.apm_572.x]
11.. Val-Bernal JF, Rubio S, Garijo MF, González-Vela MC. Extragenital subcutaneous cellular angiofibroma. Case report. APMIS. 2007;115:254–8. [pmid: 17367472]
12.. Garijo MF,Val-Bernal JF. Extravulvar subcutaneous cellular angiofibromaJ Cutan Pathol 1998;25:327–332. [doi: 10.1111/j.1600-0560.1998.tb01754.x]
12.. Garijo MF, Val-Bernal JF. Extravulvar subcutaneous cellular angiofibroma. J Cutan Pathol. 1998;25:327–32. doi:10.1111/j.1600-0560.1998.tb01754.x. [pmid: 9694623]
13.. Canales BK,Weiland D,Hoffman N,Slaton J,Tran M,Manivel JC,Monga M. Angiomyofibroblastoma-like tumors (cellular angiofibroma)Int J Urol 2006;13:177–179. [doi: 10.1111/j.1442-2042.2006.01255.x]
13.. Canales BK, Weiland D, Hoffman N, Slaton J, Tran M, Manivel JC, et al. Angiomyofibroblastoma-like tumors (cellular angiofibroma). Int J Urol. 2006;13:177–9. doi:10.1111/j.1442-2042.2006.01255.x. [pmid: 16563146]
14.. McCluggage WG,Ganesan R,Hirschowitz L,Rollason TP. Cellular angiofibroma and related fibromatous lesions of the vulva: report of a series of cases with a morphological spectrum wider than previously describedHistopathology 2004;45:360–368. [doi: 10.1111/j.1365-2559.2004.01923.x]
14.. McCluggage WG, Ganesan R, Hirschowitz L, Rollason TP. Cellular angiofibroma and related fibromatous lesions of the vulva: report of a series of cases with a morphological spectrum wider than previously described. Histopathology. 2004;45:360–8. doi:10.1111/j.1365-2559.2004.01923.x. [pmid: 15469474]
15.. Dufau JP,Soulard R,Gros P. Cellular angiofibroma, angiomyofibroblastoma and aggressive angiomyxoma: members of a spectrum of genital stromal tumours?Ann Pathol 2002;22:241–243.
15.. Dufau JP, Soulard R, Gros P. Cellular angiofibroma, angiomyofibroblastoma and aggressive angiomyxoma: members of a spectrum of genital stromal tumours? Ann Pathol. 2002;22:241–3. [pmid: 12410114]
16.. McCluggage WG,Perenyei M,Irwin ST. Recurrent cellular angiofibroma of the vulvaJ Clin Pathol 2002;55:477–479.
16.. McCluggage WG, Perenyei M, Irwin ST. Recurrent cellular angiofibroma of the vulva. J Clin Pathol. 2002;55:477–9. [pmid: 12037036]
17.. Curry JL,Olejnik JL,Wojcik EM. Cellular angiofibroma of the vulva with DNA ploidy analysisInt J Gynecol Pathol 2001;20:200–203. [doi: 10.1097/00004347-200104000-00015]
17.. Curry JL, Olejnik JL, Wojcik EM. Cellular angiofibroma of the vulva with DNA ploidy analysis. Int J Gynecol Pathol. 2001;20:200–3. doi:10.1097/00004347-200104000-00015. [pmid: 11293169]
18.. Micheletti AM,Silva AC,Nascimento AG,Da Silva CS,Murta EF,Adad SJ. Cellular angiofibroma of the vulva: case report with clinicopathological and immunohistochemistry studySao Paulo Med J 2005;123:250–252. [doi: 10.1590/S1516-31802005000500010]
18.. Micheletti AM, Silva AC, Nascimento AG, Da Silva CS, Murta EF, Adad SJ. Cellular angiofibroma of the vulva: case report with clinicopathological and immunohistochemistry study. Sao Paulo Med J. 2005;123:250–2. doi:10.1590/S1516-31802005000500010. [pmid: 16358102]
19.. Fetsch JF,Laskin WB,Tavassoli FA. Superficial angiomyxoma (cutaneous myxoma): a clinicopathologic study of 17 cases arising in the genital regionInt J Gynecol Pathol 1997;16:325–334.
19.. Fetsch JF, Laskin WB, Tavassoli FA. Superficial angiomyxoma (cutaneous myxoma): a clinicopathologic study of 17 cases arising in the genital region. Int J Gynecol Pathol. 1997;16:325–34. doi:10.1097/00004347-199710000-00006. [pmid: 9421071]
20.. Hameed M,Clarke K,Amer HZ,Mahmet K,Aisner S. Cellular angiofibroma is genetically similar to spindle cell lipoma: a case reportCancer Genet Cytogenet 2007;177:131–134. [doi: 10.1016/j.cancergencyto.2007.05.016]
20.. Hameed M, Clarke K, Amer HZ, Mahmet K, Aisner S. Cellular angiofibroma is genetically similar to spindle cell lipoma: a case report. Cancer Genet Cytogenet. 2007;177:131–4. doi:10.1016/j.cancergencyto.2007.05.016. [pmid: 17854668]
21.. Maggiani F,Debiec-Rychter M,Vanbockrijck M,Sciot R. Cellular angiofibroma: another mesenchymal tumour with 13q14 involvement, suggesting a link with spindle cell lipoma and (extra)-mammary myofibroblastomaHistopathology 2007;51:410–412. [doi: 10.1111/j.1365-2559.2007.02775.x]
21.. Maggiani F, Debiec-Rychter M, Vanbockrijck M, Sciot R. Cellular angiofibroma: another mesenchymal tumour with 13q14 involvement, suggesting a link with spindle cell lipoma and (extra)-mammary myofibroblastoma. Histopathology. 2007;51:410–2. doi:10.1111/j.1365-2559.2007.02775.x. [pmid: 17727484]

Figures

[Figure ID: Fig1]
Fig. 1 

Case 1. a Submucosal well demarcated, partially encapsulated mass is comprised of fibrovascular tissue (H&E, 50×), b Numerous prominent, plump endothelial cells line vessels in the background of a bland hypercellular fibroblastic element (H&E, 200×). Case 2. c Demarcated mass comprised of multiple small prominent vessels, zones of fibroblastic cellularity with desmoplasia and pockets of adipocytes (H&E, 50×). d Higher magnification showing vascularity, sclerosis and adipocytes (H&E, 200×). e Factor VIII immunoreactivity in vessels (IHC/DAB, 200×). f CD34 immunoreactivity in both endothelial cells and fibroblasts (IHC/DAB, 200×)



Article Categories:
  • Case Report

Keywords: Keywords Mesenchymal tumor, Vascular tumor, Hemangioma, Oral cavity, Angiofibroma.

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