Journal of Dentistry, 1, 150-l 52

Juvenile angiofibroma Report of a case occurring within the mouth

D. J. Stewart, M.D.s., F.D.s., F.F.D.

F. V. O’Brien, M.B., B.D.s., F.D.S.

The Queen’s University of Belfast, Northern Ireland

ABSTRACT The unusual tumour, juvenile angiofibroma, is normally found in the nasopharyngeal region of pubertal males. Rarely, it has been recorded within the mouth. In this report, there is given an account of a fibroma of this type that occurred in the mouth of a boy aged 10 years in a close relation to the dental structures.

‘ bleeding fungating swelling ’ which was thought to be due to a dental infection. The patient was of average physique, his weight was 374 kg., and height 1.36 m. The growth was a rounded swell- ing, about the size of a grape, situated on the gum directly on top of the alveolar ridge and immed- iately distal to and slightly overlying the crown of the lower left first permanent molar (Fig. Z). It

INTRODUCTION JUVENILE angiofibroma or bleeding fibroma of puberty is a relatively rare progressive growth that normally is found in the nasopharyngeal region of adolescents (most authors say exclusively in males) where it gives rise to nasal obstruction and, because of its highly vascular component, frequent epistaxis. Rarely this tumour has been reported as having occurred in areas outside the nasopharynx including the mouth (Hora and Weller, 1961; and Whitlock, 1961, for example, have each described lesions that were situated in the cheek), and in conse- quence reference is made to it in at least two modern textbooks of oral pathology (A Text- book of Oral Pathology by Shafer, Hine, and Levy, 1963 ; Thoma’s Oral Pathology, 1970). An account is presented here of some of the clinical and histological features of a fibroma of this type that was found in the mouth of a young patient situated in close relation to the dental structures.

CASE REPORT A schoolboy aged 10 years was referred by his family doctor for treatment of an intra-oral

Fig. I.-Juvenile angiofibroma in a boy aged 10 years located on the gum just distal to the first permanent molar.

was very firm and mobile and its smooth surface was covered by mucous membrane which was broken at one point on its upper aspect by a small area of ulceration caused by trauma from the opposing maxillary molar. The patient was unsure of the time of its fist appearance, but he had been aware of it for at least the previous 2 weeks because of the obstruction that it caused when he was eating. No apparent involvement of the under- lying alveolar bone was noted on radiographs.

The patient was admitted 1 week later to the Royal Belfast Hospital for Sick Children and the tumour was removed by localized resection. Its excision was accompanied by a brisk haemorrhage

Stewart and O’Brien : Juvenile Angiofibroma 151

which was controlled by pressure. Healing sub- distinctive type found almost exclusively in the sequently was uneventful and the patient was kept nasal and nasopharyngeal regions of pubertal under review for the following 3 years; there was males. no recurrence.

Pathological features DISCUSSION The specimen consisted of a rounded mass of firm In view of the uncommon nature of the lesion consistency measuring 2.5 x 1.5 x 1.5 cm. Histo- it would seem worthwhile to discuss the patho- logical examination showed a circumscribed but logical features in more detail.

Fig. 2.-A cellular field in which the fibroblasts are Fig. 3.-A richly vascular zone, containing vessels arranged in the form of a true tumour. Numerous of varying calibre, presents an angiomatous compressed slit-like vascular channels are present; appearance. H. and E. ( x 54.) one of these is arrowed. H. and E. ( x 54.)

non-encapsulated tumour beneath the epithelium composed of stellate fibroblasts, connective-tissue stroma, and numerous blood-vessels. The fibro- blasts were arranged in parallel bands and inter- lacing bundles, in many instances compressing the vascular spaces (Fig. 2). The blood-vessels, which were very numerous, varied from capillary size to lacunar vessels of large calibre (Fig. 3). They were thin-walled and under low-power magnification appeared to have a simple endothelial lining; however, in places higher magnification (Fig. 4) showed proliferating angioblasts forming an incomplete second layer. Special stains failed to reveal smooth muscle or elastic tissue in the vessel walls. The surface of the lesion was ulcerated at one point and was replaced by a zone of granula- tion tissue. Away from this superficial zone there was no evidence of inflammatory-celf infiltration. Calcification was not present. Elsewhere the tumour extended deep to the epithelium. Although the histological picture was suggestive of a true fibroma the vascularity was much greater than that usually seen in such turnours, and in areas was identical to that seen in the angiofibromata of the

Fig. 4.-An incomplete double layer of cells in a large vessel shown at top of picture. H. and E. (x 270.)

The arrangement of the cellular fibrous tissue and blood-vessels contrasts with that seen in the common fibrous epulis, where the picture varies from vascular oedematous fibrous tissue to a relatively avascular mass of mature collagen fibres. The alinement of well- formed fibroblasts and fibrocytes in parallel

152 Journal of Dentistry, Vol. 1 /No. 4

bands in the present specimen is characteristic of true fibromata. A smooth muscle origin for the cells could not be supported by special stains. The degree of vascularity prompted consideration of the diagnosis of haemangio- pericytoma but the cell type and arrangement around the blood channels did not support this.

The basic character of nasopharyngeal angiofibroma is still unsettled. Osborn (1959), in a review which embraces suggested origins of the growth, noted that while the majority of earlier writers had accepted these lesions as true neoplasms more recently there had been a move away from this belief. Willis (1953) suggested that the growths were inflammatory or allergic, and Evans (1966) likewise cast doubt on their neoplastic nature. Osborn (1959), who was impressed by the similarity of the vascular component to that of nasal erectile tissue, regarded the lesion as a mal- formation of this vascular tissue incited by hormonal changes of puberty in the male.

Hgrmii (1959), in a comprehensive review, regarded the lesion as a form of reactive hyper- plasia and cited evidence to support the possi- bility of spontaneous cure. This author found further evidence for his belief in the histo- pathological features when he compared them

to other hyperplastic tissue reactions of fibro- angiomatous pattern in which he included the granuloma pyogenicum. The findings in the present case do little to elucidate further its nature and origin.

Acknowledgements The authors have pleasure in acknowledging the helpful interest shown in this case by many pathologist colleagues, and, in particular, the advice received from Professor J. H. Jones of the Turner Dental School, Manchester.

REFERENCES EVANS, R. W. (1966), Histological Appearance of

Turnours, 2nd ed. Edinburgh: Livingstone. H;~RMK, A. R. (1959), Acta oto-lar., suppl., 146. HORA, J. F., and WELLER, W. A. (1961), Ann. Otol.

Rhinol. Lar., 70, 164. OSBORN, D. A. (1959), J. Lar. Otol., 73,295. SHAFER, W. G., HINE, M. K., and LEVY, B. M.

(1963), A Textbook of Oral Pathology. 2nd ed. London : Saunders.

Thoma’s Oral Pathology (1970), edited by GORLIN, R. J., and GOLDMAN, H. M., 6th ed. St. Louis: Mosby.

WHITLOCK, R. I. H. (1961), Br. dent. J., 111,372. WILLIS, R. A. (1953), Pathology of Turnours, 2nd

ed. London : Butterworths.