Juvenile angiofibroma of the maxillary sinus
Motoo Kitano 1, Gabriel Landini 1, Tamotsu M i m u r a 2 1Department of Oral Pathology, 22nd Department of Oral and Maxillofacial Surgery, Kagoshima University Dental School, Japan
A case report M. Kitano, G. Landini, T. Mimura: Juvenile angiofibroma o f the maxillary sinus. A case report. Int. J. Oral Maxillofac. Surg. 1992; 21." 230-232. Abstract. T h e clinicopathological features o f a rare case o f juvenile a n g i o f i b r o m a originating f r o m t h e maxillary sinus of a 13-year-old b o y are reported. This t u m o r was c o m p o s e d of a n g i o m a t o u s a n d fibrous structures. T h e analysis o f nucleolar organizer regions ( N O R ) o f the various c o m p o n e n t s o f this t u m o r indicated t h a t the f u n d a m e n t a l elements were the fibroblastic cells, r a t h e r t h a n the vascular endothelial cells.
Juvenile a n g i o f i b r o m a 3 is a n unusual, destructive b u t essentially b e n i g n tumor, mostly seen in the n a s o p h a r y n x o f adolescent males. Its occurrence within the maxillary sinus as a p r i m a r y t u m o r is rare 4'7. A l t h o u g h this t u m o r has distinct clinical a n d h i s t o p a t h o l o g i c a l features, there is some difference in o p i n i o n a b o u t its origin. BRUNNER2 a n d RINGERTZ ~° r e g a r d e d this lesion as a fibrous tumor. Some a u t h o r s 8'9 p r o p o s e d a vascular origin, owing to the vascularity of the tumor. R e s e a r c h carried o u t at the electron microscopical level i n d i c a t e d its fibrous origin 1,3. This r e p o r t describes a case of j u v e n ile a n g i o f i b r o m a arising in the maxilla as a n expansile a n d destructive sinus mass. O u r p u r p o s e is to describe the clinicopathological features a n d to discuss briefly the origin o f this rare tumor.
occurred during the surgical procedure. The dominating histopathological feature of the biopsy specimen was its high vascularity, and numerous vessels of various shapes and sizes were embedded in a fibrous tissue (Fig. 2). The vessels were mostly thin-walled, usually of no more than the endothelial cell thickness, and most endothelial cells appeared hypertrophied. Thrombi were occasionally present. Hemorrhagic loci, which tended to be associated with an aggregation of granulation tissue, were widely scattered. The collagenous tissue showed varying degrees of fiber density and cellularity. In some areas, it appeared edematous and myxomatous with an accumulation of acid mucopolysaccharides. Mitotic figures were rarely seen in either endothelial or fibroblastic cells, and atypical mitoses were never observed. The histopathological diagnosis was juvenile angiofibroma. Radiotherapy 11 was started after the biopsy. Partial necrosis with inhibition of growth of the tumor was noted after a total dose of 20 Gy (6°Co). Finally, partial maxil-
Case report A 13-year-old Japanese boy noticed a painless swelling of the palatal mucosa of the upper left premolar area while the neighboring teeth became loose. When he presented with a palatal swelling measuring 25 x 20 x 15 mm, slight tenderness and severe halitosis were noted. Neither ophthalmic nor nasal signs were observed. Radiographically, a radiopaque mass with unclear margins filled the lower two-thirds of the maxillary sinus, and the floor and lateral walls of the sinus were resorbed (Fig. 1). Scintigrams with 6VGa and 99Tc revealed an aggregation of isotopes in only the left maxillary sinus. There were no other abnormal findings on physical and laboratory examinations. Biopsy was carried out via the anterior wall of the maxillary sinus. Severe bleeding
Key words: juvenile angiofibroma; maxillary sinus; nucleolar organizer regions (NOR)
Accepted for publication 15 April 1992
lectomy was carried out 1 month after admission. Histopathological features of the operation specimen were similar to those of the biopsy specimen. The postoperative course was satisfactory and approximately 3 years after the operation, the patient is free from local recurrence or metastasis. Five micron sections from formalin-fixed paraffin blocks of the biopsy and operation specimens were stained with a siliver nitrate colloidal solution using a previously described method for visualization of the nucleolar organizer regions (NOR) 5. More than 100 cells from each of the fibrous areas, vascular endothelium and granulation tissue associated with hemorrhage were analyzed. The number of darkly impregnated granules, the NOR, in each nucleus was counted, adjusting the focus throughout the depth of the nuclei (Fig. 3) and subsequently the data was analyzed using a Student's t-test (Table 1). The arithmetic mean NOR count for the fibroblastic cells was higher than both the endothelial cells and the cells of the granulation tissue areas, with highly significant differences (p
Motoo Kitano 1, Gabriel Landini 1, Tamotsu M i m u r a 2 1Department of Oral Pathology, 22nd Department of Oral and Maxillofacial Surgery, Kagoshima University Dental School, Japan
A case report M. Kitano, G. Landini, T. Mimura: Juvenile angiofibroma o f the maxillary sinus. A case report. Int. J. Oral Maxillofac. Surg. 1992; 21." 230-232. Abstract. T h e clinicopathological features o f a rare case o f juvenile a n g i o f i b r o m a originating f r o m t h e maxillary sinus of a 13-year-old b o y are reported. This t u m o r was c o m p o s e d of a n g i o m a t o u s a n d fibrous structures. T h e analysis o f nucleolar organizer regions ( N O R ) o f the various c o m p o n e n t s o f this t u m o r indicated t h a t the f u n d a m e n t a l elements were the fibroblastic cells, r a t h e r t h a n the vascular endothelial cells.
Juvenile a n g i o f i b r o m a 3 is a n unusual, destructive b u t essentially b e n i g n tumor, mostly seen in the n a s o p h a r y n x o f adolescent males. Its occurrence within the maxillary sinus as a p r i m a r y t u m o r is rare 4'7. A l t h o u g h this t u m o r has distinct clinical a n d h i s t o p a t h o l o g i c a l features, there is some difference in o p i n i o n a b o u t its origin. BRUNNER2 a n d RINGERTZ ~° r e g a r d e d this lesion as a fibrous tumor. Some a u t h o r s 8'9 p r o p o s e d a vascular origin, owing to the vascularity of the tumor. R e s e a r c h carried o u t at the electron microscopical level i n d i c a t e d its fibrous origin 1,3. This r e p o r t describes a case of j u v e n ile a n g i o f i b r o m a arising in the maxilla as a n expansile a n d destructive sinus mass. O u r p u r p o s e is to describe the clinicopathological features a n d to discuss briefly the origin o f this rare tumor.
occurred during the surgical procedure. The dominating histopathological feature of the biopsy specimen was its high vascularity, and numerous vessels of various shapes and sizes were embedded in a fibrous tissue (Fig. 2). The vessels were mostly thin-walled, usually of no more than the endothelial cell thickness, and most endothelial cells appeared hypertrophied. Thrombi were occasionally present. Hemorrhagic loci, which tended to be associated with an aggregation of granulation tissue, were widely scattered. The collagenous tissue showed varying degrees of fiber density and cellularity. In some areas, it appeared edematous and myxomatous with an accumulation of acid mucopolysaccharides. Mitotic figures were rarely seen in either endothelial or fibroblastic cells, and atypical mitoses were never observed. The histopathological diagnosis was juvenile angiofibroma. Radiotherapy 11 was started after the biopsy. Partial necrosis with inhibition of growth of the tumor was noted after a total dose of 20 Gy (6°Co). Finally, partial maxil-
Case report A 13-year-old Japanese boy noticed a painless swelling of the palatal mucosa of the upper left premolar area while the neighboring teeth became loose. When he presented with a palatal swelling measuring 25 x 20 x 15 mm, slight tenderness and severe halitosis were noted. Neither ophthalmic nor nasal signs were observed. Radiographically, a radiopaque mass with unclear margins filled the lower two-thirds of the maxillary sinus, and the floor and lateral walls of the sinus were resorbed (Fig. 1). Scintigrams with 6VGa and 99Tc revealed an aggregation of isotopes in only the left maxillary sinus. There were no other abnormal findings on physical and laboratory examinations. Biopsy was carried out via the anterior wall of the maxillary sinus. Severe bleeding
Key words: juvenile angiofibroma; maxillary sinus; nucleolar organizer regions (NOR)
Accepted for publication 15 April 1992
lectomy was carried out 1 month after admission. Histopathological features of the operation specimen were similar to those of the biopsy specimen. The postoperative course was satisfactory and approximately 3 years after the operation, the patient is free from local recurrence or metastasis. Five micron sections from formalin-fixed paraffin blocks of the biopsy and operation specimens were stained with a siliver nitrate colloidal solution using a previously described method for visualization of the nucleolar organizer regions (NOR) 5. More than 100 cells from each of the fibrous areas, vascular endothelium and granulation tissue associated with hemorrhage were analyzed. The number of darkly impregnated granules, the NOR, in each nucleus was counted, adjusting the focus throughout the depth of the nuclei (Fig. 3) and subsequently the data was analyzed using a Student's t-test (Table 1). The arithmetic mean NOR count for the fibroblastic cells was higher than both the endothelial cells and the cells of the granulation tissue areas, with highly significant differences (p
