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PubMed2003

[Chromosomal alterations in juvenile angiofibromas].

INTRODUCTION: Despite their benign histological appearance, juvenile angiofibromas, which occur mainly in adolescent males, have a locally aggressive growth pattern. beta-catenin-mutations represent their only known genetic abnormality. MATERIAL AND METHODS: Angiofibroma tissue from seven patients was available for comparative genomic hybridization (CGH). RESULTS: In six out of the seven angiofibromas, CGH detected various abnormalities on 18 different chromosomes. Frequent chromosomal gains were observed on chromosomes 4q, 6q, and 8q. In four out of seven angiofibromas a complete loss of the chromosome Y was detected. CONCLUSIONS: CGH is a suitable method for the examination of angiofibromas for genetic alterations. Considering the sex distribution of this neoplasm, the frequent loss of chromosome Y is of particular interest.

Adolescent↗
PubMed2005

[Long-term follow up of angiofibromas treated with CO2 laser in 23 patients with tuberous sclerosis].

INTRODUCTION: Tuberous sclerosis is an autosomal dominant disease in which hamartomas form in multiple organs. Cutaneous changes are one of the primary characteristics of this disease. These include angiofibromas (AF), a common form of presentation that causes significant cosmetic and medical problems. The CO2 laser has been used satisfactorily in treating these lesions, but there are few studies that evaluate its long-term results. The aim of our study is to assess the long-term response of the treatment of angiofibromas. METHODS: A retrospective study was carried out on 23 patients with angiofibromas treated with CO2 laser. The patients were treated between 1991 and 2000, inclusive, with continuous or superpulsed CO2 laser. We classified the angiofibromas by size, initial treatment results and patients' ages (< 20 years and 20 years or older). RESULTS: Ages ranged from 12 to 39 years, with a median age of 22.5 years. After treatment, patients were followed up for a period of six months to 10 years. In the long-term analysis, we found that 30.1 % maintained the initial result, and 60.9 % showed different degrees of recurrence, with a mean recurrence time of 3 years. When we analyzed the long-term results by the size of the angiofibromas, initial result and patients' ages, we found no statistically significant differences among the different groups. The survival analysis of the age groups, with Kaplan-Meier curves, showed that the youngest patients (< 20 years) had earlier recurrences than the older ones (logarithmic range 4.01 and p < 0.05). CONCLUSIONS: CO2 laser treatment achieves good short-term results. On the other hand, one of the biggest problems is recurrence over the long term; this is probably due to the fact that, because of their nature, these lesions cannot be eliminated permanently. This work coincides with earlier studies which found no factors that would make it possible to predict the recurrence of the lesions. However, we can conclude that recurrence takes place at a later date in older patients, and therefore they have better cosmetic results over the long term.

Adolescent↗
PubMed2005

Extranasopharyngeal angiofibroma of the cheek.

Angiofibromas rarely localize in extranasopharyngeal sites. The most common site for extranasopharyngeal angiofibromas is the maxillary sinus. The ethmoid and sphenoid sinuses, nasal septum, middle and inferior turbinates, conjunctiva, molar and retromolar region, and larynx are other sites where extranasopharyngeal angiofibromas have been reported. Only one case of buccal extranasopharyngeal angiofibroma has been reported to date. We present a case of buccal extranasopharyngeal angiofibroma that was excised completely following embolization and we also review the literature.

Adolescent↗
PubMed2007

Recurrent and residual juvenile angiofibromas.

INTRODUCTION: In the surgical management of juvenile nasopharyngeal angiofibromas the possibility of recurrences and residual tumours is always there. This study was undertaken to predict the prognostic factors determining recurrence of juvenile nasopharyngeal angiofibroma and to find out the usual sites of these tumours. MATERIAL AND METHODS: The medical records of 95 patients with histologically proven juvenile nasopharyngeal angiofibroma were reviewed retrospectively. The commonest surgical approach used was a combined transpalatal and transmaxillary approach with a lazy S incision. A conservative lateral infratemporal approach was used in three cases. RESULTS: Complete removal of the juvenile nasopharyngeal angiofibroma was achieved in 78 (82 per cent) of the cases in a single operation. A residual tumour was found in 17 (18 per cent) cases and recurrences occurred in 13 (13.7 per cent) cases. CONCLUSIONS: Extensions into the pterygoid fossa and basisphenoid, erosion of the clivus, intracranial extensions medial to the cavernous sinus, invasion of the sphenoid diploe through a widened pterygoid canal, feeders from the internal carotid artery, a young age and a residual tumour were risk factors found associated with recurrence of juvenile nasopharyngeal angiofibroma.

Angiofibroma↗
PubMedDate not supplied

Endoscopic and KTP laser-assisted surgery for juvenile nasopharyngeal angiofibroma.

Juvenile nasopharyngeal angiofibroma is a highly vascular tumor arising from the area around the sphenopalatine foramen. Various radical and extended radical surgeries have been advocated to surgically excise both extranasopharyngeal and nasopharyngeal juvenile angiofibromas. However angiofibromas involving the nasopharynx, nose, and sphenoid with minimal lateral extension via the sphenopalatine foramen can also be adequately managed endoscopically either alone or with 1 of the traditional approaches. Nine cases of juvenile nasopharyngeal angiofibroma were successfully managed between January, 1999, and March, 2001, by preoperative selective embolization of the internal maxillary artery with or without external carotid artery clamping, followed by endoscopic excision. Two of the 9 cases underwent KTP/532 laser-assisted endoscopic excision, whereas the transpalatal approach was used along with the endoscope in another 2 cases. The patients remained free of disease after a median follow-up period of 17 months. We report our preliminary experience in endoscopic and KTP laser-assisted excision of juvenile nasopharyngeal angiofibroma.

Adolescent↗
PubMed2004

[Extranasopharyngeal angiofibroma of the nasal cavity and paranasal sinuses].

BACKGROUND: Angiofibromas commonly arise in the nasopharynx in young male patients. Diagnosis is widely based on radiographic imaging to avoid ill-advised biopsy which may result in brisk bleeding. This study was undertaken to evaluate the incidence, clinical features and complications that may occur during the process of diagnosis and surgical therapy of angiofibromas outside the nasopharynx. METHODS AND PATIENTS: Case report of a 13-year-old female patient and review of the literature. RESULTS: Our patient received multi-agent chemotherapy elsewhere due to a misdiagnosed angiofibroma. Computed Tomography (CT) revealed a maxillary tumor which was repeatedly biopsied. Hypervascularity was excluded by arteriography and the lesion removed after lateral rhinotomy. The data of 42 patients were analyzed including our own case. 32 patients were male, 10 female. The majority became symptomatic aged 19 years or younger (71.4 %). The maxilla was most commonly affected (38 %), less frequently the ethmoid, nasal cavity or septum, beside others. In 38 patients, symptoms developed within 12 months or less (average: 8.5 months). Epistaxis, nasal obstruction and facial swelling were reported for most patients. Brisk bleeding occurred in 10 patients during tumor removal and resulted from biopsies in 11 of 20 patients. Angiography detected hypervascularity in 3 of 4 patients. There was no case with lethal outcome. CONCLUSION: Extranasopharyngeal angiofibromas of the nasal cavity or paranasal sinuses should be included in the differential diagnosis of nasal tumors. Compared to nasopharyngeal angiofibromas, more female patients are involved, symptoms develop more quickly but hypervascularity is less common. Signs of questionable hypervascularity in Computed Tomography and Magnetic Resonance Imaging (MRI) should indicate arteriography prior to surgical procedures. Preoperative embolization of hypervascular lesions during arteriography will reduce the risk of brisk bleeding during biopsy or surgical tumor removal.

Adolescent↗
PubMed2006

Endoscopic surgery for juvenile nasopharyngeal angiofibroma: where are the limits?

PURPOSE OF REVIEW: This article will examine recent publications on the endoscopic management of juvenile nasopharyngeal angiofibromas. RECENT FINDINGS: The use of an endoscopic approach to resect small juvenile nasopharyngeal angiofibromas is supported by excellent results from a number of operative series published in recent years. Large juvenile nasopharyngeal angiofibromas continue to present a considerable surgical challenge, with most being resected by traditional open approaches. However, the results achieved after the endoscopic resection of large tumours have been reported for a small number of cases. This review focuses on the outcome of these cases, which would seem to compare favourably to open series, and the advances in instrumentation and techniques, that facilitate the endoscopic removal of large juvenile nasopharyngeal angiofibromas. SUMMARY: Improvements in preoperative assessment and preparation, operative technique, and instrumentation potentially enable the endoscopic removal of most juvenile nasopharyngeal angiofibromas.

Adolescent↗
PubMed1998

Extravulvar subcutaneous cellular angiofibroma.

Cellular angiofibroma is a rare distinctive mesenchymal neoplasm of the vulva or perineal region. We report here one unique extravulvar case. A 43-year-old woman presented with an asymptomatic tumor, 7 cm in diameter, located in the subcutaneous tissue of the chest below the left submammary sulcus. Histologically, the lesion was composed of uniform spindled stroma cells, numerous thick-walled vessels, and scarce mature adipocytes. An additional feature was the presence of prominent perivascular lymphoid aggregates. The stromal cells were positive for vimentin and negative for CD34 and muscle, epithelial, myoepithelial, or neural markers. Although nasopharyngeal angiofibroma or the group of acral angiofibromas have a concurrent heading, cellular angiofibroma should not be mistaken with them. Differential diagnosis of this distinctive tumor especially includes aggressive angiomyxoma, angiomyofibroblastoma, superficial angiomyxoma, vascular myxolipoma, and other tumors with spindle cells reminiscent of those in angiofibroma.

Adult↗
PubMed2004

[99mTc-MIBI imaging of nasopharyngeal angiofibroma].

OBJECTIVE: To explore a noninvasive, effective and preoperative method of diagnosis for nasopharyngeal angiofibroma. METHOD: The craniocervical examination oncotropic by 99mTc-MIBI imaging was performed in 3 patients with nasopharyngeal angiofibroma. RESULT: Early 99mTc-MIBI imaging was weakly positive and late its imaging was radioactively defective in 3 cases of nasopharyngeal angiofibroma. 99mTc-MIBI imaging of the angiofibroma was significantly different from that of malignant and benign tumors. CONCLUSION: 99mTc-MIBI oncotropic imaging is of qualitatively diagnostic value for nasopharyngeal angiofibroma before operation, because 99mTc-MIBI imaging was of certain features.

Adolescent↗
PubMed1997

Multiple facial angiofibromas and collagenomas in patients with multiple endocrine neoplasia type 1.

OBJECTIVES: To evaluate patients with multiple endocrine neoplasia type 1 (MEN 1) for cutaneous manifestations. DESIGN: Survey during a 3-year period. SETTING: The National Institutes of Health, a tertiary referral research hospital in Bethesda Md. PATIENTS: A consecutive sample of 32 individuals with previously diagnosed MEN1 who were not preselected for the presence of skin lesions were examined for cutaneous abnormalities. None of the patients or family members were diagnosed as having tuberous sclerosis. INTERVENTIONS: Lesions were identified by clinical appearance, photographed, and confirmed histologically. MAIN OUTCOME MEASURE: To determine the frequency of skin lesions in patients with MEN1. RESULTS: Multiple facial angiofibromas were observed in 28 (88%) of the patients with MEN1, with 16 patients (50%) having 5 or more. Angiofibromas were clinically and histologically identical to those in individuals with tuberous sclerosis. Collagenomas were observed in 23 patients (72%). Also observed were cafe au lait macules in 12 patients (38%), lipomas in 11 patients (34%), confetti-like hypopigmented macules in 2 patients (6%), and multiple gingival papules in 2 patients (6%). CONCLUSIONS: Multiple angiofibromas, collagenomas, lipomas, confetti-like hypopigmented macules and multiple gingival papules are cutaneous manifestations of MEN1 and should be looked for in both family members of patients with MEN1 and individuals with hyperparathyroidism of other MEN1-associated tumors. Multiple angiofibromas can no longer be considered pathognomonic for tuberous sclerosis. The observation of angiofibromas in individuals without tuberous sclerosis necessitates further biochemical testing for MEN1.

Adolescent↗
PubMed1991

The size of small polydisperse circular DNA (spcDNA) in angiofibroma-derived cell cultures from patients with tuberous sclerosis (TSC) differs from that in fibroblasts.

Cell cultures were derived from angiofibromas of three patients with tuberous sclerosis (TSC), from the unaffected skin of these patients, and from the skin of five healthy donors. The length distributions of the small polydisperse circular DNA (spcDNA) fraction of these cell cultures were then analyzed. Nearly half the spcDNA molecules from the angiofibroma cultures were longer than 0.4 micron, whereas only about 7% exceeded this threshold in the spcDNA preparations from the skin fibroblast cultures. The percentage of the larger size class of spcDNA showed an increase at higher numbers of in vitro passages in all three types of cultures, but this effect was much more conspicuous in the angiofibroma-derived cultures than in those from the skin fibroblasts. An age-dependent increase in the overall amount of spcDNA was only seen in the angiofibroma-derived cultures. Our earlier finding of elevated amounts of spcDNA in angiofibroma cultures was confirmed in cultures from an additional TSC patient.

Adult↗
PubMed1996

Preoperative embolization of paragangliomas and angiofibromas. Measurement of intratumoral arteriovenous shunts.

OBJECTIVE: To determine if technetium Tc 99m-labeled macroaggregated albumin selective angioscintigraphy enables estimation of microparticle-arteriovenous shunt in branchial paragangliomas and nasopharyngeal angiofibromas. DESIGN: Technetium Tc 99m-labeled macroaggregated albumin particles with calibrated diameters between 25 and 50 microns were administered during selective catheterization of the feeding arteries in 4 branchial paragangliomas (glomus tumors) and 4 nasopharyngeal angiofibromas. Preliminary measures of the activity of the tumor region and of the lungs were obtained in the angiography suite using a hand-held detector. Without changing the position of the microcatheter, definitive embolization was performed. After endovascular treatment, scintigraphy was performed and the pulmonary shunt index was calculated with the use of a gamma camera and a dedicated computer program. RESULTS: The pulmonary shunt index varied between 0% and 36% for branchial paragangliomas (glomus tumors) and between 82% and 95% for angiofibromas. CONCLUSIONS: Our results provide new evidence that the incidence of microparticle-arteriovenous shunt in nasopharyngeal angiofibromas is much higher than in paragangliomas.

Adolescent↗
PubMed1993

Nasopharyngeal angiofibroma in patients with familial adenomatous polyposis.

Four patients with nasopharyngeal angiofibroma and familial adenomatous polyposis are reported here. Nasopharyngeal angiofibroma was 25 times more frequent in our patient population with familial adenomatous polyposis than in an age-matched hospital population. The association of these two rare conditions suggests that nasopharyngeal angiofibroma is an extracolonic manifestation of adenomatous polyposis. In addition, somatic mutation of the adenomatous polyposis coli gene, which causes adenomatous polyposis when mutated in the germline, could play a role in the pathogenesis of sporadic nasopharyngeal angiofibroma.

Adenomatous Polyposis Coli↗
PubMed2005

A case of angiofibroma originating from the inferior nasal turbinate.

Angiofibromas are nasopharyngeal tumors mostly seen in adolescent males. They account for less than 0.5% of all head and neck neoplasms. Extranasopharyngeal angiofibromas, although rare, have been reported. We report a case of extranasopharyngeal angiofibroma arising from the inferior turbinate of a 62-year-old male. The tumor was excised using the Denker approach. The patient is currently free of relapse 10 months after surgery. Previous extranasopharyngeal angiofibromas research was reviewed.

Adult↗
PubMed2001

Combined hamartoma of the retina and retinal pigment epithelium associated with juvenile nasopharyngeal angiofibroma.

PURPOSE: To report a patient who had concomitant combined hamartoma of the retinal pigment epithelium and retina and juvenile nasopharyngeal angiofibroma. METHOD: Case report. RESULTS: An 18-year-old man presented with a juvenile nasopharyngeal angiofibroma and a combined hamartoma of the retina and retinal pigment epithelium in the ipsilateral eye. The diagnosis of juvenile nasopharyngeal angiofibroma was confirmed by histopathology, and the diagnosis of combined hamartoma was established by typical ophthalmoscopic and fluorescein angiographic findings. CONCLUSION: Juvenile nasopharyngeal angiofibroma may be associated with combined hamartoma of the retina and retinal pigment epithelium.

Adolescent↗
PubMed1999

Giant cell angiofibroma of the orbit and eyelid.

PURPOSE: To report the clinicopathologic features of a newly recognized tumor, giant cell angiofibroma. DESIGN: Observational case series. MAIN OUTCOME MEASURES: Clinical and histopathologic features of giant cell angiofibroma. METHODS: Light and electron microscopy and immunohistochemistry of five cases of giant cell angiofibroma. RESULTS: A total of five patients (4 women and 1 man) are described: two presented with a painless mass in the eyelid, two with a mass in the orbit, and one presented with a conjunctival lesion. All lesions were well demarcated with no capsule and were composed of blood vessels, a patternless spindle-shaped cell proliferation with a solid and pseudovascular appearance, and multinucleated giant cells. Both spindle-shaped and giant tumor cells were intensely positive for CD34 and vimentin. CONCLUSION: Giant cell angiofibroma resembles solitary fibrous tumor and giant cell fibroblastoma and should be considered in the differential diagnosis of spindle-cell tumors in the eyelid, orbit, and conjunctiva.

Aged↗
PubMed2001

Extranasopharyngeal angiofibroma arising from the nasal septum.

Extra nasopharyngeal origin of angiofibroma is very rare. The nasal septum is a very rare site of extra nasopharyngeal angiofibroma with only two cases reported in the medical literature. We report here a case of a vascular mass arising from the nasal septum of an 8 year old boy. Histopathology confirmed it to be a case of angiofibroma. A review is also made of the other reported cases of angiofibroma arising from the nasal cavity. The likely theory of origin of the tumor and the management is also discussed.

Angiofibroma↗
PubMedDate not supplied

Endoscopic resection of nasopharyngeal angiofibromas by combined transnasal and transoral routes.

PURPOSE: To determine the role of endoscopic surgery in decreasing intraoperative bleeding, morbidity, and hospitalization period of juvenile nasopharyngeal angiofibroma resection and to describe combined endoscopic transnasal and transoral approaches. PATIENTS AND METHODS: Twelve cases of juvenile nasopharyngeal angiofibroma diagnosed by endoscopic examination, computed tomography, and angiography were selected for endoscopic resection. Tumor staging ranged from stage I(A) to II(B). Ten patients underwent preoperative selective arterial embolization, and in 1 case selective arterial ligation was used. In general, the tumors were approached endoscopically through nasal and oral cavities with 0 degrees and 30 degrees 4-mm telescopes without any incision and no packing at their termination. RESULTS: The patients were followed by endoscopy and computed tomography. There was a dramatic decrease in intraoperative bleeding and postoperative morbidity. No early postoperative complications were seen. Two recurrences were observed in 12 patients up to a mean follow-up of 15 months. CONCLUSIONS: Minimal bleeding, decreased morbidity, and shorter hospitalization period were the main reasons that prompted us to use endoscopic technique for the removal of juvenile nasopharyngeal angiofibroma. Adding transoral endoscopic approach to the transnasal endoscopic approach provides 2-sided exposure and appreciate access to angiofibroma.

Adolescent↗