Paratesticular cellular angiofibroma: a case report.

INTRODUCTION: Paratesticular cellular angiofibroma is a rare benign mesenchymal tumor. The optimal management is surgical resection due to the difficulty of preoperative accurate diagnosis. CASE PRESENTATION: A 51-year-old Japanese male visited our hospital complaining of asymptomatic left scrotal swelling. Physical examination revealed a nontender elastic paratesticular mass (5.5 cm in diameter). Although testicular germ cell tumor was ruled out clinically, the possibility of malignant potential remained for the tumor. Since the patient consented to complete resection, a transinguinal radical orchiectomy was performed. The pathological diagnosis revealed cellular angiofibroma. The patient recovered without perioperative complications, and no apparent recurrence was observed at 5 years after surgery. CONCLUSION: The pathological findings were compatible for cellular angiofibroma. The tumor was successfully resected, and no apparent recurrence was observed at 5 years after surgery.

A Rare Case of Angiofibroma Presenting as an Endolymphatic Sac Tumor.

A 30-year-old man presented with minute-long episodes of vertigo and severe autophony. CVEMP showed a decreased threshold when testing the left side, potentially indicating SSCD. A subsequent MRI demonstrated a multi-lobulated, cystic mass in the temporal bone and the radiological diagnosis at that time was ELST. Tumor excision was performed, and microscopic examination of the excised material revealed fibrovascular tissue without signs of papillary or cystic projections. The conclusion of the histological assessment rendered a diagnosis of angiofibroma. We were unable to find a previous report of ENA originating around the endolymphatic sac. Laryngoscope, 134:1897-1900, 2024.

Preoperative Embolization of Primary Juvenile Nasopharyngeal Angiofibroma: Is Embolization of Internal Carotid Artery Branches Necessary?

PURPOSE: To determine the effects of blood supply from internal carotid artery (ICA) on the surgical outcomes of primary juvenile nasopharyngeal angiofibroma (JNA) after transarterial embolization (TAE). METHODS: A retrospective analysis was performed on primary JNA patients who underwent TAE and endoscopic resection in our hospital between December 2020 and June 2022. The angiography images of these patients were reviewed, and then they were divided into ICA + external carotid artery (ECA) feeding group and ECA feeding group according to whether the ICA branches were part of the feeding arteries. Tumors in ICA + ECA feeding group were fed by both ICA and ECA branches, while tumors in ECA feeding group were fed by ECA branches alone. All patients underwent tumor resection immediately after ECA feeding branches embolization. None of the patients underwent ICA feeding branches embolization. Data on demographics, tumor characteristics, blood loss, adverse events, residual and recurrence were collected, and case-control analysis was performed for the two groups. Differences in characteristics between the groups were tested using Fisher's exact and Wilcoxon tests. RESULTS: Eighteen patients were included in this study: nine in ICA + ECA feeding group and nine in ECA feeding group. The median blood loss was 700 mL (IQR 550-1000 mL) in ICA + ECA feeding group versus 300 mL (IQR 200-1000 mL) in ECA feeding group, with no significant statistical difference (P = 0.306). Residual tumor was found in one patient (11.1%) in both groups. Recurrence was not observed in any patient. There were no adverse events from embolization and resection in either group. CONCLUSION: The results of this small series suggest that the presence of blood supply from ICA branches in primary JNA has no significant effect on intraoperative blood loss, adverse event, residual and postoperative recurrence. Therefore, we do not recommend routine preoperative embolization of ICA branches. LEVEL OF EVIDENCE: Level 4, Case-control.

Combined endoscopic endonasal and sublabial transmaxillary approaches for resection of intracranially extended juvenile nasopharyngeal angiofibroma.

BACKGROUND: Juvenile nasopharyngeal angiofibromas (JNAs) are rare, benign, and locally invasive nasopharyngeal tumors. Endoscopic endonasal resection is effective, non-invasive, with low complication rates. Until recently, endoscopic resection was not suitable for intracranially invasive tumors. PURPOSE/METHOD: We describe resection steps of an intracranially extending JNA with a combined endoscopic endonasal and endoscopic-assisted sublabial transmaxillary approaches. Indications, advantages, and approach-specific complications are also discussed. The main surgical steps are shown in an operative video. CONCLUSION: Surgical excision of JNAs by a combined endoscopic endonasal and sublabial transmaxillary approaches represents a safe and effective treatment for selected intracranially invasive JNA.

Anatomic Considerations Guiding Single Versus Multiportal Endoscopic Approaches for Resection of Juvenile Nasopharyngeal Angiofibroma: Cases Series With Graded Multicorridor Resections.

BACKGROUND: Juvenile nasopharyngeal angiofibromas (JNAs) are characterized by expansive and destructive growth, often invading the midline/paranasal sinuses, pterygopalatine fossa, and infratemporal fossa and can extend into the orbit, cavernous sinus, or intracranially. OBJECTIVE: To evaluete the major benefits of the extended endoscopic endonasal approach (EEA) for JNA resection as compared with more traditional and invasive transpalatal and transfacial approaches. When JNAs extend into lateral anatomic compartments, the optimal operative trajectory often requires additional approach strategies or surgical staging. METHODS: We retrospectively reviewed 8 cases of large JNAs arising in symptomatic adolescent boys (University of Pittsburgh Medical Center Stages II, III, and V) and discuss anatomic and tumor considerations guiding the decision of a pure EEA vs combined EEA and sublabial transmaxillary approach (Caldwell-Luc). RESULTS: A pure extended EEA was used in 6 JNA cases (UPMC Stages II-III); a multiportal EEA + Caldwell-Luc maxillotomy was used in 2 cases. One of the 2 patients (UPMC Stage V) previously treated with multiportal EEA + Caldwell-Luc maxillotomy underwent staged left temporal/transzygomatic craniotomy, obtaining gross total resection. Seven patients ultimately underwent complete removal without recurrence. One patient with a small residual JNA (UPMC II) underwent stereotactic radiosurgery without progression to date. CONCLUSION: JNAs with lateral extension into the infratemporal fossa often benefited from additional lateral exposure using a Caldwell-Luc maxillotomy. Cases with significant skull base and/or dural involvement may undergo staged surgical treatment; temporalis + transzygomatic craniotomy is often useful for second-stage approaches for residual tumor in these lateral infratemporal or intracranial regions. SRS should be considered for residual tumor if additional surgery is not warranted.

MRI features of sinonasal tract angiofibroma/juvenile nasopharyngeal angiofibroma: Case series and systematic review.

BACKGROUND AND PURPOSE: To comprehensively summarize the radiological characteristics of sinonasal tract angiofibroma (STA) (commonly known as juvenile nasopharyngeal angiofibroma). METHODS: Forty-four lesions from 41 cases provided by 33 study articles identified through a systematic review and 13 lesions from 13 cases from our institution associated with patients with STA who underwent MRI were included in the review study, carried out by two board-certified experienced radiologists. RESULTS: The study participants were all male patients with a mean age of 15.6 years at the time of diagnosis. All of them presented with nasal cavity lesions (100%), predominantly in the nasopharynx (98.2%). The sphenopalatine foramen/pterygopalatine fossa was involved in 76.0%, and compressive shift of the posterolateral wall of the maxillary sinus was present in more than half (57.9%). T2-weighted imaging signal intensity was heterogeneous with mixed high and iso intensities as compared to skeletal muscle (100%). T1-weighted imaging showed partial high signal intensity in 61.1% of the cases. Flow void and intense enhancement were present in almost all cases. Cystic/nonenhancement changes on contrast-enhanced MRI were relatively common (40.8%). The mean apparent diffusion coefficient value (2.07 × 10-3  mm2 /second) and some quantitative dynamic contrast-enhanced MRI parameters were high. There was a significant difference in the frequency of residual/recurrent lesions based on the presence of MRI findings of skull base invasion (p = .017) and intracranial extension (p = .003). CONCLUSIONS: We summarized the MRI findings of STA that can facilitate timely diagnosis and appropriate management.

Bulky Recurrent Juvenile Nasopharyngeal Angiofibroma.

Juvenile nasopharyngeal angiofibroma is a rare and highly vascularized tumor that accounts for 0.05 to 0.5% of all head and neck neoplasms. The aim of this work was to present a case of a large recurrent juvenile nasopharyngeal angiofibroma coexisting with a facial lipoma in a 16-year-old boy. The patient was referred to our institution because of frequent unilateral epistaxis. Computed tomography revealed a hypervascular tumor with ethmoidal cell destruction and spread to the nasopharynx. Operative treatment of nasal cavity tumors was carried out using a transpalatal approach. After 6 months, the recurrence of the angiofibroma was verified radiologically. Primary as well as secondary surgical procedures were assisted with an endoscopic procedure. Accurate preoperative assessment and staging are essential for choosing a surgical procedure. The primary treatment is surgical excision. Early diagnosis, accurate staging, adequate treatment, and regular postoperative follow-up are essential in the treatment of these lesions.

Preoperative Transarterial Embolization of Advanced Juvenile Nasopharyngeal Angiofibroma Using n-Butyl Cyanoacrylate: Case-Control Comparison with Microspheres.

PURPOSE: To evaluate the efficacy and safety of transarterial embolization (TAE) with n-butyl cyanoacrylate (nBCA) for juvenile nasopharyngeal angiofibroma (JNA). MATERIALS AND METHODS: A retrospective review was performed on patients with JNA who underwent TAE and endoscopic resection between 2020 and 2022. Patients embolized with nBCA were identified, and those embolized with microspheres were set as the control group. Data on demographics, symptoms, tumor characteristics, blood loss, adverse events, residual disease, and recurrence were collected, and case-control analysis was performed for the 2 groups. Differences in characteristics between the groups were tested using the Fisher exact and Wilcoxon tests. A generalized linear model (GLM) was used to analyze the univariate and multivariate influences on blood loss. RESULTS: Twenty patients were included in this study: 13 in the microsphere group and 7 in the nBCA group. The median blood loss was 400 mL (interquartile range [IQR], 200-520 mL) in the nBCA group and 1,000 mL (IQR, 500-1,000 mL) in the microsphere group (P = .028). The GLM confirmed lower blood loss in the nBCA group (relative risk, 0.58 [0.41-0.83]; P = .01). A residual tumor was found in 1 patient in each group (7.7% vs 14.3%; P = 1.000). Recurrence was not observed in any patient. None of the patients experienced adverse events during embolization. CONCLUSIONS: TAE of advanced JNA with nBCA glue is safe and effective and can significantly reduce intraoperative blood loss compared with microspheres.

Nasal Septal Angiofibroma.

Juvenile nasopharyngeal angiofibroma is a benign vascular tumor seen predominantly in adolescent males in the second decade of life. Extranasopharyngeal angiofibroma includes vascular fibrous masses that occur outside the nasopharynx. The diagnosis of an angiofibroma is based on the clinical presentation and imaging, with biopsies being avoided to avoid excessive bleeding. Computed tomography scan is considered sufficient for the diagnosis of extranasopharyngeal angiofibroma as it clearly delineates and identifies the tumor.

Middle turbinate angiofibroma in an adolescent boy.

Bleeding nasal mass in adolescent boys has customarily been attributed to Juvenile nasopharyngeal angiofibroma. However, little is known regarding the extranasopharyngeal origin of angiofibroma, as highlighted in this case report of a 15-year-old boy who presented with recurrent epistaxis and nasal obstruction. On constructing a working diagnosis of nasal haemangioma, the patient was taken up for endoscopic excision under general anaesthesia. Intraoperative endoscopic findings and histopathological examination revealed the diagnosis of middle turbinate angiofibroma, which is an extremely rare extranasopharyngeal angiofibroma. Consequently, this represents the third described case of a juvenile angiofibroma arising from the middle turbinate.

Incidental finding of juvenile angiofibroma from pre-orthodontic radiographs: two case reports and a literature review.

This case series presents two asymptomatic cases of juvenile angiofibroma which were initially incidentally identified in pre-orthodontic radiographs. Juvenile angiofibroma is an uncommon, locally aggressive benign, vascular neoplasm with invasive growth patterns. Due to the hypervascularity of these tumours, there are biopsy associated risks and multi-slice computed tomography, magnetic resonance imaging and angiography are usually employed for diagnosis. Early pre-symptomatic identification of this lesion facilitates early management and limiting potential life-threatening complications. This highlights the importance of thorough interpretation of dental radiographs, including the evaluation of structures which are not in the primary region of interest. © 2022 Australian Dental Association.

Negative 68Ga-Prostate-Specific Membrane Antigen PET/CT Scan on a Recurrent Juvenile Nasopharyngeal Angiofibroma.

ABSTRACT: We present the case of a left-sided recurrent juvenile nasopharyngeal angiofibroma (JNA) in a 16-year-old boy who underwent endoscopic excision 2 years back. The preoperative contrast-enhanced MRI and histopathology were in favor of JNA. The follow-up 68Ga-prostate-specific membrane antigen (PSMA) PET/CT scan at 3-month interval revealed complete clearance of disease. Currently, the patient presented with recurrent bleeding and nasal endoscopic examination, and contrast-enhanced MRI revealed a presence of a recurrent tumor. The tumor did not show any uptake on 68Ga-PSMA PET/CT, and postoperative histopathology evaluation confirmed the presence of JNA. Although regarded as a criterion-standard imaging modality for assessment of recurrent or residual JNA, 68Ga-PSMA PET/CT is never 100% specific.

Angiofibroma of Soft Tissue on MRI and FDG PET/CT Image.

ABSTRACT: Angiofibroma of soft tissue is a rare benign mesenchymal neoplasm. We reported a case of angiofibroma of soft tissue in a 61-year-old woman. MRI examination indicated a soft tissue mass in the right lower thigh, which showed isosignal intensity on T1-weighted imaging and heterogeneous high signal intensity on T2-weighted imaging. This lesion had intense tracer uptake, with an SUVmax of 7.24 on FDG PET/CT. Our case revealed that when soft tissue masses with intense FDG uptake on PET/CT are encountered, angiofibroma of soft tissue should be considered in the differential diagnosis.

Cellular angiofibroma of orbit- a rare tumor at the rarest location.

Cellular angiofibroma (CAF) is a rare, site-specific benign tumor of mesenchymal origin, which is exclusive of the genital region except very few reported cases of extragenital CAF. Very recently the first case report of orbital CAF has been published and we are presenting the second documented case of CAF in this unusual location, in a 37-year-old man. Magnetic resonance imaging with T2 weighted image revealed a hyper-intense globular mass in the superomedial extraconal region of the left orbit. Histopathology of the excised mass showed numerous bland spindle cells embedded in wispy collagen, suggesting spindle cell tumor. On immunohistochemistry, the specimen was strongly positive to CD34 but negative to desmin, SMA, S-100, C-kit, ERG, EMG, CD31, and CD63, that further confirmed the diagnosis of CAF. CAF is a rare mesenchymal tumor and to the best of the authors' knowledge, this would be the second documented case of orbital CAF.