Abstract
Background
Juvenile nasopharyngeal angiofibroma (JNA) is a rare, benign but locally aggressive vascular tumor affecting adolescent males. While endoscopic resection with preoperative embolization remains the treatment of choice, recurrence remains a challenge, often driven by incomplete excision or poor follow-up adherence.
Case Presentation
We report two cases of recurrent JNA in adolescent males, each highlighting a different modifiable risk factor for recurrence. Case 1 involved a 21-year-old with prior resection and poor postoperative follow-up, who re-presented with Radkowski Stage IIIA disease involving the central skull base. Case 2 involved an 18-year-old initially misdiagnosed with a nasal polyp, later confirmed as JNA after recurrence within 4 months of surgery. Both patients underwent preoperative embolization followed by endoscopic endonasal resection using a transnasal–transpterygoid approach. Histopathology confirmed recurrent JNA in both cases. Postoperative MRI at 3 months showed no evidence of residual or recurrent disease.
Conclusion
These cases underscore the critical role of accurate initial diagnosis, proper staging, and structured postoperative surveillance in reducing recurrence risk in JNA. Multidisciplinary care, patient education, and imaging-guided follow-up protocols are essential, especially in resource-constrained settings where diagnostic delays and adherence challenges are prevalent.
Keywords: juvenile nasopharyngeal angiofibroma, endoscopic resection, recurrent JNA, case report
Introduction
Juvenile nasopharyngeal angiofibroma (JNA) is a rare, benign, yet locally aggressive vascular tumor that accounts for approximately 0.05% of all head and neck neoplasms, with a striking predilection for adolescent males between the ages of 10 and 20 years.1 It is important to distinguish JNA from other nasopharyngeal masses, most notably nasopharyngeal carcinoma. Whereas nasopharyngeal carcinoma typically arises in older adults, is frequently associated with Epstein–Barr virus, presents with cervical lymphadenopathy, and shows infiltrative margins with nodal and distant metastatic potential, JNA occurs almost exclusively in adolescent males, does not metastasise, and characteristically appears as an avidly enhancing, well-circumscribed mass centred on the sphenopalatine foramen, often with anterior bowing of the posterior maxillary wall (Holman–Miller sign).2 Though histologically benign, JNA exhibits invasive growth patterns, most commonly originating at the sphenopalatine foramen and extending into adjacent anatomical structures such as the pterygopalatine fossa, maxillary sinuses, and skull base.3
The hallmark clinical features of JNA include unilateral nasal obstruction, reported in approximately 80% to 90% of cases, and spontaneous, recurrent epistaxis, affecting up to 95% of patients.3 Depending on the extent of invasion, patients may also present with facial swelling, proptosis, or neurological deficits. Imaging modalities such as contrast-enhanced CT and MRI are crucial for diagnosis and surgical planning.3,4 Disease extent is most commonly classified using the Radkowski staging system, which stratifies tumours from Stage I (confined to the nasal cavity and nasopharynx, with IB denoting paranasal sinus extension), through Stage II (extension into the pterygopalatine/pterygomaxillary fossa and adjacent spaces), to Stage III (skull-base erosion and intracranial extension, subdivided into IIIA [minimal] and IIIB [extensive, including cavernous sinus involvement]). Higher stages correlate with greater surgical difficulty and recurrence risk.5
Surgical resection, particularly via the endoscopic approach, remains the mainstay of treatment and has shown favorable outcomes even in advanced stages.6 Preoperative embolization has become a standard adjunct to reduce intraoperative blood loss and facilitate more complete tumor removal.7 A defining feature of JNA is its rich vascular supply, derived predominantly from the internal maxillary artery, which renders the tumour prone to profuse intraoperative haemorrhage. This hypervascularity represents the principal surgical challenge and underlies the need for careful perioperative planning, including haemodynamic optimisation and assured availability of blood products.2,4,7 Nevertheless, tumor recurrence remains a significant challenge, with reported rates ranging from 15% to 50%, particularly in cases with intracranial extension, incomplete resection, or poor postoperative compliance.8 Recurrence most often reflects regrowth of residual tumour at sites that are difficult to access and clear endoscopically — particularly the basisphenoid and cancellous bone of the pterygoid base, the pterygopalatine and infratemporal fossae, and regions of cavernous sinus or skull-base extension.4,9 Incomplete resection at these locations, advanced stage at presentation, and inadequate postoperative surveillance are therefore the principal drivers of recurrence. While surgical techniques are well-studied, modifiable factors such as patient adherence to surveillance and interdisciplinary coordination are underreported in recurrence prevention.
We present two cases of recurrent JNA in adolescent males that illustrate two distinct, modifiable contributors to recurrence: one demonstrating how advanced-stage disease compounded by poor postoperative follow-up permitted undetected regrowth, and the other showing how diagnostic delay and initial misdiagnosis precipitated early recurrence. Through imaging, surgical, and histopathological correlations, we emphasize the necessity of structured surveillance protocols and patient engagement in mitigating recurrence risk.
Case Presentation
Case 1
A 21-year-old male presented to our institution in November 2024 with complaints of nasal obstruction, crusting, and infrequent episodes of epistaxis. He had a prior diagnosis of juvenile nasopharyngeal angiofibroma (JNA), which had been surgically treated at an external center at age 18.
The patient reported chronic nasal obstruction of uncertain onset. In the winter of 2020, he experienced a significant episode of epistaxis following forceful nose blowing, which necessitated nasal packing. Thereafter, moderate-volume, trauma-induced epistaxis occurred intermittently, particularly during wrestling activities. He denied facial swelling, visual disturbances, headaches, or neurological symptoms but noted diminished olfactory sensation.
In early 2022, another traumatic episode of epistaxis prompted a contrast-enhanced CT scan (not available for review) at an outside facility, which revealed a nasopharyngeal mass. He subsequently underwent external carotid artery ligation followed by endoscopic excision on April 27, 2022. Histopathology confirmed juvenile nasopharyngeal angiofibroma. However, no postoperative imaging was performed, and the patient failed to adhere to recommended follow-up visits and nasal care instructions.
Two years later, in November 2024, the patient re-presented. Physical examination was unremarkable, with no facial deformities or neurological deficits. A contrast-enhanced CT scan performed at our institution demonstrated a soft-tissue mass in the left nasopharyngeal region, extending into the pterygopalatine fossa as shown in Figure 1A and B. MRI revealed a mass filling the sphenoid sinus and involving the central skull base, with extension into the left masticator space and elevation of the sellar floor, without definite cavernous sinus invasion, consistent with Radkowski Stage IIIA JNA as shown in Figure 2. The short disease-free interval and lesion size raised concern for residual tumor, although this could not be definitively established due to the absence of prior postoperative imaging.
Figure 1.
Preoperative ct (bone window) of Case 1 demonstrating left pterygopalatine fossa involvement: (A) Axial ct image (bone window) at the level of the maxillary sinuses and nasopharynx. Soft tissue fills the left pterygopalatine fossa behind the posterior wall of the left maxillary sinus, with effacement of the normal retroantral fat plane, together with soft-tissue opacification of the left posterior nasal cavity. The contralateral (right) pterygopalatine region is preserved for comparison. (B) Coronal ct image (bone window) at the level of the posterior nasal cavity and pterygopalatine region. The normal fat-filled pterygopalatine fossa cleft is effaced and replaced by soft tissue on the left, in contrast to the preserved right side.
Figure 2.
Preoperative MRI of Case 1 demonstrating central skull-base extension (radkowski Stage IIIA): coronal non-contrast T1-weighted MR image at the sellar/parasellar level. A lobulated, intermediate-signal soft-tissue mass fills the sphenoid sinus and involves the central skull base, elevating the floor of the sella and extending inferiorly into the nasopharynx and laterally toward the left masticator space. No definite invasion of the cavernous sinus or middle cranial fossa is identified. Findings are consistent with Radkowski Stage IIIA disease.
Preoperative laboratory investigations revealed mild anemia (hemoglobin: 11.3 g/dL), with a normal coagulation profile. The patient underwent embolization followed by endoscopic endonasal resection, which lasted approximately 2 hours with an estimated blood loss of 400 mL. No intraoperative or postoperative complications occurred.
The resected specimen consisted of multiple firm, gray-brown tissue fragments measuring 8.5 × 6 × 2 cm as shown in Figure 3. Histopathological analysis revealed mature fibrous stroma with plump fibrocytes, hyalinized areas, numerous sinusoidal-type vessels (some containing mixed thrombi), focal stromal edema, and inclusions of destructive osteoid tissue, confirming juvenile nasopharyngeal angiofibroma as shown in Figure 4.
Figure 3.
The resected specimen consisting of multiple firm, gray-brown tissue fragments measuring 8.5 × 6 × 2 cm.
Figure 4.
Histopathology of patient 1 showing sinusoidal vessels and thrombi.
A postoperative MRI at 3 months demonstrated no evidence of residual or recurrent tumor as shown in Figure 5. The patient reported substantial symptomatic improvement, including restored nasal airflow, decreased crusting, and no recurrence of epistaxis. He remained adherent to follow-up and expressed satisfaction with the surgical outcome, reporting a significantly improved quality of life.
Figure 5.
Post-op MRI showing no recurrence.
Case 2
An 18-year-old male was referred to our institution in March 2025 with progressive nasal obstruction and dyspnea. His symptoms had begun three years earlier and had significantly worsened over the preceding two years. He was initially presumed to have a nasal polyp and underwent surgical resection in November 2024 at an outside facility. Postoperative histopathology confirmed juvenile nasopharyngeal angiofibroma (JNA), although no formal staging or follow-up imaging was conducted at that time.
Over the subsequent months, his symptoms persisted and progressed, prompting referral to our centre. Physical examination was unremarkable, and laboratory tests including complete blood count and coagulation profile were within normal limits. Nasal endoscopy demonstrated a vascular mass in the posterior nasal cavity.
MRI revealed a T2-hyperintense mass centred in the nasopharynx and left pterygopalatine fossa, containing multiple internal flow voids (“salt-and-pepper” appearance) characteristic of a hypervascular tumour, without evidence of skull-base erosion or intracranial extension, consistent with Radkowski Stage II JNA as shown in Figure 6A and B.
Figure 6.
(A and B) Preoperative MRI of Case 2 demonstrating a left nasopharyngeal/pterygopalatine mass without intracranial extension. (A) Coronal T2-weighted MR image (non-contrast) showing a T2-hyperintense soft-tissue mass centred in the nasopharynx and left pterygopalatine fossa, with multiple internal flow voids producing a “salt-and-pepper” appearance characteristic of a hypervascular tumour. No skull-base erosion or intracranial extension is present. (B) Axial T2-weighted MR image (non-contrast) at the level of the nasopharynx demonstrating the heterogeneous left nasopharyngeal and pterygopalatine/parapharyngeal mass with internal flow voids. The brainstem and posterior skull base are uninvolved, confirming the absence of intracranial extension.
The patient underwent preoperative embolization followed by endoscopic endonasal resection, as described in the Surgical Technique section. The operation lasted 2 hours with an estimated intraoperative blood loss of 350–400 mL and was completed without complications.
The resected tumor measured 7 × 4.5 × 2 cm as shown in Figure 7. Histological examination revealed fibrous connective tissue with sinusoidal vascular channels, stromal edema, hyalinized areas, and focal lymphoid aggregates, confirming recurrent JNA as shown in Figure 8.
Figure 7.
The resected tumor measuring 7 × 4.5 × 2 cm.
Figure 8.
Microscopy showing fibrous connective tissue with sinusoidal vessels, stromal edema, hyalinized areas, and focal lymphoid infiltrates.
Postoperatively, the patient experienced marked symptomatic improvement, particularly in nasal airflow. Follow-up MRI performed at 3 months showed no evidence of residual or recurrent tumor as shown in Figure 9. Details of both cases were summarized as shown in Table 1.
Figure 9.
Axial T2-weighted MRI at 3 months postoperatively demonstrating no evidence of residual or recurrent juvenile nasopharyngeal angiofibroma.
Table 1.
Clinical, Imaging, Surgical, and Outcome Comparison of Two Patients with Recurrent Juvenile Nasopharyngeal Angiofibroma
| Parameter | Patient 1 | Patient 2 |
|---|---|---|
| Age at recurrence | 21 | 18 |
| Symptom onset | ~3 years before recurrence | ~3 years before recurrence |
| Initial symptoms | Nasal obstruction, epistaxis | Nasal obstruction only |
| First surgery | April 2022 (external facility) | November 2024 (external facility) |
| Time to recurrence | ~2.5 years | ~4 months |
| Recurrence symptoms | Crusting, bleeding, nasal obstruction | Nasal clogging, dyspnea |
| Post-op compliance | Poor | Moderate |
| CT findings | Extension into pterygopalatine fossa | Maxillary & sphenoid sinus involvement |
| MRI findings | Radkowski Stage IIIA, central skull-base involvement | Radkowski Stage II, no intracranial extension |
| Surgery performed | Endoscopic + embolization | Endoscopic + embolization |
| Operative duration | 2 hours | 2 hours |
| Estimated blood loss | 400 mL | 350–400 mL |
| Histopathology | Vascular fibrous tissue with thrombi | Sinusoidal vessels, lymphoid infiltrates |
| Post-op follow-up | MRI: No recurrence at 3 months | MRI: No recurrence at 3 months |
| Outcome | Symptom-free, adherent | Symptom-free, adherent |
Surgical Technique
Both patients underwent endoscopic endonasal resection under general anesthesia following preoperative embolization of the tumor-feeding vessels. A binostril, four-handed technique was employed. The procedure began with a wide middle meatal antrostomy and posterior septectomy to optimize visualization and instrumentation. A transnasal–transpterygoid corridor was subsequently established through a medial maxillectomy, providing direct access to the pterygopalatine fossa and infratemporal extension of the tumor. Intraoperative image guidance was utilized throughout the procedure to maintain precise anatomical orientation and to define tumor boundaries. Early identification and control of the vascular pedicle were achieved by careful exposure and bipolar coagulation of feeding vessels within the pterygopalatine fossa, supplemented by topical hemostatic agents. Following devascularization, meticulous circumferential dissection was performed along the tumor capsule, separating it from surrounding structures, including the skull base and masticator space, while preserving adjacent critical neurovascular anatomy. Once fully mobilized, the tumor was delivered en bloc through the nasal cavity without intratumoral debulking or fragmentation. Final inspection confirmed complete macroscopic resection. Hemostasis was secured using bipolar cautery and oxidized regenerated cellulose. No intraoperative cerebrospinal fluid leak or major vascular injury occurred. Nasal packing was not required, and the nasal cavities were allowed to heal by secondary intention.
Discussion
Juvenile nasopharyngeal angiofibroma (JNA) management is complicated by its locally aggressive behavior and propensity for recurrence. Our cases underscore two critical, often interconnected challenges: diagnostic delays in low-resource settings and non-compliance with postoperative surveillance—factors disproportionately impacting adolescents in underserved regions.
Diagnostic Delays in Low-Resource Settings
Case 2 exemplifies systemic challenges pervasive in resource-constrained environments. Initial misdiagnosis as a nasal polyp and unstaged surgery reflect limited access to specialized expertise and advanced imaging. In many low- and middle-income countries (LMICs), MRI availability is markedly limited, forcing reliance on clinical suspicion alone.10 Furthermore, the cost of contrast-enhanced CT/MRI (often exceeding monthly household incomes) delays definitive diagnosis, as seen in our patient, who presented with advanced recurrence four months postoperatively. Cultural perceptions, such as stigmatization of chronic nasal symptoms or prioritization of acute care over preventive follow-up, further compound delays.11
Non-Compliance and Structural Barriers
Case 1’s loss to follow-up mirrors adherence gaps reported in LMICs, where postoperative follow-up in low-resource settings is frequently incomplete. Travel costs, lack of insurance, and competing socioeconomic priorities (eg, education, employment) disproportionately affect adolescent males, the primary JNA demographic. In rural regions, centralized specialist care necessitates long-distance travel, creating logistical and financial burdens that discourage compliance.
Interventions for Resource-Constrained Contexts
Beyond their individual clinical courses, both cases expose modifiable, system-level failure points that are amenable to targeted intervention in resource-constrained settings. Because the principal barriers—diagnostic delay and loss to follow-up—are logistical and financial rather than strictly technical, the most impactful measures are those that shorten the path to diagnosis and sustain surveillance without requiring repeated specialist visits. Telemedicine-based follow-up, which has shown feasibility for remote monitoring of head and neck cancer patients, could enable symptom triage and virtual review while reducing the travel and cost burdens that drove non-adherence in Case 1.12 Such efforts are most effective when paired with earlier case-finding: training community health workers to recognise sentinel symptoms such as unilateral nasal obstruction and recurrent epistaxis has improved early detection of paediatric tumours in comparable settings and could counter the kind of misclassification seen in Case 2.13 Complementary structural measures—subsidised or partnership-based access to contrast-enhanced CT and MRI, together with culturally tailored counselling that reframes follow-up as integral rather than optional—address the affordability and health-literacy gaps underlying both presentations. Most concretely, we advocate a structured post-resection surveillance protocol comprising MRI at 3, 6, and 12 months, with annual imaging thereafter for high-risk tumours (intracranial extension or incomplete resection), converting follow-up from an ad hoc process into a defined standard. Prospective evaluation of these context-adapted pathways represents an important direction for future work.
Recurrence Rates in Context
Our cases align with broader recurrence trends. A meta-analysis by Reyes et al comparing surgical approaches reported recurrence rates of 13% after endoscopic vs 28% after open resection in patients without intracranial compromise (p<0.02), with no significant difference between approaches once intracranial involvement was present—underscoring the higher-risk category into which Case 1 fell.14 A systematic review of exclusive endoscopic resection similarly estimated a pooled recurrence rate of approximately 10% and a residual-tumour rate of 7.7%, and found that preoperative embolization reduced mean intraoperative blood loss from 774 mL to 415 mL.15 These data reinforce that intracranial extension and residual disease—both relevant to Case 1—are principal determinants of recurrence. Consistent with this, Fang et al identified sphenoid bone invasion and surgical approach as independent risk factors for recurrence and reported improved relapse-free survival with endoscopic surgery.8
Case 1 exemplifies the consequences of disrupted postoperative follow-up. Because no postoperative imaging was performed and the patient did not adhere to scheduled visits, residual or early recurrent tumour at difficult-to-clear sites went undetected until it had progressed to advanced disease—illustrating how inadequate surveillance, rather than surgical failure alone, can drive late presentation. In contrast, Case 2 illustrates how diagnostic inaccuracy—initial misclassification as a nasal polyp and unstaged surgery—precipitated rapid recurrence within four months. Together, these cases stress that recurrence is shaped not only by tumour anatomy but also by health-system and patient-related factors.
Advancements in Surgical Management
Endoscopic resection with preoperative embolization has become the cornerstone of JNA treatment. Both patients underwent this approach, achieving gross total excision with minimal morbidity, consistent with outcomes reported by Lee et al6 Emerging adjuncts like image-guided navigation and radiofrequency ablation16 may further enhance precision, particularly in complex recurrences with skull base involvement. Modern embolic agents (eg, Onyx, tris-acryl gelatin) reduce intraoperative blood loss by 60–70% compared to non-embolized cases.4 However, the optimal embolization strategy for recurrent JNA remains debated, warranting comparative studies stratified by tumor stage.
Role of Imaging and Histopathology
MRI remains indispensable for staging and surveillance. Both cases demonstrated classic radiological markers (eg, Holman-Miller sign) and MRI’s superiority in delineating intracranial extension. Advanced techniques like DCE-MRI and DWI, though not utilized here, show promise in differentiating fibrosis from active tumor17 and should be integrated into future protocols. Histopathologically, our cases revealed sinusoidal vasculature and fibrous stroma, consistent with JNA’s hallmark features.18 Intriguingly, vascular thrombi in Case 1 and inflammatory infiltrates in Case 2 suggest heterogeneous tumor biology, potentially influencing recurrence patterns—a hypothesis needing further exploration.
Limitations
This report is subject to limitations inherent in retrospective case series. The small sample size restricts generalizability, and the absence of immediate postoperative MRI in Case 1 limits our ability to definitively distinguish between residual and recurrent tumor. In addition, post-contrast MRI sequences were not available for either case, which limited full characterisation of tumour enhancement and vascularity; diagnosis rested on the characteristic non-contrast imaging features together with histopathological confirmation. Furthermore, although follow-up was sufficient to confirm short-term remission, long-term outcomes remain unknown. Selection bias is possible, as both patients were referred following suboptimal initial management. Despite these limitations, the cases offer valuable clinical insights into potentially modifiable contributors to recurrence, particularly the role of follow-up adherence and timely imaging — factors often underrepresented in larger, technique-focused studies.
Conclusion
Recurrent juvenile nasopharyngeal angiofibroma remains a complex clinical entity in which both anatomical factors and care-delivery dynamics play a pivotal role. Through these two adolescent cases, we demonstrate how patient non-compliance and systemic diagnostic delay can contribute to recurrence, even after initial surgical intervention. Favourable outcomes in both instances were achieved through multidisciplinary management, endoscopic resection, and preoperative embolization. Beyond the technical success of surgery, however, these cases highlight three modifiable priorities: accurate initial staging to guide the extent of resection, standardised imaging-based surveillance protocols to detect residual or recurrent disease before it becomes advanced, and system-level strategies—such as earlier case-finding and improved access to cross-sectional imaging—to reduce diagnostic delay in resource-constrained settings. Embedding these measures into routine practice, alongside comprehensive patient education, is essential to reducing recurrence and ensuring long-term disease control.
Acknowledgments
We sincerely thank the patients and their families for their cooperation and for providing informed consent to share their medical information and images for the purpose of this publication.
Funding Statement
This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.
Statement on AI Use
Artificial intelligence (AI)-assisted technologies, particularly Claude Opus 4.8, were used solely to support the preparation of this manuscript by enhancing its clarity, coherence, and editorial quality. The AI tools were not involved in the interpretation of results or any part of the scientific analysis. All authors independently reviewed and verified the manuscript to ensure the accuracy and integrity of the content.
Data Sharing Statement
All data generated or analyzed during this study are included in this article. Further inquiries can be directed to the corresponding author.
Ethical Statement
This study was reviewed by the Institutional Ethics Committee of AKFA Medline University Hospital, Tashkent, Uzbekistan, which determined that formal ethical approval was not required for this case report. Written informed consent was obtained from both patients for publication. No experimental procedures were performed.
A preprint version of this manuscript has been posted on SSRN (Social Science Research Network) and is available at: https://papers.ssrn.com/sol3/papers.cfm?abstract_id=5989573
Consent for Publication
Written and oral informed consent was obtained from both patients for publication of the case details and accompanying images. Copies of the consent forms are available for review by the journal’s editorial office upon request.
Disclosure
The authors declare no conflicts of interest relevant to this publication.
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Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.
Data Availability Statement
All data generated or analyzed during this study are included in this article. Further inquiries can be directed to the corresponding author.