Nasal septal angiofibroma: less vascular behaviour than observed in radiological imaging, a rare case in a 51-year-old woman.

Angiofibroma of the head and neck is classically found in the nasopharynx of an adolescent male. We present a rare case where an angiofibroma was diagnosed in the anterior nasal septum of a 51-year-old woman, the eighth nasal septal angiofibroma reported in a female. This case highlights how an angiofibroma in an older woman has a less vascular behaviour than one in an adolescent male, even though radiological enhancement may suggest that the lesion is highly vascularised. Nasal septal angiofibromas can be managed by biopsy and removed by simple endoscopic resection with minimal haemorrhage. Although very rare, extranasopharyngeal angiofibroma/nasal septal angiofibroma should be included in the differential diagnosis when patients of all ages present with nasal obstruction secondary to a nasal mass, with or without epistaxis.

One-Stage Reconstruction of the Large Lower Nose Defect Involving 2 Subunits With Lateral Nasal Artery Pedicle Nasolabial Flap.

BACKGROUND: Large nose defect reconstructions involving the tip and alar subunits are still difficult to achieve in one stage. This study aimed to investigate the size of the nasal tip and alae, observe the distribution patterns of the lateral nasal artery and present our clinical experience with the lateral nasal artery pedicle nasolabial (LNAPN) flap. METHODS: From June 2019 to January 2020, lower nose parameters from 60 subjects were measured by a 3-dimensional scan, and the cranial digital subtraction angiogram results of 20 patents without vascular malformation were retrospectively analyzed. The case series consisted of four patients with nasal tip and alar defects who underwent surgery with LNAPN flaps from December 2018 to June 2019. Outcomes and complications were followed-up at 6 to 12 months. RESULTS: For the 3-dimensional scan study, the mean area of the 2 subunits involving the tip and alae was 7.986 ±â€Š1.728 cm. In the digital subtraction angiogram data, the lateral nasal artery was identified among all facial arteries and had a mean diameter of 1.0 ±â€Š0.20 mm. Sixteen people (80%) had a rich connection between the lateral nasal artery and the transverse facial artery. All cases revealed satisfactory aesthetic outcomes by reconstruction of large defects in one stage. The largest flap of 4.2 cm × 3.0 cm in our study showed complete healing. CONCLUSIONS: The LNAPN flap represents a feasible, flexible, efficient, and easily mastered reconstructive method for large nose defects involving the tip and alar subunits.

Extended Central Artery Perforator Propeller Flap for Large Nasal Defects.

Various local flaps have been defined for small skin defects of the nose. However, the repair of large nasal defects is only possible with flaps allowing a large tissue transfer, such as a free flap, forehead flap, and nasolabial flap. In this study, large nasal defects were reconstructed with extended central artery perforator propeller (CAPP) flaps in an attempt to describe a single-stage procedure as an alternative technique to the median forehead flap. Thirteen large nasal skin defects, including dorsum and nasal sidewall and/or dome, were repaired with a CAPP flap between January 2015 and March 2018. A total of 13 patients aged 19 to 92 years were included. The mean follow-up period was 14.9 months. Pathological diagnoses were basal cell carcinoma in 5 patients, squamous cell carcinoma in 6 patients, and trauma in 2 patients. Defect size ranged between 3 × 3 and 4 × 5 cm. Flap size ranged between 3 × 7 and 5 × 10 cm. No major complications including total flap failure, hematoma, or infection were observed. However, a partial flap necrosis occurred in 1 patient. In 3 patients, scar revision surgery was performed at the postoperative period. In conclusion, CAPP flap use is a safe and reliable option to repair large nasal defects. This flap is able to cover large nasal defects including dorsal, dome, and nasal sidewall defects in a single-stage procedure. Requiring no pedicle separation, this flap is an alternative option to the conventional median forehead flap.

Differentiation of squamous cell carcinoma and inverted papilloma using non-invasive MR perfusion imaging.

OBJECTIVES: To investigate the diagnostic value of tumour blood flow (TBF) obtained with pseudocontinuous arterial spin labelling for the differentiation of squamous cell carcinoma (SCC) and inverted papilloma (IP) in the nasal or sinonasal cavity. METHODS: We retrospectively analysed the cases of 33 patients with SCC and 8 patients with IP in the nasal or sinonasal cavity. Pseudocontinuous arterial spin labelling scanning was performed for all patients using a 3.0-T MR unit. Quantitative TBF values were measured by two neuroradiologists by respectively delineating the whole-tumour regions of interest, and the mean of them was determined as TBF value in each patient. Additionally, the presence of imaging findings of convoluted cerebriform pattern (CCP) on MR T2 weighted images was determined in all patients. As a subgroup analysis, patients with IP were divided into aggressive and non-aggressive IPs depending on their progression range. First, an intraclass correlation coefficient (ICC) of TBF values between two neuroradiologists was determined. Next, a statistical comparison of the TBF value by a Mann-Whitney U test between the patients with SCC and IP was performed. Additionally, the comparison by an ANOVA with a post hoc test of Tukey's method among the SCC, non-aggressive IP and aggressive IP groups was also performed. If significance was observed, the diagnostic accuracy to differentiate SCCs from IPs was calculated. Diagnostic accuracy by CCP findings alone and by the combination of CCP findings and TBF were also assessed. RESULTS: The ICC of TBF values between two neuroradiologists was 0.82. The mean TBF values in the patients with SCC, all patients with IP, those with aggressive IP and those with non-aggressive IP were 141.2 ± 33.1, 77.8 ± 31.5, 109.4 ± 16.7 and 58.8 ± 19.9 ml 100 g⁻¹ min⁻¹, respectively. A significant difference was observed between SCC and IP (p < 0.001), SCC and non-aggressive IP (p < 0.01) and non-aggressive IP and aggressive IP (p < 0.01). The diagnostic accuracy values obtained with receiver operating characteristic curve analysis for the differentiation of SCC from IP and for SCC from non-aggressive IP were 0.90 and 0.92, respectively. The diagnostic accuracy was elevated (0.95 from 0.88) by adding the TBF value to CCP findings. CONCLUSIONS: The pseudocontinuous arterial spin labelling technique can be a useful non-invasive diagnostic tool to differentiate SCC from IP in nasal or sinonasal cavity.

Overexpression of angiomotin in sinonasal inverted papilloma.

BACKGROUND: Although inverted papilloma (IP) is one of the most common sinonasal tumors, its etiology and factors associated with tumor progression have not been fully determined. Generally, tumorigenesis or tumor growth requires angiogenesis to feed tumor cells. Angiomotin is a recently discovered protein that regulates migration and tubule formation in endothelial cells. It has been reported that angiomotin affects angiostatin (circulating inhibitor of angiogenesis), resulting in promotion of angiogenesis. Thus, we evaluated the expression and distribution of angiomotin in sinonasal IP, compared to normal control tissue. METHODS: The study included 10 subjects with sinonasal IP and 5 normal controls. Ethmoid sinus mucosa obtained during reduction of blowout fractures was used as a normal control. Reverse transcriptase-polymerase chain reaction (RT-PCR), real-time PCR, immunohistochemistry, and Western blot analysis were used to assess the expression, intensity, and distribution of angiomotin in tissues. RESULTS: Positive bands for angiomotin were seen in all specimens by RT-PCR. The expression level of angiomotin was significantly upregulated in IP tissues versus normal sinus mucosa by real-time PCR. Immunohistochemistry revealed positive reactions on endothelial cells of capillaries and small vessels within the tumor and normal tissues, but the positivity was significantly stronger in IP. Western blot analysis showed that expression levels of angiomotin were increased in IP compared to normal sinus mucosa. CONCLUSION: Angiomotin, a novel protein in angiogenesis, was overexpressed in IP. Although it is not an etiological or initiating factor in tumor development, it seems to be associated with progression and growth of IP via promoting angiogenesis.

[Correlation of VEGF expression with microvessel and microlymphatic density in sinonasal squamous cell carcinoma].

OBJECTIVE: To study the expression of VEGF in sinonasal squamous cell carcinoma and its correlations with microvessel density (MVD), microlymphatic vessel density (MLVD). METHOD: The expression of VEGF, MVD and MLVD in 41 cases of sinonasal squamous cell carcinoma were detected by immunohistochemical technique. RESULT: In the sinonasal squamous cell carcinoma, the positive rate of VEGF was 82.9% (34/41). The over expression of VEGF was related with tumor invasion, histological grading and lymphatic metastasis (P < 0.05). The MVD of cases with positive VEGF expression was significantly higher than those without VEGF expression (P < 0.05), but was not statistical difference in MLVD (P > 0.05). CONCLUSION: VEGF may participate in the metastasis of sinonasal squamous cell carcinoma through promoting vascularization in the tumors, but not promoting MLVD.

[Expression of fascin and correlation with MVD in sinonasal inverted papilloma].

OBJECTIVE: To investigate the expression of Fascin in sinonasal inverted papilloma (SNIP) and the relationship between Fascin expression and microvessel density (MVD). METHOD: The expression of Fascin and CD34 were determined by immunohistochemistry in 30 cases of SNIP, 12 cases of nasal squamous cell carcinoma (NSCC) and 10 cases of normal inferior turbinate (IT) tissues. Meanwhile, CD34, measure of MVD, was counted at the same time. RESULT: Fascin was expressed in 17 of 30 (56.6%) cases of SNIP, 10 of 12 (83.3%) cases of NSCC and 2 of 10 (20.0%) cases of normal IT tissues. Expression of Fascin was significantly increased in SNIP and NSCC compared to normal tissues (P < 0.01). MVD counts was 23.64 +/- 2.74, 45.10 +/- 5.45, 74.76 +/- 7.32, respectively (P < 0.01). There was a positive correlation between the expression of Fascin and MVD counts. CONCLUSION: The high expression of Fascin and angiogenesis are significantly associated with biological behaviour of SNIP. Fascin may play an important role in the pathogenesis of SNIP and NSCC and synergic role with angiogenesis in progression of SNIP.

Fibro-osseous lesion of the middle turbinate: ossifying fibroma or fibrous dysplasia?

Benign fibro-osseous tumours of the head and neck region seldom appear in the midface and nasal cavity. Correct differential diagnosis of fibro-osseous tumours is crucial for adequate therapy, as their clinical aggressiveness seems to differ. The rather uncommon case of a 14-year-old boy with a tumour of the middle turbinate is presented. The radiologic appearance of the tumour, on computed tomography and magnetic resonance imaging scans, was consistent with fibrous dysplasia. Angiography revealed extensive vascularisation of the tumour from both the internal and external carotid arteries. To avoid ipsilateral blindness following embolisation, a superselective embolisation of the supplying blood vessels was performed. The tumour was completely resected via an endoscopic approach. Histopathology revealed an ossifying fibroma. This case emphasises the importance of interpretation of the clinical, radiological and histological features before planning definitive treatment. Moreover, when fibro-osseous tumours are suspected, the possibility of extensive, complicated vascularisation must be considered. This case underwent radical resection, with no recurrence after four years' follow up.

Meningioma of the paranasal sinus: a case report.

Extracranial meningioma of the paranasal sinus is a very rare condition of unclear etiology. Surgical excision is currently the only curative treatment but sometimes entails difficulties because of the complicated anatomic loci of the tumor. For the case reported here, we used a navigation system in endoscopic surgery for an extracranial meningioma of the paranasal sinus and were able to remove the tumor without complications by a transnasal approach alone. This is the first report of the use of such a system with endoscopic surgery for this condition. We discuss the clinical presentation, diagnosis, and treatment of this case as well as present a review of the literature.

Inflammatory myofibroblastic tumor of the nasal cavity.

Inflammatory myofibroblastic tumor (IMT) is a rare tumor with a variable natural history and biologic behavior, ranging from completely benign to malignant with fatal outcome. We report a case of benign IMT in the left nasal cavity with radiologic features mimicking angiofibroma. We also demonstrate the hypervascular nature of this disease on angiography and the contribution of preoperative embolization in assisting surgical excision and minimizing the potential uncontrolled intraoperative bleeding.

Endoscopic sinus surgery for the treatment of vascular tumors.

BACKGROUND: This study points out the effectiveness of the endoscopic approach for the treatment of vascular lesions such as angiofibroma, hemangioma, and hemangiopericytoma involving the nose and paranasal sinuses. METHODS: We performed a retrospective study at an academic tertiary referral center. Thirteen patients, diagnosed with vascular tumors of the nose and paranasal sinuses were treated endoscopically between February 1996 and July 2003. All patients underwent endonasal endoscopic surgery. Preoperative angiography with embolization was performed in all but two cases. RESULTS: The follow-up of this series varied from 6 to 75 months (mean, 23 months); only one recurrence (8%) was observed in the juvenile angiofibroma group encountered 20 months postoperatively. This recurrence was again treated endoscopically. The average intraoperative blood loss for the removal of the juvenile angiofibroma group was 300 mL and it was 100 mL for the other vascular tumors. CONCLUSION: Endoscopic treatment alone is an effective approach for the removal of selected cases of vascular tumors. Even in the presence of a lesion with limited intracranial extension, the tumor still may be amenable to an endoscopic approach alone. On the contrary, this is not true if the intracranial extension receives feeding vessels from the internal carotid arterial system.

[Aetiology of Angiofibromas: Reflection on their Specific Vascular Component]. / Die vaskuläre Komponente: Gedanken zur Entstehung des Angiofibroms.

BACKGROUND: Even though numerous theories have speculated either on the vascular or fibrous tumour component as tissue of origin, aetiology of angiofibroma still remain unclear. Histological investigations recently led Beham and coworkers to the assumption that angiofibromas have to be considered as vascular malformations. METHODS: After giving a literature review of the various theories on tumour origin the proposal to consider angiofibromas as vascular malformations is discussed on an embryological base. Taking typical clinical features of the tumour and knowledge of vasculogenesis into account a new explanation for origin of the vascular tumour component is presented. RESULTS: The vascular component of angiofibromas can be explained embryologically due to incomplete regression of the first branchial arch artery (vascular atavism). This vessel arises regularly between embryological day 22 and 24 and recedes during regular development completely until delivery via temporary formation of a vascular plexus. In the late stages of embryological development remnants of the plexus are found at the area of the sphenopalatine foramen, the typical site of angiofibroma origin. Incomplete regression of the vascular plexus of the former first branchial arch artery may form the vascular component of an angiofibroma arising due to growth stimulation at the time of adolescents. CONCLUSIONS: Incomplete regression of the first branchial arch artery presenting an atavism is suited to explain the vascular tumour component of angiofibromas considering main tumour characteristics (origin in the posterior nasal cavity close to the sphenopalatine foramen, main blood supply from the maxillary artery with possible feeders arising from the internal carotid artery). Our embryological contributions support to define angiofibromas as vascular malformations.

Merkel cell carcinoma: a rare cause of hypervascular nasal tumor.

Cutaneous neuroendocrine carcinoma, first described in 1972, is an aggressive disease usually occurring in sun-exposed skin. Other sites have been described, however; such tumors occasionally occur within the nasal fossa. A high rate of metastasis (>30%) explains the poor prognosis. Descriptions of the imaging features of these tumors, mainly located in cutaneous region, are rare. We therefore present the imaging features of two cases of Merkel cell carcinoma involving the sinonasal region, suggestive of a hypervascular tumor.

[Nasal/nasopharyngeal T/NK-cell lymphomas with evident angiogenic activity].

OBJECTIVE: To search the difference of the angiogenic activity between T/NK-cell lymphomas and B-cell lymphomas. METHODS: The T/NK-cell was stained immunohistochemically by CD56 and CD45 RO, the B-cell was by CD20, the microvessel endothelium was by CD34. Sixty-one cases were studied to examine the immunophenotype and to estimate the intratumoral microvessel density (iMVD). RESULTS: Forty in 61 cases (65.6%) expressed the immunophenotype of both T-cell and Natural Killer cell, were diagnosed T/NK-cell lymphomas. Fifteen in 61 cases (24.6%) expressed the immunophenotype of B-cell. There was a significant difference in incidence between the two types of lymphomas (chi 2 = 22.4, P < 0.01). The mean of iMVD/HPF was 24.1 +/- 8.7 in 40 cases of T/NK-cell lymphomas, while the mean of iMVD/HPF was 14.9 +/- 3.8 in 15 cases of B-cell lymphomas. The difference (t = 3.7, P < 0.05) was significant. CONCLUSION: The angiogenic activity of T/NK-cell lymphomas is much more than that of B-cell lymphomas. This study shows nasal/nasopharyngeal T/NK-cell lymphomas are with evident angiogenic activity.

Embolization of neurosurgical lesions involving the ophthalmic artery.

OBJECTIVE: A number of anteriorly located cranial base and extracranial lesions receive their vascular supply wholly or in part from the ophthalmic artery, and embolization of the ophthalmic artery can be helpful in the management of these lesions, either as the primary treatment or as an adjunct to surgery. We present situations in which the embolization of lesions involving the ophthalmic artery was performed to effect a partial or total cure of the lesion. METHODS: Twelve patients underwent a total of 15 embolization attempts on lesions involving the ophthalmic artery. Four patients had arteriovenous malformations of the orbit, four had dural arteriovenous fistulae, two had orbital meningiomas, one had a planum sphenoidale meningioma, and one had a juvenile nasal angiofibroma. In each case, a Tracker No. 18 microcatheter (Target Therapeutics, Inc., Fremont, CA) was navigated into the ophthalmic artery using a steerable guidewire and digital road mapping. Embolic agents included polyvinyl alcohol particles ranging from 350 to 1500 microm in diameter, 2-mm platinum microcoils, and n-butyl-cyanoacrylate. In 12 of 15 cases, lidocaine and amytal provocation tests were conducted before any attempt at embolization to assess the role of the ophthalmic artery in vision. RESULTS: Embolization was successfully performed in the 14 situations in which it was attempted. Positive results of two lidocaine/amytal tests were noted. In one case, embolization was not attempted. In the other case, a larger caliber embolic agent (2-mm platinum coils) was used. A single transient decrease in visual acuity lasting 4 days was the only embolization-related complication. CONCLUSION: Proper case selection, judicious use of embolic agents, and use of provocative testing can result in safe embolization of lesions supplied by the ophthalmic artery.

Intraosseous hemangioma of the vomer: a case report.

We describe an unusual case of intraosseous hemangioma of the vomer. The lesion had a characteristic radiographic appearance, especially on the computed tomographic scan. The two most interesting points in this case were the rarity as a site for the occurrence of hemangioma and the successful extirpation of the tumor via the Le Fort 1 osteotomy approach without embolization of the feeding vessels. The benefits of this approach included the wide surgical field afforded deep in the midface region, which was important for controlling bleeding, as well as the complete extirpation of the tumor.

CD34-reactive fibrous papule of the nose.

In human skin, the CD34 antigen is expressed on endothelium, periadnexal cells, and a population of reticular dermal interstitial cells. CD34 expression is characteristic of dermatofibrosarcoma protuberans and several other neoplasms, but not of typical fibrous papules of the nose. We describe a 16-year-old white girl with a slowly growing papule on the nose. Histopathology showed a dermal tumor with a superficial component of branched, thin-walled blood vessels and a deeper component of benign-appearing, spindle-shaped cells. These cells uniformly and strongly expressed CD34, but not factor XIIIa or markers of melanocytic, neural, muscular, vascular, or histiocytic differentiation. We consider this lesion a CD34-reactive fibrous papule. This benign tumor must be clearly distinguished from dermatofibrosarcoma protuberans, which also is composed of bundles of CD34-reactive spindle-shaped cells in most cases but has locally aggressive behavior.

Direct intratumoral embolization of juvenile angiofibroma.

INTRODUCTION: Embolization is a well-established technique that facilitates the subsequent surgical removal of vascularized tumors such as juvenile angiofibroma. However, there is risk of a neurological accident during angiography and tumor embolization from the internal carotid artery. Direct intratumoral embolization may help prevent these potential side effects. METHOD: A group of 7 patients with juvenile angiofibroma vascularized through the branches of the internal carotid artery underwent direct tumoral embolization under general anesthesia. An injection made slowly with an intranasal or lateral percutaneous route with either a mixture of cyanoacrylate, lipiodal, and tungsten powder. Continuous radiographic control was used. RESULTS: This technique induced a marked devascularization and necrosis of the tumor. The technique provided useful perioperative visualization of the tumor. No neurologic sequelae were encountered. CONCLUSION: Direct intratumoral embolization deserves further consideration. This might be especially important in tumors with extracranial extension, cavernous sinus involvement, or those with small or multiple recurrences.