[Cerebrospinal fluid rhinorrhea secondary to ethmoidal carcinoma: case report]. / Rinorrea de líquido cefalorraquídeo secundaria a carcinoma etmoidal: reporte de caso.

BACKGROUND: The first report of cerebrospinal fluid rhinorrhea (CSFR) was described in 1679. In 1826 it was reported that one of the possible causes of CSFR was a fistula between the subarachnoid space and the nasal cavity. In 1903, chemical analysis of the fluid was proposed as a diagnostic criterion. In Mexico there has been 32 case reports. CASE REPORT: Forty-nine years old female with a history of nasal polyposis, profuse rhinorrhea and cephalea who attends the allergy department with the suspicion of allergic rhinitis. After anamnesis and physical evaluation, CSFR was suspected. Chemical analysis of the fluid, head CT and biopsy of nasal polyp were performed. An etmoidal fistula associated with carcinoma was confirmed. CONCLUSIONS: Spontaneous fistulas are rare but can erosionate the bone and adjacent tissues. Diagnosis is based on the clinical findings, patient's history and complementary studies such as beta-2-transferrin determination in nasal fluid.

ANTECEDENTES: En 1679 se describió el primer caso de rinorrea de líquido cefalorraquídeo. En 1826 se reportó como causa una fistula entre el espacio subaracnoideo y la cavidad nasal. Para 1903 se propuso el análisis químico como criterio diagnóstico. En México sólo se han reportado 32 casos de rinorrea de líquido cefalorraquídeo. REPORTE DE CASO: Paciente femenina de 49 años, con antecedente de poliposis nasal, rinorrea abundante y cefalea, quien acudió a consulta para descartar rinitis alérgica. Luego de la anamnesis y la exploración física se sospechó de fuga de líquido cefalorraquídeo secundaria a fístula nasal. Con la histoquímica de moco, tomografía de cráneo y biopsia del pólipo nasal se estableció el diagnóstico de fístula etmoidal secundaria a carcinoma. CONCLUSIÓN: La fístulas espontáneas son excepcionales, pueden erosionar el hueso y los tejidos adyacentes. El diagnóstico se establece con la historia clínica y los antecedentes médicos, además de estudios complementarios y la determinación de Beta-2-transferrina en moco.

The implications of concomitant mucosal inflammation on clinical manifestations and outcomes of sinonasal inverted papilloma.

PURPOSE: This study examines the impact of concomitant mucosal inflammation on clinical manifestations and long-term outcomes of Inverted Papilloma (IP). METHODS: This retrospective cohort study was conducted in five tertiary medical centers. The included patients underwent an attachment-oriented surgical resection for IP with a minimum follow-up of 3 years. RESULTS: Of 185 patients with IP, 65 patients (35.1%) had synchronous mucosal inflammation with polypoid changes. The mean age was 56.7 years, and 69% were males. Most tumors originated from the maxillary sinus. Age, gender, Krouse stage, and tumor attachment site did not differ between the mucosal inflammation and IP-only groups. IP recurrence rate was twofold in the patients with mucosal inflammation (15.4% vs. 7.5%, p = 0.092). However, the difference was not significant, with a similar median time to recurrence between the two groups [15.5 (3-36) months vs. 16(6-96) months, p = 0.712]. In revision cases, IP recurred only in patients with mucosal inflammation (19% vs. 0%, p = 0.07). This group had a significantly worse 5-years recurrence-free survival than revision cases without mucosal inflammation (80.6% vs. 100%, p = 0.04). CONCLUSIONS: IP in the setting of mucosal inflammation might be associated with a higher recurrence rate, predominantly after revision surgery. Otolaryngologists should consider this during these patients' diagnosis, surgical planning, and follow-up.

Preoperative Prediction of Sinonasal Inverted Papilloma-associated Squamous Cell Carcinoma (IP-SCC).

INTRODUCTION: Sinonasal inverted papillomas (IP) can undergo transformation into IP-squamous cell carcinomas (IP-SCC). More aggressive treatment plan should be established when IP-SCC is suspected. Nevertheless, inaccuracy of the preoperative punch biopsy results to detect IP-SCC from IP raises the need for an additional strategy. The present study aimed to investigate significant clinicoradiological remarks associated with IP-SCC than IP. MATERIAL AND METHODS: Postoperative surgical specimens obtained from patients with confirmed IP or IP-SCC at a single tertiary medical center from 1997 to 2018 were retrospectively evaluated. Patients' demographic and clinical characteristics, preoperative in-office punch biopsy results, and preoperative computed tomography (CT) or magnetic resonance images were reviewed. Univariate and multivariate analyses were performed to assess the odds ratio (OR) associated with IP-SCC. The area under the curve (AUC) in the receiver Operating Characteristic (ROC) curve was calculated in the prediction model to discriminate IP-SCC from IP. RESULTS: The study included 44 IP-SCC and 301 patients with IP. The diagnostic sensitivity of in-office punch biopsy to detect IP-SCC was 70.7%. Multivariate analysis showed that factors significantly associated with IP-SCC included tobacco smoking >10PY (adjusted-OR [aOR]: 4.1), epistaxis (aOR: 3.4), facial pain (aOR: 4.2), bony destruction (aOR: 37.6), bony remodeling (aOR: 36.3), and invasion of adjacent structures (aOR: 31.6) (all p < 0.05). Combining all significantly related clinicoradiological features, the ability to discriminate IP-SCC from IP reached an AUC of 0.974. CONCLUSION: IP patients with a history of tobacco smoking, facial pain, epistaxis, and bony destruction, remodeling, or invasion of an adjacent structure on preoperative images may be at higher risk for IP-SCC. LEVEL OF EVIDENCE: 3 Laryngoscope, 133:2502-2510, 2023.

Sinonasal diffuse large B-cell lymphoma in a patient with Wiskott-Aldrich syndrome: A case report and literature review.

Wiskott-Aldrich syndrome (WAS) is a rare primary immunodeficiency disease with a predisposition towards autoimmunity and lymphoproliferative diseases. Non-Hodgkin lymphoma (NHL) is reported to be the predominant form of malignant tumor in WAS sufferers. Diffuse large B-cell lymphoma (DLBCL) is one of the most common types of NHL while it is uncommon to occur in paranasal sinuses and especially when associated with WAS. In this article, we report a unique case of WAS associated with DLBCL in paranasal sinuses and review the major publications of WAS-related lymphomas that occurred in the head and neck area. This study extends the available therapies for WAS-related lymphomas and emphasizes the significance of recognition for sinonasal lymphomas in WAS patients presenting with sinusitis.

The incidence of myocardial infarction and stroke in head and neck cancer patients.

Various treatment modalities are used for head and neck cancer (HNC). This study analyzed the incidence and risks of myocardial infarction (MI) and stroke by cancer site and treatment modality in 22,737 patients newly diagnosed with HNC registered in the Korean National Health Insurance Service database in 2007-2013. An additional 68,211 patients without HNC, stroke, or MI were identified as the control group. The risks for MI (hazard ratio [HR] = 1.38, 95% confidence interval [CI] 1.24-1.53), stroke (HR = 1.48, 95% CI 1.37-1.60), and mortality (HR = 5.30, 95% CI 5.14-5.47) were significantly higher in the HNC group. Analysis by cancer site showed the risk of MI and mortality was highest in hypopharynx cancer, while the risk of stroke was highest in nasopharynx and paranasal sinus cancer. Analysis by treatment modality showed the highest risks for MI (HR = 1.88, 95% CI 1.31-2.69) and mortality (HR = 2.95, 95% CI 2.75-3.17) in HNC patients receiving chemotherapy (CT) alone, while HNC patients receiving CT with surgery had the highest risk for stroke (HR = 1.81, 95% CI 1.14-2.88). Careful attention to MI and stroke risks in HNC patients is suggested, especially those who received both CT and radiotherapy.

Genomic Sequencing of Cancer-related Genes in Sinonasal Squamous Cell Carcinoma and Coexisting Inverted Papilloma.

BACKGROUND: The genetic basis of sinonasal inverted papilloma (SNIP)-derived squamous cell carcinoma (SCC) has not yet been well characterized. AIM: To characterize the genetic abnormalities of SNIP and SNIP-derived SCC and to uncover their differences. MATERIALS AND METHODS: Mutations of 409 genes were analyzed using amplicon targeted sequencing in a total of six papilloma/carcinoma samples from four patients with SNIP-derived SCC. RESULTS: The genes that were mutated in multiple cases were epidermal growth factor receptor (EGFR) (3/6), cyclin-dependent kinase inhibitor 2A (CDKN2A) (3/6), lysine methyltransferase 2D (KMT2D) (3/6), tumor protein p53 (TP53) (3/6), neurofibromin 1 (NF1) (3/6), phosphodiesterase 4D interacting protein (PDE4DIP) (3/6), cytochrome P450 family 2 subfamily D member 6 (CYP2D6) (2/6), fms-related receptor tyrosine kinase 4 (FLT4) (2/6) and myosin heavy chain 9 (MYH9) (2/6). Of the two cases analyzed in the papilloma-oncology carcinoma pair, one did not have any common mutations; the other showed a staged functional deletion of TP53 during the process of malignant transformation from SNIP to SCC. CONCLUSION: CDKN2A, KMT2D, NF1, PDE4DIP, CYP2D6, FLT4, and MYH9 were identified as candidate novel SNIP-derived SCC-related genes.

Sinonasal cancer encroaching the orbit: Ablation or preservation?

BACKGROUND: Encroachment on the orbital cavity represents a challenge in the management of sinonasal cancer. Criteria guiding orbital preservation lack univocal consensus. Stage of orbital involvement is best assessed through magnetic resonance imaging (MRI). METHODS: Patients affected by orbit-encroaching sinonasal cancer with available preoperative MRI, receiving surgery-based treatment at the University of Brescia between May 2005 and October 2018 were included. All cases were reviewed by expert radiologists and pathologists. Diagnostic performance of MRI was calculated using pathological information as reference. Survival analysis was performed. RESULTS: The study included 123 patients. The orbit was abutted in 53 (43.1%) patients, whereas orbital invasion reached the periorbit in 18 (14.6%), extraconal fat and/or medial lacrimal sac in 29 (23.6%), extrinsic ocular muscles in 7 (5.7%), intraconal compartment in 4 (3.3%), and orbital apex in 12 (9.8%). Seventy-six (61.8%) patients received orbit-sparing surgery, 47 (38.2%) underwent orbital ablation (OA). Accuracy of MRI in detecting involvement by cancer was ≥80.0% for the orbital wall, extraconal fat, and muscles, and <80.0% for the periorbit and intraconal compartment. Previous surgery, neoadjuvant chemotherapy, and perineural invasion decreased MRI accuracy. Age, histology, tumor grade, pT category, N status, perineural invasion, orbital invasion stage, and need for OA were found to affect prognosis. Five-year orbital dysfunction-free survival was 92.8%. CONCLUSION: Conservative management of sinonasal cancers encroaching the orbit is feasible. MRI is essential to preoperatively stage orbital invasion, yet with some limitation. Given the dismal prognosis despite aggressive surgery, neoadjuvant non-surgical therapies should be considered in patients requiring OA.

Inverted Papilloma Completely Obstructing Anterior Nasal Orifice.

Sinonasal inverted papilloma is a benign lesion that occurs in the nasal cavity and paranasal sinuses. Fungiform papillomas have been described as arising from the septum or the nasal vestibule, while inverted and cylindrical papillomas have been characterized as developing from an attachment site on the lateral nasal wall or a mucosal surface in the paranasal sinuses. Here, we present a rare case of an inverted papilloma that completely obstructed the anterior nasal orifice.

Orbital cellulitis secondary to giant sino-orbital osteoma: A case report.

BACKGROUND: Although osteoma is a common benign tumor of the paranasal sinuses, its orbital extension is not common. Secondary orbital cellulitis has rarely been reported in association with sino-orbital osteoma. CASE: A 30-year-old woman presented with left side proptosis, orbital pain and inflammation. Orbital CT scan showed a well-defined giant osteoma in the superonasal part of the left orbit originating from the left ethmoidal sinus associated with opacity of the ipsilateral ethmoidal sinus and infiltration of orbital soft tissue. After treatment by systemic antibiotics, osteoma was resected with combined external and endoscopic surgery and the patient recovered uneventfully. CONCLUSION: Sino-orbital osteoma may manifest primarily as orbital cellulitis and needs early surgical intervention.

Giant osteomas: Clinical results and surgical approach from ophthalmic point of view.

PURPOSE: To present clinical ocular manifestations, radiological features and surgical results of giant paranasal osteomas involving the orbit. DESIGN: Retrospective, interventional, case series. METHODS: The study included patients treated for giant paranasal osteoma involving the orbit. Total or subtotal excision of the tumor was performed via external approach. Clinical characteristics including presenting symptoms, radiologic investigation, histopathology and details of the surgery were recorded. RESULTS: Of the six patients included; four were male and two were female. Mean age was 46.8 years (range 12-70 years). Five patients had unilateral, one patient had bileteral disease. The presenting complaints included complete (n = 2/6) or partial (n = 4/6) limitation of eye movements depending on the location and size of the tumor, diplopia (n = 5/6), vision loss (n = 2/6) exophthalmos (n = 6/6) and reduced pupillary light reflex (n = 2/6). The presumed origin of the tumor was frontoethmoidal region (n = 2/6), frontal sinus (n = 3/6) and ethmoid sinus (n = 1/6). Total resection was achieved in three of the patients whereas partial resection was achieved in remaining three patients due to risk of incremental neurological damage. Mild ptosis was observed in all patients during the postoperative period (temporary, n = 4; permanent, n = 2). CONCLUSION: Despite the benign nature of osteomas, severe functional impairment including vision loss due to compression of the optic nerve and diplopia might occur in case of orbital involvement. Osteomas with an orbital extension > %50 of the bony orbit volume are more prone to cause irreversible visual loss. Surgical resection is still the mainstay of therapy.

Frontal recess osteoma causing severe headache.

Frontal sinus osteomas are benign and well-defined masses that may cause various symptoms when they press on the surrounding tissues. Paranasal sinus osteoma is seen as a well-defined mass showing high opacity in Computed tomography. In this case study, we presented here a 53-year-old frontal recess osteoma with severe headache filling the left frontal recess in the context of the literature.

Inverted papilloma is associated with greater radiographic inflammatory disease than other sinonasal malignancy.

BACKGROUND: The pathogenesis of inverted papilloma (IP) has not been fully elucidated. However, chronic paranasal sinus inflammation has been anecdotally observed in sites distant from tumor obstruction in IP patients, suggesting an association between inflammation and IP tumorigenesis. This study assesses the association between sinonasal inflammation found in IP and compares this to the level of inflammation observed in other sinonasal tumors. METHODS: A retrospective chart review was performed identifying patients with unilateral IP. Pertinent clinical data was obtained and comparative analysis of preoperative computed tomography (CT) imaging and histopathology was performed. A sample of unilateral, sinonasal, non-IP and non-squamous cell tumors was used as the control. The Lund-Mackay scoring system was used to assess radiologic sinonasal inflammation both ipsilateral and contralateral to the tumor. RESULTS: Seventy-one patients were included; 58.9% of patients with IP had evidence of contralateral sinusitis at the time of presentation. In the control group, 26.7% had evidence of contralateral inflammation. When comparing contralateral sinus inflammation between the 2 study groups, the IP patients had significantly higher Lund-Mackay scores than the control group (1.9 vs 0.26, p < 0.001). When comparing ipsilateral sinus inflammation, no significant difference was found in Lund-Mackay scores (5.44 vs 4.00, p < 0.184). CONCLUSION: In this study, unilateral IPs were associated with a higher level of contralateral sinonasal inflammation when compared to control. This suggests that IP may be associated with inflammation that is independent of obstruction by the tumor. Further studies are needed to better understand the temporal relationship between chronic inflammation and tumorigenesis.

Comorbidity burden and nonclinical factors associated with sinonasal cancer all-cause mortality.

OBJECTIVE: To describe comorbidity burden and nonclinical factors associated with all-cause mortality of sinonasal cancer in the United States. METHODS: The National Cancer Database (2004-2013) was queried for adult cases of sinonasal cancer (n = 10,518). Outcome of interest was all-cause mortality. Independent variables included comorbidity score and nonclinical factors such as age, gender, race, facility type, distance to facility, insurance, and income. Survival analysis was conducted via multivariable extended Cox regression with Heaviside adjustments. RESULTS: Patients were mostly (79%), male (61%), and mean age of diagnosis was 63.5 years. Approximately one in five patients (18.7%) had a major comorbid condition (Charlson-Deyo score ≥ 1) at diagnosis. After adjusting for clinical factors, increasing comorbidity score was associated with a corresponding increase in hazard of mortality (aHR comorbidity score of 1 = 1.25; 95% CI, 1.16, 1.35), (aHR score of 2+ = 1.61; 95%, CI 1.41, 1.83). Hazard of mortality was also associated with being male (aHR = 1.11; 95% CI, 1.04, 1.17); black (aHR = 1.13, 95% CI, 1.03, 1.24); uninsured (aHR = 1.45; 95% CI, 1.25, 1.68) or on Medicaid (aHR = 1.50; 95% CI, 1.33, 1.69); residence in zip codes with lower median income quartile (aHR < $30,000 = 1.17; 95% CI, 1.06, 1.29); and treatment at community cancer programs (aHR = 1.14, 95% CI 1.01, 1.28). CONCLUSION: Comorbid disease is associated with all-cause sinonasal cancer mortality, and after accounting for known clinical factors, significant differences in mortality persist based on disparity-driven, nonclinical factors. LEVEL OF EVIDENCE: NA Laryngoscope, 130:1443-1449, 2020.

Use of Dermaflage®, a silicone-based skin camouflage product, for treatment of medial canthal fistula.

Medial canthal fistulas present a therapeutic challenge, given that the risk factors for development of this and similar complications also contribute to their difficulty to treat. Following surgical, chemotherapy and radiation treatment of a poorly differentiated sinonasal carcinoma, a 61 year old man failed internal and external rotational flap repair and declined further operative procedures; he was satisfied with a novel conservative treatment. This report describes use of Dermaflage®, a silicone-based skin camouflage and topical cutaneous filler product, to both resolve the symptoms of tearing and discharge through the fistula and mask the defect from the casual observer.

Surgical Management of a Recurrent Orbit-Eroding Mucocele Associated With Frontal-Ethmoidal Sinus Inverted Papilloma.

The development of orbit-eroding mucocele associated with inverted papilloma has been rarely reported., Here, the authors present a case and surgical management of a patient with orbit-eroding mucocele associated with inverted papilloma who declined craniotomy. A combined approach utilizing frontal endoscopic sinus surgery and external sub-brow anterior orbitotomy was used to explore, drain, and excise the mucocele and inverted papilloma. Gelatin sponges soaked in gentamicin were used to cover the exposed dura and to protect the orbital content from the frontal sinus cavity.A fronto-ethmoidal sinus inverted papilloma associated with recurrent orbit-eroding mucocele was excised by combined transnasal endoscopic and external sub-brow anterior orbitotomy approach using stereotactic navigation.

High CT attenuation values relative to the brainstem may predict squamous cell carcinoma arising from inverted papilloma.

Background: A diagnostic indicator for differentiating squamous cell carcinomas (SCCs) from inverted papillomas (IPs) has not been established. Objectives: This study aimed to evaluate whether CT attenuation values relative to those of the brainstem (relative CT number) could be useful in differentiating IPs from SCCs. Material and Methods: Consecutive patients who were pathologically diagnosed with IP or SCC between 2007 and 2017 were retrospectively identified. Relative CT numbers were compared between the two patient groups. The factors with predictive power for differentiating IPs from SCCs were identified by univariate and multivariate logistic regression analyses. Results: Fifty-four sinonasal tumour cases were finally analysed (IP, 25 cases; SCC, 29 cases). Relative CT numbers were significantly higher in SCC than in IP (p < .001). The univariate logistic regression analysis showed BMI, relative CT number, and disease duration to have predictive value for differentiating IPs from SCCs. In the multivariate logistic regression analysis, only the relative CT number had predictive value for distinguishing IP from SCC (odds ratio, 1.97), with a relative CT number of ≥1.4 being significantly associated with SCC. Conclusions: High relative CT numbers could potentially be used to identify SCCs, and their measurement could provide a basis for differentiating IPs from SCCs.

Computed tomography findings in patients with primarily unknown causes of severe or recurrent epistaxis.

OBJECTIVE: In addition to rhinoscopy, computed tomography of paranasal sinuses (CT) may be performed on patients with primary unknown cause of severe epistaxis (SE) or recurrent epistaxis (RE) to further assess the potential cause of bleeding. The aim of this study was to evaluate CT findings during the work-up of intractable epistaxis patients. METHODS: 6937 patients were treated in our emergency department with acute epistaxis between 2009-2018. 304/6937 patients underwent CT and rhinoscopy due to intractable SE or RE. 33 patients presented with head trauma prior to epistaxis and were excluded from the final analysis. In 271 cases the primary causes of SE (n = 252) or RE (n = 19) remained unknown. Two observers retrospectively evaluated CT scans for potential sources of epistaxis. Disagreement was settled by consensus. CT and rhinoscopy findings were compared. RESULTS: In 247/271 (91.1%) SE patients no related pathology was found on CT. A possible cause for epistaxis was found in all RE patients, but only in 5/252 (1.9%) patients with SE. Most tumours (10/11) and inflammatory conditions (9/10) were found in patients with RE. In three SE cases, a tumour was suspected on CT, from which two suspicions were refuted during rhinoscopy. CT revealed 10 cases of inflammatory conditions of the sinus and anatomical variant as potential cause of bleeding. CONCLUSION: For patients with unknown causes of epistaxis, supplementary CT imaging may be a useful diagnostic add-on to rhinoscopy in the event of RE, tumour suspicion or inflammation of the paranasal sinuses. However, in most cases of first-time SE, CT does not necessarily add to the diagnosis. In these cases, the marginal benefit of CT needs to be weighed carefully against its risks.