Long-term effects of middle ear pressure therapy with the EFET01 device in patients with Ménière's disease and delayed endolymphatic hydrops in Japan.

BACKGROUND: Long-term efficacy of middle ear pressure therapy (MEPT) with the EFET01 device in patients in Japan with definitive Ménière's disease (MD) and delayed endolymphatic hydrops (DEH) was evaluated. OBJECTIVE: To examine the effects of reducing vertigo attacks and improving hearing of MD and DEH patients by using MEPT with the EFET01 device for two years after treatment. MATERIAL AND METHODS: A retrospective study was conducted of 32 MD patients and 2 DEH patients treated by MEPT with the EFET01 device from December 2018 to April 2021. According to Japan Society for Equilibrium Research (JSER) guidelines, patients were investigated for the frequency of vertigo attacks and change in hearing levels during a period of 6 months before to 18-24 months after start of treatment. RESULTS: The frequency of vertigo attacks significantly decreased in both MD and DEH patients, and hearing level has remained stable in the majority of our patients after treatment. CONCLUSION: Our study clarified that MEPT with the EFET01 device was effective in controlling vertigo symptoms of MD and DEH. It should be considered a safe option for patients failing medical treatment. SIGNIFICANCE: The efficacy of MEPT with the EFET01 was shown over a 2-year follow-up period.

Monitoring pressure changes in the inner ear induced by middle ear pressure therapy with an EFET01 device in guinea pigs.

BACKGROUND: As a low-cost, portable, handheld air pressure generation tool not requiring a ventilation tube, the EFET01 device has shown clinical effectiveness for intractable Ménière's disease (MD) patients in Japan. However, no animal studies have investigated changes in inner ear pressure (PI) when applying this device. OBJECTIVE: To determine the PI properties in response to middle ear pressure therapy (MEPT) induced by the EFET01 in guinea pigs. MATERIAL AND METHODS: In seven healthy guinea pigs, bi-phasic pressure pulses from -5 to 12 cm H2O were delivered to the external ear canal and transmitted to the middle and inner ear cavities with an intact tympanic membrane. Hydrostatic pressure change in the inner ear perilymphatic compartment was measured by a servo-controlled micropipette system. RESULTS: From eight successful ears, pressure changes in the middle ear slightly decreased and were instantly transferred to the inner ear. The EFET01 produces a bi-phasic positive/negative pressure pulse, which is approximately twice as large as the monophasic pressure pulse. CONCLUSION: Our study clarified the EFET01's ability to transmit pressure and verified its effectiveness in MD patients as observed in clinical studies. SIGNIFICANCE: The PI properties in guinea pig response to MEPT with the EFET01 device were investigated.

Estimated number and prevalence of patients with delayed endolymphatic hydrops in Japan: a nationwide survey.

BACKGROUND: Delayed endolymphatic hydrops (DEH) is a rare disease, and the actual number of patients in Japan remains unknown. OBJECTIVE: To investigate the number and prevalence of patients with DEH in Japan. METHODS: In total, 781 departments of otolaryngology in Japan were selected for survey by stratified random sampling according to the total number of hospital beds. We sent questionnaires to the target departments and collected data regarding the number of patients with DEH who visited those departments in 2019. RESULTS: The overall response rate was 68.0% (531 departments). The estimate number of patients with DEH in Japan was 962, and the prevalence was calculated to be 0.8 per 100,000 population. CONCLUSION: Patients with DEH were extremely rare in Japan. SIGNIFICANCE: This may be the first nationwide epidemiological study on the number and prevalence of patients with DEH in Japan or in the world.

Application of machine learning in the diagnosis of vestibular disease.

Machine learning is considered a potential aid to support human decision making in disease prediction. In this study, we determined the utility of various machine learning algorithms in classifying peripheral vestibular (PV) and non-PV diseases based on the results of equilibrium function tests. A total of 1009 patients who had undergone our standardized neuro-otological examinations were recruited. We applied five supervised machine learning algorithms (random forest, adaboost, gradient boosting, support vector machine, and logistic regression). After preprocessing the data, optimizing the hyperparameters using GridSearchCV, and performing a final evaluation on the test set using scikit-learn, we evaluated the predictive capability using various performance metrics, namely, accuracy, F1-score, area under the receiver operating characteristic curve, precision, recall, and Matthews correlation coefficient (MCC). All five machine learning algorithms yielded satisfactory results; the accuracy of the algorithms ranged from 76 to 79%, with the support vector machine classifier having the highest accuracy. In cases where the predictions of the five models were consistent, the accuracy of the PV diagnostic results was improved to 83%, whereas it increased to 85% for the non-PV diagnostic results. Future research should increase the number of patients and optimize the classification methods to obtain the highest diagnostic accuracy.

A 14-year nationwide epidemiological analysis of delayed endolymphatic hydrops in Japan.

BACKGROUND: Delayed endolymphatic hydrops (DEH) is an inner ear disease that causes recurrent vertigo in the ipsilateral ear or fluctuating hearing in the contralateral ear due to endolymphatic hydrops secondary to preceding deafness. There are few reports of large, multicentre studies investigating the clinical-epidemiological characteristics of DEH. OBJECTIVE: This study aimed to clarify the characteristics of DEH in Japan. METHODS: Clinical data on 662 patients with DEH were analysed by nationwide, multicentre surveys conducted by the Peripheral Vestibular Disorders Research Group of Japan. RESULTS: The proportion of ipsilateral DEH (IDEH) was slightly higher than that of contralateral DEH (CDEH) at 55.4%. The time delay between onset of precedent deafness and onset of DEH was significantly longer for CDEH than for IDEH. The most common cause of precedent deafness was a disease of unknown cause with onset in early childhood (33.1%). Epidemiological characteristics were not significantly different between CDEH with and without vertigo. CONCLUSION: DEH appearing to be caused by viral labyrinthitis has a high rate of onset within 40 years of precedent deafness. Clinical and epidemiological characteristics of IDEH, CDEH with vertigo, and CDEH without vertigo were very similar. SIGNIFICANCE: The clinical-epidemiological characteristics of DEH in Japan were clarified.

Novel Application of an Ultrasonic Bone Aspirator for Endoscopic Modified Medial Maxillectomy.

Background: Endoscopic modified medial maxillectomy (EMMM) is a surgical technique developed to approach maxillary sinus lesions, such as papilloma and postoperative maxillary cyst, while preserving the postoperative nasal morphology and nasal function. In this technique, a diamond burr is used to remove the bone, which may damage adjacent soft tissue. We developed EMMM using an ultrasonic bone aspirator (UBA) instead of a conventional diamond burr. The purpose of this study was to clarify the effectiveness of the UBA in EMMM in comparison to the conventional diamond burr technique in terms of operative time, intraoperative complications, postoperative symptoms, and recurrence. Methods: The medical records of all patients who underwent EMMM at Toyama University Hospital between June 2014 and December 2021 were reviewed. Patients who met the inclusion criteria were separated into Group 1, in which the UBA was used for EMMM, and Group 2, in which a drill with a diamond burr was used. Data on patient demographics, operation time, frequency of intraoperative complications and postoperative symptoms, and recurrence were statistically compared between the two groups. Results: There were no significant differences between the two groups in the patient demographic data, operative time, postoperative symptoms, or frequency of recurrence. The frequency of intraoperative damage to adjacent soft tissues was significantly lower in patients who underwent EMMM with the UBA in comparison to those who underwent EMMM with a conventional diamond burr. Conclusion: The application of the UBA to EMMM can improve surgical safety and facilitate surgical procedures.

12-month effect of middle ear pressure therapy with the EFET01 device for intractable definite Meniere's disease and delayed endolymphatic hydrops after certification by the public health insurance system in Japan.

BACKGROUND: Middle ear pressure therapy (MEPT) is effective for intractable vertigo in patients with definite Meniere's disease (MD) and treatment-refractory delayed endolymphatic hydrops (DEH). Four-month MEPT with the EFET01®, an MEPT device developed in Japan and covered by national health insurance since September 2018, has shown efficacy. However, efficacy and safety after 12 months of treatment, which is appropriate for determining the therapeutic effect of MEPT devices, is unclear. OBJECTIVES: Examine the therapeutic effect of 12-month MEPT using the ETET01®. MATERIAL AND METHODS: Patients underwent MEPT using the EFET01® from September 2018 to July 2021. Thirty-three patients followed for >12 months were enrolled in this retrospective study. Clinical data were evaluated in the first and second 6-month treatment periods. Data from the second 6-month period were compared with data from an MEPT study using a different device. RESULTS: MEPT with the EFET01® significantly improved vertigo in the first period, with further improvement in the second period. The efficacy and safety were comparable to MEPT with other devices. CONCLUSIONS: MEPT with the EFET01® is effective for intractable vertigo in patients with definite MD and DEH, and 12-month follow-up is recommended. SIGNIFICANCE: The efficacy of 12-month MEPT with the EFET01® was demonstrated.

Efficiency of a novel middle ear pressure device for intractable definite Meniere's disease and delayed endolymphatic hydrops after certification by the public health insurance system in Japan.

BACKGROUND: Middle ear pressure therapy (MEPT) is effective in treating intractable vertigo in patients with definite Meniere's disease (MD) and delayed endolymphatic hydrops (DEH) refractory to conservative treatment. A novel middle ear pressure device, the EFET01®, which requires no transtympanic ventilation tubes, was developed in Japan, approved by the Japanese Ministry of Health, Labour and Welfare, and has been used under Japanese national health insurance since September 2018. OBJECTIVES: To examine short-term therapeutic effect of MEPT using the ETET01® compared with previous clinical trial results. METHODS: Patients selected according to Japan Society for Equilibrium Research (JSER) guidelines underwent MEPT using the EFET01 from September 2018 to July 2021, and 44 patients were enrolled in this retrospective study. Clinical data analysed at 4 months after the start of MEPT were compared with those of the previous clinical trial for the EFET01. RESULTS: MEPT using the EFET01 showed the same therapeutic efficacy as that of the previous clinical trial, i.e. improvement in the intensity and frequency of vertigo with no effect on hearing, even under JSER guidelines for proper use of MEPT. CONCLUSION: MEPT using the EFET01 provided an effective treatment option for intractable vertigo in patients with definite MD and DEH.

Flap suturing endonasal dacryocystorhinostomy assisted by ultrasonic bone aspirator.

BACKGROUND: In the external dacryocystorhinostomy (DCR), a sutured anastomosis technique performed between the nasal mucosal and lacrimal sac flaps reported by Dupuy-Dutemps and Bourguet was the gold standard and was believed to lead to the success of the surgery. However, because of the small working space, a flap suturing technique has not been completely established in endonasal DCR (END-DCR). OBJECTIVES: The effect of the modified flap suture anastomosis technique using a Sonopet ultrasonic bone aspirator was retrospectively compared to that using a diamond burr in patients with nasolacrimal duct obstruction. MATERIALS AND METHODS: One hundred ten patients underwent the modified flap suturing technique using the Sonopet, and 30 patients were operated on using a diamond burr. RESULTS: Successful patency of the lacrimal ostium (LO) was obtained in all patients in both groups. The rates of successful suturing during the operation and of a large diameter of the LO 3 months after the operation were significantly higher in patients in whom the Sonopet rather than the burr was used. CONCLUSIONS AND SIGNIFICANCE: The Sonopet might offer similar surgical outcome to the traditional microdrill DCR and is a safer means of bone removal in END-DCR in the small working space.

Localized Amyloidosis of the Nasal Mucosa: A Case Report and Review of the Literature.

Amyloidosis is a disorder of protein folding in which various proteins automatically aggregate into a highly abnormal fibrillar conformation. Amyloidosis is classified into systemic and localized forms depending on whether the abnormal proteins deposited in several different organs or only a single organ. In localized amyloidosis of the head and neck regions, laryngeal amyloidosis is common; however, localized amyloidosis of the nose is extremely rare. We herein report a case of localized amyloidosis of the nose and review the relevant literature on localized sinonasal amyloidosis. A 41-year-old man presented with a history of severe nasal obstruction, which had persisted for two decades. Nasal endoscopy and imaging studies showed extensive thickening of the bilateral nasal mucosa and diffuse submucosal deposition of calcification. After histopathological and systemic examinations, he was diagnosed with localized amyloidosis of the nasal mucosa. Septoplasty and bilateral inferior turbinoplasty, which consisted of mucosal resection using an ultrasonic bone curette, was performed and his symptoms markedly improved. Localized sinonasal amyloidosis has a good prognosis and surgical resection should be selected as a first-line treatment; however, clinicians should recognize the high probability of recurrence.

Long-term effect of transtympanic intermittent pressure therapy using a tympanic membrane massage device for intractable meniere's disease and delayed endolymphatic hydrops.

BACKGROUND: A 12-month follow-up study showed that middle ear pressure treatment with a transtympanic membrane massage (TMM) device had a similar effect to a Meniett device. OBJECTIVES: The effects of pressure treatment with a TMM device were retrospectively compared to the effects of treatment with a Meniett device in patients with Meniere's disease (MD) and delayed endolymphatic hydrops (DEH) who were followed for a minimum of 24 months. MATERIALS AND METHODS: Twenty-seven patients were treated with the TMM device and 14 patients were treated with a Meniett device. The insertion of a transtympanic ventilation tube was necessary for the Meniett device but not for the TMM device. RESULTS: In patients treated with the TMM and Meniett devices, the frequency of vertigo significantly improved at 19-24 months after treatment. The distribution of vertigo at 19-24 months after treatment did not differ between the patients treated with the two types of devices. Pressure treatment for 8 months or more was suitable to achieve remission. CONCLUSIONS AND SIGNIFICANCE: Middle ear pressure treatment for 8 months or more with a TMM or Meniett device was equally effective and provided minimally invasive treatment options for intractable MD and DEH.

Effects of transtympanic intermittent pressure therapy using a new tympanic membrane massage device for intractable Meniere's disease and delayed endolymphatic hydrops: a prospective study.

BACKGROUND: The retrospective study showed that the effect of the middle ear pressure treatment by the tramstympanic membrane massage (TMM) device was similar to that of the Meniett device. OBJECTIVES: The new TMM device named EFET device was prospectively evaluated in patients with Meniere's disease (MD) and delayed endolymphatic hydrops (DEH) and we compared the effects to the Meniett device. MATERIALS AND METHODS: 23 ears of 19 patients were treated with an EFET device, and 17 ears of 15 patients were treated with the Meniett device. All patients suffering from intractable MD and DEH were treated for 4 months. The insertion of a transtympanic ventilation tube was necessary for the Meniett device, but not the EFET device. RESULTS: In patients treated by the EFET and Meniett devices, the frequency of vertigo significantly improved after treatment. The distribution of vertigo outcomes at 4 months after treatment did not differ between patients treated with the both devices. CONCLUSIONS AND SIGNIFICANCE: Middle ear pressure treatment by the EFET device is effective and provides minimally invasive options for intractable MD and DEH like the Meniett device.

Clinicopathological Features of Thyroid-Like Low-Grade Nasopharyngeal Papillary Adenocarcinoma: A Case Report and Review of the Literature.

Thyroid-like low-grade nasopharyngeal papillary adenocarcinoma (TL-LGNPPA) is an extremely rare neoplasm of the nasopharynx. Accordingly, its clinical and pathological characteristics are not well-known. We report a case of TL-LGNPPA and review the relevant literature on TL-LGNPPA. A 38-year-old Japanese woman presented with a history of nasal obstruction that had persisted for 1 month after symptoms of a common cold (e.g., low-grade fever, sore throat, and fatigue). A pedunculated tumor of ~20 mm in diameter was found on the posterior edge of the nasal septum. The tumor was endoscopically resected. Based on careful histopathological and immunohistochemical examinations, it was diagnosed as TL-LGNPPA. At 5 years after surgery, the patient remained disease-free. TL-LGNPPA has a very good prognosis, and complete resection with a sufficient safety margin is recommended as the first-line treatment. The morphological characteristics and immunohistochemical findings, especially TTF-1 positivity and thyroglobulin negativity, are important for the diagnosis.

Case Report: Response to Intra-arterial Cisplatin and Concurrent Radiotherapy Followed by Salvage Surgery in a Patient With Advanced Primary Sinonasal Low-Grade Non-intestinal Adenocarcinoma.

Background: The clinical usefulness of concurrent chemoradiotherapy before surgery in the treatment of primary, locally advanced sinonasal low-grade, non-intestinal type adenocarcinoma (LG non-ITAC) is unclear. Methods: We present the first case report of the efficacy of super-selective intra-arterial cisplatin (CDDP) infusion concurrent with conventional fractionated radiotherapy (RT) for LG non-ITAC in a Japanese patient. Results: A white, rugged-marginal mass that was histopathologically diagnosed as LG non-ITAC occupied the right nasal cavity. Based on the imaging findings, including computed tomography, magnetic resonance imaging, and positron emission tomography, the tumor was diagnosed as T4aN0M0, stage IVa. After treatment, the nasal tumor disappeared leaving only a small bulge in the medial wall of the middle turbinate. The patient also underwent right transnasal ethmoidectomy performed as salvage surgery. A histopathological examination revealed that the lesion was replaced by granulation tissue with lymphocytic infiltration and hemosiderin-laden macrophages, and no viable tumor cells remained. In the seven years after treatment, the patient has not experienced any local recurrence or regional or distant metastasis. Conclusions: Super-selective intra-arterial CDDP infusion concurrent with conventional fractionated RT followed by salvage surgery might be useful for the management of sinonasal LG non-ITAC.

Lacrimal Drainage System Obstruction Following Inferior Turbinate Surgery.

Objective: To provide information on the clinical characteristics and management of an uncommon acquired nasolacrimal drainage system obstruction (NLDSO). Methods: A patient was treated with microdebrider-assisted endoscopic marsupialization for a cystic lesion located in the synechiae between the inferior turbinate and the lateral wall of the inferior meatus. Results: A 21-year-old woman suffered from epiphora and purulent discharge 1 week after inferior turbinate surgery. Endoscopy reveled synechiae between the inferior turbinate and the lateral wall of the inferior meatus. Computed tomography-dacrocystography (CT-DCG) showed enlargement of the nasolacrimal duct and cystic accumulation of contrast medium from the lacrimal orifice posteriorly. The patient was treated with nasal endoscopic marsupialization of the cyst and then powered inferior turbinoplasty. Six months after the operation, both endoscopy and CT scan showed a widely patent left inferior meatus and nasolacrimal orifice. Conclusions: We report the first case of post-operative NLDSO following inferior turbinate surgery. Nasal endoscopy and CT-DCG are useful to diagnose the location of a NLDSO. Otorhinolaryngologists should be aware that surgery may lead to the formation of a NLDSO and that endoscopic marsupialization is a curative treatment for these distal-end NLDSO. NLDSOs are caused by synechiae, which are a complication of inferior turbinate surgery.

Safety management in urgent endonasal trans-sphenoidal surgery for pituitary adenoma during the COVID-19 pandemic in Japan - A case report.

We report a 72-year-old woman who required urgent endonasal trans-sphenoidal surgery (eTSS) because of progressive visual field disturbance due to pituitary adenoma, in whom we conducted reverse-transcriptase-polymerase-chain-reaction (RT-PCR) for COVID-19 and chest CT before eTSS. We took care of her by following the rule for suspected infection patient, and safely completed her treatment without medical staff infection. Under COVID-19 pandemic state, essentially careful management including RT-PCR test and chest CT should be taken for the high infection risk surgeries to avoid the outbreak through the hospital. And the cost of RT-PCR test for the patients should be covered by the government budget.

Cerebral Hemodynamic Responses to the Sensory Conflict Between Visual and Rotary Vestibular Stimuli: An Analysis With a Multichannel Near-Infrared Spectroscopy (NIRS) System.

Sensory conflict among visual, vestibular, and somatosensory information induces vertiginous sensation and postural instability. To elucidate the cognitive mechanisms of the integration between the visual and vestibular cues in humans, we analyzed the cortical hemodynamic responses during sensory conflict between visual and horizontal rotatory vestibular stimulation using a multichannel near-infrared spectroscopy (NIRS) system. The subjects sat on a rotatory chair that was accelerated at 3°/s2 for 20 s to the right or left, kept rotating at 60°/s for 80 s, and then decelerated at 3°/s2 for 20 s. The subjects were instructed to watch white stripes projected on a screen surrounding the chair during the acceleration and deceleration periods. The white stripes moved in two ways; in the "congruent" condition, the stripes moved in the opposite direction of chair rotation at 3°/s2 (i.e., natural visual stimulation), whereas in the "incongruent" condition, the stripes moved in the same direction of chair rotation at 3°/s2 (i.e., conflicted visual stimulation). The cortical hemodynamic activity was recorded from the bilateral temporoparietal regions. Statistical analyses using NIRS-SPM software indicated that hemodynamic activity increased in the bilateral temporoparietal junctions (TPJs) and human MT+ complex, including the medial temporal (MT) area and medial superior temporal (MST) area in the incongruent condition. Furthermore, the subjective strength of the vertiginous sensation was negatively correlated with hemodynamic activity in the dorsal part of the supramarginal gyrus (SMG) in and around the intraparietal sulcus (IPS). These results suggest that sensory conflict between the visual and vestibular stimuli promotes cortical cognitive processes in the cortical network consisting of the TPJ, the medial temporal gyrus (MTG), and IPS, which might contribute to self-motion perception to maintain a sense of balance or equilibrioception during sensory conflict.

Prognostic Factors for Postoperative Visual Acuity in Patients with Rhinogenic Optic Neuropathy.

PURPOSE: To examine the prognostic factors related to postoperative visual acuity in patients with rhinogenic optic neuropathy. STUDY DESIGN: Retrospective observational study. MATERIALS AND METHODS: We retrospectively studied the medical records for 15 eyes of 15 patients who underwent surgery for the treatment of rhinogenic optic neuropathy between 31 January 2010 and 30 April 2018 at Toyama University Hospital. The patient age, sex, preoperative and postoperative visual acuity, duration from visual deficit to surgery, use of steroids, type of rhinogenic optic neuropathy, and the part of sinus lesion were analyzed. We also examined postoperative visual acuity for patients whose preoperative visual acuity was less than light perception. RESULTS: The type of optic neuropathy was sinusitis in 7 cases, mucocele in 5 cases, and pyocele in 3 cases. Visual acuity was improved in 9 cases and deteriorated or unchanged in 6 cases. Patients in the improvement group were significantly younger than those in the nonimprovement group (p=0.01). In univariate regression analysis, preoperative visual acuity and type of optic neuropathy significantly related to postoperative visual acuity. Finally, 4 of the 15 cases (27%) had preoperative visual acuity less than light perception, but there was an improvement in postoperative visual acuity in 2 of 4 cases (50%). CONCLUSIONS: Preoperative visual acuity is the predictive factor for postoperative visual acuity in patients with rhinogenic optic neuropathy, but even if the preoperative visual acuity is less than light perception, it can be improved by surgical treatment.

Third Epidemiological Analysis of Nasopharyngeal Carcinoma in the Central Region of Japan from 2006 to 2015.

The present study aimed to clarify the incidence and clinical outcomes of nasopharyngeal carcinoma (NPC) in the Chubu region of Japan from 2006 to 2015, compared with previous reports. A retrospective analysis was conducted based on medical records from 40 hospitals located in the Chubu region in the central Japanese main island, with a population of around 22.66 million individuals. This study was designed in line with to two previous clinical studies into NPC conducted in the same area of Japan. We recruited NPC patients diagnosed in hospitals across this area over a 10-year period (2006-2015) using a questionnaire about sex, age, primary site, clinical symptoms, pathology, Union for International Cancer Control (UICC) staging, serological exam, treatment, and survival. A total of 620 NPC patients were identified. The age-standardized incidence of NPC from 2006 to 2015 was 0.27 per 100,000 individuals per year. There were no significant differences between this study and the previous two studies conducted in the same area of Japan. The five-year overall survival rate for all patients was 75.9%, while those for patients with stages I, II, III, and IVA were 97%, 91%, 79%, and 68%, respectively. The age-standardized annual incidence of NPC in the present study was 0.27 per 100,000 individuals per year, which was relatively low and stable. The five-year overall survival rate for all NPC patients was significantly improved in this decade compared with previous studies. The smoking rates in male and female NPC patients were 64.5% and 18.8%, respectively, thereby suggesting the involvement of smoking in the incidence of NPC.

Lymphoepithelial carcinoma of the maxillary sinus: A case report and review of the literature.

RATIONALE: Most cases of lymphoepithelial carcinoma (LEC) occur in the nasopharynx, and LEC in the sinonasal tract is extremely rare; thus, the clinical characteristics of sinonasal LEC are not well known. PATIENT CONCERNS: A 63-year-old Japanese man presented with a three-week history of left cheek pain, nasal obstruction and cheek swelling. DIAGNOSES: Enhanced CT and MRI revealed a tumor of the left maxillary sinus that invaded the left orbit and hard palate, with multiple swollen left cervical lymph nodes. Open biopsy was performed, and the specimen was diagnosed as LEC. INTERVENTIONS: Alternating chemoradiotherapy (ALCRT) followed by salvage surgery was performed. OUTCOMES: Our patient has been disease free for 5 years. LESSONS: A literature review of the epidemiology, etiology, clinical course and management of sinonasal LEC is highlighted. We believe ALCRT followed by salvage surgery to be a highly optimal treatment for sinonasal LEC from the viewpoint of a balance between quality of life and a high curative effect.