A pain in the ear: Two case reports of nervus intermedius neuralgia and narrative review.

OBJECTIVE: Present two cases of nervus intermedius neuralgia (NIN) in which patients described unilateral deep ear pain as their primary complaint and present a summary of NIN cases reported in the literature. BACKGROUND: The nervus intermedius is a tiny branch of the facial nerve that, with neuralgia, can present as a deep ear pain (NIN). The International Classification of Headache Disorders, 3rd edition, (ICHD-3) criteria for an NIN diagnosis include a unilateral deep ear pain with possible radiation that occurs in paroxysms with sharp pain and a tactile trigger. METHODS: A PubMed search was conducted for NIN and geniculate neuralgia. Two patients recently diagnosed with NIN at a single clinic were selected for case reports to highlight the variability of symptom presentation. RESULTS: The two cases reported here and the 127 cases reported in the literature show a wider range of presentations than included in the ICHD-3 criteria, including variable pain radiation sometimes diagnosed as concurrent trigeminal, glossopharyngeal, or occipital neuralgia. Pain was reported as constant or paroxysmal, as well as dull, sharp, or neuralgiform with inconsistent presence of triggers. While ICHD-3 does mention reported taste change, lacrimation, and salivation, the literature reports a much wider range of potential features associated with NIN. Optimal medical treatment is unclear given the predominance of surgical reporting of positive response to microvascular decompression, nerve sectioning, or other procedures. The two cases described here were successfully managed medically. CONCLUSION: NIN can present as described in the ICHD-3, but a more variable presentation may be possible. More studies are needed to clarify presentation, optimal medical treatment, and surgical indications for patients with NIN, especially when patients have no clear neurovascular conflict on neuroimaging.

Skull base osteomyelitis: Comprehensive analysis and a new clinicoradiological classification system.

OBJECTIVE: Skull baseosteomyelitis (SBO) is a rare phenomenon that typically occurs in diabetic or immunocompromised patients, causing significant morbidity and mortality. This study aimed to analyze a single institution's treatment results in SBO patients and propose anew integrated clinicoradiological classification system. METHODS: The medical records of 32 SBO patients that were treated at a tertiary care center between 2006 and 2017 were retrospectively reviewed. A scoring system based on anatomical involvement according to MRI was created. Subsequently, the scoring system was integrated with cranial nerve dysfunction status and a clinical grading system (CGS) was proposed. RESULTS: Among the 32 patients, 78.1% were diabetic and 63% had cranial nerve dysfunction at presentation. Bone erosion based on CT was greater in the patients without regression (P = 0.046). The regression rate decreased from clinical grade (CG)1 to CG3 (P = 0.029). Duration of hospitalization increased as CG increased (P = 0.047). Surgery had no effect on regression status at the time of discharge (P = 0.41). The 1-year, 2-year, and 5-year overall survival rates were 82.2%, 70.8%, and 45.8%, respectively. CG was significantly correlated with overall survival but not with disease-specific survival (log-rank; P = 0.017, P = 0.362, respectively). CONCLUSION: SBO continues to pose a challenge to clinicians, and causes significant morbidity and mortality. The proposed new classification system can be an option for grouping SBO patients according to clinical and radiological findings, helping clinicians estimate prognosis.

The receptive field for visceral pain referred orofacially by the vagus nerves.

BACKGROUND: The nociceptive receptive field of the vagus nerves in animals includes virtually the entire thoracic, abdominal and laryngopharyngeal regions. However, the role of the vagus nerves in the transmission of visceral pain in humans, with the exception of pain from coronary artery diseases, is believed to be insignificant. AIM: The purpose of this report is to map out the clinical visceral pain receptive field of the vagus nerves relative to its nociceptive counterpart in animals. MATERIALS AND METHODS: The PubMed database and PMC were searched for case reports of patients with orofacial pain believed by the author(s) of the article to be referred from underlying non-cardiac thoracic, laryngopharyngeal or abdominal diseases. Reports of diseases for which non-neural explanations for the orofacial spread of pain were suggested were excluded. RESULTS: A total of 52 case reports of jaw pain and/or otalgia referred from laryngopharyngeal and noncardiac thoracic sources were discovered. In addition, a multicenter prospective study found that 25.8% of more than 3,000 patients with thoracic aortic dissection experienced pain in the head and neck region. In stark contrast, no case reports of orofacially referred pain from abdominal diseases were found. DISCUSSION: The results indicate that the laryngopharyngeal and thoracic portions of the vagal receptive field are capable of referring pain orofacially while the abdominal portion is not. The roles of the somatotopic organization of the trigeminal sub nucleus caudalis and neuromodulation in this referral of pain were discussed. CONCLUSION: Referred orofacial pain can lead to delayed diagnosis and poorer outcome in visceral diseases.

Administration of 1% topical voriconazole drops was effective and safe in the treatment of refractory otomycosis without tympanic membrane perforation.

BACKGROUND: Refractory otomycosis is a common condition that is difficult to treat. OBJECTIVES: This study aimed to evaluate the effectiveness of 1% topical voriconazole drops in the treatment of otomycosis. METHODS: This retrospective analysis was conducted from November 2017 to November 2019. Patients who had refractory otomycosis without tympanic membrane perforation confirmed by microbial culture and fluorescent staining were included in the study. All patients were treated with 1% topical voriconazole drops hourly at daytime for 2 weeks. Evaluation of effectiveness was conducted 1 month after the completion of topical voriconazole treatment. Before and after topical voriconazole treatment, hearing tests were performed in all patients. RESULTS: Fifty-five patients were included in this study. The reasons for refractoriness were resistant recurrence to imidazole drugs (50 cases, 90.9%) and difficulty in cleaning the external auditory canal (5 cases, 9.1%). The most common strain was Aspergillus terreus (50.9%), followed by Aspergillus flavus (29.1%), Aspergillus niger (10.9%), and Aspergillus fumigatus (9.1%). After 2 weeks of treatment with 1% topical voriconazole drops, otomycosis in all patients was resolved. There was no significant change in bone conduction before and after topical voriconazole treatment (paired t-test, P = 0.5023; linear correlation analysis, R2 = 0.98; equation, y = 1.003x-0.284). Adverse effects, such as blurred vision and phototoxicity, were not observed in any patient. CONCLUSIONS: Administration of 1% topical voriconazole drops was effective and safe in the treatment of refractory otomycosis without tympanic membrane perforation within 2 weeks.

Temporal Bone Chondroblastoma: Systematic Review of Clinical Features and Outcomes.

OBJECTIVE: Chondroblastoma is an uncommon benign neoplasm of cartilaginous origin usually involving the long bones. The temporal bone is a rare location for this tumor. The clinical profile, optimal medical and surgical management, and outcomes of treatment for temporal bone chondroblastoma remain unknown. METHODS: We performed a systematic review of the SCOPUS, PubMed, and CENTRAL databases for case reports and case series on patients with histopathologically proven temporal bone chondroblastoma. Data on demographics, clinical manifestation, surgical management, adjuvant treatment, and outcome on last follow-up were collected. RESULTS: A total of 100 cases were reported in the literature, including one described in the current study. The mean age of patients was 42.3 years (range, 2-85 years), with a slight male predilection (1.3:1). The most common clinical manifestations were otologic symptoms (e.g., hearing loss [65%], tinnitus, and otalgia) and a palpable mass. Surgical excision was performed in all cases, with gross total excision achieved in 58%. Radiation therapy was performed in 18% of cases, mostly as adjuvant treatment after subtotal excision. There were no deaths at a median follow-up of 2 years. Among the patients with detailed status on follow-up, 58% had complete neurologic recovery, 38% had partial recovery, and 4% had progression of symptoms as a result of tumor recurrence. CONCLUSIONS: Temporal bone chondroblastoma has a distinct clinical profile from chondroblastoma of long bones. Surgery is the mainstay of treatment, and radiation therapy may be given after subtotal excision. Outcomes are generally favorable after treatment.

Hemorrhage within the tympanic membrane without perforation.

BACKGROUND: Hemotympanum refers to both the presence of blood in the middle ear cavity and to ecchymosis of the tympanic membrane (TM), and a systematic study of intra-TM (iTM) hemorrhage without bleeding in the middle ear cavity has not been conducted. The goals of our study were to analyze the causes of iTM hemorrhage without TM perforation or bleeding in the middle ear cavity, and to demonstrate the clinical characteristics of the disease. METHODS: This Case series study included five patients with iTM hemorrhage between August 2014 and August 2017. An iTM hemorrhage was diagnosed when otoendoscopic examination demonstrated minor bleeding behind the intact TM, a hemorrhage was observed between the TM annulus and the epidermal layer, and temporal bone computed tomography revealed thickening of the TM without soft tissue density within the tympanic cavity or temporal bone fracture. Initial symptoms, and serial findings of otoendoscopy and pure tone audiometry (PTA) were investigated. RESULTS: iTM hemorrhage developed due to blunt head trauma in two patients, descent barotrauma during scuba diving in two patients, and spontaneous epistaxis in one patient. Otalgia and ear fullness were the most common symptoms, but PTA showed no or minimal conductive hearing loss in all patients. CONCLUSIONS: An iTM hemorrhage may develop after blunt head trauma, barotrauma due to scuba diving, or spontaneous epistaxis; otological symptoms included otalgia, tinnitus, and aural fullness. An iTM hemorrhage resolved spontaneously without specific treatment, usually within 1 month.

An Integrative Model Accounting for the Symptom Cluster Triggered After an Acoustic Shock.

Acoustic shocks and traumas sometimes result in a cluster of debilitating symptoms, including tinnitus, hyperacusis, ear fullness and tension, dizziness, and pain in and outside the ear. The mechanisms underlying this large variety of symptoms remain elusive. In this article, we elaborate on the hypothesis that the tensor tympani muscle (TTM), the trigeminal nerve (TGN), and the trigeminal cervical complex (TCC) play a central role in generating these symptoms. We argue that TTM overuse (due to the acoustic shock), TTM overload (due to muscle tension), and ultimately, TTM injury (due to hypoxia and "energy crisis") lead to inflammation, thereby activating the TGN, TCC, and cortex. The TCC is a crossroad structure integrating sensory inputs coming from the head-neck complex (including the middle ear) and projecting back to it. The multimodal integration of the TCC may then account for referred pain outside the ear when the middle ear is inflamed and activates the TGN. We believe that our model proposes a synthetic and explanatory framework to explain the phenomena occurring postacoustic shock and potentially also after other nonauditory causes. Indeed, due to the bidirectional properties of the TCC, musculoskeletal disorders in the region of the head-neck complex, including neck injury due to whiplash or temporomandibular disorders, may impact the middle ear, thereby leading to otic symptoms. This previously unavailable model type is experimentally testable and must be taken as a starting point for identifying the mechanisms responsible for this particular subtype of tinnitus and its associated symptoms.

Comparison of clinical outcomes of three different packing materials in the treatment of severe acute otitis externa.

OBJECTIVE: To analyse the clinical outcomes of biodegradable synthetic polyurethane foam versus ribbon gauze and ear wick in the treatment of severe acute otitis externa. METHODS: Ninety-two adults with severe acute otitis externa were randomly assigned to groups receiving ear wick (n = 28), ribbon gauze (n = 34) or biodegradable synthetic polyurethane foam (n = 30). Clinical efficacy, in terms of otalgia, oedema, erythema and tenderness of the external auditory canal, was assessed before packing was applied and at follow up on the 3rd and 7th days of presentation. RESULTS: All packing materials were associated with improved otalgia and oedema on the 3rd day; however, there were significant differences between biodegradable synthetic polyurethane foam and the other packing materials, and there was no significant reduction in tenderness in the biodegradable synthetic polyurethane foam group on the 3rd day. In the ribbon gauze and ear wick groups, improvements in all clinical efficacy scores were statistically significant for all pairwise comparisons. CONCLUSION: The three packing materials were all quite effective in treating severe acute otitis externa, but ear wick and ribbon gauze were superior to biodegradable synthetic polyurethane foam for relieving signs and symptoms, especially on the 3rd day.

The possible effect of human menopausal gonadotropin on the audio-vestibular system.

OBJECTIVE: Human menopausal gonadotropin (HMG) is one of the commonest drugs used for ovarian stimulation with no reports on the audio-vestibular system. This study aims to examine HMG on the hearing profile of patients planning intracytoplasmic sperm injection (ICSI). METHODS: This prospective study was conducted from June 2016 to June 2017 in a tertiary referral hospital. The audio-vestibular system of a total of 30 patients was evaluated using pure tone audiometry, distortion product otoacoustic emissions (DPOAEs in the form of a DP-gram) and Vestibular-evoked myogenic potential (VEMP) immediately before therapy and at the day 10 after therapy. Audio-vestibular adverse effects including hearing loss, tinnitus, vertigo, and otalgia were also considered. RESULTS: Significant elevations in hearing thresholds were found on comparing thresholds at the day 10 at the onset of the study. The elevations were mostly at frequencies (1000, 2000 and 8000Hz) and did not affect speech perception. For DPOAE, significant differences were observed at all F2 frequencies on comparing both amplitudes and signal to noise ratios. Otologic complaints were significant for tinnitus and hearing loss. CONCLUSION: Significant auditory and vestibular adverse effects may result from HMG therapy, indicating the importance of prompt monitoring of auditory functions in these patients.

Manifestation of granulomatosis with polyangiitis in head and neck.

OBJECTIVES: Granulomatosis with polyangiitis is a rare autoimmune disease of the group of antineutrophil cytoplasmic antibody (ANCA)-associated vasculitides. Involvement of the ear, nose and throat (ENT)-region is only described in a few case series and case reports. The objective of this study is to systematically characterise the ENT-involvement in a large series of GPA patients. METHODS: GPA patients examined in the Department of Otorhinolayngology of the Christian-Albrechts-University of Kiel between 1990 and 2012 were included. Diagnosis was based on histological, serological and clinical parameters. GPA patients were examined in a standardised way based on the Ear Nose and Throat Activity Score (ENTAS) or its precursor. Medical history, ENT examination, diagnostic findings (ear, nose) and cranial radiology were documented cumulatively. RESULTS: A total of 230 GPA patients were included in this study. Over 95% of them showed ENT-involvement. 59% of the patients showed nasal obstructions, 57% a loss of smell. A hearing loss was diagnosed in 23% of the patients, 50% involvement in MR or CT scans and 15% showed laryngeal involvement. CONCLUSIONS: The data of the largest monocentric study presented here demonstrate a frequent ENT-involvement in GPA patients. Rhinological and ontological manifestations are most common.

Retroauricular pain caused by Eagle syndrome: A rare presentation due to compression by styloid process elongation.

Eagle syndrome is a rare condition presenting with retroauricular pain (usually as main symptom) associated with dysphagia, headache, neck pain on rotation and, much rarelier, stroke. This occurs due to styloid process elongation. Sometimes, there is also styloid ligament calcification, which can cause compression of nerves and arteries and the symptoms above. Treatment can be conservative with pain modulators (e.g. pregabalin) or infiltrations (steroids or anesthetics drugs). In refractory cases, surgical approach aiming to reduce the size of the styloid process can be performed. We present a rare case of Eagle syndrome (documented by computed tomography) with good response to clinical treatment.

Retroauricular pain caused by Eagle syndrome: A rare presentation due to compression by styloid process elongation / Dor retroauricular causada pela síndrome de Eagle: uma apresentação rara por compressão pelo alongamento do processo estiloide

Summary Eagle syndrome is a rare condition presenting with retroauricular pain (usually as main symptom) associated with dysphagia, headache, neck pain on rotation and, much rarelier, stroke. This occurs due to styloid process elongation. Sometimes, there is also styloid ligament calcification, which can cause compression of nerves and arteries and the symptoms above. Treatment can be conservative with pain modulators (e.g. pregabalin) or infiltrations (steroids or anesthetics drugs). In refractory cases, surgical approach aiming to reduce the size of the styloid process can be performed. We present a rare case of Eagle syndrome (documented by computed tomography) with good response to clinical treatment.

Resumo A síndrome de Eagle é uma condição rara na qual ocorre dor retroauricular (usualmente é o principal sintoma) associada a disfagia, cefaleia, cervicalgia durante a rotação da cabeça e, mais raramente, a AVC. Isso ocorre por conta do alongamento do processo estiloide e, às vezes, há também calcificação do ligamento estiloide. Essas estruturas podem comprimir nervos e artérias causando os sintomas citados. O tratamento pode ser conservador com moduladores da dor, como pregabalina, ou com infiltrações (corticoides ou drogas anestésicas). Em casos refratários, cirurgia para reduzir o tamanho do processo estiloide pode ser realizada. É apresentado um caso raro de síndrome de Eagle (documentado com tomografia computadorizada) com boa resposta ao tratamento clínico.

Effects of a sensory branch to the posterior external ear canal: coughing, pain, Ramsay Hunt's syndrome and Hitselberger's sign.

BACKGROUND: A distinct nerve innervating the external auditory canal can often be identified in close relation to the facial nerve when gradually thinning the posterior canal wall. This nerve has been attributed to coughing during cerumen removal, neuralgic pain, Hitselberger's sign and vesicular eruptions described in Ramsay Hunt's syndrome. This study aimed to demonstrate the origin and clinical impact of this nerve. METHODS AND RESULTS: In patients with intractable otalgia or severe coughing whilst inserting a hearing aid, who responded temporarily to local anaesthesia, the symptoms could be resolved by sectioning a sensory branch to the posterior canal. In a temporal bone specimen, it was revealed that this nerve is predominantly a continuation of Arnold's nerve, also receiving fibres from the glossopharyngeal nerve and facial nerve. Histologically, the communicating branch from the facial nerve was confirmed. CONCLUSION: Surgeons should be aware of the posterior auricular sensory branch and its clinical implications.

Frequency of temporomandibular disorder signs and symptoms among call center employees.

OBJECTIVE: The current study aimed to investigate the frequency of signs and symptoms of temporomandibular disorders (TMD) among call center employees. METHODS: Workers from four call centers (CC) were invited to participate in this study. The examination was based on the Research Diagnostic Criteria for Temporomandibular Disorder (RDC/TMD). The tenderness of the masticator muscles was assessed on both sides by bilateral palpation. The mobility of the mandible was measured with a plastic millimeter ruler on opening, lateral excursions, and protrusion. RESULTS: In total, 124 call center employees with a mean age of 28.61 ± 4.71 (between 22 and 47 years of age) were enrolled in this study. There was no statistically significant relation between the numbers of calls answered in a day and teeth clenching, teeth grinding, earache, tinnitus, or pain on yawning/chewing. Only protrusive movement pain and joint noise were significantly higher among employees who answered more than 140 calls/day (p < 0.01). An evaluation of the TMD signs and symptoms in relation to job stress level revealed that job stress level significantly affected the incidence of headaches among call center employees (p < 0.01). CONCLUSION: The present study demonstrated no statistically significant relation between TMD signs and symptoms and call center employees except protrusive movement pain and joint noise. This relation was seen only in the employees who answered more than 140 calls per day. Headache, teeth clenching, and TMJ noise were the signs and symptoms encountered most often in this study. Multicentered studies in different geographic locations should be conducted to eliminate the limitation of this study.

Otological symptoms and audiometric findings in patients with temporomandibular disorders: Costen's syndrome revisited.

OBJECTIVE: Otological symptoms (otalgia, subjective hearing loss, blocked ear sensation, tinnitus and vertigo) associated with temporomandibular disorders are documented features of Costen's syndrome. However, the origin of these symptoms and the causes of hearing loss are unknown. This study aimed to characterise hearing loss in a large number of patients with temporomandibular disorders. The causes of these symptoms were explored in patients with otological symptoms and normal audiometric findings. METHODS: A prospective case study and literature review were performed. The audiometric features of 104 temporomandibular disorder patients were compared with those of 110 control participants. RESULTS: A large proportion of temporomandibular disorder patients had several otological symptoms. Twenty-five per cent of unilateral or bilateral temporomandibular disorder patients had either unilateral (ipsilateral) or bilateral hearing loss; respectively, which was usually mild (p = 0.001). Hearing loss was predominantly sensorineural. CONCLUSION: The main cause of otological symptoms (apart from otalgia) and of audiometric findings in temporomandibular disorder patients is postulated to be an altered middle-ear to inner-ear pressure equilibrium.