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1.
Orthod Craniofac Res ; 27(4): 635-644, 2024 Aug.
Article in English | MEDLINE | ID: mdl-38512245

ABSTRACT

OBJECTIVE: To investigate the effects of congenital unilateral first permanent molar occlusal loss (CUMOL) on the morphology and position of temporomandibular joint (TMJ). MATERIALS AND METHODS: Cone-beam computed tomography (CBCT) images of 37 patients with CUMOL (18 males and 19 females, mean age: 13.60 ± 4.38 years) were divided into two subgroups according to the status of second molar (G1: the second molar not erupted, n = 18, G2: second molar erupted, n = 19). The control group consisted of 33 normal occlusion patients (9 males and 24 females, mean age: 16.15 ± 5.44 years) and was divided into 2 subgroups accordingly (G3: the second molar had not erupted, n = 18, G4: the second molar had erupted and made contact with the opposing tooth, n = 15). Linear and angular measurements were used to determine the characteristics of TMJ. RESULTS: In G1, the condyle on the side of the CUMOL shifts posteriorly, with significant side differences observed in Anterior space (AS, P < .05) and Posterior space (PS, P < .05). However, with the eruption of the second permanent molars, in G2, the condyle on the CUMOL side moves posteriorly and inferiorly. This results in significant lateral differences in the AS (P < .05), PS (P < .05), and Superior space (SS, P < .05). Additionally, there is an increase in the thickness of the roof of the glenoid fossa (TRF) on the CUMOL side (P < .05), and a decrease in the inclination of the bilateral articular eminences (P < .05). CONCLUSIONS: CUMOL can affect the position and the morphology of the condyle and was associated with the eruption of the second permanent molars. Before the eruption of the second permanent molars, CUMOL primarily affects the position of the condyle. After the emergence of the second permanent molars, CUMOL leads to changes in both the condyle's position and the morphology of the glenoid fossa.


Subject(s)
Cone-Beam Computed Tomography , Molar , Temporomandibular Joint , Humans , Cone-Beam Computed Tomography/methods , Female , Male , Retrospective Studies , Molar/diagnostic imaging , Adolescent , Temporomandibular Joint/diagnostic imaging , Temporomandibular Joint/pathology , Child , Mandibular Condyle/diagnostic imaging , Mandibular Condyle/pathology , Mandibular Condyle/abnormalities , Temporal Bone/diagnostic imaging , Temporal Bone/pathology , Young Adult
2.
Eur Arch Otorhinolaryngol ; 281(2): 731-735, 2024 Feb.
Article in English | MEDLINE | ID: mdl-37555931

ABSTRACT

PURPOSE: This study is to evaluate the duration of facial nerve enhancement in gadolinium-enhanced temporal bone MRI after the onset of acute facial palsy. METHODS: Gd-enhanced MRI imagines were examined in 13 patients with idiopathic acute facial palsy within 14 days after the onset. The degree of facial nerve function was measured according to the House-Brackmann (H-B) grading system at their first visit at outpatient clinic. The follow-up MRI was taken about 16.5 months (7-24 months) after onset of disease. The degree of facial nerve enhancement was measured with signal intensity (SI) which was quantitatively analyzed using the region-of-interest (ROI) measurements for each segment of the facial nerve. SI was statistically analyzed by comparing SI values of contralateral site and ipsilateral site using the paired t test with SPSS program. RESULTS: The gadolinium enhancement was statistically increased at labyrinthine segment and geniculate ganglion area of facial nerve at initial temporal bone MRI. The gadolinium enhancement was statistically decreased at all the segments of facial nerve except tympanic segment (p < 0.05) at follow-up MRI. CONCLUSIONS: The facial nerve enhancement in Gd-enhanced MRI images prolonged more than 21 months of the onset. The newly developed pathologic lesions of acute facial palsy especially occur at the site of labyrinthine and geniculate ganglion.


Subject(s)
Bell Palsy , Facial Paralysis , Humans , Facial Paralysis/diagnostic imaging , Facial Paralysis/etiology , Facial Paralysis/pathology , Facial Nerve/diagnostic imaging , Facial Nerve/pathology , Contrast Media , Gadolinium , Bell Palsy/diagnostic imaging , Temporal Bone/diagnostic imaging , Temporal Bone/pathology , Magnetic Resonance Imaging/methods
3.
Eur Arch Otorhinolaryngol ; 281(6): 2967-2974, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38165436

ABSTRACT

OBJECTIVE: This study investigates the importance of bone density, surface area, and diameter of anatomical structures of the superior semicircular canal (SSC), lateral semicircular canal (LSC), posterior semicircular canal (PSC), utricle, and saccule in patients diagnosed with superior semicircular canal dehiscence (SSCD). MATERIALS AND METHODS: The bone density, surface area, and diameter of SSC, LSC, PSC, utricle, and saccule were measured and compared between the SSCD group and control group. Fifteen ears in the SSCD group and 60 ears in the control group were evaluated. Additionally, within the SSCD group, the dehiscent and healthy sides were evaluated independently. RESULTS: SSC's bone density was significantly lower in the SSCD group compared to the control group (p = 0.008). No significant differences were found in surface area and diameter between the groups (p > 0.05). While most of the anatomical structures showed no significant difference in bone density between dehiscent and healthy ears (p > 0.05), SSC bone density was significantly lower in affected ears (p = 0.000) in SSCD group. CONCLUSION: Based on the data obtained in this study, bone density and anatomical structure may be useful in patients diagnosed with SSCD.


Subject(s)
Bone Density , Semicircular Canal Dehiscence , Semicircular Canals , Humans , Female , Male , Middle Aged , Semicircular Canals/pathology , Semicircular Canals/diagnostic imaging , Semicircular Canals/anatomy & histology , Adult , Semicircular Canal Dehiscence/pathology , Semicircular Canal Dehiscence/diagnostic imaging , Aged , Case-Control Studies , Tomography, X-Ray Computed , Temporal Bone/diagnostic imaging , Temporal Bone/anatomy & histology , Temporal Bone/pathology , Saccule and Utricle/pathology , Saccule and Utricle/diagnostic imaging
4.
Int Tinnitus J ; 27(2): 146-153, 2024 Mar 21.
Article in English | MEDLINE | ID: mdl-38507628

ABSTRACT

Opacification in the middle ear and mastoid region can stem from a wide range of factors. In terms of diagnostic imaging, CT is the primary tool due to its exceptional spatial resolution, particularly for examining the temporal bone and ossicles. MRI complements this by offering detailed soft tissue lesion characterization and assessing involvement in the inner ear and cranial nerves. This study focuses on inflammatory causes of opacification in the middle ear and mastoid, with an emphasis on the utility of CT and MRI. This comprehensive review aimed to provide a practical framework for considering potential differential diagnoses.


Subject(s)
Ear, Middle , Tomography, X-Ray Computed , Humans , Tomography, X-Ray Computed/methods , Ear, Middle/diagnostic imaging , Ear, Middle/pathology , Temporal Bone/diagnostic imaging , Temporal Bone/pathology , Mastoid/diagnostic imaging , Mastoid/pathology , Magnetic Resonance Imaging/methods
5.
Audiol Neurootol ; 28(2): 94-105, 2023.
Article in English | MEDLINE | ID: mdl-36265460

ABSTRACT

INTRODUCTION: Dizziness is a common disease. However, approximately 10-40% of patients were diagnosed unknown dizziness even though general, neurological, and otological examinations were performed. The aim of this otopathological study was to investigate the histopathology of the peripheral vestibular system of patients who suffered from undiagnosed dizziness. METHODS: Eighteen temporal bone specimens from 9 patients with undiagnosed dizziness and 20 temporal bone specimens from age-matched 10 normal controls were selected. Cases with a history of dizziness and vertigo caused by particular peripheral vestibular disease and central etiology were excluded. Specimens of the vestibular system were carefully assessed by light microscopy. The basophilic deposits adhered to cupulae of the semicircular canals and the wall of the labyrinth were investigated. Scarpa's ganglion cell counts in the vestibular nerves were performed. RESULTS: Fifteen ears of 9 patients had the findings of vestibular pathology such as a basophilic deposit on cupula (8 ears), on canal wall (7 ears), vestibular nerve loss (8 ears), or vestibular atelectasis (2 ears). Unclear pathological findings such as crista neglecta, subepithelial deposits of the crista ampullaris, and adhesion of the cupula to dark cell area were demonstrated. The mean size of basophilic deposits seen in the patients (mean: 191 µm) was larger than that of latent deposits seen in the normal controls (mean: 101 µm; p = 0.01). CONCLUSIONS: We demonstrated some peripheral vestibular pathological findings such as deposit within the semicircular canal, vestibular nerve loss, and vestibular atelectasis and suggested the possible diagnosis of dizziness (benign paroxysmal positional vertigo, presbyvestibulopathy, vestibular atelectasis). These findings will provide a better insight into the multiple etiologies of the unknown dizziness in the elderly.


Subject(s)
Dizziness , Vestibule, Labyrinth , Humans , Aged , Dizziness/diagnosis , Benign Paroxysmal Positional Vertigo/complications , Benign Paroxysmal Positional Vertigo/diagnosis , Benign Paroxysmal Positional Vertigo/pathology , Temporal Bone/pathology , Semicircular Canals
6.
Ear Hear ; 44(4): 661-669, 2023.
Article in English | MEDLINE | ID: mdl-36763469

ABSTRACT

OBJECTIVES: The author's objective was to evaluate sex and race representation in temporal bone histopathology studies. DESIGN: PubMed, Embase, Cochrane, Web of Science, and Scopus were searched for studies written in English examining temporal bone histopathology specimens from U.S.-based institutions from January 1, 1947, to September 1, 2021. Two authors then performed "snowballing" by reviewing references from the initial search and included the studies that fulfilled the inclusion criteria. For each study, the following information was collected: publication details, study design, funding, institution from where temporal bone specimens were procured, number of study specimens, and donor demographical information. RESULTS: The authors found that out of 300 studies, 166 (55%) report sex while only 15 (5%) reported race information. Over the past 70 years, the ratio of studies reporting sex to those that do not has increased from 1.00 to 2.19 and the number of female temporal bone histopathology subjects relative to male has increased from 0.67 to 0.75. Over 90% of studies that do report this information feature participant racial compositions that do not reflect the diversity of the U.S. population. CONCLUSIONS: Studies of temporal bone histopathology often do not report participant sex or race. The reporting of participant sex and the inclusion of specimens from female donors have both increased over time. However, temporal bone histopathology study cohorts are not representative of the racial diversity of the U.S. population. The otolaryngology community must strive to build temporal bone histopathology libraries that are representative of the diverse U.S. population.


Subject(s)
Temporal Bone , Female , Humans , Male , Research Design , United States , Temporal Bone/pathology , Racial Groups , Sex
7.
Am J Otolaryngol ; 44(5): 103952, 2023.
Article in English | MEDLINE | ID: mdl-37329696

ABSTRACT

OBJECTIVE: To report 20 years of natural history data for a facial paraganglioma and provide a comprehensive review of the existing literature. PATIENT: 81-year-old female with a remote history of cardiac arrest while under anesthesia who elected to observe her facial paraganglioma for 20 years. INTERVENTIONS: Observation, clinical documentation, radiographic surveillance. MAIN OUTCOME MEASURES: Tumor progression, patient symptomatology, and review of management options. RESULTS: The initial presentation of the facial paraganglioma was facial spasm. Over the course of observation, symptoms progressed to include complete facial nerve paralysis, pulsatile tinnitus, and otalgia on the affected side. Radiologic surveillance demonstrated incremental growth and erosion of surrounding structures, including the posterior external auditory canal, stylomastoid foramen, and lateral semicircular canal with near-dehiscence. Twenty-four cases of facial paraganglioma were identified in the extended literature search and are summarized herein. CONCLUSIONS: This unique case contributes to the scarce literature surrounding facial paragangliomas by reporting the extended natural history of this disease.


Subject(s)
Cranial Nerve Neoplasms , Facial Nerve Diseases , Facial Paralysis , Paraganglioma , Humans , Female , Aged, 80 and over , Cranial Nerve Neoplasms/diagnosis , Paraganglioma/diagnostic imaging , Paraganglioma/surgery , Facial Nerve Diseases/etiology , Facial Nerve Diseases/complications , Facial Paralysis/etiology , Temporal Bone/pathology
8.
Am J Otolaryngol ; 44(4): 103880, 2023.
Article in English | MEDLINE | ID: mdl-37003029

ABSTRACT

BACKGROUND: Metastatic disease to the temporal bone is rare. Even more uncommonly, it can be the first manifestation of an underlying malignancy. Patients typically present late in the disease process with non-specific symptoms of hearing loss, facial nerve palsy and otorrhea. CASE: A 62-year-old Chinese female presented with right facial weakness, which had near-complete improvement in response to pulse prednisolone. On examination, she had a right temporal swelling and right mild-severe conductive hearing loss. A computed tomography scan showed a destructive lesion centred in the squamous temporal bone, with an associated soft tissue component. Positron emission tomography scan revealed bony and lung metastases, but no distinct hypermetabolic primary site. An incisional biopsy unexpectedly returned as metastatic lung adenocarcinoma. CONCLUSION: Although rare, it is important for otolaryngologists to be aware of the insidious nature of temporal bone metastases and possible atypical clinical and radiological features, to facilitate timely workup and initiation of treatment.


Subject(s)
Adenocarcinoma of Lung , Adenocarcinoma , Bone Neoplasms , Facial Paralysis , Lung Neoplasms , Female , Humans , Middle Aged , Adenocarcinoma/diagnostic imaging , Adenocarcinoma/therapy , Adenocarcinoma of Lung/complications , Adenocarcinoma of Lung/pathology , Lung Neoplasms/pathology , Bone Neoplasms/diagnostic imaging , Bone Neoplasms/pathology , Temporal Bone/diagnostic imaging , Temporal Bone/pathology
9.
Br J Neurosurg ; 37(3): 302-303, 2023 Jun.
Article in English | MEDLINE | ID: mdl-31979993

ABSTRACT

INTRODUCTION: Cholesteatomas are slowly progressive and well demarcated lesions but if not treated, they can spread to the inner ear and brain and lead to serious neurological complications. CASE REPORT: A 59-year old male was referred to our practice. His initial imaging showed that a huge intracranial mass has spread to the neck. Beyond surgical exploration, a sizable temporal bone lesion with extensive keratinisation was observed which later on histopathological exam turned out to be a cholesteatoma. CONCLUSIONS: Cholesteatomas may be aggressive lesions by rapidly growing and gross destructive features. In such cases, more attempts should be given to ruling out any malignant neoplastic tissues.


Subject(s)
Cholesteatoma, Middle Ear , Cholesteatoma , Male , Humans , Middle Aged , Cholesteatoma, Middle Ear/diagnostic imaging , Cholesteatoma, Middle Ear/surgery , Cholesteatoma, Middle Ear/complications , Cholesteatoma/diagnostic imaging , Cholesteatoma/surgery , Cholesteatoma/complications , Temporal Bone/pathology , Neck
10.
J Craniofac Surg ; 34(1): 120-125, 2023.
Article in English | MEDLINE | ID: mdl-36000749

ABSTRACT

Encephaloceles rarely develop following traumatic skull fractures. Given their low incidence, the clinical presentations and management strategies of these lesions are confined to case reports and limited case series. A systematic literature review was performed using PubMed, Ovid, and Web of Science databases to identify relevant articles using Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. A total of 37 articles met inclusion criteria, including the case presented herein. These articles reported 52 traumatic encephaloceles. Mean patient age was 25.3 years (range 6 mo-66 y) with a male predominance (63%, 33/52). The most common bony defects resulting in encephalocele formation were the orbital roof (52%, 27/52), ethmoid (35%, 18/52), and sphenoid (10%, 5/52). Mean time from traumatic injury to initial presentation was 21.3 months (range 0 d-36 y) with a bimodal distribution split between immediately following the traumatic injury (57%, 26/46) or in a delayed manner (43%, 20/46). Common presentations of orbital roof, frontonasal, and temporal bone encephaloceles were exophthalmos (85%, 23/27), cerebrospinal fluid rhinorrhea (71%, 17/24), and hearing loss (100%, 4/4), respectively. Operative approach, repair technique, and materials used for encephalocele reduction were highly variable. Surgical intervention afforded definitive symptomatic improvement or resolution in the majority of cases (89%, 42/47). Clinical outcomes did not differ between orbital, frontonasal, or temporal bone encephaloceles ( P =0.438). Traumatic encephaloceles are a rare entity with diverse presenting symptomatology dependent upon the location of fracture dehiscence. Surgical intervention affords symptomatic improvement in the majority of cases irrespective of encephalocele location, time to presentation, or operative approach.


Subject(s)
Cerebrospinal Fluid Rhinorrhea , Hearing Loss , Humans , Male , Child , Female , Encephalocele/diagnostic imaging , Encephalocele/etiology , Encephalocele/surgery , Temporal Bone/pathology , Orbit/pathology , Hearing Loss/complications
11.
J Craniofac Surg ; 34(7): e628-e630, 2023 Oct 01.
Article in English | MEDLINE | ID: mdl-37236622

ABSTRACT

Giant cell tumor (GCT) is a benign tumor that originates from undifferentiated mesenchymal cells of the bone marrow. The craniums as well as temporal bone are extremely rare locations for GCTs. Clinical, radiological, and anatomical diagnosis of this locally aggressive disease poses a major challenge in clinical practice. In this article, we present a clinical study for a 35-year-old female who was found to have left-sided temporal bone GCT with extension to middle cranial fossa and temporomandibular joint (TMJ) with its clinical features and management.


Subject(s)
Giant Cell Tumor of Bone , Skull Base Neoplasms , Female , Humans , Adult , Giant Cell Tumor of Bone/diagnostic imaging , Giant Cell Tumor of Bone/surgery , Skull Base/pathology , Skull Base Neoplasms/diagnostic imaging , Skull Base Neoplasms/surgery , Temporal Bone/diagnostic imaging , Temporal Bone/pathology , Cranial Fossa, Middle/diagnostic imaging
12.
Surg Radiol Anat ; 45(2): 121-135, 2023 Feb.
Article in English | MEDLINE | ID: mdl-36596940

ABSTRACT

PURPOSE: There are only limited anatomical data on nerves, veins, and arteries in the temporal bone. More detailed anatomical data are required to improve planning of treatments targeting the temporal bone region. Herein, we performed a detailed analysis of the facial canal (FC) and the related carotid artery and vein. METHODS: We examined the bony structure of the middle ear and FC, jugular foramen, and carotid canal in 30 Japanese elderly donor cadavers. Three-dimensional reconstruction of the canal structure was achieved using cone beam computed tomography, while macroscopic and histological analyses were also performed. RESULTS: The FC form was classified as either straight (28%) or bent (72%). There were significant differences in the diameter of the FC and the distance between the internal jugular vein, other FC branches, and the FC. Principal component analysis (PCA) was performed for the FC using 29 factors. Two principal components significantly explained 30.9% (component 1, 18.6%; component 2, 12.3%) of the FC. Histological observation showed numerous ganglion cells and shrunken neurons in the geniculate ganglion of the facial nerve of elderly samples. CONCLUSION: FC diameter is an important contributor to the relationship between the FC and the jugular foramen. The FC and the internal jugular vein are located close to each other, which is useful information for the trans-canal surgery of the otology. Furthermore, the geniculate ganglion contains numerous ganglion cells and shrunken neurons, which may affect the FC structure during bone matrix remodeling with aging.


Subject(s)
Facial Nerve , Temporal Bone , Humans , Aged , Temporal Bone/diagnostic imaging , Temporal Bone/pathology , Facial Nerve/diagnostic imaging , Ear, Middle , Cone-Beam Computed Tomography , Geniculate Ganglion
13.
Surg Radiol Anat ; 45(6): 747-756, 2023 Jun.
Article in English | MEDLINE | ID: mdl-37024734

ABSTRACT

BACKGROUND: The degree of mastoid pneumatization of the temporal bone (TB) has been implicated in the pathogenesis of TB diseases and surgical implications, and planning of a few otologic surgeries. However, there is lack of consensus in the classification of the degree of pneumatization. This study aimed to suggest a simple, quick, and less-burden classification system for assessing and rating the degree of pneumatization by comparing two levels of TB computed tomographs (CTs) using the SS as a reference in an inter-observer assessment among otologists. METHODS: This was a randomized pilot survey among otologists. A questionnaire consisting of different axial CTs of TB taken at two levels: the level of malleoincudal junction (MIJ) and the level of lateral semicircular canal (LSCC), with different pneumatization patterns, was used to assess participants' impressions of the degree of pneumatization. The terms "hypo-," "moderate," "good," and "hyper-" pneumatization were listed as options to rate their impressions on the degree of mastoid pneumatization of the TB images using the SS as a reference structure. Likert scale was used to assess their level of agreement or disagreement with using SS as a reference in evaluating mastoid pneumatization. RESULTS: Participants who correctly rated images taken at the level of LSCC according to their respective degree of pneumatization were significantly higher (p < 0.05) regardless of their year of experience compared to those that correctly rated corresponding images taken at the level of MIJ. A 76% positivity in their level of agreement with the use of sigmoid sinus in evaluating mastoid pneumatization was observed on the Likert-scale chart. CONCLUSION: Findings from this study suggest that evaluating air cells around the SS at the level of LSCC on CTs could be easier in assessing and classifying the degree of mastoid pneumatization.


Subject(s)
Mastoid , Temporal Bone , Humans , Mastoid/diagnostic imaging , Temporal Bone/diagnostic imaging , Temporal Bone/pathology , Cranial Sinuses , Tomography, X-Ray Computed/methods
14.
Gen Dent ; 71(6): 42-46, 2023.
Article in English | MEDLINE | ID: mdl-37889243

ABSTRACT

Zygomatic air cell defects (ZACDs) are asymptomatic defects present in the zygomatic process and articular eminence of the temporal bone. This defect is considered a normal anatomical variant and can be detected on routine panoramic images. The objective of this study was to determine the prevalence, dominant type, and location of ZACDs in a North American population. A total of 1724 panoramic images of adult patients were analyzed by a third-year dental student in conjunction with an oral and maxillofacial radiologist. Patient demographics (age and sex) as well as ZACD location (unilateral right/left or bilateral) and appearance (unilocular or multilocular) were evaluated. Statistical analyses included the Clopper-Pearson method to calculate the 95% CI, Student t test for independent samples, and Fisher exact test. The prevalence of ZACDs in this study population was 2.1%. A total of 24 women (2.3%) and 12 men (1.7%) were affected, with no statistically significant difference between the sexes. The mean (SD) age of patients presenting with a ZACD was 56.6 (17.7) years, while that of patients without a ZACD was 53.0 (16.3) years, with no statistically significant difference between groups. Among the 36 patients with ZACDs, 28 (1.6%) had unilateral and 8 (0.5%) had bilateral lesions. One patient with bilateral ZACDs had a unilocular lesion on the right side and a multilocular lesion on the left, so there were a total of 31 unilocular lesions (1.8%) and 6 multilocular lesions (0.3%). The results of this study indicate that the prevalence of ZACDs in the study population was 2.1%, without any statistically significant differences based on the sex or age of the patient or laterality or appearance of the lesions.


Subject(s)
Air , Temporal Bone , Adult , Male , Humans , Female , Middle Aged , Prevalence , Radiography, Panoramic/methods , Temporal Bone/pathology , North America
15.
Audiol Neurootol ; 27(3): 249-259, 2022.
Article in English | MEDLINE | ID: mdl-34965531

ABSTRACT

OBJECTIVES: Hypothesized causes of vestibular neuritis/labyrinthitis include neuroinflammatory or vascular disorders, yet vascular disorders of the inner ear are poorly understood. Guided by known microvascular diseases of the retina, we developed 2 hypotheses: (1) there exist vascular vulnerabilities of artery channels in cases of hypothetical nerve swelling for the superior, inferior, and vestibulocochlear artery and (2) there are arteriovenous crossings that could compromise vascular flow in disease states. METHODS: Two fully mounted and stained temporal bones were used to render three-dimensional reconstructions of the labyrinth blood supply. Using these maps, areas of potential vascular compression were quantified in 50 human temporal bones. RESULTS: Although inner ear arteries and veins mostly travel within their own bony channels, they may be exposed (1) at the entrance into the otic capsule, and (2) where the superior vestibular vein crosses the inferior vestibular artery. At the entry into the otic capsule, the ratio of the soft tissue to total space for the superior vestibular artery was significantly greater than the inferior vestibular artery/cochleovestibular artery (median 44, interquartile range 34-55 vs. 14 [9-17], p < 0.0001). CONCLUSIONS: Three-dimensional reconstruction of human temporal bone histopathology can guide vascular studies of the human inner ear. Studies of retinal microvascular disease helped identify areas of vascular vulnerability in cases of hypothetical nerve swelling at the entrance into the otic capsule and at an arteriovenous crossing near the saccular macula. These data may help explain patterns of clinical findings in peripheral vestibular lesions.


Subject(s)
Labyrinth Diseases , Vestibule, Labyrinth , Humans , Temporal Bone/pathology , Vestibule, Labyrinth/pathology
16.
BMC Pediatr ; 22(1): 87, 2022 02 12.
Article in English | MEDLINE | ID: mdl-35151291

ABSTRACT

BACKGROUND: Juvenile xanthogranuloma (JXG) is a kind of non-Langerhans cell histiocytosis, usually with skin lesions as the main manifestation. It rarely occurs in other tissues or organs and even more rarely is it found in the skull. Here, we report a case of xanthogranuloma derived from the temporal bone that was not present in any other parts of the body. CASE PRESENTATION: A 15-year-old boy had an unaccountable right migraine for 7 months. A mass with tenderness was located behind his right ear. The mass gradually increased in size, and his headache continued. Eventually, he came to our hospital for treatment. A computed tomography (CT) scan and magnetic resonance imaging (MRI) revealed a cystic solid mass in the right temporo-occipital region with skull destruction. The clinical diagnosis was haemangiopericytoma and skull-derived tumour. Haematological and biochemical results were as follows: alanine aminotransferase (ALT) 7 U/L; aspartate aminotransferase (AST) 12 U/L; basophil percentage (BASO%) 1.2%; normal coagulation. The patient was successfully treated with total surgical resection of the tumour. Postoperative histopathology examination showed xanthogranuloma, and his prognosis after surgery was good. CONCLUSIONS: Because JXG rarely occurs in the skull and lacks typical imaging findings, an accurate diagnosis is difficult. The diagnosis of this disease mainly depends on pathological examination and immunohistochemistry. If feasible, many intracranial lesions can be cured through complete resection.


Subject(s)
Xanthogranuloma, Juvenile , Adolescent , Head/pathology , Humans , Magnetic Resonance Imaging , Male , Temporal Bone/diagnostic imaging , Temporal Bone/pathology , Tomography, X-Ray Computed , Xanthogranuloma, Juvenile/diagnostic imaging , Xanthogranuloma, Juvenile/surgery
17.
Childs Nerv Syst ; 38(8): 1615-1619, 2022 08.
Article in English | MEDLINE | ID: mdl-35102523

ABSTRACT

BACKGROUND: Solid variant aneurysmal bone cysts (SVABCs) are a rare but well-described subtype of ABCs. While classic ABCs are readily identified radiographically, SVABCs lack these characteristic radiographic features and thus have a wide differential diagnosis on presentation (including Ewing sarcoma, Langerhans cell histiocytosis, osteosarcoma, metastasis, and giant cell tumor). Genomic/molecular analyses are often necessary for the diagnosis of SVABCs, with USP6 rearrangements being a characteristic finding. We present two cases in which genomic analysis was critical in the diagnosis of SVABCs and revealed unique gene fusions that may provide insight into SVABC pathogenesis. CASE DESCRIPTIONS: Two 13-year old male children presented to our institution with new mass lesions involving the craniofacial skeleton. Magnetic resonance imaging (MRI) in both cases revealed predominantly solid, avidly enhancing masses, one of the squamous portion of the temporal bone, and the other arising from the sphenopalatine foramen with extension into the ipsilateral maxillary and ethmoid sinuses. Histopathology displayed predominantly solid morphology, and next generation sequencing (NGS) revealed a FAT1-USP6 gene fusion in the temporal lesion, and a MIR22HG-USP6 gene fusion in the maxillofacial lesion, the latter of which was not identified on fluorescence in situ hybridization (FISH). These findings were most consistent with a diagnosis of SVABC in each case. CONCLUSIONS: These two cases highlight novel gene fusions in atypically located SVABCs and emphasize the ability of NGS to more accurately and consistently identify USP6 gene fusions, particularly in SVABCs that may otherwise be indistinguishable from alternative pathologies.


Subject(s)
Bone Cysts, Aneurysmal , Adolescent , Bone Cysts, Aneurysmal/diagnostic imaging , Bone Cysts, Aneurysmal/genetics , Genomics , Humans , In Situ Hybridization, Fluorescence , Male , Proto-Oncogene Proteins/genetics , Radiopharmaceuticals , Temporal Bone/pathology , Ubiquitin Thiolesterase/genetics
18.
Childs Nerv Syst ; 38(2): 455-460, 2022 02.
Article in English | MEDLINE | ID: mdl-34115176

ABSTRACT

Gorham Stout disease (GSD) is a rare disease characterized by the proliferation of endothelial lined vessels and replacement of bone by fibrous tissue. The main imaging features are progressive osteolysis and cortical resorption. Temporal bone involvement is rare but presents as a destructive bone lesion that may be misinterpreted as more common lytic processes in the pediatric population, such as infection or Langerhans cell histiocytosis. GSD of the temporal bone is associated with cerebrospinal fluid (CSF) leaks, may present with otorrhea, and can mimic other causes of ear drainage. Here, we report the clinical course, imaging features, and outcomes of a 3-year-old girl with GSD of the temporal bone presenting with CSF leak initially attributed to infection.


Subject(s)
Osteolysis, Essential , Cerebrospinal Fluid Leak/diagnostic imaging , Cerebrospinal Fluid Leak/etiology , Cerebrospinal Fluid Leak/surgery , Child , Child, Preschool , Female , Humans , Osteolysis, Essential/complications , Osteolysis, Essential/diagnostic imaging , Osteolysis, Essential/pathology , Temporal Bone/diagnostic imaging , Temporal Bone/pathology
19.
J Oral Maxillofac Surg ; 80(3): 545-552, 2022 Mar.
Article in English | MEDLINE | ID: mdl-34852246

ABSTRACT

PURPOSE: Controversy exists among head and neck surgical specialties regarding management of Langerhan's Cell Histiocytosis (LCH). The purpose of this study was to evaluate diagnosis, management, and treatment outcomes in children with LCH of the head and neck. METHODS: This is a retrospective cohort study of children with LCH of the head and neck who presented to Children's Healthcare of Atlanta hospital from 2009 to 2021. The independent variables were demographic information, lesion locations, clinical presentation, radiographic findings, diagnostic workup, treatment, and length of follow-up. The patients were grouped based on these variables. The outcome variable was disease reactivation. Descriptive statistics were calculated. RESULTS: There were 3 presentations of LCH of the head and neck. Group 1 presented as a lesion in 1 system without CNS risk (SS-). There were 24 patients with an average age of 10 years. Lesions were located in calvaria and/or mandible. Majority of the patients were treated with only debridement. Two of the patients experienced reactivation. Group 2 presented as a lesion in 1 system with CNS risk (SS+). There were 30 patients with an average age of 6 years. Common locations were temporal bone and/or orbit. These patients present with recurrent ear infections and ptosis. Majority of the patients were treated with chemotherapy (n = 28). One patient had disease reactivation. Group 3 presented with multisystem involvement. There were 13 patients with an average age of 2 years. LCH was found in skin and the lymphatic system. Imaging demonstrated extracranial organ involvement. All of them were treated with chemotherapy. There was 40% reactivation of LCH. CONCLUSIONS: Treatment of LCH depends on presentation. SS- subgroup can be adequately treated via surgical debridement. SS+ and multisystem groups benefit from an early disease diagnosis and require chemotherapy.


Subject(s)
Histiocytosis, Langerhans-Cell , Child , Child, Preschool , Head/diagnostic imaging , Head/pathology , Histiocytosis, Langerhans-Cell/drug therapy , Histiocytosis, Langerhans-Cell/therapy , Humans , Neck/pathology , Retrospective Studies , Temporal Bone/pathology
20.
Am J Otolaryngol ; 43(2): 103348, 2022.
Article in English | MEDLINE | ID: mdl-34973661

ABSTRACT

Capillary hemangiomas are benign vascular lesions that are common in head and neck, but hemangiomas of jugular foramen and temporal bone are rare with only a few cases reported in the literature. We present a case report of this rare disease entity highlighting the subtle radiographic nuances that can benefit clinicians when encountered with similar unusual clinical scenario. Although radiographic features of capillary hemangioma can be distinctive, they are not specific. In this case, the lack of significant involvement of the jugular bulb and the absence of the typical pattern of osseous erosion of the jugular carotid spine led to the alternative diagnosis of a rare capillary hemangioma of the jugular foramen.


Subject(s)
Hemangioma, Capillary , Jugular Foramina , Paraganglioma , Hemangioma, Capillary/diagnostic imaging , Hemangioma, Capillary/surgery , Humans , Temporal Bone/pathology
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