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1.
Mol Med Rep ; 23(5)2021 05.
Artigo em Inglês | MEDLINE | ID: mdl-33760111

RESUMO

Cholesteatoma constitutes an acquired benign epidermal non­permanent bone lesion that is locally destructive and patients often relapse. Inflammasomes, which mediate the maturation and production of IL­18 and IL­1ß, resulting in pyroptosis, have been documented to serve a core function in multiple inflammatory conditions. Absent in melanoma 2 (AIM2) is an inflammasome that identifies cytoplasmic DNA and has previously been reported as a pivotal modulator of inflammatory responses. Therefore, the present study aimed to determine the expression levels of AIM2 in human cholesteatoma tissues, and elucidate its function in modulating cytokine production. The expression levels of IL­18, apoptosis­associated speck­like protein containing a CARD (ASC), IL­1ß, AIM2 and caspase­1 were markedly elevated in cholesteatoma tissues. Protein expression levels of AIM2, caspase­1 and ASC were localized in the cellular cytoplasm, primarily in the granular and prickle­cell layers in the cholesteatoma epithelium. Induction using IFN­Î³, as well as cytoplasmic DNA markedly activated the AIM2 inflammasome and elevated the release of IL­18 and IL­1ß in human cholesteatoma keratinocytes. IFN­Î³ was found to enhance poly(dA:dT)­induced pyroptosis of cells and cytokine production. The results of the present study revealed that AIM2 expressed in human cholesteatoma serves a vital function in the inflammatory response by initiating the inflammasome signaling cascade in cholesteatoma.


Assuntos
Neoplasias Ósseas/genética , Colesteatoma/genética , Proteínas de Ligação a DNA/genética , Interleucina-18/genética , Interleucina-1beta/genética , Animais , Apoptose/efeitos dos fármacos , Neoplasias Ósseas/patologia , Proteínas Adaptadoras de Sinalização CARD/genética , Caspase 1/genética , Colesteatoma/patologia , Citocinas/biossíntese , Citocinas/genética , Citoplasma/genética , DNA/genética , Regulação Neoplásica da Expressão Gênica/efeitos dos fármacos , Humanos , Inflamassomos/genética , Interferon gama/genética , Interleucina-18/biossíntese , Interleucina-1beta/biossíntese , Queratinócitos/metabolismo , Neoplasias/genética , Neoplasias/patologia , Poli dA-dT/farmacologia , Piroptose/efeitos dos fármacos , Piroptose/genética
2.
Biomed Res Int ; 2021: 6644897, 2021.
Artigo em Inglês | MEDLINE | ID: mdl-33778077

RESUMO

Objective: Cholesteatoma is a clinically heterogeneous disease, with some patients showing spontaneous regression, while others experiencing an aggressive, lethal disease. Cholesteatoma in children can be divided into two types: congenital and acquired. Identifying good prognostic markers is needed to help select patients who will require immediate surgical intervention. Matrix metalloproteinase-2 (MMP2) was previously reported to play an important role in cholesteatoma progression, by promoting bone destruction and keratinocyte infiltration. Herein, we analyzed MMP2 mRNA expression level in cholesteatoma using RNA-in situ hybridization in formalin-fixed, paraffin-embedded (FFPE) tissue samples. Methods: Sixty patients with cholesteatoma under 15 years old, who underwent their primary surgery at Aichi Medical University's Otolaryngology Department, were analyzed for MMP2 expression level, using RNA-in situ hybridization. Results: There were no significant differences in MMP2 mRNA expression level between congenital cholesteatoma and acquired cholesteatomas. In congenital cholesteatoma, higher MMP2 signals were observed in the open type than in the closed type (p < 0.001). In acquired cholesteatoma, higher MMP2 signals were observed in the pars tensa than in the pars flaccida (p < 0.001). MMP2 mRNA expression level was almost exclusively found in the fibroblasts or in the inflammatory cells in the stroma, but not in the epithelium. Conclusion: Our study reveals that MMP2 mRNA expression level is strongly associated with the subtypes of cholesteatoma. The findings suggest that the level of expression of MMP2 mRNA may be related to the pathogenesis and aggressive features of cholesteatoma.


Assuntos
Colesteatoma/congênito , Regulação Enzimológica da Expressão Gênica , Regulação Neoplásica da Expressão Gênica , Metaloproteinase 2 da Matriz/biossíntese , Proteínas de Neoplasias/biossíntese , RNA Mensageiro/biossíntese , RNA Neoplásico/biossíntese , Adolescente , Criança , Pré-Escolar , Colesteatoma/classificação , Colesteatoma/enzimologia , Colesteatoma/patologia , Feminino , Humanos , Masculino
4.
Laryngoscope ; 131(4): E1282-E1285, 2021 04.
Artigo em Inglês | MEDLINE | ID: mdl-33030245

RESUMO

Extra- and intracranial complications of cholesteatoma are rare in the new era of antibiotics. The rarity of the severe complications and the atypical symptoms cause difficulties to set up proper diagnosis and treatment. We report about a 76-year old patient with external auditory canal cholesteatoma causing Bezold abscess and sigmoid sinus thrombosis. The patient underwent canal wall up mastoidectomy and mastoid obliteration with abdominal fat and Bezold abscess drainage. Systemic antibiotic and anticoagulant therapy were applied. The treatment resulted in fast improvement in the general condition of the patient, and the control magnetic resonance imaging (MRI) showed no recurrence of the disease. Laryngoscope, 131:E1282-E1285, 2021.


Assuntos
Abscesso/etiologia , Colesteatoma/complicações , Trombose dos Seios Intracranianos/etiologia , Abscesso/diagnóstico por imagem , Abscesso/cirurgia , Idoso , Antibacterianos/uso terapêutico , Otorreia de Líquido Cefalorraquidiano , Colesteatoma/patologia , Meato Acústico Externo/diagnóstico por imagem , Meato Acústico Externo/patologia , Meato Acústico Externo/cirurgia , Feminino , Cefaleia , Humanos , Processo Mastoide/patologia , Trombose dos Seios Intracranianos/diagnóstico por imagem , Trombose dos Seios Intracranianos/cirurgia
5.
PLoS One ; 15(10): e0240216, 2020.
Artigo em Inglês | MEDLINE | ID: mdl-33031450

RESUMO

Middle ear cholesteatoma is a destructive disease in which inflammation plays an important role in development and progression, and there are currently no biomarkers predicting prognosis or recurrence. Cylindromatosis (CYLD), a tumor suppressor deubiquitinase, serves as a negative regulator of inflammation expressed in tissues including the middle ear. To determine the clinical significance of CYLD in acquired cholesteatoma, we evaluated CYLD expression in acquired cholesteatoma tissue by immunostaining and analyzed its correlation with clinicopathological characteristics. Our immunohistochemical analysis revealed that CYLD expression levels were varied in the tissues of acquired cholesteatoma patients. The relative expression levels of CYLD in cholesteatoma exhibited a significant correlation with the grade of otorrhea (R = 0.532, p = 0.039). Moreover, the period of epithelialization was also significantly associated with the relative expression levels of CYLD (R = 0.720, p = 0.002). In addition, CYLD expression tended to be lower in the group with recurrence. These results suggest that low CYLD expression correlates with postoperative recovery of acquired cholesteatoma, while potentially affecting the induction of recurrence. This is the first report showing that low CYLD expression correlates with accelerated disease recovery, and suggests a new aspect of CYLD as a prognostic predictor of acquired cholesteatoma.


Assuntos
Colesteatoma/metabolismo , Colesteatoma/patologia , Enzima Desubiquitinante CYLD/metabolismo , Regulação Enzimológica da Expressão Gênica , Adolescente , Adulto , Idoso , Colesteatoma/diagnóstico , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Prognóstico
6.
World Neurosurg ; 139: 223-225, 2020 07.
Artigo em Inglês | MEDLINE | ID: mdl-32305611

RESUMO

BACKGROUND: Osteosarcoma is a common malignant bone tumor that occurs in children or adolescents but rarely in the skull. Epidermoid cysts, also known as cholesteatomas, represent approximately 0.2%-1.8% of all intracranial tumors. The occurrence of osteosarcoma with an epidermoid cyst is extremely rare. CASE DESCRIPTION: A 41-year-old woman had both osteosarcoma and cholesteatoma in the left cerebellopontine angle. We resected the 2 tumors using the suboccipital retrosigmoid approach, and she received radiotherapy and chemotherapy after the surgery. One year after surgery, the patient is healthy and has recovered well. CONCLUSIONS: Osteosarcomas and epidermoid cysts should be completely resected to prevent tumor recurrence and aseptic meningitis. Postoperative osteosarcoma treatment should include radiotherapy and chemotherapy to improve the survival rate of patients. It is hoped that this report will help clinicians in diagnosis and treatment of patients with similar conditions.


Assuntos
Encefalopatias/patologia , Neoplasias Cerebelares/patologia , Ângulo Cerebelopontino/patologia , Colesteatoma/patologia , Osteossarcoma/patologia , Adulto , Encefalopatias/complicações , Neoplasias Cerebelares/complicações , Colesteatoma/complicações , Cisto Epidérmico/complicações , Cisto Epidérmico/patologia , Feminino , Humanos , Osteossarcoma/complicações
7.
Neuroradiol J ; 33(3): 210-215, 2020 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-32336206

RESUMO

BACKGROUND: Suspected cholesteatoma recurrence is commonly investigated with magnetic resonance imaging (MRI) of the temporal bone. Non-echo planar diffusion-weighted imaging (non-EP DWI) has become the sequence of choice. PURPOSE: To assess the agreement between an MRI protocol incorporating both non-EP DWI and contrast-enhanced sequences, and a shortened protocol without contrast-enhanced sequences in the assessment of suspected cholesteatoma recurrence. MATERIALS AND METHODS: One hundred consecutive MRIs, consisting of T2-weighted, non-EP DWI and pre- and post-contrast T1-weighted sequences, were reviewed by two radiologists at a tertiary referral centre. Agreement between the two protocols was assessment by means of a weighted Cohen kappa coefficient. RESULTS: We found a near perfect agreement between the two protocols (kappa coefficient with linear weighting 0.98; 95% confidence interval 0.95-1.00). There were two cases in which the two protocols were discordant. In both cases, the lesion measured <3 mm and images were degraded by artefact at the bone-air interface. The shortened protocol without post-contrast sequences yielded a 32% reduction in acquisition time. CONCLUSION: When non-EP DWI is available, contrast-enhanced sequences can be omitted in the vast majority of cases without compromising diagnostic accuracy. Contrast-enhanced sequences may provide additional value in equivocal cases with small (<3 mm) lesions or in cases where images are degraded by artefact.


Assuntos
Doenças Ósseas/diagnóstico por imagem , Colesteatoma/diagnóstico por imagem , Imagem de Difusão por Ressonância Magnética/métodos , Interpretação de Imagem Assistida por Computador/métodos , Osso Temporal/diagnóstico por imagem , Adolescente , Adulto , Idoso , Doenças Ósseas/patologia , Colesteatoma/patologia , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Recidiva , Estudos Retrospectivos , Osso Temporal/patologia , Adulto Jovem
8.
Scanning ; 2020: 9371516, 2020.
Artigo em Inglês | MEDLINE | ID: mdl-32158510

RESUMO

Bone erosion is considered a typical characteristic of advanced or complicated cholesteatoma (CHO), although it is still a matter of debate if bone erosion is due to osteoclast action, being the specific literature controversial. The purpose of this study was to apply a novel scanning characterization approach, the BSE 3D image analysis, to study the pathological erosion on the surface of human incus bone involved by CHO, in order to definitely assess the eventual osteoclastic resorptive action. To do this, a comparison of BSE 3D image of resorption lacunae (resorption pits) from osteoporotic human femur neck (indubitably of osteoclastic origin) with that of the incus was performed. Surface parameters (area, mean depth, and volume) were calculated by the software Hitachi MountainsMap© from BSE 3D-reconstructed images; results were then statistically analyzed by SPSS statistical software. Our findings showed that no significant differences exist between the two groups. This quantitative approach implements the morphological characterization, allowing us to state that surface erosion of the incus is due to osteoclast action. Moreover, our observation and processing image workflow are the first in the literature showing the presence not only of bone erosion but also of matrix vesicles releasing their content on collagen bundles and self-immuring osteocytes, all markers of new bone formation on incus bone surface. On the basis of recent literature, it has been hypothesized that inflammatory environment induced by CHO may trigger the osteoclast activity, eliciting bone erosion. The observed new bone formation probably takes place at a slower rate in respect to the normal bone turnover, and the process is uncoupled (as recently demonstrated for several inflammatory diseases that promote bone loss) thus resulting in an overall bone loss. Novel scanning characterization approaches used in this study allowed for the first time the 3D imaging of incus bone erosion and its quantitative measurement, opening a new era of quantitative SEM morphology.


Assuntos
Doenças Ósseas/patologia , Reabsorção Óssea/patologia , Colesteatoma/patologia , Bigorna/patologia , Osteoclastos/patologia , Osteogênese/fisiologia , Doenças Ósseas/metabolismo , Reabsorção Óssea/metabolismo , Colesteatoma/metabolismo , Colágeno/metabolismo , Feminino , Colo do Fêmur/metabolismo , Colo do Fêmur/patologia , Humanos , Imageamento Tridimensional/métodos , Bigorna/metabolismo , Osteoclastos/metabolismo , Osteócitos/metabolismo , Osteócitos/patologia , Pós-Menopausa/metabolismo , Pós-Menopausa/fisiologia
9.
Perm J ; 242020.
Artigo em Inglês | MEDLINE | ID: mdl-32097113

RESUMO

INTRODUCTION: Cholesteatomas are lined by squamous epithelium, contain keratin debris, and can cause bony erosion. Although commonly found in the middle ear space and mastoid, cholesteatomas may develop in adjacent structures including the paranasal sinuses. Frontal sinus cholesteatoma (FSC) is a rare condition with fewer than 30 reported cases. The aims of this study are to describe the clinical presentation, diagnostic imaging, and endoscopic treatment of FSC and to review the literature focusing on the pathogenesis, diagnosis, and historical and contemporary treatments of FSC. CASE PRESENTATION: A 45-year-old man presented with a 1-week history of right eyelid and forehead swelling. Results of computed tomography scans and magnetic resonance images revealed a right frontal sinus lesion of soft-tissue density with bony dehiscence along the superior orbit and posterior table. He underwent right-sided endoscopic sinus surgery at a tertiary care center in January 2017. Intraoperatively, the frontal sinus contained keratin debris suggestive of FSC. This suspicion was confirmed postoperatively by pathologic analysis after subtotal resection. DISCUSSION: The pathogenesis of frontal sinus cholesteatoma varies based on its cause (congenital vs acquired). Clinical diagnosis remains challenging but is aided by nasal endoscopy, computed tomography, and magnetic resonance imaging. Historically, FSC has been managed by total extirpation through open approaches, which can entail substantial morbidity. With sophisticated endoscopic sinus instrumentation and image guidance, FSC can be successfully treated via an endoscopic approach. Serial débridements and washouts in an outpatient setting may adequately manage the residual disease in the postoperative period.


Assuntos
Colesteatoma/patologia , Seio Frontal/patologia , Doenças dos Seios Paranasais/patologia , Colesteatoma/diagnóstico por imagem , Colesteatoma/cirurgia , Endoscopia/métodos , Humanos , Imageamento por Ressonância Magnética , Masculino , Pessoa de Meia-Idade , Doenças dos Seios Paranasais/diagnóstico por imagem , Tomografia Computadorizada por Raios X
10.
Acta Otolaryngol ; 140(4): 286-288, 2020 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-31928292

RESUMO

Background: Mastoid development, tympanic sinus depth, and residual disease after surgery for congenital cholesteatoma are probably related, but these relationships have not been examined in detail.Aims/objectives: This study aimed to clarify the relationships between the abovementioned factors. Materials and Methods: The subjects were 31 patients with congenital cholesteatoma (stage III or IV in Potsic's staging system) that underwent mastoidectomy. The cross-sectional area of the mastoid air cells was measured as described previously. Tympanic sinus depth was classified into A-C using Marchioni et al.'s system.Results: Patients with deep tympanic sinuses or residual disease exhibited significantly greater mastoid air cell development. However, little residual disease was found in the mastoid air cells. Conversely, residual disease was observed more frequently in the patients with deep tympanic sinuses.Conclusions and significance: After surgery for congenital cholesteatoma, residual disease is more likely to occur in patients with marked mastoid growth, possibly because they have deep tympanic sinuses. Cases in which congenital cholesteatoma spreads to the mastoid air cells are classified as stage IV in Potsic's system, but our findings indicate that invasion into a deep tympanic sinus is more important than invasion into the mastoid air cells.


Assuntos
Colesteatoma da Orelha Média/patologia , Colesteatoma/congênito , Orelha Média/patologia , Processo Mastoide/patologia , Adolescente , Criança , Pré-Escolar , Colesteatoma/patologia , Colesteatoma/cirurgia , Colesteatoma da Orelha Média/cirurgia , Feminino , Humanos , Masculino , Mastoidectomia , Estudos Retrospectivos
11.
Cells ; 9(1)2020 01 14.
Artigo em Inglês | MEDLINE | ID: mdl-31947538

RESUMO

Cholesteatoma is a severe non-cancerous lesion of the middle ear characterized by massive inflammation, tissue destruction, and an abnormal growth of keratinized squamous epithelium. We recently demonstrated the presence of pathogenic stem cells within cholesteatoma tissue, unfortunately their potential roles in regulating disease-specific chronic inflammation remain poorly understood. In the presented study, we utilized our established human in vitro cholesteatoma stem cell model for treatments with lipopolysaccharides (LPS), tumor necrosis factor α (TNFα), and the TLR4-antagonist LPS from R. sphaeroides (LPS-RS) followed by qPCR, western blot, and immunocytochemistry. Middle ear cholesteatoma stem cells (ME-CSCs) showed a significantly increased expression of TLR4 accompanied by a significantly enhanced LPS-dependent pro-inflammatory gene expression pattern of TNFα, IL-1α, IL-1ß, IL-6, and IL-8 compared to non-pathogenic control cells. LPS-dependent pro-inflammatory gene expression in ME-CSCs was driven by an enhanced activity of NF-B p65 leading to a TNFα-mediated feed-forward-loop of pro-inflammatory NF-B target gene expression. Functional inactivation of TLR4 via the TLR4-antagonist LPS-RS blocked chronic inflammation in ME-CSCs, resulting in a nearly complete loss of IL-1ß, IL-6, and TNFα expression. In summary, we determined that ME-CSCs mediate the inflammatory environment of cholesteatoma via TLR4-mediated NF-B-signaling, suggesting a distinct role of ME-CSCs as drivers of cholesteatoma progression and TLR4 on ME-CSCs as a therapeutic target.


Assuntos
Colesteatoma/patologia , Inflamação/patologia , Lipopolissacarídeos/farmacologia , Rhodobacter sphaeroides/química , Células-Tronco/patologia , Receptor 4 Toll-Like/antagonistas & inibidores , Colesteatoma/genética , Meato Acústico Externo/patologia , Orelha Média/patologia , Regulação da Expressão Gênica/efeitos dos fármacos , Humanos , Inflamação/genética , Mediadores da Inflamação/metabolismo , Modelos Biológicos , NF-kappa B/metabolismo , Transdução de Sinais/efeitos dos fármacos , Pele/patologia , Esferoides Celulares/patologia , Células-Tronco/efeitos dos fármacos , Receptor 4 Toll-Like/metabolismo , Fator de Necrose Tumoral alfa/farmacologia
12.
Turk J Med Sci ; 50(1): 155-162, 2020 02 13.
Artigo em Inglês | MEDLINE | ID: mdl-31800200

RESUMO

Background/aim: Surgical success is related with many factors belonging to both the patient and the disease. This study aims to analyse the preoperative and intraoperative characteristics, the postoperative results, and the factors affecting the surgical success in different types of chronic otitis media (COM). Materials and methods: A total of 1510 ears of 1398 patients who underwent COM surgery were included in the study. Postoperative results were obtained from 376 ears of 356 patients who had been followed after surgery. The demographic characteristics of the patients, such as age and sex, operative findings, preoperative audiological examination results, and final audiometric and otoscopic examination findings, were retrospectively obtained from the archives of the department. Results: The most frequent diagnosis was simple COM (39.9%), and the most frequently performed surgery was tympanoplasty without mastoidectomy (46.6%). The overall hearing success rate was found to be 75.8%. Postoperative hearing success was significantly associated with the chronic otitis subgroup, ossicular pathologies, and the condition of the middle ear mucosa. Postoperative graft take rate was found to be 78.6%. Graft success was statistically significantly higher in patients with normal middle ear mucosa. Performing mastoidectomy, the presence of patency in aditus ad antrum, and being a paediatric case had no impact on graft success. Conclusion: Factors affecting the success of COM surgery include age, chronic otitis subgroup, location and size of perforation, the condition of the middle ear mucosa, and the level of the ossicular disease. These factors should be known and an appropriate treatment plan should be prepared.


Assuntos
Colesteatoma/cirurgia , Otite Média/cirurgia , Complicações Pós-Operatórias/etiologia , Adolescente , Adulto , Idoso , Audiometria , Biópsia por Agulha Fina , Criança , Colesteatoma/etiologia , Colesteatoma/patologia , Doença Crônica , Orelha Média/patologia , Orelha Média/cirurgia , Feminino , Seguimentos , Perda Auditiva/etiologia , Humanos , Masculino , Mastoidectomia , Pessoa de Meia-Idade , Otite Média/etiologia , Otite Média/patologia , Otoscopia , Fatores de Risco , Timpanoplastia , Adulto Jovem
13.
J Laryngol Otol ; 134(12): 1096-1102, 2020 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-33407963

RESUMO

OBJECTIVE: To correlate pre-operative computed tomography findings, intra-operative details and surgical outcomes with cholesteatoma recurrence in revision tympanomastoidectomy. METHODS: This retrospective, non-randomised, single-institution cohort study included 42 patients who underwent pre-operative computed tomography imaging and revision surgery for recurrent chronic otitis media. Twelve disease localisations noted during revision surgery were correlated with pre-operative temporal bone computed tomography scans. A matched pair analysis was performed on patients with similar intra-operative findings, but without pre-operative computed tomography scans. RESULTS: Pre-operative computed tomography identified 25 out of 31 cholesteatoma recurrences. Computed tomography findings correlated with: recurrent cholesteatoma when attic opacification and ossicular chain involvement were present; and revision surgery type. Sinodural angle disease, posterior canal wall erosion and dehiscent dura were identified as predictors of canal wall down tympanomastoidectomy. Patients with pre-operative computed tomography scans had a higher rate of cholesteatoma recurrence, younger age at diagnosis of recurrent disease, more revision surgical procedures and less time between previous and revision surgical procedures (all p < 0.05). CONCLUSION: Pre-operative imaging and intra-operative findings have important clinical implications in revision surgery for chronic otitis media. Performing pre-operative computed tomography increases diagnosis accuracy and reduces the time required to diagnose recurrent disease.


Assuntos
Colesteatoma/diagnóstico por imagem , Colesteatoma/cirurgia , Mastoidectomia/métodos , Ventilação da Orelha Média/métodos , Tomografia Computadorizada por Raios X/estatística & dados numéricos , Adolescente , Adulto , Idoso , Idoso de 80 Anos ou mais , Criança , Colesteatoma/patologia , Doença Crônica , Terapia Combinada/métodos , Feminino , Humanos , Cuidados Intraoperatórios , Masculino , Mastoidectomia/estatística & dados numéricos , Pessoa de Meia-Idade , Ventilação da Orelha Média/estatística & dados numéricos , Otite Média/diagnóstico por imagem , Otite Média/cirurgia , Período Pré-Operatório , Recidiva , Reoperação/métodos , Estudos Retrospectivos , Osso Temporal/diagnóstico por imagem , Tomografia Computadorizada por Raios X/métodos , Resultado do Tratamento
14.
Rev. ORL (Salamanca) ; 11(2): 1-9, 2020. ilus, tab
Artigo em Espanhol | IBECS | ID: ibc-193766

RESUMO

INTRODUCCIÓN Y OBJETIVOS: El colesteatoma de conducto auditivo externo (CCAE) es un proceso patológico poco frecuente, caracterizado por la invasión de tejido escamoso en un área del canal auditivo, que progresa hasta la destrucción ósea. Debido a la escasez de casos publicados hemos considerado de utilidad esta revisión. MATERIAL Y MÉTODOS: Estudio prospectivo y observacional desde el año 2000, de 18 pacientes diagnosticados clínicamente de CCAE en nuestro servicio de ORL. RESULTADOS: El porcentaje de hombres y mujeres es similar, con una edad media de diagnóstico de 60 años. La localización más frecuente es póstero-inferior y en la mayor parte de casos el origen es primario. La otalgia, la otorrea y la hipoacusia fueron los síntomas principales, siendo menos habitual el prurito y excepcional la debilidad facial. En general el tratamiento es conservador, pero resultó necesario el abordaje quirúrgico en un tercio de pacientes mediante canaloplastia, siguiendo el esquema expuesto, o la mastoidectomía, en función de la extensión de las lesiones. CONCLUSIONES: Aunque desconocemos los mecanismos patogénicos responsables de la formación y desarrollo del CCAE, la inclusión de queratina entre el epitelio y el hueso, con la participación del periostio, parecen ser los desencadenantes del proceso. El diagnóstico es clínico y su extensión determina el empleo de un tratamiento local o quirúrgico, que suele ser resolutivo


INTRODUCTION: External auditory canal cholesteatoma (EACC) is a rare entity characterized by the invasion of squamous tissue in the auditory canal, that progresses to bone destruction. Due to the scarcity of published cases of EACC, we have deemed useful this review. MATERIAL AND METHODS: Prospective and observational study from the year 2000 to the present in 18 patients clinically diagnosed with EACC in our ORL service. RESULTS: The percentage of men and women is similar, with a mean age at diagnosis of 60 years. The most frequently location was postero-inferior, and in most cases, the origin is primary. Otalgia, otorrhea and subjective hearing loss were the main symptoms, being less common the pruritus and exceptional facial weakness. Overall, the treatment is conservative, but it was necessary surgical treatment on one-third of the patients, performing canaloplasty or mastoidectomy, according to the extent of injuries. CONCLUSIONS: Although we do not know the genesis and pathogenic mechanisms responsible of EACC formation and development, the inclusion of keratin between the epithelium and bone, with the participation of the periosteum, seems to be the trigger of this process. The diagnosis is clinical, and its extension determines the use of a local or surgical treatment, which is usually resolutive


Assuntos
Humanos , Masculino , Feminino , Adulto , Pessoa de Meia-Idade , Idoso , Colesteatoma/diagnóstico por imagem , Colesteatoma/patologia , Procedimentos Cirúrgicos Otológicos/métodos , Colesteatoma/cirurgia , Otopatias/cirurgia , Estudos Prospectivos , Dor de Orelha/etiologia , Endoscopia/métodos , Mastoidectomia
15.
J Int Adv Otol ; 15(3): 386-390, 2019 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-31846916

RESUMO

OBJECTIVES: Since its introduction in 2002, the staging system for congenital cholesteatoma, as defined by Potsic, has been used widely owing to its simplicity and predictability. The aim of the present study was to analyze the clinical characteristics and surgical treatment outcomes of congenital cholesteatoma and to correlate them with postoperative recurrence. MATERIALS AND METHODS: A retrospective chart review was performed for 38 patients who were diagnosed with congenital cholesteatoma and who underwent surgical resection between August 2007 and November 2014 at the Department of Otorhinolaryngology of our hospital. RESULTS: The mean age of the patients was 7.9±8.2 years, and the number of males and females was 20 and 18, respectively. The mean follow-up period was 30±26 months. Of the 38 patients, 9 (24%) had residual or recurrent cholesteatoma during follow-up examination after primary surgery. The results showed that the size (≥4 mm) of the lesion was significantly correlated with residual or recurrent disease (p=0.026). The disease extent (single vs. multiple quadrant involvement), type (closed vs. open type), and stage; applied surgical method (with mastoidectomy vs. without mastoidectomy); and preoperative hearing level (normal vs. abnormal) were not significantly correlated with residual or recurrent disease. CONCLUSION: The disease extent, type, and stage; applied surgical methods; and preoperative hearing level were not significantly correlated with residual or recurrent disease. The size (≥4 mm) of congenital cholesteatoma was significantly correlated with residual or recurrent disease.


Assuntos
Colesteatoma/congênito , Mastoidectomia , Adolescente , Criança , Pré-Escolar , Colesteatoma/patologia , Colesteatoma/cirurgia , Feminino , Seguimentos , Audição , Humanos , Masculino , Processo Mastoide/patologia , Processo Mastoide/cirurgia , Período Pós-Operatório , Recidiva , Estudos Retrospectivos , Resultado do Tratamento
16.
J Int Adv Otol ; 15(3): 391-395, 2019 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-31846917

RESUMO

OBJECTIVES: Petrous bone cholesteatoma is a rare pathologic entity and may be a difficult surgical challenge because of potential involvement of the facial nerve, carotid artery, dura mater, otic capsule and superior petrosal or lateral sinus. The objective of this article is to present the endoscope-assisted surgery for petrous bone cholesteatoma. MATERIALS AND METHODS: Eight patients (nine ears) who underwent endoscope-assisted petrous bone surgery for cholesteatoma. Pure tone audiogram, magnetic resonance imaging were performed at preoperatively, and at approximately 12 months postoperatively. RESULTS: Endoscope assisted surgery was performed in 8 patients and 9 ears. Of these patients, 6 were male and 2 were females. Median age was 19,5 (range 7-52) years. Hearing was able to preserved in 8 ears (8/9). Recurrence disease was observed one ear in long term follow up (1/9). In another one patient, cholesteatoma pearl was removed in the office. CONCLUSION: Endoscope-assisted surgery can allow removal of cholesteatoma of petrous apex with preserving hearing. It also provides to remove the cholesteatoma via transmastoid approach for perilabyrinthine space as "minimally invasive surgery" instead of middle fossa approach that is standard surgical procedure. In apical and peri-labyrinthine cholesteatomas, endoscopes allow to preserve hearing with middle fossa approach instead of trans-otic/ trans-labyrinthine/trans-cochlear approach.


Assuntos
Doenças Ósseas/cirurgia , Colesteatoma/cirurgia , Endoscopia/métodos , Audição , Osso Petroso/patologia , Adolescente , Adulto , Doenças Ósseas/patologia , Doenças Ósseas/fisiopatologia , Criança , Colesteatoma/patologia , Colesteatoma/fisiopatologia , Orelha Interna/fisiopatologia , Orelha Interna/cirurgia , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Período Pós-Operatório , Estudos Retrospectivos , Resultado do Tratamento , Adulto Jovem
17.
BMJ Case Rep ; 12(11)2019 Nov 19.
Artigo em Inglês | MEDLINE | ID: mdl-31748359

RESUMO

Cholesteatoma of the paranasal sinus is a very rare condition. As in the tympanomastoid region where cholesteatomas are a common entity, the paranasal sinus cholesteatomas also tend to erode the surrounding bony structures. Because of the extensive bony erosion, this condition often masquerades as a chronic granulomatous or a malignant lesion. Clinical presentation can be quite varied like facial deformities, visual and neurological deficits. Radiological findings are also non-specific making a preoperative diagnosis challenging. Histopathological examination is the only confirmatory investigation. We present a patient with frontal cholesteatoma who presented with forehead swelling of 1 month duration. Since the diagnosis could be obtained only intraoperatively, the patient required multiple surgeries. Frontal sinus cholesteatomas often require a combined endoscopic and external approach to ensure complete disease clearance. Periodic follow-up is essential to rule out recurrence.


Assuntos
Colesteatoma/patologia , Seio Frontal/patologia , Cefaleia/etiologia , Neoplasias dos Seios Paranasais/diagnóstico por imagem , Adulto , Colesteatoma/diagnóstico por imagem , Colesteatoma/cirurgia , Diagnóstico Diferencial , Endoscopia/métodos , Feminino , Seio Frontal/diagnóstico por imagem , Cefaleia/diagnóstico , Humanos , Imageamento por Ressonância Magnética/métodos , Neoplasias dos Seios Paranasais/patologia , Tomografia Computadorizada por Raios X/métodos , Resultado do Tratamento
18.
Otol Neurotol ; 40(10): 1306-1312, 2019 12.
Artigo em Inglês | MEDLINE | ID: mdl-31634284

RESUMO

OBJECTIVES: To determine the feasibility of endoscopic-assisted surgery for lesions involving the perilabyrinthine recesses, and develop and validate a technique for this kind of lesions that increases the possibility of preserving the labyrinth and cochlea. STUDY DESIGN: Observational study. SETTING: Referral center. CASES: Five patients who underwent endoscopy-assisted surgery for lateral skull base pathology involving the perilabyrinthine recesses between July 2010 and March 2016 were reviewed. INTERVENTIONS: Clinical data of the five patients were collected. MAIN OUTCOMES MEASURES: Hearing level and recurrence. RESULTS: Three of the five patients (three women, two men) had petrous bone cholesteatomas, one a facial neurofibroma, and one a petrous bone cholesterol granuloma. Complete excision with labyrinth and cochlea preservation was achieved using an endoscopic-assisted technique in all patients. Four had preoperative conductive hearing loss and one was totally deaf. In one patient, the postoperative air-bone gap was 30 dB lower than preoperatively levels, one stayed the same, and one was 30 dB because of closing of the external ear canal. After an average follow-up of 62.9 months, no patient had recurrence. CONCLUSIONS: Endoscope-assisted surgery is feasible for lesions involving the perilabyrinthine recesses and increases the likelihood to preserve the structure and function of labyrinth/cochlea, which may provide chances for artificial hearing devices implantation.


Assuntos
Colesteatoma/cirurgia , Endoscopia/métodos , Granuloma/cirurgia , Neurofibroma/cirurgia , Osso Petroso/cirurgia , Base do Crânio/cirurgia , Adolescente , Adulto , Idoso , Colesteatoma/patologia , Orelha Interna/fisiologia , Estudos de Viabilidade , Feminino , Seguimentos , Granuloma/patologia , Humanos , Masculino , Microscopia , Recidiva Local de Neoplasia , Neurofibroma/patologia , Estudos Retrospectivos , Adulto Jovem
19.
Otol Neurotol ; 40(8): e803-e811, 2019 09.
Artigo em Inglês | MEDLINE | ID: mdl-31348131

RESUMO

INTRODUCTION: Partial epithelial-mesenchymal transition (p-EMT) is a process by which epithelial cells partially lose their intercellular adhesion and change to obtain migration ability. The transcription factor p63 regulates the expression of cadherin family and induces epithelial cell proliferation. In this study, we hypothesized that p-EMT under p63 expression may be a key factor in epithelial cell growth in middle ear cholesteatoma. METHODS: Specimens were surgically excised from patients with congenital cholesteatoma (CC) (n = 48), acquired middle ear cholesteatoma (AC) (n = 120), and normal skin tissue (n = 34). We analyzed immunohistochemically for the EMT marker (N-cadherin), adherence junction marker (E-cadherin), and tight junction marker (claudin-1, claudin-4, occludin). We also examined the labeling index (LI) of p63 and Proliferating cell nuclear antigen (PCNA) (late S phase marker), and Snail expression as a mobility marker. RESULTS: The expression of p63 (CC 51.0 ±â€Š7.4%, AC 50.0 ±â€Š5.9%) was significantly higher in the thickened epithelium of CC and AC compared with normal skin tissue (p < 0.0001). The loss of E-cadherin was observed (CC 50.0%, AC 55.8%) but the expression patterns in the tight junction were almost normal. N-cadherin was partially detected in the basal and upper layer of epithelium in CC and AC. In contrast to that of normal skin tissue, the LI of PCNA was significantly higher in AC (p < 0.0001). The positive rate of Snail was significantly higher in CC (p < 0.0001). CONCLUSION: This study indicates that p-EMT via the p63 signaling pathway might plays an essential role in epithelial growth in AC and CC formation, although tight junction formation and terminal differentiation were not affected in those processes.


Assuntos
Colesteatoma da Orelha Média/patologia , Colesteatoma/congênito , Transição Epitelial-Mesenquimal/fisiologia , Fatores de Transcrição/metabolismo , Proteínas Supressoras de Tumor/metabolismo , Adulto , Colesteatoma/metabolismo , Colesteatoma/patologia , Colesteatoma da Orelha Média/metabolismo , Epitélio/metabolismo , Epitélio/patologia , Feminino , Humanos , Masculino , Pessoa de Meia-Idade
20.
J Assoc Res Otolaryngol ; 20(5): 449-459, 2019 10.
Artigo em Inglês | MEDLINE | ID: mdl-31254133

RESUMO

Cholesteatoma starts as a retraction of the tympanic membrane and expands into the middle ear, eroding the surrounding bone and causing hearing loss and other serious complications such as brain abscess and meningitis. Currently, the only effective treatment is complete surgical removal, but the recurrence rate is relatively high. In rheumatoid arthritis (RA), osteoclasts are known to be responsible for bone erosion and undergo differentiation and activation by receptor activator of NF-κB ligand (RANKL), which is secreted by synovial fibroblasts, T cells, and B cells. On the other hand, the mechanism of bone erosion in cholesteatoma is still controversial. In this study, we found that a significantly larger number of osteoclasts were observed on the eroded bone adjacent to cholesteatomas than in unaffected areas, and that fibroblasts in the cholesteatoma perimatrix expressed RANKL. We also investigated upstream transcription factors of RANKL using RNA sequencing results obtained via Ingenuity Pathways Analysis, a tool that identifies relevant targets in molecular biology systems. The concentrations of four candidate factors, namely interleukin-1ß, interleukin-6, tumor necrosis factor α, and prostaglandin E2, were increased in cholesteatomas compared with normal skin. Furthermore, interleukin-1ß was expressed in infiltrating inflammatory cells in the cholesteatoma perimatrix. This is the first report demonstrating that a larger-than-normal number of osteoclasts are present in cholesteatoma, and that the disease involves upregulation of factors related to osteoclast activation. Our study elucidates the molecular basis underlying bone erosion in cholesteatoma.


Assuntos
Osso e Ossos/patologia , Colesteatoma/patologia , Osteoclastos/fisiologia , Ligante RANK/fisiologia , Transdução de Sinais , Artrite Reumatoide/complicações , Diferenciação Celular , Humanos , Interleucina-1beta/análise , Osteoclastos/citologia , Ligante RANK/genética , RNA Mensageiro/análise
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