RESUMEN
It is generally believed that the microbiome plays a role in the pathophysiology of chronic rhinosinusitis (CRS), though its exact contribution to disease development and severity remains unclear. Here, samples were collected from the anterior nares, nasopharynx, and maxillary and ethmoid sinuses of 190 CRS patients and from the anterior nares and nasopharynx of 100 controls. Microbial communities were analyzed by Illumina sequencing of the V4 region of 16S rRNA. The phenotype and patient characteristics were documented, and several serum inflammatory markers were measured. Our data indicate a rather strong continuity for the microbiome in the different upper respiratory tract (URT) niches in CRS patients, with the microbiome in the anterior nares being most similar to the sinus microbiome. Bacterial diversity was reduced in CRS patients without nasal polyps compared to that in the controls but not in CRS patients with nasal polyps. Statistically significant differences in the presence/absence or relative abundance of several taxa were found between the CRS patients and the healthy controls. Of these, Dolosigranulum pigrum was clearly more associated with URT samples from healthy subjects, while the Corynebacterium tuberculostearicum, Haemophilus influenzae/H. aegyptius, and Staphylococcus taxa were found to be potential pathobionts in CRS patients. However, CRS versus health as a predictor explained only 1 to 2% of the variance in the microbiome profiles in an adonis model. A history of functional endoscopic sinus surgery, age, and sex also showed a minor association. This study thus indicates that functional studies on the potential beneficial versus pathogenic activity of the different indicator taxa found here are needed to further understand the pathology of CRS and its different phenotypes. (This study has been registered at ClinicalTrials.gov under identifier NCT02933983.)IMPORTANCE There is a clear need to better understand the pathology and specific microbiome features in chronic rhinosinusitis patients, but little is known about the bacterial topography and continuity between the different niches of the upper respiratory tract. Our work showed that the anterior nares could be an important reservoir for potential sinus pathobionts. This has implications for the diagnosis, prevention, and treatment of CRS. In addition, we found a potential pathogenic role for the Corynebacterium tuberculostearicum, Haemophilus influenzae/H. aegyptius, and Staphylococcus taxa and a potential beneficial role for Dolosigranulum Finally, a decreased microbiome diversity was observed in patients with chronic rhinosinusitis without nasal polyps compared to that in healthy controls but not in chronic rhinosinusitis patients with nasal polyps. This suggests a potential role for the microbiome in disease development or progression of mainly this phenotype.
Asunto(s)
Microbiota , Nariz/microbiología , Sinusitis/microbiología , Sinusitis/fisiopatología , Adulto , Enfermedad Crónica , Análisis por Conglomerados , ADN Bacteriano/química , ADN Bacteriano/genética , ADN Ribosómico/química , ADN Ribosómico/genética , Femenino , Humanos , Masculino , Persona de Mediana Edad , Nasofaringe/microbiología , Senos Paranasales/microbiología , Filogenia , ARN Ribosómico 16S/genética , Análisis de Secuencia de ADN , Adulto JovenRESUMEN
PURPOSE: To report the case of an atypical sino-orbital inflammatory myofibroblastic tumor. METHODS: Case report. RESULTS: A 71-year-old man presented slowly progressive painless diplopia followed by unilateral proptosis of the left eye with slight edema of the upper eyelid. Visual acuity was 0.4 with a relative afferent pupillary defect (RAPD). Abduction and depression of the left eye were limited. Imaging showed a left sino-orbital tumor with mass effect on the medial rectus muscle resulting in proptosis. The lesion showed intracranial extension through the superior orbital fissure with bone erosion and sclerotic bone reaction. Endoscopic ethmoidal, maxillary, and orbital biopsies revealed an inflammatory myofibroblastic tumor. Full blood count showed an eosinophilia while antineutrophil cytoplasmic antibodies and C-reactive protein were negative. To date, 7 cases of orbital myofibroblastic tumor have been described, though none of the prior studies have described bone invasion. CONCLUSIONS: This erosive sino-orbital inflammatory myofibroblastic tumor is a novel finding mimicking a malignant tumor.
Asunto(s)
Encefalopatías/patología , Miofibroblastos/patología , Órbita/patología , Seudotumor Orbitario/patología , Enfermedades de los Senos Paranasales/patología , Administración Oral , Anciano , Biopsia , Encefalopatías/diagnóstico por imagen , Encefalopatías/tratamiento farmacológico , Diplopía/diagnóstico , Endoscopía , Exoftalmia/diagnóstico , Glucocorticoides/uso terapéutico , Humanos , Imagen por Resonancia Magnética , Masculino , Metilprednisolona/uso terapéutico , Órbita/diagnóstico por imagen , Seudotumor Orbitario/diagnóstico por imagen , Seudotumor Orbitario/tratamiento farmacológico , Enfermedades de los Senos Paranasales/diagnóstico por imagen , Enfermedades de los Senos Paranasales/tratamiento farmacológico , Tomografía Computarizada por Rayos X , Agudeza VisualRESUMEN
We describe the surgical technique of temporary removal of the posterior auditory canal wall with reconstruction and report the outcome of using this technique as a treatment method for cholesteatoma in a case series. In 32 cases of cholesteatoma surgery a technique of temporary removal of the posterior bony wall was applied. During primary surgery the posterior auditory canal wall was removed using an oscillating saw. For the purpose of reconstruction, the canal wall was repositioned and fixed using two titanium microplates (n = 26). In case the canal wall could not be reconstructed with osteosynthesis, either glass-ionomeric cement (BioCem™) was used for fixation (n = 4) or fibrin glue (Tissucol™) (n = 2) to support the posterior wall. The outcome includes the healing process in the first postoperative month, the absence of residual or recurrent disease and the successful reconstruction of the posterior auditory canal wall as evaluated during second-look surgery. When microplates where used, we saw healing problems of the canal skin in about 4% of patients. Recurrent cholesteatoma was found in 4 cases (14%), residual cholesteatoma in 8 ears (25%). In the osteosynthesis group, successful reconstruction was achieved in 25 patients (96%). In 3 out of 4 patients of the glass-ionomeric cement group (75%) excessive granulation tissue developed with extensive bony lysis. Temporary removal of the posterior auditory canal wall offers potential for the control of cholesteatoma. Our first results suggest that osteosynthesis allows for a good anatomical and functional reconstruction.
Asunto(s)
Placas Óseas , Colesteatoma del Oído Medio/cirugía , Conducto Auditivo Externo/cirugía , Fijación Interna de Fracturas/métodos , Apófisis Mastoides/cirugía , Procedimientos de Cirugía Plástica/métodos , Adolescente , Adulto , Anciano , Niño , Preescolar , Estudios de Cohortes , Femenino , Humanos , Masculino , Persona de Mediana Edad , Estudios Retrospectivos , Adulto JovenRESUMEN
Transtympanic Electrocochleography (TT-ECoG) and the glycerol test can aid in the diagnostic process of Ménière's disease (MD). Measurement of travelling wave velocity (TWV) has been proposed as an alternative to TT-ECoG to detect endolymphatic hydrops. We assessed the feasibility and obtained normative data of the TWV test in the diagnosis of MD, and compared the test results in MD patients with their symptoms and their TT-ECoG results. The TWV test records two runs of auditory brainstem responses to clicks that are ipsilaterally masked with two different high pass filtered noise bands. The resulting latency difference of peak V was used as a measure of the TWV. The 95% confidence interval of this latency difference was defined in a group of 28 normals. Subsequently, the test was performed in nine MD patients. After the TWV test, seven of these patients underwent a TT-ECoG. The symptoms of MD patients at the time of testing and their TT-ECoG results were compared with TWV test results. The 95% confidence interval of the latency difference of peak V in the control group was 0.190-0.668 ms. A latency difference <0.190 ms indicates an increase in TWV, and thus reflects a positive test. In nine MD patients, we found a clear correlation between the result of the TWV test and TT-ECoG. The TWV test result did not significantly correlate with a single symptom. Our findings suggest that the TWV test can be useful to detect endolymphatic hydrops, but further experiments with larger patient groups are needed to confirm the diagnostic value of the TWV test.
Asunto(s)
Audiometría de Respuesta Evocada/métodos , Hidropesía Endolinfática/diagnóstico , Potenciales Evocados Auditivos del Tronco Encefálico/fisiología , Enfermedad de Meniere/diagnóstico , Adulto , Audiometría de Tonos Puros , Hidropesía Endolinfática/fisiopatología , Estudios de Factibilidad , Femenino , Humanos , Masculino , Enfermedad de Meniere/fisiopatologíaRESUMEN
Otosclerosis is a progressive hearing loss characterized by an abnormal bone homeostasis of the otic capsule that leads to stapes fixation. Although its etiology remains unknown, otosclerosis can be considered a complex disease. Transforming growth factor-beta 1 (TGF-beta1) was chosen for a case-control association study, because of several non-genetic indications of involvement in otosclerosis. Single nucleotide polymorphism (SNP) analysis in a large Belgian-Dutch sample set gave significant results (P = 0.0044) for an amino acid changing SNP, T263I. Analysis of an independent French population replicated this association with SNP T263I (P = 0.00019). The results remained significant after multiple testing correction in both populations. Haplotype analysis and the results of an independent effect test using the weighted haplotype (WHAP) computer program in both populations were both compatible with SNP T263I being the only causal variant. The variant I263 is under-represented in otosclerosis patients and hence protective against the disease. Combining the data of both case-control groups for SNP T263I with a Mantel-Haenszel estimate of common odds ratios gave a very significant result (P = 9.2 x 10(-6)). Functional analysis of SNP T263I with a luciferase reporter assay showed that the protective variant I263 of TGF-beta1 is more active than the WT variant T263 (P = 1.6 x 10(-6)). On the basis of very low P-values, replication in an independent population and a functional effect of the protective variant, we conclude that TGF-beta1 influences the susceptibility for otosclerosis, and that the I263 variant is protective against the disease.
Asunto(s)
Otosclerosis/genética , Polimorfismo de Nucleótido Simple , Factor de Crecimiento Transformador beta1/genética , Sustitución de Aminoácidos , Estudios de Casos y Controles , Susceptibilidad a Enfermedades , Variación Genética , Genética de Población , Genotipo , Haplotipos , Humanos , Modelos Biológicos , Otosclerosis/etnología , Otosclerosis/etiología , Fenotipo , Análisis de Secuencia de ADN , Población BlancaRESUMEN
Congenital nasal pyriform aperture stenosis has been described as an unusual cause of neonatal nasal obstruction. Clinical suspicion is based on respiratory distress, cyclic cyanosis, apneas, and feeding difficulties. A bony overgrowth of the maxillary nasal processes is thought to be responsible for this deformity. This anomaly has been reported as an isolated feature or can be associated with craniofacial or central nervous system anomalies. Surgery is indicated in cases of severe respiratory distress, feeding difficulties, and when conservative methods fail.