An unusual case of lingual tonsillar hypertrophy.
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ABSTRACT:
SUBMITTER: Hope N
PROVIDER: S-EPMC4856252 | biostudies-other | 2016 May
REPOSITORIES: biostudies-other
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Project description:BackgroundTonsils provide an innovative in vivo model for investigating immune response to infections and allergens. However, data are scarce on the differences in tonsillar virus infections and immune responses between patients with tonsillar hypertrophy or recurrent tonsillitis. We investigated the differences in virus detection and T cell and interferon gene expression in patients undergoing tonsillectomy due to tonsillar hypertrophy or recurrent tonsillitis.MethodsTonsils of 89 surgical patients with tonsillar hypertrophy (n = 47) or recurrent tonsillitis (n = 42) were analysed. Patients were carefully characterized clinically. Standard questionnaire was used to asses preceding and allergy symptoms. Respiratory viruses were analysed in tonsils and nasopharynx by PCR. Quantitative real-time PCR was used to analyse intratonsillar gene expressions of IFN-α, IFN-β, IFN-γ, IL-10, IL-13, IL-17, IL-28, IL-29, IL-37, TGF-β, FOXP3, GATA3, RORC2 and Tbet.ResultsMedian age of the subjects was 15 years (range 2-60). Patients with tonsillar hypertrophy were younger, smoked less often, had less pollen allergy and had more adenovirus, bocavirus-1, coronavirus and rhinovirus in nasopharynx (all P < 0.05). Only bocavirus-1 was more often detected in hypertrophic tonsils (P < 0.05). In age-adjusted analysis, tonsillar hypertrophy was associated with higher mRNA expressions of IL-37 (P < 0.05).ConclusionsIntratonsillar T cell and interferon gene expressions appeared to be relatively stable for both tonsillar hypertrophy and recurrent tonsillitis. Of the studied cytokines, only newly discovered anti-inflammatory cytokine IL-37, was independently associated with tonsillar hypertrophy showing slightly stronger anti-inflammatory response in these patients.
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Project description:The correlation between tonsil microbiome and tonsillar hypertrophy has not been well established. Given that oral dysbiosis is related to several metabolic diseases and that tonsillar hypertrophy leads to disordered breathing during sleep and obesity in children, it is necessary to investigate the relationship between the oral microbiome and tonsillar hypertrophy. After 16S rRNA amplicon sequencing of tonsillectomy samples, we evaluated the correlation between the tonsil microbiome and biochemical blood indices in pediatric patients who underwent tonsillectomy. Groups are classified into two categories: based on BMI, and grades 2, 3, and 4 based on tonsil size. Children with obesity and tonsillar hypertrophy have similar microbiome compositions and induce comparable changes in microbiome abundance and composition, confirming the association from a metagenomic perspective. In addition, obesity and tonsillar hypertrophy demonstrated a strong correlation with the Proteobacteria to Firmicutes (P/F) ratio, and among various biochemical indicators, alanine aminotransferase (ALT) levels increase with obesity and tonsillar hypertrophy, indicating a possible association of tonsil microbiome and liver metabolism. These novel findings demonstrate the significance of the tonsil microbiome and suggest the need for tonsil regulation, particularly during childhood.
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Project description:The comprehension of unconventional immune functions of tonsillar B cells, their role in tolerance induction and protective immune responses, is crucial to unveil the dynamic interactions of the upper aero digestive tract with polymicrobial commensal flora and pathogens, in health and disease. Here, we describe the kinetics of IL10 intracellular expression and compare it with that of cytokines known to be produced by tonsillar B cells. Additionally, we detected a relevant proportion of IL17-expressing tonsillar B cells, which has not previously been reported. We immunophenotyped tonsillar IL10-expressing B cells (B10) and observed IL10 production in activated B cells at every developmental stage. Finally, we identified a relationship between decreased B10 percentages, increased proportion of the germinal centre (GC) population and hypertrophied tonsils (HT). Our findings provide greater insight into the role of B10 in GC reactions and characterized their involvement in the pathogenesis of tonsillar dysfunction.
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Project description:TNFRSF13B/TACI defects have been associated with CVID pathogenesis and/or phenotype, especially the development of benign lymphoproliferation and autoimmunity. Our purpose was to investigate the role of TNFRSF13B/TACI defects in the pathogenesis of two common lymphoproliferative disorders, namely, sarcoidosis and tonsillar hypertrophy (TH). 105 patients (71 with sarcoidosis and 34 with TH, including 19 without infectious causative and 15 due to Haemophilus influenzae) were analyzed for TNFRSF13B/TACI defects. Two out of 19 TH patients without infectious cause (10.5%) and 2 patients with sarcoidosis (2.8%) displayed rare TNFRSF13B/TACI defects (I87N, L69TfsX12, E36L, and R202H, resp.). Both mutations identified in TH patients have been assessed as deleterious for protein function, while the patient with the R202H mutation and sarcoidosis exhibited also sIgG4D. Our study further supports the notion that TNFRSF13B/TACI defects alone do not result in CVID but may be also found frequently in distinct clinical phenotypes, including benign lymphoproliferation and IgG subclass deficiencies.
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Project description:IntroductionOral-pharyngeal reflexes demonstrate a wide range of complexities due to their intricate synaptic pattern. Most of the reflexes are protective in nature such as preventing aspiration. These reflexes in oral cavity affect the muscles and can be evoked either in isolation or in combination in order to achieve a specific response. Certain sensory inputs induce an entire motor behavior pattern as seen in lingual hypoglossal reflex.Materials and methodsA case report depicting this reflex have been presented. To our knowledge, this is the first case documented in a living human. Along with this, cadaveric studies have been also done in fifteen specimens.ResultsExtralingual anastomosis between lingual nerve and hypoglossal nerve were found in six specimens.ConclusionHereby, we present a clinical paper of this unusual phenomenon which has not been documented in the literature.
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Project description:INTRODUCTION: Benign idiopathic tonsillar hypertrophy (HBI) may affect a child's quality of life and sleep. Several studies have sought to relate the clinical features of HBI with the infectious and/or immunologic changes that occur. OBJECTIVE: To increase the knowledge of the etiology of HBI. DATA SYNTHESIS: From 2012 to 2013 we conducted a retrospective observational study of 101 children with HBI who underwent tonsillectomies at Ambulatory ENT General Hospital of the East Zone of São Paulo City, a region with a poor socioeconomic population. Preoperative serologic results were available to confirm mononucleosis, cytomegalovirus, anti-streptolysin O (ASLO) and immunoglobulins. The mean patient age was 5.8 years (55% male, 45% female). Using the Mann-Whitney U test, we identified significant gender differences in the parameters of immunoglobulins (Ig) M (IgM), IgA, and IgE. Forty-seven percent of the patients had increased ASLO levels, and 37% had increased IgE levels. CONCLUSION: An evaluation of a patient's serologic parameters and laboratory results may be relevant to the etiology and prevention of HBI. Based on the results obtained from the study sample, the identification of etiologic agents and causative factors remain a public health challenge that affects the quality of life of children.
| S-EPMC4297036 | biostudies-other
Project description:STUDY OBJECTIVES:The goals of this study were to (1) evaluate the degree of decisional conflict (DC) experienced by caregivers of children with obstructive sleep apnea (OSA) without tonsillar hypertrophy; and (2) describe the association between DC, quality of life (QOL), and OSA severity. METHODS:This study comprised children evaluated in the multidisciplinary upper airway center at the Cincinnati Children's Hospital Medical Center from December 2014 to May 2016. Caregivers were asked to complete surveys (Pediatric Quality of Life Inventory 4.0 [PedsQL], OSA-18, Epworth Sleepiness Scale, Family Impact Questionnaire, Decisional Conflict Scale, CollaboRATE scale, and SURE questionnaire) during a clinic visit. Polysomnography data were collected. Analysis included Kruskal-Wallis, Wilcoxon rank-sum, and regression testing. RESULTS:Caregivers of 76 children participated; 16 (21.1%) had high DC. There were no significant differences in demographics between those with low and high DC; the low DC group had a higher obstructive apnea-hypopnea index (13.2 versus 12.3 events/h; P = .013). Overall and disease-specific QOL, sleepiness, family impact scores, and DC did not differ by OSA severity except for the PedsQL physical subcategory (P = .02). DC was associated with the total PedsQL (P = .043) on univariate regression; however, this did not persist (P = .61) after controlling for demographic variables. DC scores correlated well with CollaboRATE and SURE throughout the analysis (P < .001). CONCLUSIONS:The proportion of caregivers of children with OSA without tonsillar hypertrophy who experienced a high level of DC regarding their child's treatment was 21.1%. Neither DC nor OSA severity was related to QOL in children with OSA. The briefer SURE or CollaboRATE scales were adequate tools to measure DC in these children.
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Project description:Introductionand Importance: Acute ischemic stroke is a rare event in children with leukemia, yet with long-term morbidity, substantial health, and economic cost. Central nervous system (CNS) leukemic involvement and chemotherapy-related stroke are the most common causes. Hyperleukocytosis induced stroke is very rarely reported.Case presentationA 2-year-old male child presented with hyperleukocytosis (leukocyte count was 320x109/L). Bone marrow evaluation revealed T-cell acute lymphoblastic leukemia. He was treated with dexamethasone, vincristine and daunorubicin, and on day 4 of chemotherapy, he developed abnormal movements, altered mental status, limb weakness and mutism. Magnetic resonance imaging of the brain showed multifocal infarctions involving left pons and both cerebellar hemispheres causing tonsillar herniation with restricted diffusion and mild hydrocephalus but no leptomeningeal enhancement or leukemic infiltrates. Magnetic resonance angiography did not show any arterial stenosis. He was intubated, sedated and managed conservatively with dexamethasone. Cytologic analysis of cerebrospinal fluid showed no blasts. Thrombophilia work up was negative. Five weeks later, the patient had significant improvement in overall neurologic status. He is free of leukemia. MRI showed interval resolution of previous infarcts.Clinical discussionHyperviscosity secondary to hyperleukocytosis was considered to be the most likely explanation for this patient stroke after excluding thrombophilia and leukemic infiltration. Prompt management with hydration and careful chemotherapy resulted in good outcome in our patient.ConclusionThis case demonstrate the value of early recognition and prompt management of posterior circulation ischemic stroke in children with leukemia and hyperleukocytosis at presentation.
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Project description:Untreated tonsillar phlegmon is a life-threatening condition commonly caused by Streptococcus pyogenes and Fusobacterium necrophorum, among other pathogens. Here, using specific laboratory tools, we detected Methanobrevibacter smithii in addition to S. pyogenes. This unprecedented observation questions the role of methanogens in phlegmon and the optimal treatment of this mixed infection.
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Project description: Not available
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