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Association between socioeconomic deprivation, ethnicity and health outcomes in preschool children with recurrent wheeze in England: a retrospective cohort study. 英格兰反复喘息学龄前儿童的社会经济贫困、种族和健康结果之间的关系:一项回顾性队列研究。
IF 9 1区 医学
Thorax Pub Date : 2024-07-02 DOI: 10.1136/thorax-2023-221210
David Lo, Claire Lawson, Clare Gillies, Sharmin Shabnam, Erol A Gaillard, Hilary Pinnock, Jennifer K Quint
{"title":"Association between socioeconomic deprivation, ethnicity and health outcomes in preschool children with recurrent wheeze in England: a retrospective cohort study.","authors":"David Lo, Claire Lawson, Clare Gillies, Sharmin Shabnam, Erol A Gaillard, Hilary Pinnock, Jennifer K Quint","doi":"10.1136/thorax-2023-221210","DOIUrl":"https://doi.org/10.1136/thorax-2023-221210","url":null,"abstract":"<p><strong>Background: </strong>Preschool-aged children have among the highest burden of acute wheeze. We investigated differences in healthcare use, treatment and outcomes for recurrent wheeze/asthma in preschoolers from different ethno-socioeconomic backgrounds.</p><p><strong>Methods: </strong>Retrospective cohort study using data from the Clinical Practice Research Datalink linked to Hospital Episode Statistics in England. We reported number of acute presentations and hospitalisations stratified by index of multiple deprivation (IMD) and ethnicity; and factors associated with treatment non-escalation, and hospitalisation rates using multivariable logistic and Poisson regression models.</p><p><strong>Results: </strong>194 291 preschool children were included. In children not trialled on asthma preventer medications, children from the most deprived IMD quintile (adjusted OR 1.67; 95% CI 1.53 to 1.83) and South Asian (1.77; 1.64 to 1.91) children were more likely to have high reliever usage and where specialist referral had not occurred, the odds of referral being indicated was higher in the most deprived quintile (1.39; 1.28 to 1.52) and South Asian (1.86; 1.72 to 2.01) children compared with the least deprived quintile and white children, respectively.Hospitalisation rates for wheeze/asthma were significantly higher in children from the most deprived quintile (adjusted IRR 1.20; 95% CI 1.13 to 1.27) compared with the least, and in South Asian (1.57; 1.44 to 1.70) and black (1.32; 1.22 to 1.42) compared with white children.</p><p><strong>Conclusions: </strong>We identified inequalities in wheeze/asthma treatment and morbidity in preschool children from more deprived, and non-white backgrounds. A multifaceted approach to tackle health inequality at both the national and local levels, which includes a more integrated and standardised approach to treatment, is needed to improve health outcomes in children with preschool wheeze/asthma.</p>","PeriodicalId":23284,"journal":{"name":"Thorax","volume":null,"pages":null},"PeriodicalIF":9.0,"publicationDate":"2024-07-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141493543","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Journal club 期刊俱乐部
IF 1 1区 医学
Thorax Pub Date : 2024-07-01 DOI: 10.1136/thorax-2024-221967
Timothy J Davies
{"title":"Journal club","authors":"Timothy J Davies","doi":"10.1136/thorax-2024-221967","DOIUrl":"https://doi.org/10.1136/thorax-2024-221967","url":null,"abstract":"In 2015 the WHO set ambitious targets to tackle the global epidemic of Mycobacterium Tuberculosis (TB), including increased detection, reduced mortality, and decreased economic burden of disease, however, the WHO update from September 2023 reports that none are on track for their 2025 milestones (https://reliefweb.int/report/world/global-tuberculosis-report-2023). This situation is not new- globally all three of the 2020 milestones were missed despite some country specific successes. However, the pandemic has had a major impact, stalling, or outright reversing pre-pandemic progress. For example, overall incidence estimates had declined year-on-year between 2010 and 2020 from 11.4 million (95% CI, 8.94,14.10) to 10 million (95% CI, 9.40,10.70). However, in 2021 and 2022, global incidence estimates increased to 10.3 million (95% CI, 9.64,11.00) and 10.6 million (95% CI, 9.87,11.40) respectively. Similar trends were seen in mortality, although the initial increase was in 2020 with 1.37 million estimated deaths compared with 1.32 million in 2019, with the number returning back to pre-pandemic levels (1.30 million, 95% CI 1.18 to 1.43) in 2022. The timing discrepancy reflects the delayed onset of the disease post infection, compared with the more immediate effects of reduced access to treatment. Shockingly, they estimate that 500 000 excess TB deaths occurred compared with if pre-pandemic trends had been maintained. As it stands, the total reduction in incidence rate and TB deaths in 2022 compared with 2015 were 8.7% and 19% respectively, a long …","PeriodicalId":23284,"journal":{"name":"Thorax","volume":null,"pages":null},"PeriodicalIF":10.0,"publicationDate":"2024-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141319845","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Sleep-disordered breathing in children and adults with intellectual disability: mind the gap! 智障儿童和成人的睡眠呼吸障碍:注意差距!
IF 1 1区 医学
Thorax Pub Date : 2024-06-27 DOI: 10.1136/thorax-2023-220032
Renata L Riha, Ankur Singh, Elizabeth A Hill, Hazel Evans, David O'Regan
{"title":"Sleep-disordered breathing in children and adults with intellectual disability: mind the gap!","authors":"Renata L Riha, Ankur Singh, Elizabeth A Hill, Hazel Evans, David O'Regan","doi":"10.1136/thorax-2023-220032","DOIUrl":"https://doi.org/10.1136/thorax-2023-220032","url":null,"abstract":"Background In adults and children with intellectual disability (ID), sleep -disordered breathing (SDB) is thought to be common. However, large epidemiological studies are lacking, and there are few studies on optimal methods of investigation and even fewer randomised, controlled intervention trials of treatment. Method Peer-reviewed publications from various databases were examined in line with search terms relevant to ID and SDB spanning the years 200-2024. Results Findings suggest that, due to comorbid conditions, children and adults with ID may experience both an increased risk of SDB, as well as lower frequency of diagnosis. SDB can compromise the emotional, physical and mental health of individuals with ID. Appropriate treatment when tolerated leads to an improvement in health and well-being and several studies emphasized the importance of consistent follow-up of people with ID - something that is not universally occurring during childhood, in the transition to adulthood and during adulthood itself. As the most frequently occurring form of ID worldwide, we use Down syndrome as a specific example of how diagnosing and treating SDB can lead to improved outcomes. Conclusions This review highlights the importance of identifying SDB in this heterogenous population, recognising the multi-faceted, deleterious consequences of untreated SDB in people with ID, and presents some strategies that can be harnessed to improve diagnosis and management. Until further ID-specific research is available, we urge flexibility in the approach to people with ID and SDB based in guidelines and standard practice developed for the typically developing population.","PeriodicalId":23284,"journal":{"name":"Thorax","volume":null,"pages":null},"PeriodicalIF":10.0,"publicationDate":"2024-06-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141462526","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Long-term impact of ivacaftor on mortality rate and health outcomes in people with cystic fibrosis 伊伐卡夫多对囊性纤维化患者死亡率和健康状况的长期影响
IF 1 1区 医学
Thorax Pub Date : 2024-06-27 DOI: 10.1136/thorax-2023-220558
Christian A Merlo, Teja Thorat, Maral DerSarkissian, Lisa J McGarry, Catherine Nguyen, Yuqian M Gu, Joe Healy, Jaime L Rubin, M Alan Brookhart
{"title":"Long-term impact of ivacaftor on mortality rate and health outcomes in people with cystic fibrosis","authors":"Christian A Merlo, Teja Thorat, Maral DerSarkissian, Lisa J McGarry, Catherine Nguyen, Yuqian M Gu, Joe Healy, Jaime L Rubin, M Alan Brookhart","doi":"10.1136/thorax-2023-220558","DOIUrl":"https://doi.org/10.1136/thorax-2023-220558","url":null,"abstract":"Background Ivacaftor (IVA) has been shown to improve lung function and other clinical outcomes in people with cystic fibrosis (CF). A decade of real-world IVA availability has enabled the examination of long-term outcomes with this treatment. This retrospective, longitudinal cohort study investigated the impact of IVA on mortality rate and health outcomes among people with CF in the US. Methods Data from the US CF Foundation Patient Registry from January 2010 to December 2019 were analysed. The IVA-treated cohort included people with a CF transmembrane conductance regulator ( CFTR ) gating mutation (excluding R117H ); age-matched comparator cohort included people with a F508del and a minimal function CFTR mutation who had no prior CFTR modulator treatment. Baseline characteristics were balanced between cohorts using standardised mortality ratio weighting generated from propensity scores. Outcomes of interest were overall survival, lung transplant, percent predicted forced expiratory volume in 1 s (ppFEV1), body mass index (BMI), pulmonary exacerbations (PEx), outpatient visits and hospitalisations. Findings Over a maximum follow-up of 7.9 years, the IVA-treated cohort (N=736) had lower rates of mortality (hazard ratio [HR] (95% CI): 0.22 (0.09 to 0.45)), lung transplant (HR: 0.11 (95% CI 0.02 to 0.28)), PEx (rate ratio: 0.49 (95% CI 0.42 to 0.55)) and all-cause hospitalisations (rate ratio: 0.50 (95% CI 0.43 to 0.56)) as well as better lung function (mean difference in ppFEV1: 8.46 (95% CI 7.34 to 9.75)) and higher BMI/BMI z -scores (mean difference 1.20 (95% CI 0.92 to 1.71) kg/m2 and 0.27 (95% CI 0.25 to 0.40), respectively) than the comparator cohort (N=733). Interpretation Our analysis suggests that IVA provides sustained clinical benefits in people with CF over a follow-up period of approximately 8 years. These findings reinforce the existing real-world evidence that IVA can slow disease progression and decrease the healthcare burden of CF over the long term. Data are available on reasonable request. The data supporting the findings of this study are available from the US CFFPR at <https://www.cff.org/researchers/patient-registry-data-requests>. The US CFFPR collects and manages its own data and maintains processes for researchers to request summarised data. Restrictions may apply to the availability of these data, which were used under the licence agreement for this study.","PeriodicalId":23284,"journal":{"name":"Thorax","volume":null,"pages":null},"PeriodicalIF":10.0,"publicationDate":"2024-06-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141462461","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
A structural and metabolic framework for classifying pre-clinical tuberculosis infection phenotypes using 18F-FDG PET-CT: a prospective cohort analysis following M. tuberculosis exposure. 利用 18F-FDG PET-CT 对临床前结核感染表型进行分类的结构和代谢框架:结核杆菌暴露后的前瞻性队列分析。
IF 9 1区 医学
Thorax Pub Date : 2024-06-24 DOI: 10.1136/thorax-2024-221470
Jee Whang Kim, Sonam Vadera, Meedya Sharifpour, Amrita Bajaj, Anver Kamil, Pranabashis Haldar
{"title":"A structural and metabolic framework for classifying pre-clinical tuberculosis infection phenotypes using 18F-FDG PET-CT: a prospective cohort analysis following <i>M. tuberculosis</i> exposure.","authors":"Jee Whang Kim, Sonam Vadera, Meedya Sharifpour, Amrita Bajaj, Anver Kamil, Pranabashis Haldar","doi":"10.1136/thorax-2024-221470","DOIUrl":"https://doi.org/10.1136/thorax-2024-221470","url":null,"abstract":"<p><p>Tuberculosis (TB) control efforts are limited by ineffective characterisation of tuberculosis infection (TBI) -a heterogeneous spectrum of pre-clinical infection states, invisible to tools of routine clinical screening, that are associated with variable risk of progression to TB disease. In this prospective study, we use positron emission tomography-CT (PET-CT) as a high-resolution imaging modality to characterise and classify structural and metabolic features observed in 16 asymptomatic household TB contacts with normal chest radiographs. We identify four feature patterns that associate with distinct clinical and microbiological outcomes, supporting potential utility of PET-CT for objective classification of TBI phenotypes.</p>","PeriodicalId":23284,"journal":{"name":"Thorax","volume":null,"pages":null},"PeriodicalIF":9.0,"publicationDate":"2024-06-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141447181","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Pulmonary sarcoidosis: differences in lung function change over time. 肺肉样瘤病:肺功能随时间变化的差异。
IF 9 1区 医学
Thorax Pub Date : 2024-06-21 DOI: 10.1136/thorax-2023-221309
Michelle Sharp, Kevin J Psoter, Ali M Mustafa, Edward S Chen, Nancy W Lin, Stephen C Mathai, Nisha A Gilotra, Michelle N Eakin, Robert A Wise, David R Moller, Meredith C McCormack
{"title":"Pulmonary sarcoidosis: differences in lung function change over time.","authors":"Michelle Sharp, Kevin J Psoter, Ali M Mustafa, Edward S Chen, Nancy W Lin, Stephen C Mathai, Nisha A Gilotra, Michelle N Eakin, Robert A Wise, David R Moller, Meredith C McCormack","doi":"10.1136/thorax-2023-221309","DOIUrl":"https://doi.org/10.1136/thorax-2023-221309","url":null,"abstract":"<p><strong>Introduction: </strong>Given the heterogeneity of sarcoidosis, predicting disease course of patients remains a challenge. Our aim was to determine whether the 3-year change in pulmonary function differed between pulmonary function phenotypes and whether there were differential longitudinal changes by race and sex.</p><p><strong>Methods: </strong>We identified individuals seen between 2005 and 2015 with a confirmed diagnosis of sarcoidosis who had at least two pulmonary function test measurements within 3 years of entry into the cohort. For each individual, spirometry, diffusion capacity, Charlson Comorbidity Index, sarcoidosis organ involvement, diagnosis duration, tobacco use, race, sex, age and medications were recorded. We compared changes in pulmonary function by type of pulmonary function phenotype and for demographic groups.</p><p><strong>Results: </strong>Of 291 individuals, 59% (173) were female and 54% (156) were black. Individuals with restrictive pulmonary function phenotype had significantly greater 3-year rate of decline of FVC% (forced vital capacity) predicted and FEV<sub>1</sub>% (forced expiratory volume in 1 s) predicted course when compared with normal phenotype. We identified a subset of individuals in the cohort, highest decliners, who had a median 3-year FVC decline of 156 mL. Black individuals had worse pulmonary function at entry into the cohort measured by FVC% predicted, FEV<sub>1</sub>% predicted and diffusing capacity for carbon monoxide % predicted compared with white individuals. Black individuals' pulmonary function remained stable or declined over time, whereas white individuals' pulmonary function improved over time. There were no sex differences in rate of change in any pulmonary function parameters.</p><p><strong>Summary: </strong>We found significant differences in 3-year change in pulmonary function among pulmonary function phenotypes and races, but no difference between sexes.</p>","PeriodicalId":23284,"journal":{"name":"Thorax","volume":null,"pages":null},"PeriodicalIF":9.0,"publicationDate":"2024-06-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141437519","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Mucus clears from the trachea in a helix: a new twist to understanding airway diseases. 粘液以螺旋状从气管中清除:了解气道疾病的新转折。
IF 1 1区 医学
Thorax Pub Date : 2024-06-14 DOI: 10.1136/thorax-2023-221052
David Abelson, James Di Michiel, Clayton Frater, Mark Pearson, Robert Russo, Martin Wechselberger, Alice Cottee, Lucy Morgan
{"title":"Mucus clears from the trachea in a helix: a new twist to understanding airway diseases.","authors":"David Abelson, James Di Michiel, Clayton Frater, Mark Pearson, Robert Russo, Martin Wechselberger, Alice Cottee, Lucy Morgan","doi":"10.1136/thorax-2023-221052","DOIUrl":"10.1136/thorax-2023-221052","url":null,"abstract":"<p><strong>Background: </strong>Mucociliary clearance (MCC) is critical to lung health and is impaired in many diseases. The path of MCC may have an important impact on clearance but has never been rigorously studied. The objective of this study is to assess the three-dimensional path of human tracheal MCC in disease and health.</p><p><strong>Methods: </strong>Tracheal MCC was imaged in 12 ex-smokers, 3 non-smokers (1 opportunistically imaged during acute influenza and repeated after recovery) and 5 individuals with primary ciliary dyskinesia (PCD). Radiolabelled macroaggregated albumin droplets were injected into the trachea via the cricothyroid membrane. Droplet movement was tracked via scintigraphy, the path of movement mapped and helical and axial models of tracheal MCC were compared.</p><p><strong>Measurements and main results: </strong>In 5/5 participants with PCD and 1 healthy participant with acute influenza, radiolabelled albumin coated the trachea and did not move. In all others (15/15), mucus coalesced into globules. Globule movement was negligible in 3 ex-smokers, but in all others (12/15) ascended the trachea in a helical path. Median cephalad tracheal MCC was 2.7 mm/min ex-smokers vs 8.4 mm/min non-smokers (p=0.02) and correlated strongly to helical angle (r=0.92 (p=0.00002); median 18<sup>o</sup> ex-smokers, 47<sup>o</sup> non-smokers (p=0.036)), but not to actual speed on helical path (r=0.26 (p=0.46); median 13.6 mm/min ex-smokers vs 13.9 mm/min non-smokers (p=1.0)).</p><p><strong>Conclusion: </strong>For the first time, we show that human tracheal MCC is helical, and impairment in ex-smokers is often caused by flattened helical transit, not slower movement. Our methodology provides a simple method to map tracheal MCC and speed in vivo.</p>","PeriodicalId":23284,"journal":{"name":"Thorax","volume":null,"pages":null},"PeriodicalIF":10.0,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139913572","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Actinomycosis mimicking metastatic lung malignancy. 模仿转移性肺部恶性肿瘤的放线菌病。
IF 1 1区 医学
Thorax Pub Date : 2024-06-14 DOI: 10.1136/thorax-2024-221556
Daniel Sims, Anthony Kerry, Kim Billingham
{"title":"Actinomycosis mimicking metastatic lung malignancy.","authors":"Daniel Sims, Anthony Kerry, Kim Billingham","doi":"10.1136/thorax-2024-221556","DOIUrl":"10.1136/thorax-2024-221556","url":null,"abstract":"","PeriodicalId":23284,"journal":{"name":"Thorax","volume":null,"pages":null},"PeriodicalIF":10.0,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140917301","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Challenging the gold standard: the limitations of molecular assays for detection of Mycobacterium tuberculosis heteroresistance. 挑战金标准:检测结核分枝杆菌异抗性的分子测定的局限性。
IF 9 1区 医学
Thorax Pub Date : 2024-06-14 DOI: 10.1136/thorax-2023-220202
Sarah N Danchuk, Ori E Solomon, Thomas Andreas Kohl, Viola Dreyer, Ivan Barilar, Christian Utpatel, Stefan Niemann, Dick van Soolingen, Richard Anthony, Jakko van Ingen, Joy S Michael, Marcel A Behr
{"title":"Challenging the gold standard: the limitations of molecular assays for detection of <i>Mycobacterium tuberculosis</i> heteroresistance.","authors":"Sarah N Danchuk, Ori E Solomon, Thomas Andreas Kohl, Viola Dreyer, Ivan Barilar, Christian Utpatel, Stefan Niemann, Dick van Soolingen, Richard Anthony, Jakko van Ingen, Joy S Michael, Marcel A Behr","doi":"10.1136/thorax-2023-220202","DOIUrl":"10.1136/thorax-2023-220202","url":null,"abstract":"<p><strong>Objectives: </strong>Heteroresistant infections are defined as infections in which a mixture of drug-resistant and drug-susceptible populations are present. In <i>Mycobacterium tuberculosis</i> (<i>M. tb</i>), heteroresistance poses a challenge in diagnosis and has been linked with poor treatment outcomes. We compared the analytical sensitivity of molecular methods, such as GeneXpert and whole genome sequencing (WGS) in detecting heteroresistance when compared with the 'gold standard' phenotypic assay: the agar proportion method (APM).</p><p><strong>Methods: </strong>Using two rounds of proficiency surveys with defined monoresistant BCG strains and mixtures of susceptible/resistant <i>M. tb</i>, we determined the limit of detection (LOD) of known resistance associated mutations.</p><p><strong>Results: </strong>The LOD for rifampin-R (RIF-R) detection was 1% using APM, 60% using GeneXpert MTB/RIF, 10% using GeneXpert MTB/RIF Ultra and 10% using WGS. While WGS could detect mutations beyond those associated with RIF resistance, the LOD for these other mutations was also 10%. Additionally, we observed instances where laboratories did not report resistance in the majority population, yet the mutations were present in the raw sequence data.</p><p><strong>Conclusion: </strong>The gold standard APM detects minority resistant populations at a lower proportion than molecular tests. <i>Mycobacterium bovis</i> BCG strains with defined resistance and extracted DNA from <i>M. tb</i> provided concordant results and can serve in quality control of laboratories offering molecular testing for resistance. Further research is required to determine whether the higher LOD of molecular tests is associated with negative treatment outcomes.</p>","PeriodicalId":23284,"journal":{"name":"Thorax","volume":null,"pages":null},"PeriodicalIF":9.0,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11187393/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139576526","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Prevalence of lung cysts in adolescents and adults with a germline DICER1 pathogenic/likely pathogenic variant: a report from the National Institutes of Health and International Pleuropulmonary Blastoma/DICER1 Registry. 带有种系 DICER1 致病性/可能致病性变异的青少年和成人肺囊肿患病率:美国国立卫生研究院和国际胸膜肺大疱瘤/DICER1 登记处的报告。
IF 9 1区 医学
Thorax Pub Date : 2024-06-14 DOI: 10.1136/thorax-2023-221024
Alexander T Nelson, Lauren M Vasta, Dave Watson, Jung Kim, Anne K Harris, Ana F Best, Laura A Harney, Ann G Carr, Nicole Frederickson, Louis P Dehner, Christian P Kratz, Kelly N Hagedorn, William A Mize, Alexander Ling, Yoav H Messinger, D Ashley Hill, Kris Ann P Schultz, Douglas R Stewart
{"title":"Prevalence of lung cysts in adolescents and adults with a germline <i>DICER1</i> pathogenic/likely pathogenic variant: a report from the National Institutes of Health and International Pleuropulmonary Blastoma/<i>DICER1</i> Registry.","authors":"Alexander T Nelson, Lauren M Vasta, Dave Watson, Jung Kim, Anne K Harris, Ana F Best, Laura A Harney, Ann G Carr, Nicole Frederickson, Louis P Dehner, Christian P Kratz, Kelly N Hagedorn, William A Mize, Alexander Ling, Yoav H Messinger, D Ashley Hill, Kris Ann P Schultz, Douglas R Stewart","doi":"10.1136/thorax-2023-221024","DOIUrl":"10.1136/thorax-2023-221024","url":null,"abstract":"<p><strong>Background: </strong>Pleuropulmonary blastoma (PPB), the hallmark tumour associated with <i>DICER1</i>-related tumour predisposition, is characterised by an age-related progression from a cystic lesion (type I) to a high-grade sarcoma with mixed cystic and solid features (type II) or purely solid lesion (type III). Not all cystic PPBs progress; type Ir (regressed), hypothesised to represent regressed or non-progressed type I PPB, is an air-filled, cystic lesion lacking a primitive sarcomatous component. This study aims to evaluate the prevalence of non-progressed lung cysts detected by CT scan in adolescents and adults with germline <i>DICER1</i> pathogenic/likely pathogenic (P/LP) variants.</p><p><strong>Methods: </strong>Individuals were enrolled in the National Cancer Institute Natural History of <i>DICER1</i> Syndrome study, the International PPB/<i>DICER1</i> Registry and/or the International Ovarian and Testicular Stromal Tumor Registry. Individuals with a germline <i>DICER1</i> P/LP variant with first chest CT at 12 years of age or older were selected for this analysis.</p><p><strong>Results: </strong>In the combined databases, 110 individuals with a germline <i>DICER1</i> P/LP variant who underwent first chest CT at or after the age of 12 were identified. Cystic lung lesions were identified in 38% (42/110) with a total of 72 cystic lesions detected. No demographic differences were noted between those with lung cysts and those without lung cysts. Five cysts were resected with four centrally reviewed as type Ir PPB.</p><p><strong>Conclusion: </strong>Lung cysts are common in adolescents and adults with germline <i>DICER1</i> variation. Further study is needed to understand the mechanism of non-progression or regression of lung cysts in childhood to guide judicious intervention.</p>","PeriodicalId":23284,"journal":{"name":"Thorax","volume":null,"pages":null},"PeriodicalIF":9.0,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11179973/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140176608","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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