Research Appraisals
Evidence-based critical appraisals of the latest medical research, systematically evaluated using Oxford CEBM methodology.
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Journal of medical Internet research
Reporting of Telehealth Implementation in Cystic Fibrosis: Scoping Review Using a Novel Theory-Based Evaluation Lens
BACKGROUND: Many inductive reviews exploring telehealth and its application in health care have identified missing or inconsistently reported implementation data, calling for a standardized approach to telehealth research. OBJECTIVE: Using cystic fibrosis (CF) as a case exemplar, this study evaluated the adherence of telehealth research to standardized reporting frameworks through a theory-based evaluation lens to assess implementation reporting quality and identify knowledge gaps and strengths across the literature. METHODS: We conducted an updated systematic review of the PubMed, Scopus, and Web of Science databases using a novel deductive approach to identify relevant scientific papers available in English and focusing on the delivery of telehealth interventions to CF populations as part of or alongside routine CF care. Two relevant reporting checklists were identified in the Equator Network database (Guidelines and Checklist for the Reporting on Digital Health Implementations [iCHECK-DH] and Template for Intervention Description and Replication for Telehealth [TIDiER-telehealth]) to extract data from the papers. Each checklist category was described as being "fully reported" (score=2), "partially reported" (score=1), and "did not report" (score=0) for each paper. An overall score was calculated for adherence to the checklists. RESULTS: In total, 98 studies published between 2006 and May 2025 were included in this review, with the majority appearing during the COVID-19 pandemic (2021-2022). Most studies were conducted in a single country, predominantly the United States, Australia, and the United Kingdom, and were published in medical journals. Telehealth was variably described, with video call-based models in combination with remote monitoring being most common. The median score was 22/40 (range 11-29, 55.0% adherent) for iCHECK and 15/24 (range 6-23, 62.5% adherent) for TIDiER, demonstrating moderate overall reporting quality. For iCHECK, ≥50% of studies fully reported 6/20 categories, partially reported 9/20 categories, and did not report 3/20 categories. For TIDiER, ≥50% of studies fully reported 4/12 categories, partially reported 6/12 categories, and did not report 1/12 categories, indicating persistent gaps in intervention description despite improved partial reporting. CONCLUSIONS: Key areas, such as justification for telehealth, target populations, and outcomes, are well documented, offering valuable insights into the rationale for and outcomes associated with telehealth. However, implementation processes remain underreported, partly due to the more recent adoption of frameworks like iCHECK and TIDiER. The clinical implications of the current evidence limit the implementation of telehealth in terms of the ability to assess feasibility and readiness for adoption; understand financial implications and plan sustainably; ensure patient safety, data protection, and equity; interpret outcome data in context; and share, replicate, or scale evidence-based models of care. Strengthening the commitment to standardized telehealth reporting will ultimately support clinical decision-making and improve the effective and equitable integration of telehealth into care.
23 May 2026
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Lung transplant in people with cystic fibrosis and nontuberculous mycobacteria infection
RATIONALE: Cystic fibrosis (CF) is an inherited multi-organ disorder. People with CF (pwCF) experience recurrent and chronic lung infections and a progressive loss of lung function. PwCF with poor and rapidly declining lung function may be considered for lung transplantation (LTx), which may improve their quality of life and survival. Nontuberculous mycobacteria (NTM) can cause pulmonary disease in pwCF, and NTM infection is a poor prognostic factor in LTx. Guidelines recommend NTM infection should not automatically preclude LTx. It is important to evaluate the evidence base for LTx in pwCF and NTM pulmonary disease. OBJECTIVES: To evaluate clinical outcomes in pwCF and with NTM infection (NTM infection alone or with NTM pulmonary disease) who undergo LTx by comparing: 1. pwCF with current NTM lung infection who undergo LTx versus those with NTM infection who do not undergo LTX; 2. pwCF with current NTM lung infection who undergo LTx versus those without NTM undergoing LTx. SEARCH METHODS: We searched the Cochrane Cystic Fibrosis Trials Register, CENTRAL, MEDLINE, Embase, and PubMed as well as two ongoing trials registries. We checked references. The latest search date was 17 February 2026. ELIGIBILITY CRITERIA: We considered non-randomised studies of pwCF (any age) with or without NTM lung infection or disease being considered for LTx as well as studies of pwCF and NTM who either did or did not undergo LTx. OUTCOMES: Our critical outcomes were mortality, disseminated NTM infection post-LTx, time to chronic lung allograft dysfunction (CLAD), and quality of life at any time points reported. We additionally planned to report lung function, hospitalisations for pulmonary exacerbations, and nutritional parameters in the review. RISK OF BIAS: We assessed the risk of bias in three studies using ROBINS-I and in one study using the Joanna Briggs Institute checklist for case series. SYNTHESIS METHODS: We could only report results narratively. We used GRADE to assess the certainty of the evidence. INCLUDED STUDIES: We included four single-centre retrospective studies (388 adults). Sample sizes ranged from nine to 177 participants. Mycobacteria abscessus was the most common NTM species identified, and all studies reported infection with other pathogens. All studies compared pwCF and NTM infection to pwCF without NTM infection, all undergoing LTx. Each study reported mortality and disseminated NTM infection post-LTx; two studies recorded CLAD. No study reported quality of life, specific lung function measures (although one study commented briefly on lung function in general), hospitalisations for pulmonary exacerbations, or nutritional parameters. SYNTHESIS OF RESULTS: We analysed all NTM infections together for practical reasons and were not able to undertake a planned subgroup analysis by subspecies, but acknowledge that the prognosis and clinical trajectory of pwCF infected with different NTM may not be similar. We downgraded the certainty of the evidence due to non-randomised study design and serious risk of bias across all studies. We assessed all identified evidence as of very low certainty, such that lung transplant may have little to no effect on any of the outcomes listed below, but the evidence is very uncertain. Mortality Two studies (18 participants with NTM) reported similar survival data between NTM-positive LTx recipients and matched controls without NTM. Another study (9 participants) reported that two of five participants NTM-positive at LTx died within a few months post-LTx, whilst one of four NTM-negative participants died three years post-LTx due to chronic rejection. One study (177 participants) found that pwCF who had positive NTM cultures pre-LTx had a longer median survival duration than those who had negative cultures. This study additionally reported on survival of participants with post-LTx NTM infection, finding that the five participants who had post-LTx NTM disease had a longer mean survival duration than the 141 participants without post-LTx NTM disease. Disseminated NTM infection post-LTx In the largest study, of the 18 pwCF with NTM at the time of LTx, seven had at least one positive NTM culture, and four developed NTM disease post-LTx. Conversely, 79 of the 89 pwCF without NTM remained so post-LTx; 10 participants recorded a positive NTM culture, but none developed NTM disease. For the 39 participants without a baseline NTM culture, three participants recorded positive NTM cultures post-LTx, and one developed NTM disease. Of the remaining small studies, one reported that NTM was isolated in four of 13 participants at LTx and in three of these post-LTx. A second study reported that one out of five pwCF had NTM infection post-LTx (all were positive at LTx). The third study reported that five out of nine participants had NTM disease at LTx, and two of these five remained NTM-positive post-LTx. CLAD Two studies assessed CLAD. One study reported that none of the five NTM-positive LTx recipients developed CLAD, stating that the risk of CLAD appeared to be similar between the NTM and the comparator group. The second study stated that three out of nine LTx recipients with NTM disease developed chronic rejection or graft dysfunction. AUTHORS' CONCLUSIONS: There are no randomised trials to guide clinicians and patients or their families when making decisions regarding LTx in pwCF with NTM. The available data come from observational studies and registry data, often with few people with NTM reported. It has not been possible to pool the available data in meta-analysis, and we are very uncertain of the effect of NTM on pwCF undergoing LTx on the risk of developing NTM disease post-LTx, survival after LTx, and the development of CLAD. The studies were small and at times contradictory. In the era of highly effective modulator treatments, as some centres do not offer LTx to people with a history of NTM, there is an urgent need for more data to guide decision-making. FUNDING: This review is part of a suite of reviews on NTM funded jointly by the CF Foundation and the CF Trust. REGISTRATION: Protocol registration (2024): www.crd.york.ac.uk/PROSPERO/view/562682.
23 Apr 2026
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