Delays in diagnosis of paediatric cancers: a systematic review and comparison with expert testimony in lawsuits.

Delayed diagnosis of paediatric cancers is reported regularly and is a source of remorse for physicians and parents and a leading cause of malpractice claims. We did a systematic review of information about the distribution, determinants, and consequences of time to diagnosis of paediatric malignancies and compared these findings with those of court-appointed expert witnesses in malpractice claims in Canada and France. Time to diagnosis varied widely between tumour types in the 98 relevant studies (medians ranged from 2-260 weeks) without any significant decrease with time. Determinants of a long delay in diagnosis included older age, qualification of the first physician contacted, non-specific symptoms, histological type, and tumour localisation. Delayed diagnosis was associated with poor outcome for retinoblastoma and possibly for leukaemia, nephroblastoma, and rhabdomyosarcoma (data were insufficient for definitive conclusions). It was not associated with an adverse outcome for most CNS tumours, osteosarcoma or Ewing's sarcoma, and, paradoxically, was frequently associated with better outcomes than was short time to diagnosis in these cancers. A third of the court-appointed experts provided testimony concordant with the medical literature. The relations between delay in diagnosis and outcome are complex and probably depend more on tumour biology than on parental or medical factors.

Patient-reported outcomes in childhood head and neck rhabdomyosarcoma survivors and their relation to physician-graded adverse events-A multicenter study using the FACE-Q Craniofacial module.

INTRODUCTION: Adverse events (AE) of treatment are prevalent and diverse in head and neck rhabdomyosarcoma (HNRMS) survivors. These AEs are often reported by physicians; however, patients' perceptions of specific AE are not well known. In this study, we explored patient-reported outcomes measuring appearance, health-related quality of life (HRQOL), and facial function in HNRMS survivors. Second, we assess the relationship between physician grading of AE and patient reporting. MATERIALS AND METHODS: Survivors of pediatric HNRMS, diagnosed between 1993 and 2017, who were at least 2 years after completing treatment were invited to an outpatient clinic as part of a multicenter cross-sectional cohort study. At the outpatient clinics, survivors aged ≥8 years filled out the FACE-Q Craniofacial module; a patient-reported outcome instrument measuring issues specific to patients with facial differences. AE were systematically assessed by a multidisciplinary team based on the Common Terminology Criteria of Adverse Events system. RESULTS: Seventy-seven survivors with a median age of 16 years (range 8-43) and median follow-up of 10 years (range 2-42) completed the questionnaire and were screened for AEs. Patient-reported outcomes varied widely between survivors. Many survivors reported negative consequences: 82% on appearance items, 81% on HRQOL items, and 38% on facial function items. There was a weak correlation between physician-scored AEs and the majority of patient-reported outcomes specific for those AEs. CONCLUSIONS: Physician-graded AEs are not sufficient to provide tailored care for HNMRS survivors. Findings from this study highlight the importance of incorporating patient-reported outcome measures in survivorship follow-up.

Inadequate critical appraisal of studies in systematic reviews of time to diagnosis.

OBJECTIVE: To analyze tools used to critically appraise primary studies included in systematic reviews (SRs) of time to diagnosis (TTD). STUDY DESIGN AND SETTING: We systematically searched MEDLINE via PubMed and Web of Science for SRs of TTD published up to the end of February 2015; we identified and characterized tools used for critical appraisal and classified their items. RESULTS: From 1,936 articles identified, we included 45 SRs that aimed to summarize the available information on the length (n = 16), determinants (n = 31), and/or consequences (n = 14) of TTD. For the 23 SRs (51%) reporting a critical appraisal process, 21 different tools were used, with 232 items assessing quality of reporting (64%), risk of bias or threats to generalizability (43%), statistical issues (5%), and/or an unclear domain (0.5%); 11% were specific to TTD issues. Overall, 36% of the 45 SRs assessed risk of bias and/or threats to generalizability. CONCLUSION: Assessment of risk of bias and threats to generalizability in primary studies included in SRs of TTD is infrequent, nonstandardized and rarely concerns TTD study specificities. These findings highlight the need for guidance on critical appraisal of studies of TTD.

Quality of reporting of studies evaluating time to diagnosis: a systematic review in paediatrics.

OBJECTIVE: An ever-increasing number of studies analyses the distribution, determinants and consequences of time to diagnosis and delays. Weaknesses in their reporting can impede the assessment of the risks of bias and variation and thus create a risk of invalid conclusions and counterproductive clinical and public health efforts. This study sought to assess systematically the quality of reporting of articles about time to diagnosis in paediatrics. DESIGN: Two authors identified and analysed the quality of reporting of 50 consecutive articles assessing these intervals published from 2005 through October 2011, according to a checklist we developed of 35 items potentially associated with risks of bias and variation. MAIN OUTCOME MEASURE: Frequency of articles reporting each item. RESULTS: Symptoms that should trigger a diagnostic procedure were reported in 28% of the articles; only two articles reported whether all patients with these symptoms underwent that procedure. Only 44% of the articles defined the beginning of the illness, 46% the date of diagnosis and 60% the distribution of time to diagnosis. Two studies met the criteria for all 11 items considered essential for assessing the risks of bias and variation in this type of study. INTERPRETATION: This study identified many weaknesses in the quality of reporting of studies of time to diagnosis in paediatrics, especially for items potentially related to risks of bias and variation. This finding underlines the need for the development of new (or the refinement of existing) guidelines for reporting this type of study.