Elsevier

The Lancet Oncology

Volume 13, Issue 10, October 2012, Pages e445-e459
The Lancet Oncology

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

https://doi.org/10.1016/S1470-2045(12)70361-3Get rights and content

Summary

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.

Introduction

In high-income countries, about one child per 440 will develop cancer beforethe age of 15 years.1, 2 Cancer accounts for 19% of deaths in children older than 1 year in Europe.3 Delayed diagnosis of paediatric tumours has been reported regularly for more than 50 years4 and leads to feelings of remorse or guilt for parents and physicians, loss of confidence, and sometimes conflicts.5, 6 In some countries, more malpractice claims related to diagnostic errors are associated with cancer than with any other disease in children.7

The only review8 of delayed diagnosis in paediatric cancer so far (published in 2007) stated that “the impact of diagnostic delays on the prognosis of children with cancer is still unknown” and that “more studies are needed to investigate this potential impact”. However, this review did not follow specific methods, pooled studies from developing and developed countries, and did not include all papers published before 2007 (the authors selected only 21 of the 64 eligible studies published in English from developed countries). Since 2007, more research has been published, including that about the consequences of delayed diagnosis.

We aimed to do a systematic review of available information about the distribution, determinants, and consequences of time to diagnosis of paediatric cancers and thus to identify tumours for which long time to diagnosis is not associated with adverse outcome (information that could reduce guilt in parents and physicians about the consequences of these delays and improve the quality of the reports of court-appointed experts in malpractice claims) or more research is needed. Our secondary objective was to compare available information about the consequences of long time to diagnosis with the conclusions of court-appointed experts in malpractice claims.

Section snippets

Search strategy and selection criteria

We systematically reviewed studies until Dec 31, 2011 (inclusive) about the distribution, determinants, and consequences of time to diagnosis of paediatric cancers—specifically, leukaemias, lymphomas, brain tumours, neuroblastomas, kidney tumours, bone tumours, soft-tissue sarcomas, germ-cell tumours, and retinoblastomas. We excluded rare malignancies—ie, those accounting for less than 1·5% of the total incidence of childhood cancers (eg, hepatic tumours, malignant epithelial neoplasms, and

Data extraction and analysis

MM and J-FB independently extracted data for the distribution, determinants, and consequences of time to diagnosis (MC resolved discrepancies). Time to diagnosis (which we converted to weeks for our analysis) was defined in all studies as the interval between the first symptom attributable to the disease and final diagnosis. In some studies, it was separated into patient-associated delay (ie, the interval between onset of symptoms and first medical consultation) and doctor-associated delay (ie,

Clinical data

We identified 6412 studies; we assessed the full texts of 378 for eligibility and included 98 in our final analysis (appendix). 56% of the studies were published during or after 2000. The total sample was 22 619 patients.

Four of the ten studies in which investigators looked at time to diagnosis during the studies showed a significant decrease (brain tumours,33, 34 retinoblastoma,35 and all tumours pooled27) in the overall time to diagnosis or doctor's delay. Conversely, no significant

Discussion

We conclude, based on our review, that no adverse association and sometimes even a paradoxical positive relation exists between a long time to diagnosis and outcome for most types of CNS tumours (the extent of initial tumour invasion and survival) and osteosarcoma and Ewing's sarcoma (initial invasion and possibly survival). The relation between time to diagnosis and poor outcome for retinoblastoma (initial invasion, survival, and blindness) is well established. Because data are few and from

Conclusion

The general belief that a long delay before diagnosis of paediatric cancers leads to worse prognosis is often untrue. For most paediatric cancers, time to diagnosis probably depends most on the tumour's biology.8, 16, 20, 26, 33, 42, 108 The apparently contradictory relation between a long time to diagnosis and better outcomes that is noted in some studies does not mean that diagnosis should be delayed on purpose. Early referral to specialists could help with anxiety and distress in patients

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