In 2011 the first RCT reporting the effects of singleevent multilevel surgery2 was published in the Journal of Bone and Surgery7 and the 5-year outcomes were reported in Gait & Posture this year.8 Our study group investigated the stability of the Gross Motor Function Classification System level9 and this study suggests that RCTs are not indicated if the surgical interventions are carefully planned using 3D gait analysis and calculation of the Movement Analysis Profile and the Gait Profile Score.10 Surprisingly, these important papers were not included in this review. Hip surveillance was given a green light intervention, but it is not an intervention, itself. The fact, that painful hip dislocation remains a common problem affecting children with CP in Scandinavia and Australia11–14 and the surgical treatment options were not addressed appropriately in our opin-

ion. However, there were no papers included in this review explaining the lack of effectiveness of non-surgical interventions and the need for preventive and reconstructive surgery in the CP hip.15 It is clear the hips will continue to dislocate in children with CP if surgery is not performed. We feel strongly that this could mislead therapists and parents who may feel that surgery should not be necessary and that they have somehow failed the patient with CP when the hip does finally dislocate. We want to, again, applaud the authors for their hard work and thank them for their contribution to the field, but for all of the reasons detailed above we have serious concerns that this paper may mislead parents, therapists, and policy makers and may subsequently delay or restrict access to effective and valuable treatment options for our patients.

REFERENCES 1. Novak I, McIntyre S, Morgan C, et al. A systematic

6. Rutz E, Hofmann E, Brunner R. Preoperative botu-

review of interventions for children with cerebral palsy:

linum toxin before muscle lengthening in cerebral palsy.

the state of the evidence. Dev Med Child Neurol 2013;

J Orthop Sci 2010; 15: 647–53.

55: 885–910.

7. Thomason P, Baker R, Dodd K, et al. Single-event mul-

2. Rutz E, Baker R, Tirosh O, Brunner R. Are results after

tilevel surgery in children with spastic diplegia: a pilot

single-event multilevel surgery in cerebral palsy durable?

randomized controlled trial. J Bone Joint Surg Am 2011;

Clin Orthop Relat Res 2013; 471: 1028–38.

93: 451–60.

11. Soo B, Howard JJ, Boyd RN, et al. Hip displacement in cerebral palsy. J Bone Joint Surg Am 2006; 88: 121–9. 12. Connelly A, Flett P, Graham HK, Oates J. Hip surveillance in Tasmanian children with cerebral palsy. J Paediatr Child Health 2009; 45: 437–43. 13. H€agglund G, Lauge-Pederson H, Wagner P. Characteristics of children with hip displacement in cerebral palsy.

3. Brunner R, Baumann JU. Clinical benefit of reconstruc-

8. Thomason P, Selber P, Graham HK. Single Event Mul-

tion of dislocated or subluxated hip joints in patients

tilevel Surgery in children with bilateral spastic cerebral

with spastic cerebral palsy. J Pediatr Orthop 1994; 14:

palsy: a 5 year prospective cohort study. Gait Posture

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BMC Musculoskelet Disord 2007; 8: 101. 14. Terjesen T. The natural history of hip development

4. Smith GC, Pell JP. Parachute use to prevent death and

9. Rutz E, Tirosh O, Thomason P, Barg A, Graham HK.

15. Rutz E, Brunner R. The pediatric LCP hip plate for fix-

major trauma related to gravitational challenge: systema-

Stability of the Gross Motor Function Classification

ation of proximal femoral osteotomy in cerebral palsy

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and severe osteoporosis. J Pediatr Orthop 2010; 30:

327: 1459–61.

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726–31.

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following botulinum toxin type-A treatment in children

as a result of single event multi-level surgery in cerebral

with cerebral palsy. Dev Med Child Neurol 2013; 55: 813–20.

palsy. Gait Posture 2013; 38: 455–60.

How to bridge the gap between systematic reviews and clinical guidelines Ilona Autti-R€ am€ o1, Ann Christin Eliasson2, Hans Forssberg2,3,4 1 Research Department, The Social Insurance Institute, Helsinki, Finland; 2 Department of Women’s and Children’s Health, Karolinska Institutet, Stockholm; 3 Neuropaediatrics, Astrid Lindgren Children’s Hospital, Stockholm, Sweden; 4 Chairman, European Academy of Childhood Disability. Correspondence to: [email protected] doi: 10.1111/dmcn.12412

SIR–There is a growing demand for evidence-based recommendations for managing children with cerebral palsy 398 Developmental Medicine & Child Neurology 2014, 56: 390–406

(CP). Numerous interventions are provided within ordinary healthcare systems and diverse alternative treatments are advertised on the internet, making it difficult for parents to decide whether to adhere to current national practices or to try alternative interventions. In the absence of well-grounded recommendations, health professionals cannot help parents make accurate decisions and clinical practice is often based on traditions that differ greatly, for example, between European countries. Some clinics are still practising according to concepts invented several decades ago and have not applied new findings indicating what elements of an intervention are critical for effective

outcomes. Evidence-based information and recommendations are also required by policymakers who must decide what treatments should be funded; for example, the implementation of the EU Cross-Border Healthcare Directive requires that Member States define what healthcare services provided abroad will be reimbursed. The identified lack of evidence-based recommendations has inspired almost exponential growth in the number of highquality intervention studies and systematic reviews of CP in recent years. However, a systematic review is only a first step in the process of developing recommendations or guidelines. The need to develop evidence-based recommendations in the field of childhood disability has been on the agenda of the European Academy of Childhood Disability (EACD) for several years. In collaboration with a German effort led by Rainer Blank, recommendations for managing developmental coordination disorders (DCDs) were developed and published in DMCN.1 The process involved experts from many disciplines and professions who, together with parents and other stakeholders, performed a series of steps, starting with patient needs, and a systematic review. At the end of the process, consensus was reached by all participants. Next, the EACD intended to develop recommendations for CP using a similar procedure, but the process never gained momentum and was terminated at the last EACD annual meeting in Newcastle, UK. A major reason for terminating the process was the complexity of CP, which is a heterogeneous condition blending motor, sensory, and cognitive disorders, often accompanied by other medical symptoms, and calling for the involvement of many disciplines and professions. It turned out to be impossible to make recommendations for the entire group of cerebral palsies at different functional levels, in contrast to the more homogenous entity DCD. A group of Australian researchers, led by Iona Novak, has published a systematic review in DMCN covering all types of interventions applied to children with CP.2 They have taken on a formidable task in reporting the current level of evidence for an extensive range of CP interventions. Since their systematic review is based largely on earlier systematic reviews, they mainly confirm previous findings. This type of systematization and summarization of existing research is extremely important and the authors should be commended for their ambitious effort. The systematic review identifies areas where evidence for common interventions is lacking, prodding clinicians and researchers to undertake the research required to prove their efficacy or identify other treatments with better outcomes. However, the authors’ ambitions did not end with providing evidence summaries. To help clinicians who find it difficult to apply the results of systematic reviews in relation to their clinical practice, the authors provide clear information on whether the evidence is strong enough to justify using an intervention. They use the Grading of Recommendation Assessment, Development and Evaluation

(GRADE) system as well as the Evidence Alert Traffic Light System that results in simple ‘traffic light’ signals, such as ‘Do it’ or ‘Don’t do it’. The systematic review confirms that several treatments now used lack scientific support, signalling these with red or yellow traffic lights. As expected, proponents representing unfavourably assessed treatments and therapies have reacted strongly, defending interventions that they have learned to value based on clinical experience and certain kinds of research, such as prospective follow-up studies, often not included in systematic reviews with strict inclusion criteria. Some of these defensive claims are not convincing – ‘so many clinicians and patients cannot be wrong’ – although they rightly emphasize the importance of patient experience. However, strong arguments from several experienced clinical and research commentators raise serious concerns about how recommendations for CP interventions should be developed and disseminated. Can recommendations be made by a relatively small research group lacking expertise in several of the areas reviewed? Can they be based solely on clinical trials? Can they be published without broader consensus among researchers and clinicians? Is the paper of Novak et al. indeed simply a systematic review, as stated in the title, or does it provide recommendations intended to be used by professionals and decision-makers? Systematic reviews are conducted in order to summarize existing evidence. Evidence concerns facts (actual or asserted) intended for use in supporting a conclusion, but is not the same as the conclusion. Judgments are needed for decisions. Sackett et al.3 have stated that evidence-based medicine is the integration of the best research evidence with clinical expertise and patient values, so research evidence alone is not enough for recommendations or clinical decision-making. Commentators have noted that clinical relevance has not been taken into account by Novak et al. Clinical expertise constitutes the ability to use clinical skills and past experience to identify each patient’s unique health state, diagnosis, risks, values, and expectations as well as the benefits of potential interventions. To make a decision, or to develop recommendations, these clinical parameters must be considered in combination with the existing evidence. In addition, the patient’s values and individual preferences must be integrated into clinical decisions if they are to serve the patient best. Patient needs also must be considered when recommendations or guidelines are being developed. Such patient values, as well as the weight of clinical expertise, were not considered in this systematic review, making it an insufficient basis for recommendations or decision-making. A problem with the traffic light system as applied in this systematic review is that it does not take into account the heterogeneity of the CP population, for example, in terms of type of CP, severity level, sensory, and cognitive functions, medical comorbidities, age, and context. Guyatt et al.4 and van Tulder et al.5 have identified important questions to ask when appraising the clinical applicability Letters to the Editor

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of evidence-based research results, for example: Are the patients described in detail so that you can decide whether they are comparable to those seen in your practice? Are the interventions described well enough that you can implement them for your patients? Were all clinically relevant outcomes measured appropriately and reported fully? Are the likely treatment benefits worth the potential harms and costs? In light of these questions, it is obvious that traffic lights cannot be uniformly applied to the whole heterogeneous CP population and cannot constitute a sound basis for recommendations, best practices, or health policy decision-making. Novak et al. used the GRADE system as a framework but not as a method. GRADE is a good though cumbersome method to use when formulating clinical guidelines. When using GRADE, not all outcomes are treated as having the same value but are classified as critical, important, or not important. Ideally, outcomes ought to be classified jointly with patients. Using GRADE, one can also look at the evidence from various perspectives, ranging from that of patients to that of the funders; one must consider the harms, costs, organizational requirements, and family and clinical applicability in a given national context. In the GRADE system, evidence based on randomized controlled trials (RCTs) is methodologically regarded as ‘high-quality’ evidence, but confidence in evidence may be decreased by study limitations (efficacy vs effectiveness), inconsistency of results across studies, indirectness of evidence, and reporting bias. Evidence from lower-quality studies may be upgraded if the magnitude of an effect is very large, a dose–response relationship is found, and all plausible confounders would decrease the magnitude of an apparent treatment effect. Rehabilitation interventions are also never ‘active’ as such: change requires activity, which is based on motivation and opportunity, and motivation is based on meaningful goals. Meaningful goals must be individualized, so change cannot be captured by one-size-fits-all outcome measures. Furthermore, most RCTs cover only a short period, while CP is a lifelong disorder and rehabilitation must apply a long-term perspective. Our last concern with this systematic review is its focus on whether interventions worked. Research also needs to

focus on how to make interventions work and how to meet patient needs. RCTs will never supply all the answers when it comes to developing optimal care for children and adults with CP. We also need qualitative studies of patients’ perspectives, values, needs, and goals. We need better skills for judging and interpreting scientific studies, and when reading a research article one must ask: how much confidence do I have that the ‘cause’ of the effect has been identified? Can the clinically relevant question be answered by the study design? Many clinically relevant questions cannot be answered by RCTs – or by systematic reviews, for that matter. In summary, we think that the Novak et al. systematic review is important. From an overarching perspective, it covers all interventions studied in the entire heterogeneous group of cerebral palsies. It provides us with a list of current evidence regarding the undefined effects of various interventions. The traffic light system conveys this information in an easily graspable way, but it moves the systematic review into a grey area close to recommendations for best practice and information to support decision-making. Providing such information, however, would require a process in which evidence from the systematic review is balanced against clinical expertise and patient needs. In the case of this systematic review, such matters were not considered before publication; instead, they are now being considered in public via strong arguments voiced by leading clinical experts. One advantage of such public discussion is that it enhances awareness of the complex processes involved in developing evidence-based recommendations and guidelines. However, there is also a risk that clinicians and decision-makers may take the traffic light signals at face value. Some fear that the findings of this systematic review could be used by healthcare managers to justify healthcare cost-cutting. If so, this would represent a fundamental misunderstanding and misapplication of evidence-based medicine. The development of clinically valid patient-oriented guidelines for managing children with CP that can be applied internationally would require a much more complex process than that applied in this systematic review.

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