A systematic review of Clinical Practice Guidelines for the management of fractures in children to develop the WHO’s Package of Interventions for Rehabilitation

Abstract

INTRODUCTION

Fractures have been identified as one of the 20 major health conditions for the World Health Organization’s (WHO) Package of Interventions for Rehabilitation (PIR) — that includes also the needs of children and youth. The identification of existing interventions for rehabilitation and related evidence is a crucial step along the development of the PIR. The methods for the identification have been developed by WHO Rehabilitation Program and Cochrane Rehabilitation under the guidance of WHO’s Guideline Review Committee Secretariat.

EVIDENCE ACQUISITION

This paper is part of the “Best Evidence for Rehabilitation” (be4rehab) series, developed according to the methodology presented in the PIR introductory paper. It is a systematic review of the existing Clinical Practice Guidelines (CPGs) on fractures in pediatric population published between 2009 to 2019.

EVIDENCE SYNTHESIS

We identified seven relevant CPGs after title and abstract screening. According to inclusion/exclusion criteria and after checking for quality, publication time, multidisciplinarity and comprehensiveness, we have been able to include two CPGs: one addresses the treatment of supracondylar humerus fractures and the other provides recommendations on the treatment of diaphyseal femur fractures.

CONCLUSIONS

The selected CPGs on the management of supracondylar humerus and diaphyseal femur fractures in pediatric population include few recommendations considered as interventions for rehabilitation, of low quality of evidence and weak strength. We found several gaps in specific rehabilitative topics. High quality studies are absolutely needed to upgrade the quality of available evidence to inform future development of guidelines.

Introduction

The World Health Organization (WHO) has the strategic priority goal of achieving Universal Health Coverage (UHC), that means “all people receive quality health services that meet their needs without being exposed to financial hardship in paying for the services”.¹ UHC includes rehabilitation among the services to be provided. As part of the WHO Rehabilitation 2030 - A call for action initiative,² the WHO Rehabilitation Program is developing a Package of Interventions for Rehabilitation (PIR, formerly “Package of Rehabilitation Interventions”) to support ministries of health in integrating rehabilitation services into national health systems.³

The development of the PIR takes a stepwise approach.³ The second step, here referred to as “Best Evidence for Rehabilitation” (be4rehab), requires the identification of interventions for rehabilitation and related evidence for the health conditions selected in the first step. The WHO Rehabilitation Program and Cochrane Rehabilitation have developed the corresponding methodology under the guidance of WHO’s Guideline Review Committee Secretariat and are collaborating in conducting this step. Be4rehab includes a series of systematic reviews on Clinical Practice Guidelines (CPGs) for the different health conditions. Interventions and related evidence are identified from these. The identified interventions will undergo a consensus process before being included in the final PIR. Information related to the provision of the interventions will be added. All information will undergo a review process before developing the final version of the PIR.

Fractures are a common cause of morbidity during childhood and adolescence.⁴ Factors such as age and gender are associated with increased incidence of fractures in this population.⁴ The overall rate of pediatric fractures ranges from 12 to 36.1/1000/year⁵ with an incidence of 4.38/1000/year in children less than 5 years old⁶ and a general predominance for boys (42-64%) versus girls (27-40%).⁷ Typical injury mechanisms are falls even if about one third of fractures occurs after sport trauma.^{8, 9} Upper limb is widely involved with the distal radius as the most frequent site of fracture, representing about the 25-43% of all childhood fractures.⁹

Due to the effects on activity restriction and socioeconomical impact, pediatric fractures are a major health problem.¹⁰ Little is known about the rehabilitation strategy in this population, although children could benefit from rapid recovery of painless motion, avoiding the risk of disuse osteopenia and muscle atrophy.¹¹ Early rehabilitation programs in children may lead to a full recovery of body function, as reported, for example, after lateral condylar fractures of the humerus.¹²

The objective of this paper is to report on the results of the systematic search for CPGs relevant to rehabilitation of pediatric population with fractures, limiting the search to the following sites: humerus, radius, femur/hip, and tibia. The specific objectives are to present the topics of the recommendations and the current state of evidence available from the identified CPGs.

Evidence acquisition

This systematic review of CPGs has been developed in full compliance with the methodology presented in the introductory PIR paper.³ These stages have been followed (Figure 1):

Figure 1.

—Results of the screening process (diagram).

• systematic literature search: CPGs have been searched in the following databases: PubMed, Pedro, CINAHL, Embase, Google Scholar, Guidelines International Network (GIN), US National Guideline Clearinghouse, UK National Institute for Clinical Excellence (NICE), Australian National Health and Medical Research Council clinical practice guidelines, UK National Library for Health Guidelines Database, Scottish Intercollegiate Guidelines Network (SIGN), Canadian Medical Association Infobase of Clinical Practice Guidelines, Agence Nationale d’Accréditation et d’Évaluation en Santé (France), New Zealand Guidelines Group, eGuidelines, EBMPracticeNet, USA National Guideline Clearinghouse (NGC), WHO Guidelines, Haute Autorité de Santé (HAS, Frankrijk, Agency for Healthcare Research and Quality (AHRQ, VS), National Health Service Evidence (VK) American Academy of Orthopedic Surgeons (AAOS), British Orthopaedic Association (BOA), BOA Standards for Trauma and Orthopaedics January 2012, Société Française de Chirurgie Orthopédique et Traumatologique (SOFCOT), British Society for Surgery of the Hand (BSSH), National Health Society (NHS). Considering the heterogeneity of the rehabilitation needs of different bones fractures, we have decided to limit the search to long appendicular bones, including humerus, radius, femur/hip, and tibia. The search strategies are reported in Supplementary Digital Material 1 (Supplementary Text File 1). The search was performed on February 27^th, 2019, and included all the documents from 2009 to 2019 regarding CPGs on fractures in both children/youth and adults;

• independent abstract and full-text screening of the retrieved documents by members of the technical working group (TWG);

• independent evaluation of the CPGs quality with the “Appraisal of Guidelines for Research and Evaluation” (AGREE II) tool by 2 members of the TWG:¹³ a specific focus has been given to items 7,8,12 and 22 where the average result had to be >2 (AGREE/4), and to the items 4, 7, 8, 10, 12, 13, 15, 22 and 23 whose average sum score had to be >45 (AGREE/9);

• final selection of a maximum of 5 CPGs for each group ages children/youth and adults according to the following criteria: 1) quality 2) publication time 3) multiprofessionality 4) comprehensiveness. This decision has been reached by agreement of the whole group;

• data extraction using a standardized form, which comprises information on the recommendation (type of recommendation, dosage, target group, etc.), the strength of recommendation, and the quality of the evidence used to inform the recommendation.

With respect to the published protocol no changes have been performed. The quality check and the methodological support for this study have been provided by Cochrane Rehabilitation.

The topics addressed by each CPG for the different types of recommendation (service, assessment and intervention) have been extracted. The topics from the first CPG have been compared independently by two authors and integrated with those coming from the second. If needed, agreement by discussion was reached involving a third author. The process has been repeated for all the CPGs until final agreement on the topics was reached.

Evidence synthesis

The result of the selection process is reported in Figure 1. Our TWG identified 7 CPGs on fractures in children/youth, based on title and abstract screening.^14-20 We excluded five of them since: two were summaries of CPGs,^{14, 15} one was an editorial about a CPG,¹⁶ and two did not report on rehabilitation interventions.^{17, 18} The two included CPGs were American Academy of Orthopedic Surgeons, Treatment of pediatric supracondylar humerus fractures, 2012 (AAOS pediatric humerus),¹⁹ and American Academy of Orthopedic Surgeons, Treatment of pediatric diaphyseal femur fractures, 2009 (AAOS pediatric femur)²⁰ (Table I).

Table I. —Guidelines found and selected, and their respect of the criteria used to reach the final choice.^{19, 20}.

Included guidelines	AGREE ratings						Multiprofessional team	Topic	Publication date
	Total	Average of key items
		7	8	12	22	4, 7, 8, 10, 12, 13, 15, 22, 23
AAOS pediatric humerus19	100	7	7	7	7	63	Yes	Pediatric supracondylar humerus fracture	2012
AAOS pediatric femur20	100	7	7	7	7	63	Yes	Pediatric diaphyseal femur fracture	2009

We extracted six recommendations relevant to rehabilitation: two on services and four on interventions. The identified recommendations per type and topic (functioning domain) considered by the selected CPGs are resumed in Table II and Table III. The quality of evidence ranges from moderate to low with a weak strength of recommendation (Table IV).

Table II. —Number of recommendations per type of recommendation for each guideline.^{19, 20}.

Guideline	N. of recommendations on:
	Service	Assessment	Intervention
AAOS pediatric humerus19	1 (50%)	0 (0%)	1 (50%)
AAOS pediatric femur20	1 (25%)	0 (0%)	3 (75%)

Table III. —Number of identified recommendations per topic (functioning domain) and recommendation type (service, assessment, interventions).^{19, 20}.

Topics	Selected guidelines
	AAOS pediatric humerus19	AAOS pediatric femur20
Service recommendations
Postoperative management	We are unable to recommend for or against routine supervised physical or occupational therapy for patients with pediatric supracondylar fractures of the humerus (p 136, recommendation 11)	We are unable to recommend for or against outpatient physical therapy to improve function after treatment pediatric diaphyseal femur fractures (p 57, recommendation 11)
Assessment recommendations
Intervention recommendations
Therapeutic exercise	We are unable to recommend an optimal time for allowing unrestricted activity after injury in patients with healed pediatric supracondylar fractures of the humerus (p 141, recommendation 12)
Orthosis and prosthesis		Treatment with a Pavlik harness or a spica cast are options for infants six months and younger with a diaphyseal femur fracture (p 13, recommendation 2) We suggest early spica casting or traction with delayed spica casting for children age six months to five years with a diaphyseal femur fracture with less than 2 cm of shortening (p 15, recommendation 3) We are unable to recommend for or against early spica casting for children age six months to five years with a diaphyseal femur fracture with greater than 2 cm of shortening (p 20, recommendation 4)

Table IV. —Strength of recommendation and quality of the evidence of the selected guidelines. Since the reference scales adopted by each guideline are not directly comparable, we propose here the recommendations according to two 3-point Likert scales.^{19, 20}.

Guideline	Body of evidence			Strength of recommendation
	RCTs, Systematic reviews or meta-analyses*	Clinical studies	Expert opinion	Strong	Intermediate	Weak
AAOS pediatric humerus19	1 (50%)	0 (0%)	1 (50%)	0 (0%)	0 (0%)	2 (100%)
AAOS pediatric femur20	0 (0%)	5 (83.3%)	1 (16.7%)	0 (0%)	1 (25%)	3 (75%)

*At least one RCT or one systematic review are required to classify in this column.

As for post-operative management of pediatric supracondylar humerus and diaphyseal femur fracture, both CPGs are unable to recommend for or against routine supervised physical or occupational therapy and outpatient physical therapy to improve function, respectively (strength of recommendation inconclusive; quality of the evidence low).^{19, 20} For AAOS pediatric humerus fractures, the authors are also unable to recommend an optimal time to end restriction of activity after supracondylar humerus fracture healing (strength of recommendation inconclusive).¹⁹ For AAOS pediatric femur fractures, the authors recommend treatment with a Pavlik harness or a spica cast as options for a diaphyseal fracture in infants six months or younger (strength of recommendation weak/conditional; quality of the evidence low); while for children up to 5 years they recommend an early spica casting or traction followed by spica casting only when the shortening of the diaphyseal femur after fracture is less than 2 cm (strength of recommendation weak/conditional; quality of the evidence moderate).²⁰

Discussion

We performed the search on the CPGs for the rehabilitation of pediatric population with fractures, limiting the search to the following sites: humerus, radius, femur/hip, and tibia. We found only two CPGs regarding supracondylar humerus and diaphyseal femur fractures in this patient population that satisfied our quality criteria. Both CPGs have been primarily planned to guide the orthopedic management and therefore are not focused on rehabilitation.^{19, 20}

As for the humerus site, we have two inconclusive recommendations: one on service and the other on intervention.¹⁹ The only available evidence to draw recommendation about service derived from a single unblinded randomized controlled study which considered only patients treated in open reduction, highlighting no difference in elbow motion after 1 year between children treated twice or three times weekly with physiotherapy (passive joint and soft tissue stretching techniques, and active exercises according to Sherrington principles²¹) and those not receiving any rehabilitative intervention.²²

As for the femur site, the available CPG echoes the one on the humerus site stating that there is no available evidence to recommend or not outpatient physical therapy to improve patient function after treatment, referring the decision to the clinician for the single specific case.²⁰ Moreover, in AAOS pediatric femur, the authors give recommendations on the use of splints, casts, and traction²⁰ even though in this case there is a lack of information on the therapeutic exercise to perform during or after the use of these orthoses.

A possible reason for the few rehabilitative recommendations for children and adolescents after fracture is to be found in the high rate of spontaneous bone healing commonly observed in this population. However, a comprehensive description of functioning, according to International Classification of Functioning, Disability and Health (ICF),²³ and a selection of specific patient-reported outcome to promote the assessment of the functional recovery after fracture should be integrated within the pediatric clinical care.²⁴ The kind of treatment (surgical vs conservative), the severity of the fracture (displaced or not), and its consequences (vascular or nervous injuries) should all be considered as they can be risk factor for an incomplete or delayed recovery. Challenging management issues concerning possible complications may occur following specific fractures with relevant functional implications in pediatric population.²⁵ For example, in pediatric uncomplicated forearm fractures, a combination of fracture severity, related treatment (more invasive), and immobilization may result in slower range of motion restoration;²⁶ distal femoral epiphysis fractures might result in poor bone healing and loss of range of motion;²⁵ there might be low bone mineral mass after cast immobilization following upper limb fractures;²⁷ a possible complication of supracondylar humerus fractures might be injuries of the brachial artery and radial or median nerves that should be promptly investigated and addressed with appropriate rehabilitation interventions.²⁸

On the other hand, we know that joint stiffness is rarely reported in pediatric patients compared with adults,²⁹ and that other complications are rarely observed, making physiotherapy often unnecessary. However, CPGs focused on the possible rehabilitation needs of the pediatric population following a fracture would be useful to orientate rehabilitation health professionals in all complicated cases, needing an accurate individualized rehabilitation plan. In particular, in case of femur fractures, CPGs on rehabilitation might be useful to advise on assisted weight bearing and on the use of crutches or other walking aids, as well as to assist parents in the home management of their children.

There’s a general lack of CPGs addressing the point of view of rehabilitation health professional, with an overall low to very low quality of evidence and a weak/conditional strength of recommendation both in the adult³⁰ and in the pediatric population.

Conclusions

In this systematic review we reported a substantial lack of CPGs rehabilitation oriented and a relevant evidence gap of rehabilitation interventions targeting fractures in the pediatric population. Only two CPGs, on humerus and femur sites, have been identified. However, they are both mainly focused on the orthopedic management of the fractures. Rehabilitation appears as an unmet topic in this population and high-quality studies are mandatory to implement the current state of the art.

Supplementary Digital Material 1

Supplementary Text File 1

Search strategy