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Charateristics and Incidence

of Child Abuse in Young

Children with a Femur

Fracture – a systematic

review.

Patricia Ribeiro

11399872

University of Amsterdam-

patricia.batistaribeiro@student.uva.nl

Supervisor:

Co-assessor:

M.H.J. Loos, MD

Prof. dr. R.R. van Rijn

Department of Paediatric Surgery, AMC

Department of Radiology, AMC

m.h.loos@amc.uva.nl

r.r.vanrijn@amc.uva.nl

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Table of Contents

Table of Contents ... 3 List of Figures ... 4 List of Tables ... 4 1. Abstract ... 6 2. Introduction ... 7 3. Methods... 9 3.1 Search Strategy ... 9 3.2 Selection process ... 9

3.3 Data Collection Process ... 10

3.4 Assessment of risk of bias in individual studies ... 11

4. Results ... 13

4.1 Incidence ... 13

4.2 Age ... 14

4.3 Gender ... 14

4.4 Mechanism of injury ... 15

4.5 Location of the Fracture ... 16

4.6 Type of Fracture ... 16

4.7 Time Before Going to the Hospital ... 17

4.8 Socio-Economic Status ... 17 4.9 Additional Injuries ... 18 5. Discussion ... 20 Incidence ... 20 Age ... 20 Gender ... 20 Mechanism of injury ... 20

Location of the Fracture ... 21

Type of Fracture ... 21

Time Before Going to the Hospital ... 22

Socio-economic Status ... 22

Additional Injuries ... 22

6. Limitations ... 24

7. Conclusions and Recommendations ... 25

8. References ... 26

9. Appendix ... 29

9.1 Appendix I – Project Plan ... 29

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9.3 Appendix III – Excluded Studies List ... 37

9.4 Appendix IV – Selected Studies Information ... 47

9.4.1 Overall view of the Selected Studies ... 47

9.4.2 Methods and Results of the Selected Studies ... 51

9.4.3 Outcomes of the Selected Studies ... 65

9.4.4 Rate of the Studies ... 71

9.5 Appendix V – Additional Information ... 72

9.6 Appendix VI – Abbreviation List ... 73

List of Figures

Figure 1: Flow chart of study selection process ... 10

Figure 2: SPUTOVAMO-R questionnaire10 ... 72

Figure 3: GRADE quality assessment criteria19 ... 72

List of Tables

Table 1: Overview of Rayyan findings part I ... 9

Table 2: Overview of Rayyan findings part II ... 9

Table 3: Overall view of incidence in the selected studies ... 13

Table 4: Overview of age as a risk factor ... 14

Table 5: Overview of gender as a risk factor ... 14

Table 6: Overview of mechanism of injury as a risk factor ... 15

Table 7: Overview of location of the fracture as a risk factors ... 16

Table 8: Overview of the type of fracture as a risk factor ... 16

Table 9: Overall view of time between injury and ED, as a risk factor ... 17

Table 10: Overall view of socio-economic status as a risk factor ... 18

Table 11: Overall view of additional injuries status as a risk factor ... 18

Table 12: Excluded studies part I ... 37

Table 13: Excluded studies part II ... 46

Table 14: Overall view of the selected studies... 47

Table 15: Methods and results of the selected studies ... 51

Table 16: Outcomes of the selected studies ... 65

Table 17: Rate of the selected studies ... 71

Table 18: Ranking of definitions on child abuse18 ... 72

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1. Abstract

Introduction:

Child abuse remains a social and a public health problem in a modern society. Fractures are the second most common indicator of physical abuse, after soft tissue injuries, such as bruises and burns, particularly in infants and toddlers. There is a 30% to 50% probability that a misdiagnose can lead to relapse of the abuse and a 5% to 10% probability that this misdiagnosis can lead to the child’s death. Despite the literature that is available on this subject, the diagnosis of child abuse is still not clear due to importance of the consequences of a misdiagnose. In children, femur fractures are the number one musculoskeletal injury that requires hospitalization. Other studies evaluated that 80% of non-ambulatory infants that is admitted in the hospital with femoral fractures and 30% of children, younger than 4 years of age, that have femur fractures, are associated with physical abuse.

Aim of the study

The aim of this systematic review is to establish qualitative evidences that explore the incidence and characteristics of nonaccidental trauma in children between 0 and 4 years of age with a femur fracture

Methodology

A literature search was performed on 3 databases of studies published until December 2016. A total of 204 studies were firstly found where ten were duplicates. 194 abstracts were read and 31 were included to do a full-screen. Out of those 31, 17 were included for the systematic review. All the reviews were elected by two independent members. The quality of evidence was performed individually and compared by the two reviewers using a tool that was adapt from specific guidelines that involved mainly, three factors: study design, selection of control group and certainty of abuse and external. Data such as age, gender, mechanism of injury, placement and type of fracture, time before going to the ED, socio-economic status and additional injury were collected.

Results

The incidence of femur fractures in child abuse vary between 6.07% and 45.9%. Younger age, with unclear or unexplained mechanism of injury, with a transverse or spiral diaphyseal femur fracture and additional injuries are the main characteristics in these studies. Socio-economic factors may be a risk factor, but further analysis needs to be performed to confirm it. Gender was not considered a risk factor due to the similarity of the incidence between both males and females. Not enough data was provided regarding the time between injury and seeking the ED, thus it was not considered a risk factor.

Conclusion

An individual risk factor is not enough to provide an accurate diagnose but using the group of features found in this study may lead to a better conclusion. The incidence of all the presented risk factors varies extensively, and the selected studies were rated from very low to low.

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2. Introduction

Child abuse remains a social and a public health problem in a modern society. Fractures are the second most common indicator of physical abuse, after soft tissue injuries, such as bruises and burns, particularly in infants and toddlers. 1–8 Ideally, the evaluation of a suspected case of child abuse needs

to be performed by a multidisciplinary medical team. It is important to acquire an accurate diagnosis, otherwise there is a high probability that the physical abuse may be repeated, or the child may die from the inflicted injuries. According to previous studies, there is a 30% to 50% probability that a misdiagnose can lead to relapse of the abuse and a 5% to 10% probability that this misdiagnosis can lead to the child’s death.3,8,9

At the Emergency Department (ED), the paediatrician starts by obtaining the history from the parents and from the child, when possible, followed by a full physical evaluation. The doctor from the ED can also consult a paediatrician specialised in child abuse and the Child Abuse Assessment Team (CAAT) for further investigation. The CAAT may involve several specialists such as surgeon, radiologist, psychiatrist and psychologist, all with a subspecialty in paediatrics; a paediatrician specialized in child abuse, social worker, forensic psychiatrist as well as an advisor forensic medicine, emergency nurses and physician. In USA there is also a lawyer in the CAAT.10 Other countries, such as the Netherlands,

the team doesn’t have a lawyer but it does involve the National State Protective Services (CPS). To assess possible child abuse cases, the Dutch Health Care Inspectorate, in 2009, incorporated in Dutch Hospitals a mandatory screening on child abuse, consisting of a questionnaire (SPUTOVAMO-R - Figure 2) to help the doctors determine the presented case is suspected child abuse case. The enquiry alerts for the coherency of the history with the age and injuries of the child; if the parents maintain the history throughout the appointment; if there is any delay from the time that the injury occurred and the attendance to the hospital and if there is a satisfactory explanation; the behaviour of the parents regarding the whole incident; if the physical exam (fracture type, pattern and location) shows any kind of suspicious injuries and if there are other injuries (i.e. suspected bruises).9,10

The presence of multiple fractures in different parts of the body such as: long-bones (humerus, tibia and femur), metaphyseal corner, ribs, spine, scapula; as well as different healing stages, unproportioned swelling compared to the type of injury and separations of the epiphyses, may also be indicators of non-accidental trauma injuries (NAT).3,4,9,11

In children, femur fractures are the number one musculoskeletal injury that requires hospitalization. According to Baldwin et al (2011)9 there have been several studies trying to characterize these type of

fractures, through type and placement, as an indicator of NAT.9 Accordingly, other studies evaluated

that 80% of non-ambulatory infants that is admitted in the hospital with femoral fractures and 30% of children, younger than 4 years of age, that have femur fractures, are associated with physical abuse.7,12

It has also been suggested that the morphology of the fracture may be an indicator of NAT.4 Flaherty et

al (2014)13 classified and explained the mechanism on the injury. It is stated that the metaphyseal

femoral fractures (MF) as high specificity for children less than 1 year of age, that suffered from child abuse. The authors explained that these fractures occur by twisting and pulling the extremities of the child, as well as the result of shear force by torsion and traction of the bone. Moreover, other types of femoral fractures can be present when it comes to child abuse. Different types of diaphyseal fractures

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8 (DF) of long bones are often observed in NAT injuries: transverse fractures are caused by applying a force that bends the bone perpendicularly to itself; spiral fractures occur when there is a twist or a torsion along the long axis of the bone; oblique fractures occur when the bone is bended and twisted at the same time; buckle fractures are the result of compressed forces taken by the bone throughout its length; greenstick fractures are damage to a young and soft bone by a bending force.13 There are also a

group of four fractures, the Salter Harris fractures, where I and II are the most common ones. These fractures take place in the distal part of the bone and are associated with severe trauma. Salter Harris type I results in the displacement of the distal physis, while Salter Harris type II also extends to the metaphysis, 85% of this type of fractures is the Salter Harris type II.14

Despite the literature that is available on this subject, the diagnose of child abuse is still not clear due to importance of the consequences of a misdiagnose.15 The majority of the studies indicate that, in a

non-ambulatory child, a femur fractures is evaluated as a NAT injury. Nevertheless, there are authors that disagree when it comes to children older than 1 year. Since these children can walk already, according to Blakemore et al (1996)16, femur fractures may also occur if a child trips while exercising

or falls from a low height.

The aim of this study is to establish qualitative evidences that can explore the incidence and risk factors of nonaccidental trauma in children between 0 and 4 years of age with a femur fracture, through a systematic review of pre-existent literature.

The primary expected outcome is to identify the characteristics of child abuse in children with a femur fractures. The secondary outcomes are: to determine the incidence, mortality, as well as hospital admission, of child abuse in children with femur fracture; clinical and radiological features of femur fractures due to child abuse; and the sociodemographic of parents with a child with a femur fracture due to child abuse.

Given the expected outcomes from this systematic review, the main question is “What is the incidence

and which characteristics are correlated with femur fractures due to child abuse in children, between 0 and 4 years of age?”.

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3. Methods

3.1 Search Strategy

To create a specific search strategy, a clinical health librarian with expertise in systematic review searching was consulted. Three Databases were searched: PubMed/MEDLINE, EMBASE and Cochrane Library. Medical subject headings (MeSH) and text words related to child abuse and femur fractures in title and abstract, were included. The reference lists of included studies will be scanned to identify relevant articles not identified during the search. All data published until December 2016 were included.

The number of total hits was 295, where: • Pubmed/MEDLINE - 114 hits • Embase - 176 hits

• Cochrane Library - 5 hits

After removing the duplicates in Endnote, the final number of hits was 204. Where: • Pubmed/MEDLINE - 114

• EMBASE - 87 • Cochrane Library - 3.

For more information about the project planning and the search strategy, see Appendix 9.1 and 9.2, respectably.

3.2 Selection process

This process was divided in two parts: firstly, two review authors, Marie-Louise Loos (MR) (supervisor) and Patrícia Ribeiro (PR) (student), independently screened the articles on title and abstract using the eligibility criteria and select the relevant articles to be included in the review. This was achieved using an online software, Rayyan. Secondly, when it was indistinct to include a specific article by title and abstract, the full text was obtained. Disagreement about inclusion of studies was solved by discussion. If a disagreement would consist, the third author (co-assessor Rick van Rijn) would be consulted. Publications that were duplicated, were excluded.

Table 1: Overview of Rayyan findings part I

Options Marie-Louise Patricia Ribeiro Final decision

Included 42 46 31

Excluded 152 148 163

Conflict 52 0

Table 2: Overview of Rayyan findings part II

Options Marie-Louise Patricia Ribeiro Final Decision

Included 12 13 17

Excluded 19 18 14

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10 A flow chart to document the study selection process was made, is presented below. The list of excluded studies is in the Appendix 9.3, attended with the reason of exclusion.

Figure 1: Flow chart of study selection process

3.3 Data Collection Process

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11 • Study design, number of participants

• Participant characteristics: age, gender, zip code (socio-economic status), femur fracture (type, mechanism of injury and location in the bone), presence of other physical injury, delayed presentation, referrer, referral to child protection services, history of traumatic injury, history of child abuse, mortality.

• The data will be extracted by two reviewers (ML and PR) independently. In case of disagreement, a discussion was made until the decision was unanimous.

• During data extraction duplicates were excluded, if some included studies overlap.

• Only data concerning femur fractures was reviewed. Other types of child abuse weren’t evaluated in the results because of search selection bias.

3.4 Assessment of risk of bias in individual studies

Marie-Louise Loos (MR) (supervisor) and Patrícia Ribeiro (PR) (student), independently reviewed the studies individually for the risk of bias, using an already establish protocol. This protocol was based on the Grading of Recommendations Assessment, Development and Evaluation (GRADE) guidelines17

and a study by Maguire et al (2008)18, built for a systematic review to identify the risk factors for

non-accidental burns in children. The protocol was adapted for the present study, accordingly.

The GRADE was designed to rate the quality, as well as grading strength, of the evidences for constructing guidelines, health technology assessments (HTA) and systematic reviews.19 This system

has an important limitation when assessing the quality of evidence, given that the starting point to initially assess the evidences was based on the study design of our selected studies. Contrary to this, GRADE guidelines’ rating system for systematic reviews is based on randomized trials. In cases of child abuses, for ethical reasons, this approach is not feasible. Furthermore, the aim of this study is to find risk factors of child abuse in young children with a femur fracture, and not perform a comparison on treatments and diagnostical approaches, which is one of the purposes of GRADE guidelines. Consequently, some upgrading or downgrading factors are not applicable (purple squares in Appendix 9.5 - Figure 3).

The certainty of abuse was established as an important factor, when assessing the quality of evidence. To define certainty of child abuse, Table 18 and Table 19 (see in Appendix 9.5) were agreed upon, based and adapted from a study by Maguire et al (2008)18, where the authors rank the definitions on

child abuse and accidental injury. As a result, a tool was designed to assess the quality of evidence for the individual studies of this review.

The grading tool is based on a 1 to 4-star rating by the following standards: 1. Appropriateness of study design.

Study design Grade

prospective design 4

cross-sectional 3

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12 case series without matched controls Downgrade

2. Risk of bias

a. Appropriateness of eligibility criteria - Desirably all ED/emergency room (ER) presentations. Downgrade if:

• CPS or CAAT -referred children only Admissions only is not a reason for downgrading.

b. Selection of control group - desirably all non-abused children from the same group or a comparable group in the same setting.

Downgrade if:

• control group selected from another population

• control group consists of other abused victims without femoral fractures.

c. Certainty of abuse - define ranking of abuse (1-5) and whether abuse was excluded in the deemed accidental cases using the ABC-ranking developed by Maguire et al (2008)18.

Desirably abuse ranking 1-3 and accidental ranking A or B. Downgrade if abuse ranking 4-5 AND if accidental ranking C. d. Confounding

Downgrade if failure to adequately control confounding factors.

e. Differential in surveillance for outcome in abused and non abused groups Downgrade if that is the case.

3. Generalizability

Downgrade if generalizability is low due to e.g. cultural factors. 4. Large magnitude of effect -> rate up

5. All plausible confounders increase confidence in the estimated effect -> rate up

Rate down for: Rate up for:

- 1 factor listed under ‘risk of bias’  1 star - >1 factor listed under ‘risk of bias’  2 stars - Other factors 1 star

- large magnitude of effect

- residual confounding increases confidence in estimated effects

Any disagreement about the evaluation of the studie’s quality was solved by discussion between ML and PR. If a disagreement would consist, the third author (co-assessor Rick van Rijn) would be consulted.

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4. Results

This chapter contains the overall view, the methods and results, and outcomes of the selected studies. Information provided by the 17 selected studies was gathered and organized in tables that are present in the Appendix 9.4. The rates attributed to the selected studies are present in the Table 17Error! Reference source not found. in the Appendix 9.4.4.

The overall view of all studies is summarized in Table 14 and contains the information about the type of study, the aim of study, the country where it took place, population abused/total population, control group, setting and inclusion period of the study. (Appendix 9.4.1)

The methods and results are summarized in Table 15 with information about the population included for review/reason exclusion, age, gender, mechanism of injury, fractures’ location, type of fracture, time before going to the ED, socio-economic status, additional injury and a summary of what data gathered was gathered. (Appendix 9.4.2Error! Reference source not found.)

The outcomes are summarized in Table 16 with information about the overall conclusion, significant risk factors, no significant factors and overall conclusion consistent with existing literature. (Appendix 9.4.3)

Furthermore, the characteristics age, gender, mechanism of injury, fractures’ location, type of fracture, time before going to the ED, socio-economic status and additional injury for the abused children, will be shown individually according to each characteristic.

Furthermore, the incident and the characteristics age, gender, mechanism of injury, fractures’ location, type of fracture, time before going to the ED, socio-economic status and additional injury for the abused children, will be shown individually according to each characteristic.

4.1 Incidence

The incidence in all selected studies is represented in Table 3

Table 3: Overall view of incidence in the selected studies

Study Authors Incidence

Anderson et al (1982)20 24/117 (20.51%) abused

Baldwin et al (2011)9 70/209 (32. 9%) femur fractures from physical abuse;

139/209 femur fractures from accidental trauma Beals et al (1983)21 80 femoral fractures:

12 (15%) – abuse 12 – suspected of abuse 39 – not abuse 17 – excluded Capra et al (2013)2 24/203 (11.82%) – CPS involved: 3/24 (12.5%)– child abuse 23/203 – SCAN without CPS Dalton et al (1990)22 36/138 (26.08%) abuse cases

Gross et al (1983)23 34/74 (45.9%) abuse and suspect abuse

18/26 (69.23%) abused children under 1yr Hui et al (2008)3 14/127 (11.02%) abuse with NAT

10/60 (16.67%) abuse with NAT in patients with < 12 months Leventhal et al (1993)24 253 fractures in 215 patients, 24.2% - abuse

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14 Murphy et al (2015)4 20/95 (21.05%) patients suffer from physical abuse

Pandya et al (2009)11 SCAN database: General database:

73/500 (14.6%) – child abuse 140/985 (1.42%) – child abuse Rewers et al (2005)25 49/1139 (4.30%) – child abuse

Rex et al (2000)26 14/33 (42.42%) – child abuse

Scherl et al (2000)12 13/214 (6.07%) – child abuse

Shrader et al (2011)6 31.3% suspected NAT

Thomas et al (1991)5 25/215 (11.62%) femur fractures; 9/25 (36%) – abused children with

femoral fracture

Trokel et al (2006)27 178/2253 (7.9%) – abused cases

Wellington et al (1987)28

In 150 fractures:

22% abuse in children <5yr 39% abuse in <1yr.

4.2 Age

The majority of the selected studies were consistent with the fact that children under 1 year old with a femur fracture were suffering from child abuse. Table 4 presents an overview of the percentage of the age of abused children.

Table 4: Overview of age as a risk factor

Study Authors Age

Anderson et al (1982)20 79% of children less then 2yr had femoral fractures due to physical

abuse.

Baldwin et al (2011)9 90% of the abused children as <18 months

Beals et al (1983)21 9.6 months of mean

Capra et al (2013)2 Majority was between 18 and 23 months

Dalton et al (1990)22 46.5% <11 months; 33% 2-3yr

Gross et al (1983)23 65% <1yr; 40% 3-4yr

Hui et al (2008)3 12 months mean

Leventhal et al (1993)24 60% <1yr; 20% older than 1yr

Murphy et al (2015)4 21.5 months of mean

Pandya et al (2009)11 17.5% < 18 months;

Rewers et al (2005)25 14.5% <3yr

Rex et al (2000)26 92.85% <1yr

Scherl et al (2000)12 0.89yr of mean

Shrader et al (2011)6 90% of <1yr were referred to CPS

Thomas et al (1991)5 66.67%<11 months

Trokel et al (2006)27 39% < 1yr Children’s Hospital; 13%<1yr general’s hospital

Wellington et al (1987)28

5.6%<1yr confirmed, 33.3% <1yr suspect abuse;

4.3 Gender

Regarding the gender of the subjects in the selected studies, there was information that was missing or not fully described. Table 5 presents a summary of each study regarding this characteristic.

Table 5: Overview of gender as a risk factor

Study Authors Gender

Anderson et al (1982)20 45.83% males abused

Baldwin et al (2011)9 51.4% males abused

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15 Capra et al (2013)2 57.14% males abused

Dalton et al (1990)22 65% males

Gross et al (1983)23 No data

Hui et al (2008)3 50% males abused

Leventhal et al (1993)24 10.65% males unknown cause; 59.83% males accidental

Murphy et al (2015)4 68.42% males

Pandya et al (2009)11 60% males SCAN database; 55% males general database

Rewers et al (2005)25 62.5% males abused (0-3yr)

Rex et al (2000)26 No data

Scherl et al (2000)12 57.47% males

Shrader et al (2011)6 73% males

Thomas et al (1991)5 No data

Trokel et al (2006)27 57% males General Hospitals; 63% males Children’s hospital

Wellington et al (1987)28

60.6% males

4.4 Mechanism of injury

The mechanism of injury in a child, investigated by the authors, that results in a femur fracture can be highly variable. Some papers don’t individualize that information, rather than describing the types of mechanism presented in the studied population in general. Table 6 has information about the percentage of mechanisms of injury regarding physical abuse. When the paper doesn’t present specific data about this characteristic, “no specific data” will be mentioned and, in case the authors did not took into account this characteristic “no data” will be mentioned in the table.

Table 6: Overview of mechanism of injury as a risk factor

Study Authors Mechanism of Injury Anderson et al (1982)20 No specific data

Baldwin et al (2011)9 No specific data

Beals et al (1983)21 62.5% unknown; 16.67% diaper change; 16.67% jumped or fell from

height; 1.17%– fell while walking/running Capra et al (2013)2 No specific data

Dalton et al (1990)22 10.14% abuse and 60.14% unknown

Gross et al (1983)23 No specific data

Hui et al (2008)3 7.14% fall from tripping; 21.43% fall from low height; 14.29%

unwitnessed; 35.71% inconsistent; 21.49% unknown

Leventhal et al (1993)24 26.92% falls; 51.92% abnormality; 3.84% hit by an object; 11.54% hit

by person; 5.77% playing Murphy et al (2015)4 No data

Pandya et al (2009)11 No data

Rewers et al (2005)25 14.45% NAT; 3.31% - other

Rex et al (2000)26 No data

Scherl et al (2000)12 33.3% falls; 41.67% no history; 8.3% hit by object; 8.3% hit accidently

by person; 8.3% unknown

Shrader et al (2011)6 51% - falls; 7% - twisting while playing; 2% - trampoline; 40% -

unknown Thomas et al (1991)5 No specific data

Trokel et al (2006)27 13% abuse in General Hospitals; 29% abuse in Children’s Hospitals

Wellington et al (1987)28

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4.5 Location of the Fracture

According to the selected papers, some authors provided information regarding the location of the fracture on the femur. That information is gathered in Table 7. When there is no specific data about the fractures that resulted from physical abuse “no specific data” will be present and, in case the authors did not took into account this characteristic “no data” will be mentioned in the table.

Table 7: Overview of location of the fracture as a risk factors

Study Authors Location of the fracture Anderson et al (1982)20 No specific data

Baldwin et al (2011)9 20% proximal (sub-trochanteric region); 45.7% diaphyseal (shaft

region); 37.1% distal (distal metaphyseal region)

Beals et al (1983)21 41.67% midshaft; 20.83% proximal diaphysis; 20.83% distal

metaphysis; 16.67% distal metaphyseal chip; Capra et al (2013)2 94% diaphyseal

Dalton et al (1990)22 No data

Gross et al (1983)23 No data

Hui et al (2008)3 7.14% sub-trochanteric; 78.57% midshaft; 14.29% distal metaphyseal

Leventhal et al (1993)24 No specific data

Murphy et al (2015)4 NAT less on anteroposterior ration but more at the lateral ratio when

comparing to accidental and CPS determining accidental; Pandya et al (2009)11 No data

Rewers et al (2005)25 58.3% shaft; 18.7% distal; 16.7% shaft+distal; 6.3% shaft+proximal

Rex et al (2000)26 3.03% distal epiphyseal; 3.03% proximal metaphyseal; 6.06% distal

metaphyseal; 21.21% proximal third diaphyseal; 48.48% middle third diaphyseal; 18.18% distal third diaphyseal

Scherl et al (2000)12 No specific data

Shrader et al (2011)6 All shaft

Thomas et al (1991)5 64.28% diaphyseal spiral or oblique; 21.43% diaphyseal simple; 7.14%

greenstick; 7.14% metaphyseal Salter Harris I; (no separated data available) Trokel et al (2006)27 No data Wellington et al (1987)28 No specific data

4.6 Type of Fracture

According to the selected papers, some authors provided information regarding the type of the femur fracture. That information is gathered in Table 8. When there is no specific data about the type of fractures that resulted from physical abuse “no specific data” will be present and, in case the authors did not took into account this characteristic “no data” will be mentioned in the table.

Table 8: Overview of the type of fracture as a risk factor

Study Authors Type of fracture

Anderson et al (1982)20 29.17% transverse; 20.83% oblique; 16.67% spiral; 16.67%

metaphyseal; 8.3% bucket; 4.17% femoral neck Baldwin et al (2011)9 No data

Beals et al (1983)21 No data

Capra et al (2013)2 28.57% oblique; 14.29% spiral; 57.14% inconclusive

Dalton et al (1990)22 No specific data

Gross et al (1983)23 No data

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17 Leventhal et al (1993)24 11.1% spiral; 88.8% no data

Murphy et al (2015)4 Transverse more common than spiral

Pandya et al (2009)11 No data

Rewers et al (2005)25 No data

Rex et al (2000)26 14.28% oblique; 57.14% spiral; 7.14% greenstick; 21.42% simple

Scherl et al (2000)12 36% transverse; 36% spiral; 7% oblique; 21% unknown

Shrader et al (2011)6 60% spiral; 28% oblique; 7% transverse; 5% comminuted (suspicious of

NAT)

Thomas et al (1991)5 64.28% diaphyseal spiral or oblique; 21.43% diaphyseal simple; 7.14%

greenstick; 7.14% metaphyseal Salter Harris I; (no separated data available)

Trokel et al (2006)27 No data

Wellington et al (1987)28

No data

4.7 Time Before Going to the Hospital

According to the selected papers, some authors provided information regarding the between the time when the injury occurred and the time it took to get to the hospital. That information is gathered in Table 9. When there is no specific data about this characteristic in abuse cases “no specific data” will be present and, in case the c did not took into account this time period “no data” will be mentioned in the table.

Table 9: Overall view of time between injury and ED, as a risk factor

Study Authors Time before going to the Hospital Anderson et al (1982)20 No data Baldwin et al (2011)9 No data Beals et al (1983)21 No data Capra et al (2013)2 85.71% <5h; 14.28% >12h Dalton et al (1990)22 No data Gross et al (1983)23 No data Hui et al (2008)3 No data Leventhal et al (1993)24 No data Murphy et al (2015)4 No data Pandya et al (2009)11 No data Rewers et al (2005)25 No data Rex et al (2000)26 No data Scherl et al (2000)12 No data Shrader et al (2011)6 No data Thomas et al (1991)5 No data Trokel et al (2006)27 No data Wellington et al (1987)28 No data

4.8 Socio-Economic Status

According to the selected papers, some authors provided information regarding the socio-economic status of the family of the abuse child. That information is gathered in Table 10Table 9. When there is no specific data about this characteristic in abuse cases “no specific data” will be present and, in case the authors did not took into account this characteristic “no data” will be mentioned in the table.

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Table 10: Overall view of socio-economic status as a risk factor

Study Authors Socio-Economic Status Anderson et al (1982)20 No data

Baldwin et al (2011)9 7.1% abused children had no insurance

Beals et al (1983)21 No data

Capra et al (2013)2 No data

Dalton et al (1990)22 No data

Gross et al (1983)23 No data

Hui et al (2008)3 No data

Leventhal et al (1993)24 28.86% government insurance; 19.44% private insurance; 14.29%

self-paid; 24.78% clinical care Murphy et al (2015)4 No data

Pandya et al (2009)11 No data

Rewers et al (2005)25 Lower incomes and single motherhood have a higher risk

Rex et al (2000)26 No data

Scherl et al (2000)12 No data

Shrader et al (2011)6 65% government insurance; 23% commercial plans; 12% no insurance

Thomas et al (1991)5 No data

Trokel et al (2006)27 63% government insurance (General’s Hospital); 49% government

insurance (Children’s Hospital) Wellington et al (1987)28 No data

4.9 Additional Injuries

According to the selected papers, some authors provided information regarding the physical condition of the abused child, i.e. if there are any prior injuries, polytrauma, skeletal survey required, and radiographic studies were examined while the child was in the ED. That information is gathered in Table 11Table 9. When there is no specific data about this characteristic in abuse cases “no specific data” will be present and, in case the authors did not took into account this characteristic “no data” will be mentioned in the table.

Table 11: Overall view of additional injuries status as a risk factor

Study Authors Additional injuries Anderson et al (1982)20 No specific data

Baldwin et al (2011)9 62.3% prior injuries; 52.8% polytrauma

Beals et al (1983)21 5% brain damage; 34.61% others

Capra et al (2013)2 28.51% prior femur fracture

Dalton et al (1990)22 7.14% positive skeletal survey; 42.86% negative for skeletal survey;

8.93% positive for skull roentgenographic; 41.07% negative skull roentgenographic

Gross et al (1983)23 No data

Hui et al (2008)3 42.86% had several other fractures, multiple bruises, bilateral

subdural, haemorrhages; 15.38% positive skeletal survey; 86.61% negative skeletal survey

Leventhal et al (1993)24 31% positive skeletal survey

Murphy et al (2015)4 No data

Pandya et al (2009)11 No specific data

Rewers et al (2005)25 1.32% head injury; 0.53% polytrauma; 0.26% thorax injury; 0.09%

abdominal trauma (death with associated trauma) Rex et al (2000)26 No specific data

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19 Scherl et al (2000)12 13.3% abrasions; 20% head; 6. 7% hip; 6.7% humerus; 26.7% internal

injuries; 13.3% mandible injuries; 6.7% pelvic; 6.7% skull Shrader et al (2011)6 21% associated injuries

Thomas et al (1991)5 No data

Trokel et al (2006)27 No specific data

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20

5. Discussion

Incidence

Child abuse, being a clinical diagnosis, is a subjective evaluation depending on the physicians that are present when the child enters the ED. Even the small percentage of children that are diagnose with physical abuse with a femur fracture, may have a big impact on the incidence of this crime. It is important to keep in mind the proportion of children that walk in the emergency department every day. This heterogenicity may be linked to the different populations involving the selected studies and how the authors present their findings. Nevertheless, , the percentage of children that suffered from child abuse is between 6.07%12 and 45.9%23 (Table 3)

Age

Throughout the studies, there was a coherent finding: children younger than one year of age have a higher risk of being victims of child abuse. Although it is clear that these cases of children with femur fractures are automatically considered as physical abuse, according to Beals et al (1983)21 there are two

peaks of higher risk. In their study, they provided information that those two peaks are between zero and six months and between thirty-one and thirty-six months, i.e. between two years and seven months and three years. Dalton et al (1990)22 supported those findings by stating that 33% of abused children

with a femoral fracture in their study were also between two and three years of age. This second peak was also shown by Gross et al (1983)23 where 40% of the abuse children were between three and four

years old. (Table 4)

Gender

In most of the selected studies, there was a difference between gender where is shown that male infants are more prone to be victims of physical abuse.,2-4,9,11,12,20,22,24,25,27,28 However, the difference between

gender in each study is not significant except for two studies. Beals et al (1983)21 and Shrader et al

(2011)6 indicate there was a high percentage of males in their study, 74% and 73% respectively. Three

other studies didn’t provide information about the percentage of males and females in the population.5,23,26 Considering there is not a clear indication that there is a statistically significant

difference between gender when it comes to child abuse involving femoral fractures, this feature may not be used as a risk factor for these specific cases.(Table 5)

Mechanism of injury

There are several ways for a child to injury and produce a femoral fracture. In accidental fractures, most studies stated that the most common cause on accidental fractures are due to falls3,12,20,21,24,25,27 and motor

vehicle accident22,23. While in nonaccidental trauma the outcomes of the selected studies diverse

between inconsistent history 3,24 and unknown history of how the child was injured6,12,21,23. The

remaining studies did not provide data regarding this characteristic2,4,11,28 or just mention the percentage

of abused children20,22,25,27

According to Shrader et al (2011)6 there is a high risk of abuse due to fall (51%) as well as if the

caregivers didn’t have a clear explanation about the mechanism of injury. There is still controversy if the height of the fall can be an indication of abuse or not. In Leventhal et al (1993)24 study, the authors

were able to conclude that falls from lower height may produce fractures. Moreover, Capra et al (2013)2

studied different heights and concluded that 57.5% of the falls occurred with less than 0.61 meters, 22.5% occurred between 0.61 meters and 1.22 meters and 20% of the falls occurred within more than

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21 1.22 meters. Even though this study doesn’t differentiate the falls from accidental and abused, these data can provide information that there is a possibility that an infant can have fractures from falling from a low height. Nevertheless, Helfer et al (2000)29 studied 246 children younger than 5 years old

and only 3% present a fracture falling form a low height in which none was a femoral fracture. This information may lead to think that a history of small falls from the caregiver may be classified as a suspicious case and assess it with caution. (Table 6)

Location of the Fracture

There are several strong muscles associated with sub-trochanteric portion of the femur which makes the mechanism of the fracture lightly different from the mechanism of a shaft or intertrochanteric region. This is a very challenging part of the bone to manage and only 4-7% of the fractures occur in this region. It’s mostly correlated to the ambulatory forces that the weight of the body exerts, such as compression, tension and torsional forces. In non-ambulatory children this type of fracture is automatically considered as an indicator of physical abuse, especially because it takes a torsion and bending movement to break the bone in this area of the femur.30

The shaft of the femur is usually associated with high energy trauma, such as motor vehicle accidents or a fall from a high height. In younger children the femur contains of much more soft bone than in adults, which doesn’t need as much force to be fractured. In these children, these fractures may occur by an indirect impact essentially, by torsion.31

The abovementioned data may be a reason why there is a higher percentage of diaphyseal fractures and lower on the sub-trochanteric region, in the selected studies.2,3,5,6,9,25,26 The remaining studies didn’t

provide specific data on the abused cases or no data regarding this feature.11,12,20,22,23,27,28 Distal fractures

were also present in abuse cases but never with the highest or lowest incidence.3,5,9,21,25,26

It is also noticeable that in Beals et al (1983)21, Hui et al (2008)3 and Rex et al (2000)26there was no

significant difference in the location of the fracture in both abuse and control group. This can indicate that the location of the fracture may not be a risk factor by itself. Nonetheless, is important to know where the injury took place, so it can be correlated with the mechanism and ascertain if there is a relation between the fracture and how it was inflected, to determine if it is a case of NAT. (Table 7)

Type of Fracture

Eight studies didn’t provide information about the type of fractures or weren’t specific about the type of fracture in the abuse cases.9,11,21–23,25,27,28 There was a clear difference in incident of spiral fractures

in Thomas et al (1991)5 and Shrader et al (2011)6 studies, 64.28% and 60%, respectively, followed by

oblique fractures. In the remaining studies there was no statically significant difference between the type of injury.2,12,20,26

Dalton et al (1990)22 didn’t provide specific numbers about this characteristics but they showed that

spiral fractures are often associated with physical abuse in non-ambulatory children. Such data were also stated by Hui et al (2008)3 while referring to historically wise, but the true findings of this study

were that transverse fractures can also be an indicator of NAT. According to Murphy et al (2015)4, not

only the authors support that the transverse pattern was more common than the spiral, but also they discredit the association of NAT with spiral fractures as well, stating that there was no prove to support that fact. (Table 8)

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22 Given the contradictory outcomes from these studies, this feature cannot be used individually to diagnose NAT but may be an indicator of child abuse if it’s coherent with the explanation that the caregivers provide to justify the injury.

Time Before Going to the Hospital

Regarding the time that the caregivers took to take the child to the hospital after the injury was considered in only one study. According to Capra et al (2013)2 85.71% of the children that were

physically abused took less than five hours to be examined by professionals in the ED, and 14.28% took more than twelve hours. The lack of data about this characteristic maybe be due to the fact the family may live far away from the hospital which can lead to a long period of travel for medical care; this information may not be recorded in the emergency department of the hospital or, in case it is, the health professionals need to accept the information provided by the caregivers, since the child may not be able to express herself/himself given the early age. It is also important to keep in mind that dating a fracture is a complex process which is not always possible to do accurately, especially within a small hour’s range. This characteristic should not be taken into account for this systematic review due to the lack of data.32 (Table 9)

Socio-economic Status

The socio-economic status was a risk factor considered by few authors.6,9,24,25,27According to those

studies, children raised in a “low income family” are more prone to be abused. Rewers et al (2005)25

concluded this based on the annual income of the family, and when children were raised in a single motherhood environment, who had a higher risk of being physical abused. There is no exact way of knowing the reason behind these findings, but there are authors that relate this incidence to financial problems and the stress associated with it.33 The remaining studies addressed this risk factor by assessing to the type of insurance that is provided to the

family and, the majority of the data indicates that, in USA, there is a higher incidence on governmental insurances, such as Medicaid, which gives a estimation of the income of the family.6,24,27According to Trokel et al (2006)27,

there is also a difference between type of hospitals. The authors stated that there is a higher percentage of governmental health insurance users in General Hospitals and private health insurance in Children’s Hospitals. There is a possible connection between child abuse and socio-economic status, but further analysis is needed to confirm this directly with femoral fractures in these specific cases. (Table 10)

Additional Injuries

This characteristic varies a lot between, skeletal surveys, prior injuries and polytrauma as in associated injuries. Hui et al (2008)3 provided information about the difference in NAT and accidental injury

groups with additional injuries, 43% and 12%, respectively, concluding that this characteristic can be a risk factor of NAT. Scherl et al (2000)12 and Shrader et al (2011)6 also demonstrated that abused

children may have a different type of injuries associated with femur fracture.

Despite Leventhal et al (1993)24 did not provide specific information about the associated injuries, they

concluded that a skeletal survey may be an essential tool to help clarify doubts regarding the diagnosis, where the 31% of positive survey were performed, and eleven cases out of those 31%,the skeletal survey helped detecting an additional fracture.

In the Netherlands the current protocol states that a skeletal survey should be perform in children younger than two years old or in children between two and four years of age, if the physician determines that is necessary to reach a conclusion. After four years old, this tool is no longer informative. 34

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23 Eight studies didn’t provided data about this features, or didn’t have specific data for the abuse group.4,5,11,20,23,26–28(Table 11)

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24

6. Limitations

The selected studies vary between one and two stars, which indicates that is a very low and low rating, respectively. There was also a variety of study designs within the same topic which didn’t help extracting the data in a methodical manner. Most of the studies provided information regarding the overall population as well as individual information about the abuse group, but a few studies didn’t distinguish the data, which made the data extraction more complex.

There was not much diversity on the countries that the studies took place. The majority took place in the USA, but this doesn’t mean that can be applied to the reality of the Netherlands. Not only the risk factors can be different but, the ones that are the same may have a different impact over the population.

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25

7. Conclusions and Recommendations

Child abuse is less complex to confirm in children younger than one year old given the fact that it is not easy to fracture a femur when the individual is a non-ambulatory child. In contrast, infants between two and three years old, are starting to walk and experience falls from climbing, stepping or even running. This can be a factor that may contribute to the misdiagnose of child abuse in children within this age range.

Gender may be discarded as a risk factor due to the similar percentages that the selected studies showed regarding this feature. There is no significant statistically difference that shows that there is more incidence on one gender than the other.

None of the features described previously may be used individually to compare a non-accidental trauma from an accidental injury. Ideally, should be a group of factors that can lead to that: mechanism of injury, type and location of the fracture and additional injuries need to be related and coherent so that a child can be most accurately included or excluded has a victim of physical abuse.

There might be a correlation between NAT and socio-economic status but further analysis on this feature needs to be done before stating that it can be a feature to keep in mind when assessing a child in the ED.

The interval of time taken between the injury and the arrival at emergency department, given the selected studies, cannot be an indicator or child abuse.

The skeletal survey and radiography may be a helpful tool to diagnose NAT and it should be used whenever necessary.

It would be advised to perform studies regarding toddles between 1-5 years old, since most of studies previously mentioned could only conclude that it is automatically considered as non-accidental trauma when the admitted infant is younger than one year old, consequently, designate as child abuse.

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26

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Fractures. J Pediatr Orthop. 2007;27(7):751-753.

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Capra L, Levin A V, Howard A, Shouldice M. Characteristics of femur fractures in

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Three Years. J Pediatr Orthop. 2008;28(3):297-302.

doi:10.1097/BPO.0b013e3181653bf9.

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Murphy R, Kelly DM, Moisan A, et al. Transverse Fractures of the Femoral Shaft Are

a Better Predictor of Nonaccidental Trauma in Young Children Than Spiral Fractures

Are. J Bone Jt Surg. 2015;97(2):106-111. doi:10.2106/JBJS.N.00664.

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Thomas S, Rosenfield N, Leventhal J, Markowitz R. Long-Bone Fractures in Young

Children : Distinguishing Accidental Injuries From Child Abuse. 1991;88(3):471-476.

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Shrader MW, Bernat NM, Segal LS. Suspected Nonaccidental Trauma and Femoral

Shaft Fractures in Children. Orthopedics. 2011:1-10.

doi:10.3928/01477447-20110317-06.

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Taitz J, Moran K, Meara MO. Long bone fractures in children under 3 years of age : Is

abuse being missed in Emergency Department presentations ? 2004;16(August

2003):170-174.

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age 1 year: importance of collaboration with child protection services. J Pediatr

Orthop. 2002;22(6):740-744. doi:10.1097/00004694-200211000-00009.

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Baldwin K, Pandya NK, Wolfgruber H, Drummond DS, Hosalkar HS. Femur fractures

in the pediatric population: Abuse or accidental trauma? Clin Orthop Relat Res.

2011;469(3):798-804. doi:10.1007/s11999-010-1339-z.

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Sittig JS, Post EDM, Russel IMB, Van Dijk IAG, Nieuwenhuis EES, Van De Putte

EM. Evaluation of suspected child abuse at the ED; Implementation of American

Academy of Pediatrics guidelines in the Netherlands. Am J Emerg Med.

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Pandya NK, Baldwin K, Wolfgruber H, Christian CW, Drummond DS, Hosalkar HS.

Child abuse and orthopaedic injury patterns: analysis at a level I pediatric trauma

center. J Pediatr Orthop. 2009;29(6):618-625. doi:10.1097/BPO.0b013e3181b2b3ee.

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Scherl S a, Miller L, Lively N, Russinoff S, Sullivan CM, Tornetta P. Accidental and

nonaccidental femur fractures in children. Clin Orthop Relat Res.

2000;83(376):96-105.

13.

Flaherty EG, Perez-Rossello JM, Levine MA, Hennrikus WL. Evaluating Children

With Fractures for Child Physical Abuse. Pediatrics. 2014;133(2):e477-e489.

doi:10.1542/peds.2013-3793.

14.

Lippincott. Professional Guide To Pathophysiology. 3rd ed. (Eckman M, ed.). Wolters

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Bullock DP, Koval KJ, Moen KY, Carney BT, Spratt KF. Hospitalized cases of child

abuse in America: who, what, when, and where. J Pediatr Orthop.

2009;29(3):231-237. doi:10.1097/BPO.0b013e31819aad44.

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LC B, RT L, RN H. Role of intentional abuse in children 1 to 5 years old with isolated

femoral shaft fractures. J Pediatr Orthop. 1996;16(5):585-588.

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Guyatt GH, Oxman AD, Vist G, et al. GRADE guidelines: 4. Rating the quality of

evidence - Study limitations (risk of bias). J Clin Epidemiol. 2011;64(4):407-415.

doi:10.1016/j.jclinepi.2010.07.017.

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Maguire S, Moynihan S, Mann M, Potokar T, Kemp AM. A systematic review of the

features that indicate intentional scalds in children. Burns. 2008;34(8):1072-1081.

doi:10.1016/j.burns.2008.02.011.

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Guyatt G, Oxman AD, Akl EA, et al. GRADE guidelines: 1. Introduction - GRADE

evidence profiles and summary of findings tables. J Clin Epidemiol.

2011;64(4):383-394. doi:10.1016/j.jclinepi.2010.04.026.

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Anderson W a. The significance of femoral fractures in children. Ann Emerg Med.

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Beals RK, Tufts E. Fractured femur in infancy: the role of child abuse. J Pediatr

Orthop. 1983;3(5):583-586. doi:10.1097/01241398-198311000-00004.

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Dalton HJ, Slovis T, Helfer RE, Comstock J, Riolo S, Scheurer S. Undiagnosed abuse

in children younger than 3 years with femoral fracture.pdf. 1990;144:875-878.

23.

Gross RH, Stranger M. Causative factors responsible for femoral fractures in infants

and young children. J Pediatr Orthop. 1983;3(3):341-343.

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nsible_for_femoral.12.aspx%5Cnhttp://www.ncbi.nlm.nih.gov/pubmed/6874931.

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Leventhal JM, Thomas SA, Rosenfield NS, Markowitz RI. Fractures in young

children. Distinguishing child abuse from unintentional injuries 1993.pdf. Am J Dis

Child. 1993;174:87-92.

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Rewers A, Hedegaard H, Lezotte D, et al. Childhood Femur Fractures, Associated

Injuries, and Sociodemographic Risk Factors: A Population-Based Study. Pediatrics.

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Rex C, Kay PR. Features of femoral fractures in nonaccidental injury. J Pediatr

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Trokel M. Variation in the Diagnosis of Child Abuse in Severely Injured Infants.

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Wellington P, Bennet GC. Fractures of the femur in childhood. Injury.

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Helfer RE, Slovis T, Black M. Injuries Resulting from Small Children Falling Out of

Bed. Paedriatics. 2009;1(4):583-535.

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+shaft+cause&hl=pt-PT&sa=X&ved=0ahUKEwjX1dfntuLXAhWPC-wKHdxjCOEQ6AEIQDAD#v=onepage&q=femur fracture shaft cause&f=false.

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29

9. Appendix

9.1 Appendix I – Project Plan

Title

Risk factors for child abuse in young children and toddlers with a femur fracture; protocol for a systematic review.

Registration:

Our systematic review was registered with the International Prospective Register of Systematic Reviews (PROSPERO) on 26-3-2017 and was last updated on --- 2017 (registration number CRD42017059541).

Authors:

Corresponding author: Marie-Louise Loos1

m.h.loos@amc.uva.nl

Author affiliations:

1. Paediatric Surgical Centre, Emma Children's hospital AMC & VU Medical Centre (H7-282) Meibergdreef 9, 1105 AZ Amsterdam, The Netherlands.

University of Amsterdam. 2. Department of Radiology, AMC,

Meibergdreef 9, 1105 AZ Amsterdam, The Netherlands. University of Amsterdam.

3. Clinical Librarian, AMC

Meibergdreef 9, 1105 AZ Amsterdam, The Netherlands. University of Amsterdam.

1 Bakx R, r.bakx@amc.uva.nl

2 Rijn van, RR, r.r.vanrijn@amc.uva.nl 3Etten van, FS, f.s.vanetten@amc.uva.nl

Contributions:

RRR is the guarantor. ML drafted the manuscript and selected with student the articles. ML/student and FSE developed the search strategy. ML and student performed the risk of bias assessment. Provides statistical expertise. RB & RRR, provided critical revision of the article. RB & RRR provided final approval of the article.

Amendments:

When this protocol needs to be amend, the date and a fine description of each amendment will be given. The amendments will be added in the appendix.

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30 Support

This systematic review is funded by the “Louise Vehmeijer” foundation, Amsterdam, Netherlands (funding reference number 2016-007V). The funder is not involved in any aspect of the project, such as the design of the project’s protocol, collection of data and analysis. The funder will have no input on the interpretation or publication of the study results.

Introduction

Up to 1% of children attending the emergency department is due to non-accidental physical injury1, based upon the data of 66 studies. Fractures and skin lesions are common injuries in children, due to accidental and non-accidental causes. Several characteristics of non-accidental fractures have been described, such as young age and type and location of fracture (femur fracture, skull fracture, humeral fracture etc)2,3,4.

Children with abusive fractures are missed in 21% at initial presentation5. Worlock et al. reviewed that

85% of accidental fractures occur in children older than five years, in contrast to 80% of non-accidental fractures of which occur in children younger than 18 months6. Mean age for non-accidental fractures

was 11.7 months compared to 22.1 months in accidental fractures7. Though, overall literature describes

30% - 46% of non-accidental femur fractures in children <5 years, up to 93% in children <1 year8.

Several studies show that in abuse cases a femur fracture is the most common lower extremity fracture in young children9,10,11.

Non-accidental injury has serious health implications, it is associated with (a longer) hospital admission and increased mortality rate. Moreover, abused children have an increased risk of suffering from additional injuries like (other) fractures and burns, due to the abuse12,13. Both short and long-term

consequences can be far-reaching for the physical and mental health of children and later on in life14.

However, few articles address specifically femur fractures in children, describing several diversities of characteristics. A clear overview of the literature concerning femoral fractures in (young) children is lacking. The aim of this systematic review is to present an overview of the literature of risk factors, characteristics and incidence of non-accidental femur fractures in children.

Objectives

The aim of this review is to systematically review the literature for qualitative evidence that explores the incidence and risk factors of child abuse in children with a femur fracture.

Outcomes

The primary outcome is

1) Risk factors of (physical) child abuse in children with a femur fracture. Secondary outcomes include:

2) Incidence of child abuse in children with a femur fracture.

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31 4) Mortality due to child abuse in children with a femur fracture.

5) Hospital admission due to child abuse in children with a femur fracture.

6) Risk factors and incidence of child abuse related femur fractures on other continents. 7) Socio-demographics of parents with a child with a femur fracture due to child abuse.

9.2 Appendix II – Search Strategy

This systematic review is drafted following the PRISMA guidelines.

Eligibility criteria

Studies will be selected according to the following drafted pre-specifications.

Study design

We will include randomized controlled trials (RCTs), including cluster RCTs, controlled (non-randomized) clinical trials or cluster trials, prospective and retrospective comparative cohort studies, case-control, case series and cross-sectional studies. We will exclude case reports. Based on a previous review concerning this subject the expectation is that there will be very few or none RCTs.

We will not use any limitation concerning the number of included participants. Unpublished data will be excluded.

Language: English, Dutch, German and French articles will be included.

All data published until December 31st, 2016 will be included. Studies of non-abusive data only and/or

relating to management and treatment will be excluded.

The studies will be selected by two independent reviewers and criticized by ‘general critical appraisal guides’ and ‘designed-based quality checklists’, with this also heterogeneity will be explored and concretized whether the found articles will be suitable fora n overall conclusion. The Consort 2010 checklist will be used.

Excluded studies will be reported separately, with motivation for exclusion.

Participants

We will include studies examining young children (0-5 yr) with a femur fracture. No restriction for gender will be applied. We will include studies addressing children with other traumatic injury if data provided for a femur fracture are reported separately.

Subgroups will be framed while extracting the data. Interventions and comparators

Inapplicable. Outcomes

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32 The primary outcome is

1) Risk factors of (physical) child abuse in children with a femur fracture. Secondary outcomes include:

2) Incidence of child abuse in children with a femur fracture.

3) Clinical and radiological features of femur fracture due to child abuse. 4) Mortality due to child abuse in children with a femur fracture.

5) Hospital admission due to child abuse in children with a femur fracture.

6) Risk factors and incidence of child abuse related femur fractures on other continents. 7) Socio-demographics of parents with a child with a femur fracture due to child abuse.

In regard to our primary outcome, we will use CoreInfo criteria to determine the degree of proven presence of child abuse (table 1)15.

We will use the WHO definition of child maltreatment as child abuse “includes all forms of physical and emotional ill-treatment, sexual abuse, neglect, and exploitation that results in actual or potential harm to the child’s health, development or dignity. Within this broad definition, five subtypes can be distinguished – physical abuse; sexual abuse; neglect and negligent treatment; emotional abuse; and exploitation”. ‘Physical abuse’ and ‘neglect’ are applicable for our primary and secondary outcomes. Table 1

Criteria used to define abuse:

1 Abuse confirmed at case conference or civil or criminal court proceedings or admitted by perpetrator

2 Abuse confirmed by stated criteria including multidisciplinary assessment 3 Abuse defined by stated criteria

4 Abuse stated but no supporting detail given 5 Suspected abuse

We will extract outcomes in all data forms (dichotomous, continuous) as reported in the included studies.

Information sources

The following electronic databases will be searched: PubMed (MEDLINE) (1948 onwards), EMBASE (1980 onwards), Cochrane Library. We will use medical subject headings (MeSH) and text words related to child abuse and femur fractures in title and abstract. The reference lists of included studies will be scanned to identify relevant articles not identified during the search. Authors of included studies will be contacted when relevant information about the study is unclear.

All data published until December 2016 will be included.

An update search towards the end of the review will be performed to identify new released significant articles.

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