sbu evaluate • report 255/2016

Shaken baby syndrome The role at the medical investigation of suspected shaken baby syndrome

A systematic Overview

the State's preparation for the medical and social assessment

Report series This report belongs to the series SBU Assesses (ISSN 1400‐1403). A comprehensive scientific scientific evaluation that includes systematic overview, economic/hälsoekono‐economic analyses and review of social and ethical aspects. Subject matter experts are involved in the work with the support of the employees of the SBU, patients/users. The report is reviewed by independent experts. The degree of scientific reliability in the sam‐manvägda results are assessed on a systematic manner and report's conclusions approved by the SBU's Board.

Declaration of contents ✓ Evaluation of established method

✓ Systematic literature search ✓ Relevansgranskning ✓ Quality review ✓ Aggregate of results ✓ Evidensgradering made by SBU ✓ Evidensgradering made externally Based on a systematic literature review ✓ The consensus process

✓ Designed in collaboration with experts Patients/users involved ✓ Ethical aspects Economic aspects Societal perspective ✓ Approved by the SBU's priorities and quality group ✓ Approved by the SBU's Board

Keywords Shaken baby syndrome, Babies, Child Abuse, Triad, Subdural Haemorrhage, Cerebral Effect, Fundus Bleeding

Published October 2016

Period of validity Results that are based on a strong scientific basis continues usually to be valid for a long time to come. Other results may have had time to become stale. The branchialsmiles of all areas where the scientific basis is insufficient or limited

Order This report (# 255) can be ordered from Strömberg distribution. Phone: 08‐779 85 96 • Fax: 08‐779 96 10 • e‐mail: [email protected]

Production Graphic production of Emma Österman, SBU. Printed by Elanders Sweden AB, Mölnlycke, 2016. Cover Photo: Shutterstock

Reference number UTV2014/254

Cite this report SBU. Shaken baby syndrome – The role at the medical investigation of suspected shaken baby syndrome. Stockholm: the State's preparation for the medical and social assessment (SBU); 2016. SBU rapport nr. 255. ISBN 978‐91‐85413‐98‐0. Content

Summary of results 5

1 Objective 7

2 Background 9 What is shaken baby syndrome? 9 Symptoms and findings 10 In health care 10 Terminology 11 Investigation of the damage that can be caused by shaken baby syndrome 11 Other possible causes for the and its sub‐components 12 Triad – symptoms and findings 12 — Subdural 12 — Fundus bleeding 12 — Brain influence 13 Diagnostic methods 13 — Intracranial study 13 — Fundoscopy 14

3 Methodology for the systematic evaluation 15 Question according to PIRO 15 Inclusion and exclusion criteria 16 — Inclusion criteria 16 — Exclusion criteria 16 Methodology for selection of studies 17 Relevansgranskning 17 Quality review 17 Method for aggregate of results 18 The scientific base strength 18

4 Results 19 Flowchart of literature review 20 Evidensgraderade results 20

5 Discussion 25 Methodological issues 26 — Definition of the shaken baby syndrome 27 — Group Division of studies 27 — Circular reasoning in clinic and research 27 — Diagnostic methods 28 Comparisons with the results from other surveys 29 6 Consideration for research 31 What is needed in order to fill the gaps? 32 — International coordination 32 — Important areas of research 32 7 Project team, external reviewers, advice and Board 35 Project group 35 — Special 35 — SBU 36 External auditor 36 Bindings and conflict of interest 37 SBU's scientific advice – the Brage 37 SBU's scientific advice — Eira 37 SBU: s Board 38

8 External collaboration 39 Usually collaboration 39 Network authorities 39 The State's medical‐ethical advice (Smer) 39

9 41 glossary

10 Reference 43

Annex 1 Other possible causes for the subcomponents 51

Annex 2 Biomekanikstudier 55

Annex 3 -ethical analysis of shaken baby syndrome (Smer) 57

Annex 4 search strategies/Appendix 4 available at www.sbu.se/255 Search strategies

Annex 5 Granskningsmallar/Appendix 5 available at www.sbu.se/255 Evaluation Protocols

Annex 6 Exclusive studies and studies available at www.sbu.se/255 with low quality/Appendix 6 Excluded studies and studies with high risk of bias

Summary of the results

The systematic litteraturöversikten shows the following evidensgraderade results:

• (D) et there is limited scientific evidence for the triad 1 and thus its some components may be present at the skakvage.

• (D) et there is insufficient scientific backing for that assess the diagnosis thematic accuracy of the triad 1 in order to identify the skakvage.

Limited scientific basis (b) ygger in studies with high or medium-high quality with greatly debilitating factors at an overall assessment. Det is important to clarify that even limited scientific support for a method adds reliability or effectiveness does not mean absence of vetenskapligt support.

Osufficient scientific basis exists when studies are lacking, the available studies are of low quality or where studies of similar quality shows contradictory results.

(E) vidensvär tion bygger not on a formal evidensgradering with GRADE but at a total worthevaluation of the vetenskapliga base.

1 T re components in a whole, usually consisting of subdural haemorrhage, fundus hemorrhage and hjärnpåvyoucan.

1 Order

When suspected cases of shaken baby syndrome have the combination of subdural haemorrhage (bleeding under the hard brain membrane), fundus bleeding and various Forms of encephalopathy (encephalopathy), with symptoms such as the example with vetandesänkning, convulsions and hypoxic apnoea, along come to known as the triad. Ucrossthat evaluation aims to to answer with any certainty the triad or its subcomponents can be explained by the shaken baby syndrome of children under 1 year of age.

2 Background

M isshandel of children described in the medical literature as early as the 1800 's [1] but the knowledge began to get larger spread first far later [2,3]. Barnmisshandel can often be hidden in the family and it is a risk for the elderly is not diagnosed among other things because the child is not able to bring their own actions. Shile are over-diagnosing all consideration for,such as society's interventions and actions can lead to that a family can shatter on improper grounds. Over-diagnosing would be able to be done about health care feel to the triad always caused by shaken baby syndrome and overlook other possible causes.

What is shaken baby syndrome?

S kakvåld this means that a person shakes a child so that the child's head is thrown forward and back. Neuro surgeon Guthkelch , the 1971 forward a hypothesis about that miss trading can give rise to subdural haemorrhage but that it fi any visible external head injuries [4]. In the article described two cases in which parents are admitted to those of different reasons have shaken the child before it went down. One of the children had , moreover, fundus bleeding. The relationship between the skakvage, subdural and fundus bleeding was described by Caffey 1972 and was called then the Whiplash shaken infant syndrome [2]. Skadorna was considered to occur when the child's head by agitation were exposed for acceleration,deceleration and rotational forces. Teorin was questioned in a biomechanical study of Duhaime and Colleagure 1987 [5]. Slutsatsen in this study was that isolated shaking without direct violence, troligen not provide adequate power for that cause these injuries. Benämningen on tuntiltonnthet has later changed to Shook baby syndrome (SBS) and a variety of studies have been published on the relationship between different clinical and radiographic findings and damage caused by shaken baby syndrome [6-13].

SBS is a concept that does not exist in the Swedish language and projects the Group has chosen to use the term shaken baby syndrome for the current type of våldsinverkan and use der the term triad for the current symptoms/findings [13]. Pon later years has even the term AHT (Abusive head trauma) has come to be used in the literature, see Chapter 1 health care.

Symptoms and findings

In the scientific literature , there are various symptoms and findings are registered as explained by shaken baby syndrome. (F)for the most prevalent (subdural haemorrhage, eye the bottom bleeding and hjärnpåvercan) has the name triad used. Denna ..rt focuses on triad, see section Terminologi. Förexcept triad noted sometimes at shaken baby syndrome bruises on the body and fractures on to sample ribs and tibia (metafysfrakturer) but such damage has not been included in this IslandOverview.

In health care

(D) et is on the basis of clinical symptoms such as cramps, decreased consciousness or other symptoms of brain the influence that the child received into care. Clinical and radiological examination can initially detect finds which subdural or other Forms of hjärnpåvercan. Subduralblödning, fundus bleeding and various Forms of brain influence can get severe consequences with permanent damage to the brain and/or eyes. Syrebrist can give so serious brain damage to the child 's death. Permanent damage may consist of serious cognitive(a) and/or motor disabilities with comprehensive invyoucan on the child's health, utvelopment and future qualityTy.

V years staff are encouraged to be aware of the symptoms/findings that are included in the triad and is obliged to immediately notify the social services If they are in their operations,such knowledge may about or suspect that a child 's father badly under 14 Cape. 1 § Social Service Act. In an often -quoted article by American Academy of Pediatrics, fn 1993 prompted doctors to be careful with that attention to trauma with brain impact in babies and be well familiar with the clinical and radiology studies strategic bargain that can confirm the damage caused by shaken baby syndrome [14]. In the regional treatment program "For suspicion of physical mistreatment by diluting the child", in the Stockholm county administrative County Councils from 2011 stand [15]:

"The combination of the subdural bleeding and brain damage with oedema or bleeding speaks very starkt for that baby has been beaten on a history of trafi leak or fall from high altitude is missing. Seen in addition, bleeding in the eye the bottoms is a diagnosis of abuse from the medical point of view absolutely clear".

Aev a other regional vårdprprogrammes and Socialstyrman writing about children as father ill or at risk to endangered bad provides guidance regarding the care of infants at miss meant abuse [16.17]. U uring recent years have , however, the security with which it is possible to determine If the findings in the triad are really caused by isolated shaken baby syndrome been questioned [18-26]. Msteam articles that debated the topic shaken baby syndrome and finds/symptoms of the triad has been published in international and national journals and media. Against this backdrop comes across it as urgent to the determination of shaken baby syndrome as the cause of the symptoms/findings are based on the best possible vetenskapliga basis. (E) v in the n s grading is based , however, on assessment of groups, not individuals. (F)in order to the judiciary or social services should be able to speak out about the connection between expo exposure and disease/injury required assessments of individual cases where, Althoughthe other observations and conditions weighed in.

Terminology

(D) a English term for the triad 's 'shook baby syndrome (SBS), which aims at the symptoms/findings that may arise after the isolated shaken baby syndrome it would say shaking but to stay supported against any object. American Academy of Pediatrics advocated year 2009 a broader term, Abusive head trauma (AHT) which includes also direct violence against the head [27]. In the literature used ytterlingare a number of denominations that fully or partially overlap with terms Shook baby syndrome and Abusive head trauma, see Chapter 9 Ordförklaringar. (B) (e) t (e) (c) k n in n gar n (a) used in different ways in the scientific literature which has contributed to methodological inaccuracies in studies of the eff by suspected shaken baby syndrome. ProjektgrGroup has chosen to separate pruncheck the item to isolated shaken baby syndrome and therefore only include studies with isolated shaken baby syndrome it would say damage without signs of direct violence against (external damage ) header. Ffor abbreviations and Glossary see Chapter 9 glossary.

Investigation of the damage that can be caused by shaken baby syndrome

For to diagnose suspected brain injuries used datortomografiundersök ning (DT) and/or MRI (magnetic resonance imaging, MRI). Fundus bleeding determined by fundoscopy by ophthalmoscopy or fundus camera.

Other possible causes to the triad and its subcomponents

V id symptoms/findings of the triad must to examine whether these can have other causes than skakvage.

Symtom/bargains such as subdural haemorrhage, fundus hemorrhage and hjärnpå effect has been described after childbirth and at a number of different medical conditions such as various seizure condition, some bleeding disorders, infectious diseases, metabolic diseases, immunological diseases, bone diseases and vascular malformations, see Bannex 1.

Triad – symptoms and findings

Subdural haemorrhage It is well known that violence against your head can give rise to subdural haemorrhage. Mekanismen of an adult is to rupture of one or more bryggvener cause bleeding in the space under the hard brain membrane. Ofta is in these cases also external signs of violence against the head in the form of soft tissue bleeding but in other cases , DTsurvey show at there damage without signs on yttre vage.

V id shaking of an infant come brain moves in step with skull rforelser, but blödningsmekanismen is disputed and has foreslagits be able to originate from kapillära damage [28.29]. EINCE No direct violence against the head occurs at the isolated shaken baby syndrome seen No external signs of violence against the head such as swelling, soft tissue bleeding, krosskada or skallbensfraktur. Mjukdelsblöd tions and skull fractures are therefor symptoms/findings that are not classified as isolated skakvage.

Radiological practices have shown that the subdural haemorrhage in conjunction with vagi nal delivery can occur, but usually resorbed a such bleeding within a few weeks [22]. In the space that is created by the bleeding can it by eff (effusion of fluid) developed a so called hygrom which contains cerebrospinal fluid. Det has been discussed about a renewed bleeding in this space would be triggered spontaneously or by a slight shock to the head [30-33]. Möjligheten to a broader subaraknoidalrum would be able to increase the risk of subdural haemorrhage has also been discussed [19.34-36].

Fundus bleeding Island gonbottenblödning at shaken baby syndrome have been declared prayo on the tensile forces in the eye's vitreous Islandverförts to the retina with increased pressure in the venous blood vessels in the retina that resulted in blood vessel rupture [37-39]. Det has also been expressed that the repetitive forces at shaking with acceleration and deceleration creates draws between the vitreous and the retina , as well as a direct damage in the eyeball. Fundus bleeding has noted,time has been shown to occur even in the subdural haemorrhage as assessed have been caused by the disease and it is therefore possible that the fundus bleeding also would be able to emerge as a result of subduralblödningen. An explanation would be able to be that an increase in pressure inside the skull caused by brain swelling leads to increased pressure in the central eye vein with blodstas in the retina [40.41]. Sambandet between subdural and fundus bleeding is evidenced by studies that show that isolated the fundus bleeding is very rare [38.41]. Ögonbottenblödningar has also shown itself able to occur after normal vaginal childbirth [42].

Brain effects Brain effects (encephalopathy) may Show itself in the form of inter alia consciousness , convulsions and andningspåvercan. (D)these symptoms/findings can asko on the wear damage in the brain or cervical spinal cord and/or Brain swelling. Brain swelling and damage of brain tissue caused by sewebrist can also give hjärnpåvyoucan, and permanent damage to the brain. Even increased intracranial tryck, for example the result of Brain swelling or subdural haemorrhage, can lead to convulsions, respiratory effects and consciousness lowering [43.44]. Brain swelling can be demonstrated with both computed tomography (CT) and MRI (MRT) and be seen then as the flattening of the brain's grooves and tillklämning of brain vätskerum. (D) these permits may be temporary and go back without to cause permanent damage to the brain. (D)a serious condition can by examination with CT and MRI are seen as impaired diff entiering between the brain's white and grey matter which representerar a global irreversibel, ischemic damage.

Diagnostic methods

Intracranial study M INCE DT exploits differences in has also been suspected of x-rays of substances and tissues with different density, making use of MRI in a number of different qualities of substances and tissues and provides thereby a more abundant and often more specifi characterization of the tissue that is examined.

(B) box technologies offers the opportunity to thin cut through the entire brain with reconstructions of several planes, and also assessment of the vessels in the brain through the use of contrast in the vessels. Both technologies provide similar information about changes in liquid rooms in and around the brain, such as compression of the brain's fluid-carrying hålrum at the Brain swelling, widening of the brain's fluid-carrying cavities at the hydrocefalus, and offset by bleeding and threatening inklämning.

MRT can provide different information than DT, for example if the presence of both fresh bleeding that deposition of the breakdown products of blood (hemosiderin) and early ischemic and axonala damage in brain tissue [45.46].

M INCE an acute subdural haemorrhage of a small child consists of fresh blood, consists a subacute subdural usually by a mixture of an island in theupper liquid layer and a sediment of coagulated blood [47]. (B)lödningens development over time leads tol differenta DT och MRTavbildningsmönster. Tidsgränsen for patterns onras up to yo and duration may overlap which makes an age assessment not sure [48]. Förkalkningar can in rare cases be confused with fresh blood, particularly within the brain tissue. A bleeding on the DT has a more robust design than the image pattern you see with different MRIsequences,< b1137 > what varies and partial Islandoverlap in appearance depending on the composition and chronology of events in relati spirit to the injury. (D)isto be considered age assessment of a subdural haemorrhage with DT to be more reliable than that which can be done with MRI [49.50]. Att to åldersbedöma a subdural haemorrhage can be important for that time able to korrelera to the alleged date of injury occurrence.

(B) both CT and MRI can be used for to assess Brain swelling. Brain swelling can be seen as flattening of grooves on the brain's surface area and compression of the brain's Chamber room and öother fluid-filled rooms. DT is a safer method than MRI in evaluation of fractureis.

Fundoscopy Two methods can be used for fundoscopy. The most common is the fundus mirroring with or without dilated pupil. Pon later time have a Photo graphic method for that image eye bottom developed (Retcam). (D)his method provides the opportunity to review the findings of other analysts without knowledge about the background story and question [51,52].

V id autopsy can all the eye to be examined. Even other eye injuries than those who are in the eyes of the bottom can then be described [53, 54].

Island gonbottenblödning can normally not be assessed with the help of DT or MR.T. Ma in a recently published MRIstudy have been compared a specific image ningssekvens by ophthalmoscopy and could then show that the fundus bleeding could be seen by MRTsurvey of 83 prand percent of cases [55.56].

In this context, it is important to understand that the assessment of surveys with DT, MRT and ögonbottenundersökningar part is subjective and that the individual assessoreven experiencesdevice can affect the final evaluation/ outcome. 3

Method for the systematic evaluation

Question according to PIRO

U tvärderingen aims to to answer the following: Med what security can we say that the subdural haemorrhage, fundus hemorrhage and hjärnpåvyoucan orderis managed by isolated shaken baby syndrome (without the outer layere signs of violence)?

P (Population): (B) arn ≤ 12 months In (inndextest): Tthe Riad at suspected skakvset I R (Referenstest, gold standard(d)): (E) rkänt or witnessed documented shaken baby syndrome or other trauma O (Utfallsmått): (D) iagnostisk reliability

P rojektet has been carried out according to the method that is to be found in SBU's Handbook [57]. Inclusion and exclusion criteria

Inclusion criteria

Study design S tudietyper such as case-controloll, cohort, and registry studies as well as studies with SWquality analysis method.

Observations (E) nly studies with 10 or more cases have been included in order to reduce the risk of random errors in the selection. For possible alternative(a) explanations of the symptoms/ findings have the project team assessed that it sufficed with a published case for that question the hypothesis that the triad always has been caused by shaken baby syndrome. Articles on alternative explanations have not been controlled and is therefor not to because of the results. If a subgroup of children who were victims of shaken baby syndrome and/ or a subgroup with age ≤ 12 months (median and/or medelvärde) concluded in AHTstudy r has the project team included these. Genomsnittsåldern for children who were victims of shaken baby syndrome has been set to be 2-3 months [58] and project group has therefore defined the project to the studies of children with average age or median age ≤ 12 months.

Language Articles in English, German, French and the normalwash-up languages have been included.

Miscellaneous P rojektgruppen have agreed on the only cases of shaken baby syndrome as is witnessed or in which any admitted to have shaken the child taken with in the evaluation.

Exclusion criteria P rojektgrgroup has excluded studies with Colre than 10 cases and AHforstudies of added outer layer(e) injury to the head and/or with fractures and other injuries.

(B) iomekaniska studies and studies that deal with other possible causes to the triad that has been identified in the literature search has been handled separately and presented in (B)annex 1 and 2.

Methodology for selection of studies

M ed starting point in projektets issues was carried out systematic searches of bibliographic databases in close cooperation between SBU's information tionsspecialist and the experts in the project group. Litteraluckysincreasegiven perform des in databases PubMed, Embase and Cochrane Library to and with the 15 October 2015. Additional studies have been sought through the scientific articles ' rafterenslistor and in systematic Islandversikter. For a detailed description of the search terms and restrictions that the userused, see Bannex 4 at www. sbu.se/255.

Relevansgranskning

(D) (e) abstract lists that genesboth by the literature search were reviewed by two thing savvy independent of v.arandra. Studies that assessed rpupiladopt for your project's issues of at least one of the experts was read in full text. Full-text articles were reviewed by two experts independent of each other with respect to the inclusion criteria. Studies that at the review in full text does not meet the inclusion criteria were excluded with an indication of the main reasons for exclusion, see Bannex 6 on www.sbu.se/255. Vid disagreement was resolved the question in the first place by en this floodon between the two experts who read the article. In some cases, involve the entire des project group in the discussion and decision on inclusion or exclusion was taken in consensus.

Quality review

P å because of the special research field has projektgrGroup modifi SBU: s template for that kvalitetsgranska studies in pruncheck the item and assess the risk of systematic errors (cirkelresonemang, section Cirkelresonemang in clinic and Research ning). Mallen includes among other things the type of study (prospective study, the diagnostic study, biomekanikstudie , etc.), the study's main focus , and If the study treats subduralblödning, fundus bleeding and/or hjärnpåvyoucan. Review mallen are reported in detail in Bannex 5 on www.sbu.se/255. Enly studies of medium-high or high quality has been taken into account in the results and discussion in accordance with SBImplement method [11].

S ystematiska Islandversikter on the area has been controlled with AMSTAR [57]. Rresults in this report have been based on the primary studies and not on other systematic Islandversikter, see Chapter 5 Discussion.

Method of weighting of results

Meta-analysis is a statistical way to quantitatively sum up the results from several studies and thereby get a larger data sets and a safer rate ning of the statistical uncertainty. In order to sum up the results required that studies have been done with similar method and that analyses have been adjusted for similar lightundsfaktoryou. Because only one of the two in-depth studies have a reference group has No meta-analysis could be performed.

The scientific base strength

The strength of the scientific dossier (evidensstyr) can specify how adds litling the estimate of the results are. The vetenskapliga surface is analyzed with respect to the risk of systematic errors. These include study quality, överonformity between studies, the uncertainty in the sammanvowned the results, the risk of publikationsbias and the risk for non- Islandverförbarhet to the clinical situation.

N any meta-analysis has not been possible to do but the results are based on a merger of the included studies. Evidensvärtion bygger not on a formal evidensgradering with GRADE but on a total valuation of the scientific dossier. (B)egränsat scientific basis (b)ygger on studies with high or medium-high quality with greatly debilitating factors at an overall assessment. Insufficient vetenskapligt a basis fortrue when studies are lacking, available studies have low kvconfidentiality or where studies of similar kv, that heterosexuality is showing contradictory results. Det is important to clarify that even limited scientific scientific support for a method's reliability or an effect does not imply the absence of vetenskapligt support. 4 Results

S the increase in gave 3 773 abstract of which 1 065 was commissioned in full text. Av these exklud Ed 1 035 since they do not meet the inclusion criteria. Efter quality review remained 30 articles of which two of medium quality but no study of high quality. The main reason for that so few studies met the quality requirements were that there was a lack of documentation on the cases was recognized or witnessed cases of skakold. The results are based , therefore, on the only two studies with yourname skakvage. Any balance of the results of a meta- analysis has therefore not been possible. Projektgruppen have had a discussion on coherence in the included studies. Flow chart of literature review

Figur

e 4.1 Flowchart of literature.

Evidensgraderade results

The systematic litteraturöversikten shows the following evidensgraderade results:

• The fi insufficient scientific basis for to assess the diagnosis tito the accuracy of the triad for to identify the skakvage.

• (D) et there is limited scientific evidence for the triad , and hence its subcomponents can be present at the skakvage.

(D) (e) two included studies of moderate quality studies are with recognized cases, both from France. The study of VIncheon and Associates is a prospective study. The study is based on a registry of traumatic head injuries to children under two years of age who received the hospital between January 2001 and February 2009 in a catchment area of approximately 4 million inhabitants [59]. Materialet comprises 412 cases, of which 124 were classified as added head injury (innflicted head injury, IHI) and 288 as accident damage (Accidental trauma, AT).

In the group with added damage (IHI group) were 45 cases with recognition, of which 30 cases in which shaken baby syndrome been recognized and 15 cases where others external violence recognised. In the article missing , however, detailed descriptions of how the suspect has gone about as well as how the recognition emerged. (D)his Group children compared with 39 cases of witnessed the accident damage that owned rooms in public environment (AT group).

In the Group of inflicted injuries had 37 of 45 children (82 percent) subdural haemorrhage comparet with 17 out of 39 children (44 per cent) in the Group accident injuries. Trettiosju of 44 children (84 percent) in the Group of inflicted injuries had fundus bleeding, comparet with 6 of 35 children (17 %) in Group accident fallsskador. In the Group to suffer head injury found to be 12 of 45 children (27 prcent) had brain ischemia comparet with 1 of 39 children (3 pr%) in Group with accident damage.

Study of Adamsbaum and employeee is a retrospective observational study that includes 29 recognized cases of shaken baby syndrome (where direct violence against the head as de vits in 5 cases) as compared with 83 cases without recognition [60]. Innklusionskrite Ries in the study are subdural as shown with DT and a recognition from the suspect. Then one of the inclusion criteria in shaken baby syndromesgruppen is considered subdural haemorrhage, only the results of fundus bleeding in this study.

In the Group with recognized cases (Group A) had 24 cases (83 %) eyes the bottom bleeding. In cases where someone recognized described the shaking as violent (100 per cent) and in some cases have repeated shaking recognised (55 per cent). Intime correlation could be seen between the density of subdural and number of tryingepade shaking of the child. In this article , see detailed descriptions of how the suspect passed about in 14 of 29 cases. In the other Group (Group B) Code of your cases with children who had been shaken in order to revive the child, olycksfallsskador and children where no explanation regarding the permit specified and This group can therefore not be considered to be an acceptable reference.

(B) both Vinchon and employees as well as Adamsbaum and coworkers reported that shaken baby syndrome can lead to subdural and fundus bleeding. In the study of Vinchon and employees exhibiting group with recognized shaken baby syndrome a larger proportion of subdural haemorrhage, fundus hemorrhage and brain ischemia that may give rise to brain impact, than the group who injured at a witnessed the accident. Adamsbaum and employees compare children who were victims of recognized shaken baby syndrome with group children where the suspect not recognized which may lead to inklusionsbias in one or both of the compareda groups. (E)since only one of the two studies have a referensgrup has no meta-analysis could be performed.

The fi also other published cases that have been excluded (the wrong population, the wrong study design), but where there are detailed descriptions of the admission that speaks in the same direction as the two studies of medium-high quality [61,62]. Because the fi so few studies with medium/high quality has it not been possible to assess the diagnostic accuracy of the triad in order to identify shaken baby syndrome.

Table 4.1 Characteristics of included studies.

5 Discussion

Literature search identified only two studies of medium kvto corporal punishment, Although that relatively many met the inclusion criteria. (D) thenumber one is anmärkningsvärt with a view on the shaken baby syndrome is very seriously and get dramatic consequences for both the child and the family. Forskningsområdet is complicated, but it defends don't such as circular reasoning and a lack of recognition of data collection. Det is important to Islandversikter on the area presents the methodological deficiencies that research field characterized by.

Sstudies of Adamsbaum and employees as well as VIncheon and colleagues assessed the achievement of medium kvto corporal punishment. Alsoa If both studies have methodological limitations speaks the for to isolated shaken baby syndrome can give rise to the triad.

(D) a prospective study of Vinchon and Associates includes more than 400 cases in which 124 rating that added head trauma and 288 as accident damage. Fyrtiofem was recognized cases of added , damage of which 30 cases were recognized in isolation shaken baby syndrome (IHI group).

Trettionio case was witnessed accidents (Asuch group). The advantages with this study is that all the trauma cases as submitted to the hospital recorded exploration effective during many years. Sstudy endpoint is also a clear reference together with injuries after witnessing an accident. (D)his Group of children is , however, significantly older. Tto the study's limitations are related to the lack of detailed Description of how the tremors passed to and when they occurred. VIncheon and colleagues analyze the part components was for himself, but the authors introducerar also a different Combi nation of the triad , which consists by subdural haemorrhage, fundus bleeding and absence of visible damage on the head. Data on hjärnpåvyoucan see , however, registered in the study in the form of , inter alia, cramps, reduction of consciousness and coma. Group of isolated shaken baby syndrome will per definition to include only those cases that do not have external signs of violence while the group of accidental injury will to include cases that have signs of external violence. Projektgruppen has in a early stage contacted doctor Vinchon for to undanran opportunitya vinssa oclearhets but inte received answers on all questions. Frågorna force among other things how the fundus bleeding defined, how the authors have calculated the sensitivity, specificity and predictive(a) worthit for the triad, wasfor the valt a deviant triad (wear byaro of visible damage instead of brain effects) , as well as how the admission gone to and what that isfelt.

Study of Adamsbaum and employeee is a retrospektiv observationsstudie which includes 29 recognized cases of shaken baby syndrome. In the study reported detailed admission in 14 cases. It can however not be excluded that the among the cases without detailed admission may have tocome shake after that that child had symptoms at hjärnpåvercan. Studiegruppen is compared with a reference group consisting of 83 cases without recognition but this is not a "pure" reference group because it can access case by shaken baby syndrome even among those as not recognized. A criterion for all the cases in the study is the subdural haemorrhage, which means that only the results of fundus bleeding can be taken into account.

At review of the literature has the project team identified the other conditions or events that can give rise to the three symptoms/findings in the triad. A some of these conditions or events does not lead to permanent damage or are very rare, but shows that triad or subcomponents in the can be caused by other things than shaking. Da is , therefore, important to Islandverväga the ability of these differential diagnoses in the investigation of suspected skakvage. Decisions that are taken of social service or Court is based alsoan on other bases/other evidence than the medical assessment. In the further processing of cases way other possible causes to the triad or its subcomponents .

An analysis of studies in biomechanics, see (B)annex 2, showing conflicting results and No conclusions can be drawn about where the border runs for the forces that generate these damages on child.

Methodological problems

By the time of the vetenskapliga basis for the diagnosis of shaken baby syndrome in children under 1 year of age (mean age or median age ≤ 12 months) have shown that the published studies have methodological problems.

Definition of shaken baby syndrome P rojektet has been limited to studies in which shaken baby syndrome been deemed to be the primary cause for the resulting damage, but others studies have had a further defi tion, for example suffer head trauma. It is then not always been possible att distinguish if an injury caused by shaken baby syndrome or by direct violence against the head.

Group Division of studies A other methodological problems have been that shaken baby syndrome not always been witnessed or been recognized of the suspect which leads to a proper rating of the cases in shaken baby syndromes r and referensgrup becomes uncertain, and thus run the risk of leading to an incorrect evaluation of the relationship between the triad and shaken baby syndrome. At the same time as it is a risk for false attribution constitute this the only possibility to get insight into what that happened to the infant unless it does not see video documentation. P å because of the risk of false confessions have any recognition in these studies are treated with caution.

P rojektgruppens choice to accept only recognized cases, is thus tainted with methodological p.r.operation issues. Erkännandet would be able to be false on the basis of that it emerged through a hearing. Det would be able to be false even on the basis of that the suspect felt himself forced to admit [63-65].

Circular reasoning in clinic and research In the social service act is apparent to all who get knowledge about or suspect that a child father ill should report it to social welfare. Registration required is the staff in the health and medical careit, tandvårit, pre-school, school, socialservice and the probation service who is obliged to immediately notify the social services If they are in their activities , knowledge about or suspect that a child 's father badly (the social service act 14 . 1 c §).

In many cases, are investigating a child protection team case of suspected shaken baby syndrome, and during the years races have these teams developed criteria for when certain symptoms and findings may be linked to isolated shaken baby syndrome, after it to other possible causes of symptoms and findings have been omitted [66.67%]. SOME of these criteria are linked to the care nadshavarens believeheatis required. Vårdnadshavara deemed not reliable if he is not able to give a satisfactory explanation of how symptoms and findings , to the example of the child fallen from low altitude and not a exterior damage. Changed the data – for example, that the custodian first indicated that he/she has not shaken the baby but then says that as actually occurred, but only after it to the child stopped to breathe or become unconscious – means also to guardian's credibility diminishes. If shaking occurred as a consequence of that child suddenly a symptom (for example stopped breathing) would it however be reasonable to assume that the root cause of the child's condition arose< b1224 > already before the child was shaken, and consequently that the shaking is not been the cause of the symptoms. Ma if such a Declaration is not deemed as acceptable is the case still classified as shaken baby syndrome.

(B) arnsky ddsteamens criteria is primarily based on current clinical approaches [66.67%]. Problem occurs later if/when the criteria are not prevas prerequisite solved by researchere at systematic studies of the relationship between triadsymtomen and shaken baby syndrome. This means that the teams ' interpretations come to characterize the vetenskapliga survey and hypothesis, which in its turn leads to that the existing approach is strengthened instead of to the przvas. Ma if you already before the study proceeded from there you want to study, it would say how the link between the findings and symptoms and shaken baby syndrome look out (according to< A1115 > team's criteria), ends up being in a circular argument. Tillämpat on this way leads the reasoning to high risk för systematic error (bias) which in its turn leads < b1167 > to that man does not know what the man jämför (shaken baby syndromesgruppen may include children who have not been shaken , and the reference group could include those that have shaken). Att based on such compareelser calculate sensitivity, specifications, as well as predictive worthit, leads to incorrect conclusions. It leads also to that incidensberäkningar becomes invalid.

(F) ör to avoid this circular reasoning in the studies must study cases and reference are identified on an opposition-free way. Projektgruppen has chosen to as study cases accept only cases where someone witnessed (or through video surveillance documented) an event with shaking or where any of the detail recognized themselves have shaken baby.

Diagnostic methods (B) stimate by the time of the emergence of a subdural haemorrhage is unsafe. (D)his insecurity is also greater among children under one year of age, on the basis of the subdural haemorrhage of a child is something different to their character than in an adult. A subdural haemorrhage of a small child consists usually of a övre vätskeskikt and a sediment of coagulated blood, which if subduralblödningen is subacute can exhibit different also been suspected [47]. OSecurity is through development, ling av me DT och MRTmethods(a) reducingt anysuch comparirt med earlyre [46], but it must be continued careful at age assessment when it is different bleeding patterns that partly Islandoverlap [48].

In both controlollerade experiments as observational studies , systematic errors occur when different observatöryou don't always make the same observations and/or make different interpretations of observations. Oyouknow between different under the viewfinder in a study may vary depending by how trained observers are. (D) thenumber one applies generally in observations and assessments, and of course alsoa at examinations and assessments of symptoms/findings after suspected skakvage.

In one study showed it out for example that variation was high between investigated th e interpretation of fundus bleeding it would say it was low- coherence between different assessors [51]. Comparisons with results from other views

Projektgrrevelations have identifi seven systematic literatureIslandversikter that contains the same or partly the same question as this report [68-74]. Dessa systematic Islandversikter is included not in the profit and loss part but the project team has therereceived reviewed and assessed these as they are often mentioned in the scientific literature. All systematic Islandversikter has the project team assessed as having low quality (high risk of bias) , among other things, for that many of them are based itself on studies where a team has assessed that a child shaken if the child has triad (cirkel reasoning, section Cirkel reasoning in clinic and research). A different problem in the systematic Islandversikterna is that shaken baby syndrome is not specified, and that it used the more Island-wide term AHT but that in detail describe what that are included in the groups that compares.

6

Consideration for research

(E) INCE this of course is impossible for scientists to expose children to shaken baby syndrome, used in biomechanical studies dolls/models fitted with inbyggda measuring instrument for that examine how the mechanical forces seem on children. Rresults are not conclusive and results from animal models are of different reasons difficult to Islandverföra to the children.

Projektgrthe revelations was therefore referred to the observational studies where exposure (in this case shaking) adopted be given. Most reliable are prospective cohort studies and ideally livingthe the that included in a shaken baby syndromeskohort consist of such cases in which someone isknown shaken baby syndrome and where these arefeeling redoshown in detail and where it is shown in what circumstances the recognition has emerged.

In many of the reviewed studies are children who belong to the reference group signifikant older than children in shaken baby syndromesgruppen. The brain, bones and neck muscles look differently out of a 2 -month -old children compared with a child at 8 months. Children who are 0-2 months old can be assumed to be more sensitive to shaken baby syndrome than an older child. If we compare the two groups of children (shaken baby syndromesfall and children with olycksfallsskada) who not is age-matched can it lead to selektionsbias and incorrect conclusions. Studies with matched age groups would mean the ability to calculate the sensitivity and specificity and predictive values. (D)on the other hand, would be able to Express themselves more secure if the probability to triad caused by skakvage.

The lack of knowledge about the pathophysiology at utvecklingen of subdural and fundus bleeding in conjunction with vaginal childbirth. Adven If majo the majority of förlossningsbetingade hemorrhage did not give symptoms and disappears

(resorbed) within a few months , occasional bleeding may be converted to a hygrIf [19, 30.36]. Dessa separated water retention is surrounded by a mem brane in which it formed the small vessels, which in its turn is considered to lead to a new bleeding (reblödning) and a chronic subdural blood accumulation. Det can not be ruled out that a reblödning in some cases can give symptoms [19.36]. (D) thenumber one might be able to be a reason to be a child suddenly affected by brain effects (becomes consciousness sänkt, stop breathing and/ellyou have cramps) and that custody seas on the basis of this looking for care. Hypotetiskt seen would one such reblödning could be triggered spontaneously or at a less trauma. It is therefore urgent that it conducted research concerning the pathophysiology and natural history of subdural and fundus bleeding. Even the development of higher performancee models within biomeka technique, to the example models that take into account the impact on both the brain and cervical spine would be able to contribute to increased knowledge about shaken baby syndrome.

What is needed in order to fill the gaps?

The reasons for the gaps in this area are of different nature and need therefore be managed on different ways; from a coordination within the entire research field on what that should be researched , to that do the studies on a methodologically correct manner, as well as to fully describe how studies have been carried out.

International coordination (F) ör to improve Diagnostics in the area needed a broad coordination at international level in order to get enough big study groups. (F)orskare in the field would be able to agree on what that is most important to do research on and interact in order to allow for more comprehensive studies , as well as more similar study design so that the results can be compared. An international register över recognized and väldoku documented cases would be established.

Urgent research areas S Pecific important are studies that aim to improve the the diagnostic reliable ity by diagnostic imaging of the brain, cervical spine and eye [75]. Det needed also retreate methods for the study of the identified the natural history. Alsoa differential diagnoses such as förlossningsrelaterade bleeding in children need to be studied in order to identify the natural course [22, 36, 76,77]. Studies also are needed for to increase the understanding of the pathophysiology behind triad. Even better I lovee models of Biomechanics might be able to increase the understanding of the skakvage.

In as far as possible, subject to , of course, all the established quality criteria to be met. Viktigt is also to researchers is blinded for the suspects up komstmekanismen, and that the results be presented so that they allow calculation of diagnostic reliability. (D)a later means therefore that each individual finds must be assessed in both the study group and referensgrup. A of the reasons for that, it is difficultfor to obtain evidence on the field is that many studies are flawed description of methodology and results. Below describes some viewpoints and aspects that should be taken into account in order to study the quality should be assessed and for the meta-analyses should be carried out on a Add precaution :

S the studies should: • consist of a prospective observational studies with yourfamous and well document related cases with reliable methodology and where the risk of false confessions have been minimized

• be age-matched (study group and referensgrup)

• contain detailed accounts of how the learning materials collected, including including documentation of investigative techniques, and detailed accounting of the complementary studies which have been carried out in order to exclude the differential diagnoses

• ready view to analysts of symptoms/findings varit blinded (IE. v arit ovetande) If fyndens reason and how this blinding has been implemented

• ready view raw data, sensitivity/specificity , and confidence intervals

• use sufficiently large materials which are examined with uniform methodology

• present detailed readyviews of yourfeeling and what that isfelt and how the recognition went to.

7

Project team, external reviewers, advice and Board Project group

Experts Göran ElindEr pr ofessor in Paediatrics at Karolinska innstitutet, former senior consultant in child and adolescent medicine, Sachsska barnsjuk House, Södersjukhuset, Stockholm

andErs Siksson professor in forensic medicine at the Umeå University, Islandverläkare in forensic medicine at the forensic medicine alvisket

BouBou HallBErG with Dr., everläkare in Pediatrics and Neonatology, Karolinska innstitutet and Karolinska hospital, Stockholm

niEls lynøE professor in medical ethics and medical specialists in general medicine, Karolinska Institute, Stockholm

PIA Maly soundGrEn pr ofessor in radiology, Islandverläkare Radiology and Neuroradiology, Lund University Hospital, Lund

Måns rosén former adjunct professor of medical methodevaluationevaluation, Karolinska Institutet, Stockholm

Bearsn-Erik Erlandsson (B)annex 2 Biomekanikstudier (external ECTkande) professor in medical technology, school of technology and health, KBasic T's technical high school, Stockholm

SBU Frida MowaFIn Project Manager

MariannE HEiBErt arnlind Project Manager until september 2015

Irene EdEBErt Deputy Project Manager(e)

Hanna oloFsson information specialist

(a)nd (a) GranatH project administrator

lau r a lintaMo investigators

(a)nna BjörklövenF Communications Manager

External examiners

SBU hires external Auditors of their reports. Dessa has come with valuable comments, which in high grade contributed to to improve the report. In the final version of the report is it possible that the SBU could not accommodate all the change or amendment from the external examiners, among other things, for that they have not always been consistent. De external examiners are therefore not necessarily being the emergency behind all parts of report.

(E) xternal reviewers who have chosen to be named:

stEvEn lucas * with Dr., everläkare in children and young childhood medicine, Academic hospital, U ppsala

Tiit MatHiEsEn a visiting professor, everläkare, Neurokirurgiska clinic, Karolinska hospital, Stockholm

titti M pr ofessor , (J)legal faculty, Lund University

nils-Eric saHlin pr ofessor , Medical faculty, Lund University

arnE stray-PEdErsEn * position, Institutt for klinisk medisin , Universitetet in Oslo

innGEMs tHiBlin* pr ofessor , Department of surgical vetenskaper, Uppsala University

* S (e) opinion on www.sbu.se/255 Bindings and conflict of interest

S akkunniga and the Reviewer has in accordance with SBU: s requirements submitted declaration concerning bonds and conflict of interest. (D)these documents are available on the SBU's Cabinet. SBU has assessed that the conditions that are reported there are compatible with the requirements of objectivity and impartiality.

SBU's scientific advice – the Brage

SBU: s vetenskapliga Council has reviewed the vetenskapliga basis in the report.

lars H President, professor, vårdvetenskap, Lund University cHristEl BtsEvani registered nurse, with dr, vårdvetenskap, Malmö högskola

PE r carlsson professor, health economics, Linköping University

Bearsn-Erik Erlandsson professor, medical technology, KBasic T's technical high school, Stockholm

ARnE GErdnEr pr ofessor , social work, School of Health Sciences in Jönköping

lEnnart isElius Associate Professor, Hhealth and care inårdsdirektör, County Council of Västmanland

MussiE MsGHina Associate Professor, everläkare, Psychiatry, Karolinska University Hospital, Stockholm lars sandMan pr ofessor , ourdetik, University of Borås

Br itt-MariE stålnackE professor/Island v erläkare, rehabilitation medicine, Umeå Universitet enantE twtMan pr ofessor , tandvind, Halmstad , and Copenhagen University

SBU's scientific advice ‐ Eira

SBU: s vetenskapliga Council has reviewed the vetenskapliga basis in the report.

kjEll a.s. President, professor, Stockholm

HE #NS andErsHEd professor of psychology, Associate Professor of Criminology, Örebro University kristina BEnGts BoströM Associate Professor, Billingen ourdcentral, Beavde

cH ristina ASKrGH professor, women's clinic, Sahlgrsport for the Italian, Gothenburg anna EHrEnBErG professor, vårdvetenskap, High School DAlders theGEMs EnGlitterM pr ofessor , Psychiatry, ethics, Örebro University

nils FEltElius Associate Professor, Läkemedelsverket

y l v (a) n Associate Professor, physiotherapy, CAMTÖ, Region Islandrebro County

stEn-kE stEnBErG professor, social research, Stockholm University ka take ina stEEn carlsson file dr, health economics, IHE Lund

SBU: s Board

SBU: s the Board has reviewed and approved the results.

nina rEHnqvist President SBU: s Board proffessor, Karolinska Institutet

susanna axElaib generaldir ektör, SBU

HEiki ErkErs President, graduates SSR

Eva Franzén Research and developmentecklingschef, The State's Institutionsstyrelse

on the other hand,said Furén-tHulin Section Manager, S Sweden Municipalities and County Councils

jan-inGv s jönsson huvudsekreterare for Scientific Council for medicine, Vetenskapsrådet

Bjorn klinGE pr ofessor , Dental faculty, Malmö högskola, and Karolinska innstitutet

lars-torstA larsson Associate Professor, ofsharing a Manager, Socialstyrelsen

stEFan lindGrA professor, President, SIslands medical society

STIG nyMan President, Disability Federation sinEva riBEiro Chairperson, Vyeardcovenant

(a) GnE take von scHotinG Chairman, National winnersChurch group for knowledge management in social services (NSK's)

HEidi stEnsMyrEn President, SSweden Medical Association andErs sylvan County Director, Västerbottens Läns Landsting

Håkan sörMan ver kställande Director, S Sweden Municipalities and County Councils k arin tEnGvald professor emerita, Linköping University

8 External collaboration

Usually the interaction

P rojektledningen have met the National Association Fo Familjers Rights (RFFR) in the beginning of the pruncheck the item for to inform and take in the views.

Network agencies

R representatives from Barnombudsmannen, innspektionen for health and welfare, Kriminalvårit, Socialstyrelsen, Sweden's Municipalities and County Councils, Polismyndigheten, Rättsmedicinalverket and the Prosecutor's Office, has been offered the opportunity to hit the SBU for that get information about the report's results in conjunction with the report was released.

The State's medical‐ethical advice (Smer)

(D) a ethical analysis has been conducted of the Smore.

Glossary

AHT (eng Abusive head trauma) Head injury added by child abuse At (eng Accidental trauma) Accident trauma, accident damage Has also been suspected of X‐rays attenueras (absorbed) in the body various very depending on the tissue density ASK (eng Brain edema) Cerebral edema (Brain swelling) Cerebrospinal fluid Fluid that surrounds the brain and spinal cord (eng CSF cerebrospinal fluid) CT (eng Computed tomography) Computed tomography (x‐ray) EDH (eng Epidural Epiduralblödning, bleeding between hemorrhage, Epidural the hard meninges and skull bones hematoma) Brain edema Brain swelling

Hydrocephalus Increased amount of cerebrospinal fluid in the brain cavity ("water‐kalle") Hygrom The accumulation of fluid, can relate to education after bleeding IHI (eng Inflicted head injury) Head injury added by child abuse IHT (eng Inflicted head trauma) Head injury added by child abuse

Interdisciplinary Team with diverse professional backgrounds (multidisciplinary) who are working together in an area or on a team question MRI (Magnetic MRI resonance imaging ) MRI (Magnetic MRI (see MRI) resonance tomography) nAHI (eng Non- Head injury added by child abuse accidental head injury)

NAHT (eng Non-abusive Head trauma caused by other than child abuse, head trauma) usually by accident damage NAT (eng Non-accidental trauma) Damage added by child abuse

SAH (eng Subarachnoidal Subarachnoid haemorrhage, hemorrhage) haemorrhage between the soft meninges SBS ( Shaken baby syndrome) A syndrome consisting of three components, the triad

SDH (eng Subdural hemorrhage, Subdural haemorrhage, Subdural hematoma) haemorrhage during the hard meninges Subaraknoidalrum The room between the soft meninges

Syndrome Symptom complex, a group of symptoms that belong together and which occur together in a same and patient more frequently than that can be explained by random chance Triad Three components in a whole. At SBS refers to a triad usually consisting of subdural haemorrhage, fundus bleeding and encephalopathy

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Annex 1

Other possible causes of the subcomponents

At clinical diagnosis is made the differential diagnostic Islandpanacheownerships. In these cases the child is received with hjärnrelaterade symptoms that needver utredas . In this report has therefore the differential diagnoses that emerged in our data base search containing alternative(a) explanations for the various symptoms/findings of triad, was of itself or to the triad in its entirety noted. (D)these articles are in the rule of the case reports of individual patients without reference test and has therefore not been included in the quality control. Projektgruppen has nevertheless considered that there exists a intresse to here readyto show these sorts of Options(a) explanations to the triad.

Table 1 Disease/condition Reported Reference Other possible causes findings from number (differential diagnosis) the triad (number of of the triad and its cases, or components. cases/study population size) Reported finding from the triad Diseases or von Willebrand's disease SDH, RH [78] (1) conditions causing Delta storage pool disease SDH, PRAY, RH [79] (1) hemorrhagic symptoms Hyperfibrinogenemia RH (including [80] (1) vitreous hemorrhage)

Hemophilia A SDH/RH [81] (2) RH [82] (1) SDH Factor X deficiency SDH [83] (2)

Idiopathic ICH [84] (1) thrombocytopenic purpura Kasabach‐Merrit RH [85] (1) syndrome thrombocytopenia Hepatitis RH, ASK, SDH [86] (1)

Albers‐Schönberg disease SDH [87] (1)

Vitamin K deficiency SDH (ICH)/BE/RH [88] (3) SDH [89] (17) SDH [90] (1) SDH [91] (16) SDH, [92] (1) SDH, PRAY, RH [93] (1) SDH, ASK Menkes disease SDH [94] (1) (Copper deficiency) Unspecified RH, SDH [95] (1) [96] (3) Infections Infection RH [97] (4)

Infection with SDH (intradural [21] (10/30) or without bleeding) hypoxia

Vascular mal Aneurysm, Arterio‐ SDH (SAH)/BE/RH [98] (1) SDH formation venous malformation [99] (1) SDH, ASK [100] (1) SDH, ASK [101] (1) SDH, RH The table continues on the next page Disease/condition Reported Reference number findings from (number of cases, or the triad cases/study population size) Reported finding from the triad Prenatal and Countries RH [102] (11) birth-related injuries Delivery injury SDH (ICH)/RH [18] (2) SDH [103] (3) ICH [96] (3) SDH [56] (53) RH [104] (10) RH

Normal SDH/RH [77] (17/97) SDH delivery (or [39] (94/252) RH prenatal) [76] (32/63) SDH Prenatal trauma RH [105] (2)

Congenital SDH SDH [106] (1)

Congenital heart disease SDH [22] (66/152)

Table 1 continued

Large head size

Enlarged SAID space/External hydrocephalus/Benign enlargement of the subarachnoid spaces

SDH [31] (6/108) [32] (4/177) [107] (7) [33] (3) [96] (6)

External hydrocephalus RH, SDH [108] (1) [109] (6)

Immunological diseases

Hemo‐phagocytic lymphohistiocytosis

SDH, SAH (ICH), PRAY, RH

[113] (1)

Transplacental acquisition of anti‐Ro antibodies

SDH [114] (2)

Skeletal Osteogenesis imperfecta SDH, RH [115] (3) diseases Brittle bone disease SDH/RH [116] (20/20) SDH (11/20) RH

Other Hypoxia and resuscitation RH [117] (1/33) [118] (1)

Hypoxia SDH/intradural bleeding

[21] (20/30)

Choking and resuscitation SDH (SAH), RH [119] (1)

Resuscitation in patients with retinopathy of countries Hypernatremia and dehydration

RH [118] (2) [117] (1/33) ICH, ASK [120] (1)

Leukemia RH [95] (3)

Vaccine‐induced vitamin C deficiency

SDH, ASK [121] (2)

ASK = Brain edema; ICH = Intracranial hemorrhage; RH = Retinal hemorrhage; SAH = Subarachnoid hemorrhage; SDH = Subdural hematoma

Annex 2

Biomekanikstudier

For to study the Biomechanics behind shaken baby syndrome and shaken baby syndromesrelaterade questions have different physical and virtual models are designed as a simulation of the babies who are shaken and that aims to to measure certain effects of the mixing cycle. Trettio scientific articles about Biomechanics as identified in the search have stude rats approachinge. A few Islandversiktsartiklar gives a good understanding of the problem area, but also the understanding of genesella traumatic brain damage, regardless of age. (D)(e) the majority of articles readyshows model tests and simulations of biomechanical character and many articles readyshows , moreover, preliminary data, while getting have repeated their studies. The articles are very different in their design, ranging from case histories of various modelluppbyggnader which will be ofclear mechanisms at shaken baby syndrome. A couple of articles consists of comments to a different article. In one such example , it should be noted that the authors to a previously published article has shrimp nat error on a 10 power if an injury should be able to occur or not [122]. SOME studies demonstrate clearly contradictory results. His follows examples of this.

(D) uhaime and coworkers present a biomechanical model for shaken baby syndrome [5]. The work is regarded as a reference work that many other researchers have been deleted from and then further developed. In the article turns solid that you can not achieve the harmful effects of shaking.

Cory and (J) theones ' article [123] bygger on a biomechanical model that has modi fi after Duhaimes model [5]. Cory and co-workers ' article shows that this model is marred with irregularities and the result shows that the forces at shaking of a child many times can Islandexceed the powers which are necessary in order that the damage will occur.

The v etenskapliga substrate from the studied articles is thus diverging rande and no definitea conclusions can be drawn regarding the powers that are necessary for that damage will occur.

Annex 3 Ethical analysis of shaken baby syndrome The State's medical‐ethical advice

S tatens medical ethical advice (S) have implemented an ethical analysis of whether Council "shaken baby syndrome" in connection to Statens preparation for medical and social assessment (SBU) report on the subject.

S more has not made any own scientific assessment of the base material. The analysis is based therefore on the results that the SBU had come forward to report in and that can be summarized in the following points:

• (D)et , there is insufficient scientific backing for that assess the diagnosis TICAL accuracy of triad for that identify the skakvage.

• There are limited vetenskapligt basis for the triad (subduralblöd ning, fundus bleeding and various Forms of brain influence) and thus its subcomponents can be present at the skakvage.

• TRiad or subcomponents in the can be caused by other things than shaking.

Outline

Analysis begins with a conceptual analysis of the term shaken baby syndrome. The fortsatta ethical analysis are structured on the basis of two dimensions. In the one identified the parties who have intressen in question and in the other are identified which ethical valuen that is relevant in the context of a possible shaken baby syndromessituation. Slutligen dinskuter.as avvägningen between different central heatit and the heatdekonflikter that may occur at this balancing.

Conceptual problems

A conceptually understood issues about shaken baby syndrome is that the term as such is etiolo policy, it would say that it in itself includes the reason for the observed damage in the child . (B)the concept of shaken baby syndrome has been used as one of a small child found triad subdural haemorrhage, fundus bleeding and various Forms of brainaffectedcan. In SBImplement review of the scientific literature has been found in a limited vetenskapligt basis for the triad or its subcomponents can be present at the shaken baby syndrome, but it has also found that the triad or its part components can be caused by other things than shaking.

Es the SBImplement report is the scientific dossier insufficient " to assess the diagnostic accuracy of the triad for to identify the shaken baby syndrome".

The doctor that meets the family of the injured child has possibly also other sources than those that x-ray, neurological examination and fundoscopy offers. Det can be other injuries on the body which supports the suspicion of abuse as well as observations in conversations with health nadshavarna. Det is an ethical requirement that all this way in the physician's judgement in the face of a possible notification of concern to social welfare.

The doctor have a duty to accurately describe everything that transpired during the investigation, both the damage that emerged as the information that the guardians have to leave If the sequence of events and circumstances in Island incidentally. It is also of the utmost importance that all injuries are documented thoroughly, both for health care needs that are ahead of a possible future judicial process.

V ACH decisions in health and medical services, either out of these is of diagnostic or therapeutic grade art, based on both facts and values. Med facts referred to in this samman hang description of all rpupiladopt findings that were made with the help of physical, radiological, laboratory- based and other medical examinations of the child. It should , however, take into account that even facts may contain evaluative element, such as assessments of x-ray findings. The next step that occurs in the doctor's job is to evaluate the medical findings and the content of the stories about the events. (D) thenumber one work is of a different nature than the facts related description. His has the doctor a significant ethical responsibility that in rate expectations only based on vetenskap and proven theory,what experiences theunit. Parties

Utgångspunkten for analysis is that a small child is received to the health and medical care along with a or two guardians with damage that gives rise to a clinical suspicion If that assault can be a reason for the child's injuries. If the child's injuries include the so called triad of symptoms and findings arises the question whether this may have been caused by shaken baby syndrome. RINCE in this mode, fi the fl partners which have legitimate interests of how the situation is handled. It is the child, its guardians and caregivers in various professions. In occurring cases can also be the child's siblings to be affected by the process. In a later stage , the situation also come to include social service personnel and political officers (eg. in the social welfare board) as well as the police, prosecutors and the judiciary's different instances.

Values

(B) arnet has a very special position in the current situation of several reasons that are based on ethical values. (D)a is in these contexts for very small children. It means that the child completely lack the ability to self report on what that occurred and therefore of obvious reasons not able to defend their own interests. De damage which is present in the context where shaken baby syndrome can be suspected can be of serious art, both acute as in the longer term. Skadorna can be immediatesuch life-threatening or result in a risk of permanent legislative decisionbe ruled out, what applies to the child's utvelopment, health and future qualityTy.

A ethical analysis of shaken baby syndrome should of these reasons primarily deleted from a child 's perspective. (D)a central ethical question is how the child's intressen on best way can be exploited because there never can be acceptable that a small child becomes a victim of domestic abuse.

It is an ethical duty to the small and vulnerable child's interests are catered for by any other person. In the normal case is that parents who have the task to defend the child's interests. In a situation where the shaken baby syndrome are suspected as it is , however, often some of the guardians (or both) that may be caused to the injury. (D) thenumber one means to him/this then possibly not fulfilled their parental responsibility.

In the scenario that this outline is falling because the immediate responsibility for to protect the child's central values on the health professionals who meet the family at the hospital. In one such situation must staff Act according to their professional dizzykesetik and based on the existing legislation.

It can be about that in the first place take vital emergency medical measures that are needed by the child's health status. Barnet must have access to all emergency and other necessary medical interventions for the cure and relieve the acute injuries and for to prevent future sequelae. (D) thenumber one has , of course, the first priority in the processing.

If suspicion arises about the damage caused by the violence as is the doctor's responsibility to investigate this suspicion based on science and the beprIslandwhat experiencedevice. Det is also the physician's obligation to under 14 Cape. 1 § social services Act (2001:453) report to social welfare if there exists suspicion about that child 's father badly.

S of all in have an explicit responsibility for the protection of children in a number of different ways. (D) thenumber one follows the example of the Social Service Act and Act with specific provi sions for care of the young (1990:52). The last Act Specifies the opportunities for social action in consultation with the parents while the other teams are giving the community the opportunity to take measures to the protection of the child without custody officers consent. An outer measure is that the social welfare board may take possession of the child for care outside the home.

(B) arnkonv entionen, as for the present is on its way to be incorporated in Swedish law legislation, indicates a number of basic rights that all children have, such as the right to the protection of life and health, right to grow up in good conditions and the right to good care. (D) thenumber one document is formulated on the basis of a rights perspective but rests also on the Central ethical principles about adult society's responsibility for child's life to the protection of what that is Central values for all children.

N is the doctor will recruit guardians if the damage can be caused by any of these known external event, so is it unusual that this allowed in time (Lowenstein , 2004). It is important that the doctor in this situation not to take over the judiciary's role to determine a possible crime or accuse any single individual. Vårdnadshavs have legitimate intressen of to some of those central values are taken into account in the situation. Till these include the right to good care, which custody seas vanligen is concerned about the independence of reason or evmay intent (Leuthner 2001). (V)Moreover , it is an important value for him/them to be listened to in an adequate manner and that the handling of the situation at the hospital is done based on an impartial and unprejudiced basis with regard to of all possible causes of the observed damage.

(F) ör the nursing staff is there a Central value to meet out of respect for their professional on professional assignments , both from a medically as an ethical perspective. It is the most common genetic doctor who has to task to evaluate the likelihood of that the observed damage as the child exhibits can be the guarantee of any adult person,< b181 > usually someone of the guardians, and thus able to be characters on skak violence. (F)for the physician is that of considerable value to get the necessary notice of his decision on a possible notification of concern. A decision on the complaint is fraught with significant legislative decisionalso for both child custody seaIslands and therefore must be well founded and well thought out. One such decision should always be made in consultation with at least one other medicale.

The doctor has also an intresse of that have adequate training and expertise in the area of child abuse for being able to handle these ethical and psychological very difficult situations in a professional manner.

N is it concerns social services separates itself responsibilityarsförhållandena radically compared with health and careinthe. Decision-making mandate regarding measures without consent fall on the social welfare board politicians; in acute situations represented by its words Chairman by delegation. Udossier for the decision to be taken , however, until by perso nal within social services. (D)his staff have professional ethics rules for his work which should be taken into account in situations of current kind. (F)for social service personnel is the a Central value to safeguard the child's interests and to protect the child from threats against life , health and development. It is a Central value for the social services that the information which they receive from health and health care is medically CorrECT, well researched and formulated in such a way that conclusions if the reason for the observed damage not reported but secure foundations.

If the case, emergency or in a later stage, later Islandvergår to the police, Prosecutor and Court (s) so have these instances a similar interest when it comes to infOrma forinflation from health and health care. If and when a case ends up in Court, so it is important for the Court to have access to special scientific expertise which pronounces itself in accordance with ethical principles and applicable legal rules for certificates and opinions.

Value conflicts

(D) et there are some significant value conflicts in conjunction with shaken baby syndrome. One of the more important concerns the question of whose intressen who should weigh heaviest – child's or care nadshavarens. Utifrån a child perspective so it can not be any doubt about that child's intressen have highest priority in several respects. Barnet has for the first a need of to get his injury examined and treated at a professional international and competent way in health and health care. If the damage can be suspected to be forcaused by assault , there is an apparent need forv of protection for their lives and their health.

It or the guardians, who are suspected to be caused to the shaken baby syndrome has on the other side a legitimate intresse of to not be judged innocent (a). Hare available , therefore a potential value conflicts that can be described as an ethical dilemma in the sense that it does not have any very trouble-free solution.

(D) etta dilemma can also be expressed in terms of risks for under the respective Islandverdiagnostik. Med elderly is not diagnosed means that children who actually suffered for shaken baby syndrome not get identified and thus not receive the society's protection against further abuse or lack of HEAäxtvillkor in General. Still underdiagno stik can take place on the grounds of lack of competence or observans in health and health care or to owill or inability of staff to on a professional way utr eda suspicions about the skakvage.

Colonel erdiagnostik can occur if the doctor who meets the child with the current triad at an immediate way to value this as a proof of that shaken baby syndrome and be shaken baby syndrome is the cause of the observed damage. (D) theet tube itself thus on a confusion between a hypothesis about a possible cause of the child's injuries and an alleged safe knowledge about that it is such a clear and safe coupling between cause and effect. A such a procedure creates thus risk for that the continuation of the proceedings in a such case mainly will be characterized by a so called validation approach (Meltzer et al. in press). Med this means that further measures are made only in order to confirm the hypothesis and that the information that would be able to falsify hypo thesis is not taken into account in sufficient extent.

Both under and over-diagnosing is deeply problematic from an ethical point of view. A överDiagnostics protects many children, both those with secured shaken baby syndrome that reason as a part of other. It leads , however, to the families being split of which some on the wrong basis. (A) tt separating children from their parents/guardians are a seriousarligt surgery which only should be made if the child runs the obvious risk to endanger badly in home met. Att , even the family's other children can be disposed of can further aggravate the situation.

The Wo dekonflikt as here outlined between the child's and guardian's/nas intressen needs related to the legal principle that No innocent person should be condemned for a brott. Colonelerdiagnostik of the shaken baby syndrome , to the impact that a number of children to be protected, of which a part really been exposed to shaken baby syndrome, but that this is happening to the price of that a number of parents/guardians become frihetsberIslandvade without to be own ga to any brott. Underdiagnostik of shaken baby syndrome joints on the other side of that child as father ill have to Live left in a harmful home environments with risk of future violent incidents.

The medical controversy around shaken baby syndrome that has been going on in Sweden as well as in large parts of the world is above all if it is a secured scientific scientific support for that symtomtriaden subdural haemorrhage, fundus hemorrhage and hjärnpåvyoucan be caused by shaken baby syndrome and only by skakvage. SBU's report shows that there is scientific evidence, if than limited, for the triad can be caused by shaken baby syndrome but that there also exist other diseases and events that can result in the triad , or parts of it.

(D) etta raises the question about what your doctor can and should pronounce itself on when it comes to shaken baby syndrome. Det are ethically of the utmost importance to physicians and other healthcare professionals are observant on damage of small children who can conceivably be caused by human hand despite the fact that the guardians are denying everything . (D)a clinical investigation and treatment of the damage must be carried out with full force. (F)he question is If the doctor in a slightly later stage can Express themselves with scientific certainty about the cause of the observed damage. The doctor has, as previously pointed out, a number of different information to take into account in this assessment of the possible causes of damage. Att on the basis of the mere presence of the triad rule to this with Security caused by shaken baby syndrome must , however, be regarded as incompatible with the authorities forl l oka ren's professional ethics as applicable regulations concerning attestation (Albert et al. 2012).

(D) etta the finding does not prevent that there can be reasons to do a so called oros notification trots this uncertainty because a child's need of protection is a broader issue than the about the cause of the current damage. Conclusions

S more have in this ethical analysis of shaken baby syndrome been deleted from SBUreport konsta Ting to the vetenskapliga dossier concerning shaken baby syndrome is limited. There is limited scientific support for that on the so called triad of sym tom or its subcomponents can be present at shaken baby syndrome but the report shows that there are differential diagnoses that also can give rise to the tre symptoms/findings in the triad.

(G) (iv) et this finding is so is the term shaken baby syndrome ethically problematic because it in itself includes an etiologic findings. Smore mean that it is ethically problematic to within health and medical care with Security set to some specific damage of small children per automatics are proof of that these arose mit through shaken baby syndrome. One such over-diagnosing of shaken baby syndrome should not be present when the scientific knowledge is so limited (Riggs & Hobbs 2011).

S more mean that it also is ethically problematic with the elderly is not diagnosed in bemär kelsen to kids who actually been vulnerable for shaken baby syndrome not get identified and understood within health and medical care. Denna risk can however be limited by improved professional development of child abuse in general and shaken baby syndrome in particular within both health and health care and social services.

Smer would point out the importance of the health and health care system takes into account the obligation that the social welfare report cases where children in any way suspected danger . (D) thenumber one applies in any particular case of suspicions of child abuse of various kinds. (H)health and care staff must be able to combine a high observans of suspected shaken baby syndrome with a caution when it comes to pronounce itself on the cause of the observed damage then the vetenskapliga knowledge does not allow safe conclusions on this area.

Smer recommends that the term shaken baby syndrome not used will continue on the basis of the likelihood of confusion between the hypothesis and secure knowledge (Christian & BCap 2009). An alternative term should be etiologic neutral.

References

Alber t DM, Weisberger Blanchard J & Knox AMONG OTHERS. Ensuring appropriate specialists,such testimony for cases involving the "shaken baby". JAMA 2012; 308: 39 40.

Christian CW & Block r. Abusive head traumas in infants and children. Pediatrics 2009: 123: 1409 1411.

Leuthner SR. Ethical challenges in the care of the shaken baby. Journal of Aggrprogression, Maltreatment & Traum(a) 2001; 5: 341 347.

L owenstein LF. RECt research and views on shaking baby Sinrome. International Journal of Psychiatry in Medicine 2004; 34: 131 141. Meltzer CC, Sze G, Rommelfanger KS, Kinlaw K, Banja JD & Wolpe PR. Guidelines for the ethical use of neuroimages in medical testimony: scratchest of a multidisciplinary conference. American Journal of Neuroradiology 2014; 35: 632 637.

Riggs JE & Hobbs Gr. innfant homicide and accidental death in the United States, 1940 2005: ethics and epidemiological classification. Journal of Medical Ethics 2011; 37: 445448.

The ethical analysis has been taken up by thengemar Engström, expert in Smore, in consultation with Kjell Asplund and Chatrine Pålsson Ahlgren.

(B) If the agency decision If this text was adopted at Statens medicalethical Council regular sam provision on 26 August 2016. In the decision attended members Kjell Asplund (Chairman), Finn Bengtsson, Sven Olov Edvinsson, Katie P Ahlgra, Åsa GybergK, Barbro westerholm and Anders Åkesson. During the preparation of the case took part also the expert Lars Berge Kleber, inngemar Engström, Göran , Hermerén, Ann j.Johansson, Olle Olaib, Bengt Rönngren, Nils Eric Sahlin, Anna 'singer and Elisabet Wennlund.

(F) ör the Council, the

kjEll a.s. O interview with Chairman in Statens medical ethical advice

SBU – The State's preparation for

medical and social evaluation

Web site: www.sbu.se • twitter: @SBU_se • phone: 08‐412 32 00

report number 255 • published 2016 • isbn 978‐91‐85413‐98‐0