BMJ -Editorial - The evidence base for shaken baby syndrome Shaken Baby - We need to question the diagnostic criteria

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http://bmj.bmjjournals.com/cgi/content/full/328/7442/719

BMJ 2004;328:719-720 (27 March), doi:10.1136/bmj.328.7442.719

Editorial

The evidence base for shaken baby syndrome

We need to question the diagnostic criteria

The phrase " shaken baby syndrome " evokes a powerful image of abuse, in

which a carer shakes a child sufficiently hard to produce whiplash forces

that result in subdural and retinal bleeding. The theory of shaken baby

syndrome rests on core assumptions: shaking is always intentional and

violent; the injury an infant receives from shaking is invariably severe;

and subdural and retinal bleeding is the result of criminal abuse, unless

proved otherwise.1 These beliefs are reinforced by an interpretation of the

literature by medical experts, which may on occasion be instrumental in a

carer being convicted or children being removed from their parents. But

what is the evidence for the theory of shaken baby syndrome?

Retinal haemorrhage is one of the criteria used, and many doctors consider

retinal haemorrhage with specific characteristics pathognomonic of shaking.

However, in this issue Lantz et al examine that premise (p 754) and

conclude that it " cannot be supported by objective scientific evidence. " 2

Their study comes hard on the heels of a recently published review of the

literature on shaken baby syndrome from 1966 to 1998, in which Mark Donohoe

found the scientific evidence to support a diagnosis of shaken baby

syndrome to be much less reliable than generally thought.3

Shaken baby syndrome is usually diagnosed on the basis of subdural and

retinal haemorrhages in an infant or young child,1 although the diagnostic

criteria are not uniform, and it is not unusual for the diagnosis to be

based on subdural or retinal haemorrhages alone.w1 The website of the

American Academy of Ophthalmology states that if the retinal haemorrhages

have specific characteristics " shaking injury can be diagnosed with

confidence regardless of other circumstances. " 4 Having reviewed the

evidence base for the belief that perimacular folds with retinal

haemorrhages are diagnostic of shaking, Lantz et al were able to find only

two flawed case-control studies, much of the published work displaying " an

absence of... precise and reproducible case definition, and interpretations

or conclusions that overstep the data. " 2 Their conclusions are remarkably

similar to those of Donohoe, who found that " the evidence for shaken baby

syndrome appears analogous to an inverted pyramid, with a very small

database (most of it poor quality original research, retrospective in

nature, and without appropriate control groups) spreading to a broad body

of somewhat divergent opinions. " 3 His work entailed searching the

literature, using the term " shaken baby syndrome " and then assessing the

methods of the articles retrieved, using the tools of evidence based

inquiry. Reviewing the studies achieving the highest quality of evidence

rating scores, Donohoe found that " there was inadequate scientific evidence

to come to a firm conclusion on most aspects of causation, diagnosis,

treatment, or any other matters, " and identified " serious data gaps, flaws

of logic, inconsistency of case definition. " 3

The conclusions of Lantz et al and of Donohoe make disturbing reading,

because they reveal major shortcomings in the literature relating to a

field in which the opportunity for scientific experimentation and

controlled trials does not exist, but in which much may rest on

interpretation of the medical evidence.5

If the concept of shaken baby syndrome is scientifically uncertain, we have

a duty to re-examine the validity of other beliefs in the field of infant

injury. The recent literature contains a number of publications that

disprove traditional expert opinion in the field. A study of independently

witnessed low level falls showed that such falls may prove fatal, causing

both subdural and retinal bleeding.6 w2 A biomechanical analysis validates

that serious injury or death from a low level fall is possible and casts

doubt on the idea that shaking can directly cause retinal or subdural

haemorrhages.7 w3 An important lucid interval may be present in an

ultimately fatal head injury in an infant.8 Neuropathological studies have

shown that abused infants do not generally have severe traumatic brain

injury and that the structural damage associated with death may be

morphologically mild.9 10 What is the relevance of the craniocervical

injuries to corticospinal tracts, dorsal nerve roots, and so on that have

been described?10 11 We do not know. What is the force necessary to injure

an infant's brain? Again, we do not know.

While most abused children indisputably show the signs of violence, not all

do. No one would be surprised to learn that a fall from a two storey

building or involvement in a high speed road traffic crash can cause

retinal and subdural bleeding, but what is the minimum force required? " It

is one thing clearly to state that a certain quantum of force is necessary

to produce a subdural hematoma; it is quite another to use examples of

obviously extreme force... and then suggest that they constitute the

minimum force necessary. " 12

Research in the area of injury to infants is difficult. Quality evidence

may need to be based on finite element modelling from data on infants'

skulls, brains, and neck structures, rather than living animals. Any

studies on immature animal models, if performed, will need to be validated

against the known mechanical properties of the human infant. Pending

completion of such studies, the reviews by Lantz and Donohoe are a valuable

contribution and provide a salutary check for anyone wishing to cite the

literature in support of an opinion. Their criticisms of lack of case

definition or proper controls can be levelled at the whole literature on

child abuse. If the issues are much less certain than we have been taught

to believe, then to admit uncertainty sometimes would be appropriate for

experts. Doing so may make prosecution more difficult, but a natural desire

to protect children should not lead anyone to proffer opinions unsupported

by good quality science. We need to reconsider the diagnostic criteria, if

not the existence, of shaken baby syndrome.

J F Geddes, retired (formerly reader in clinical neuropathology, Queen

, University of London)

London (j.f.geddes@...)

J Plunkett, forensic pathologist

Regina Medical Center, 1175 Nininger Road, Hastings, MN 55033, USA

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Editorial p 720 Clinical review p 754 Letters p 766 Personal view p 775

Additional references w1-w3 are on bmj.com

Competing interests: JFG and JP have given evidence in criminal cases at

the request of both the prosecution and the defence.

References

American Academy of Pediatrics Committee on Child Abuse and Neglect. Shaken

baby syndrome: rotational cranial injuries—technical report. Pediatrics

2001;108: 206-10.[Abstract/Free Full Text]

Lantz P, Sinai S, Stanton C, Weaver R. Perimacular retinal folds from

childhood head trauma: Case report with critical appraisal of current

literature. BMJ 2004;328: 754-6.[Free Full Text]

Donohoe M. Evidence-based medicine and shaken baby syndrome. Part I:

literature review, 1966-1998. Am J Forensic Med Pathol 2003;24:

239-42.[iSI][Medline]

American Academy of Ophthalmology. Shaken baby syndrome resources.

www.aao.org/aao/education/library/shaken_baby.cfm (accessed 25 Feb 2004).

Milroy CM. Medical experts and the criminal courts. BMJ 2003;326:

294-5.[Free Full Text]

Plunkett J. Fatal pediatric head injuries caused by short-distance falls.

Am J Forensic Med Pathol 2001;22: 1-12.[CrossRef][iSI][Medline]

Ommaya AK, Goldsmith W, Thibault L. Biomechanics and neuropathology of

adult and paediatric head injury. Br J Neurosurg 2002;16:

220-42.[CrossRef][iSI][Medline]

Denton S, Mileusnic D. Delayed sudden death in an infant following an

accidental fall. Am J Forensic Med Pathol 2003;24: 371-6.[iSI][Medline]

Geddes JF, Hackshaw AK, Vowles GH, Nickols CD, Whitwell HL. Neuropathology

of inflicted head injury in children. I. Patterns of brain damage. Brain

2001;124: 1290-8.[Abstract/Free Full Text]

Geddes JF, Vowles GH, Hackshaw AK, Nickols CD, IS, Whitwell HL.

Neuropathology of inflicted head injury in children. II. Microscopic brain

injury in infants. Brain 2001;124: 1299-306.[Abstract/Free Full Text]

P, CR, Deck J, Ang LC, Ho M, Becker L. Axonal injury and the

neuropathology of shaken baby syndrome. Acta Neuropathol 1998;95:

625-31.[CrossRef][iSI][Medline]

People v ez, 51 P3d 1046 ( 2001) (R'hrg den. 2002) (cert. granted,

2002).

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