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C J Bacon, West Yorkshire Braithwaite, and E N Hey
Uncertainty in classification of repeat sudden unexpected infant deaths in Care of the Next Infant programme
BMJ 2007; 335: 129-131 [Full text]
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Rapid Responses published:

[Read Rapid Response] Sudden Infant Death Explained.
Michael D Innis   (26 July 2007)
[Read Rapid Response] Re: Sudden Infant Death Explained.
Raymond G Holder   (26 July 2007)
[Read Rapid Response] Re: Re: Sudden Infant Death Explained.
Michael D Innis   (27 July 2007)
[Read Rapid Response] Re: uncertainty in classification of repeat sudden unexpected infant deaths in Care of Next Infant programme
Robert G. Carpenter, Alison Waite, Robert Coombs, Charlotte Daman-Willems, Angela McKenzie, Jonne Huber   (3 August 2007)
[Read Rapid Response] Adverse reaction to vaccine as a category in cot death
Lisa C Blakemore-Brown   (20 August 2007)
[Read Rapid Response] Uncertainty in classification of repeat sudden unexpected deaths
Christopher Bacon, Edmund Hey   (20 August 2007)

Sudden Infant Death Explained. 26 July 2007
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Michael D Innis,
Director Medisets Internatinal
Home 4575

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Re: Sudden Infant Death Explained.

Editor,

Dr Bacon and Dr Hey say, “since distinction between sudden infant death syndrome and covert homicide can be equally difficult, it seems illogical to exclude covert homicide when there is not enough information to exclude a specific natural cause”[1], and conclude that when rib fractures are found in addition to a natural disorder, the natural disorder cannot be assumed to be the cause of death."

And, “most rib fractures in babies are posterior and indicated abuse as may some anterior fractures.”

They are referring to the postmortems of 46 infants carried out between 1989 and 1999. It would appear it was for these reasons they concluded that 12, and not 6, of the 46 deaths reported should have been included in the “probably homicide” group.

Dr Bacon and Dr Hey, and for that matter, most previous reports, from Caffey [2] to the present [3] have ignored or dismissed the possibility of a deficiency of either Vitamin C or K in these deaths.

I have previously pointed out[4], and again submit, Richard Follis Jr reported three “Sudden Death In Infants with Scurvy” and Dr Kalokerinos eliminated Sudden Death In Infants {SIDS} in his practice by administering Vitamin C prior to immunization.

Sally Clark was the latest victim of the medical profession’s reluctance or inability to understand that immunization can cause Sudden Infant Death from a precipitous fall in the blood levels of Vitamin C. Her son Harry died within a few hours of being immunized.

Both Vitamin C and Vitamin K deficiencies can cause fractures[5,6] and the claim that “posterior rib fractures indicate abuse” is speculative and has no place in a scientific discussion which involves the lives of innocent care givers.

If the sudden deaths of infants were examined more diligently, especially looking for evidence of nutritional deficiencies, fewer would be “unexplained.”

Specifically, no charge of homicide should be made in cases of sudden infant death unless the blood levels of Vitamin C, Histamine [7], Tryptase [8] , Protein Induced by Vitamin K Absence/ Abnormality (PIVKA- II) AND Undercarboxylated Osteoclcin have been shown to be normal.[9]

Any, or all the above, may be abnormal in SIDS and in the so-called Shaken Baby Syndrome (SBS). Haemorrhagic Disease of the Newborn may present as SBS[10].

Michael D Innis MBBS, DTM&H, FRCPA, FRCPath

Rererences:

1. Bacon CJ, Hey EN. Uncertainty in classifiv=ction of repeated sudden unexpected infant deaths in Care of the Next Infant programme. BMJ 2007:235;129-131

2. Caffey J, "Multiple fractures in the long bones of infants suffering from subdural hematoma." Am J Roentgenol, 1946, 56: 163-173.

3. Carpenter RG, Waite A, Coombs RC, et al; Repeat unexpected and unexplained infant deaths: natural or unnatural? Lancet 2005:365; 29-35

4. Innis MD Sudden Infant Death - Revisited http://bmj.com/cgi/eletters/328/7451/1309#169107, 19 Jun 2007

5. Nelson’s Texbook of Pediatrics. 17th Edition. Edited by Behrman RE, Kliegman RM, Jenson HB p 185 Publisher Saunders

6. DeRusso PA, Spevak MR Schwartz KB. Fractures in Biliary Atresia Misinterpreted as Child Abuse. PEDIATRICS Vol 112. No1 July 2003:pp185-188

7. Clemetson CAB. Vitamin C Vol III p 11 CRC Press Inc Boca Raton Florida

8 Zinka B, Rauch E, Buettner A, Ruëff F, Penning R. Unexplained cases of sudden infant death shortly after hexavalent vaccination. Vaccine. 2006; Jul 26;24(31-32):5779-80.

9. Conway SP. Vitamin K in cystic fibrosis. J R Soc Med 004;97(Suppl.44):48-51

10. Rutty GN, Smith CM, Malia RG. Late Form Hemorrhaagic Disease of the Newborn. A Fatal Case Report with illustrations of Investigations That May Assist in Avoiding the Mistaken Diagnosis of Child Abuse. Amer J Forensic med Pathol. 1999;20(1):48-51

Competing interests: I have given evidence for the Defence in cases of alleged child abuse and been paid for my service.

Re: Sudden Infant Death Explained. 26 July 2007
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Raymond G Holder,
Retired engineer
BH9 3NF

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Re: Re: Sudden Infant Death Explained.

I have taken an interest in SID syndrome since becoming aware of the deficiency of L Carnitine in my own case, due to post polio and statin damage, and reading Fernando Scaglia's paper Carnitine Deficiency, obtainable easily via Google. He is an Assistant Professor in pediatrics, and quotes all the measures necessary to deal with carnitine deficiency, sometimes genetic, in the newborn, often resulting in SID, and so likely to affect other siblings. His earlier version of the paper quoted tests etc to be made to avoid the possibility of legal claims by the parents.

Carnitor is shown in the BNF for primary or secondary carnitine deficiency, but the details of secondary deficiency omit the effect of surgery, valproate, penicillin, post polio and statins from the causes. It is not easily obtainable here in a reasonably priced form, the expensive packaging of Carnitor is unnecessary for normal adult use, being seven times the price which I pay for mine.

Competing interests: None declared

Re: Re: Sudden Infant Death Explained. 27 July 2007
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Michael D Innis,
Director Medisets International
Home 4575

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Re: Re: Re: Sudden Infant Death Explained.

Editor,

Raymond Holder rightly reminds us that Carnitine deficiency may cause Sudden Infant Death and indeed in Medium-chain acylCoA dehydrogenase (MCAD) deficiency, a known cause of SIDS, both plasma and tissue concentrations of Carnitine are reduced to 25-50% normal. [1].

Vitamin C is an essential cofactor for the biosynthesis of carnitine (http://www.eurekalert.org/pub_releases/2006-04/foas-vcd040306.php) and this is further support for the crucial role of Vitamin C in the Sudden Infant Death Syndrome..

Michael Innis

Reference:

1. Nelson Textbook of Pediatrics 17th Edition p 433.

Competing interests: As previously declared

Re: uncertainty in classification of repeat sudden unexpected infant deaths in Care of Next Infant programme 3 August 2007
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Robert G. Carpenter,
Hon. Professor, Medical Statistics Unit, London School of Hygiene & Tropical Medicine, London
19 Lansdowne Avenue, Orpington, Kent BR6 8JT,,
Alison Waite, Robert Coombs, Charlotte Daman-Willems, Angela McKenzie, Jonne Huber

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Re: Re: uncertainty in classification of repeat sudden unexpected infant deaths in Care of Next Infant programme

Sir,

On January 1st 2005 (1)we reported that in a review of 46 first deaths on the Care OF Next Infant, CONI, programme, i.e., the second unexpected infant death in the family, 87% (95% CI 74–95%) were natural and 13% (95%CI 5–26%) were unnatural1. The confidence limits indicate uncertainty that may be due to sampling error. These conclusions were based on detailed reviews of 29 cases, and, when these were not possible, the conclusions of the usual registration and judicial procedures. We appreciate that when the cause of death is unknown any classification may be subject to error.

Bacon(2) suggested that our report was too black and white and that it would have been more scientific to have included an element of uncertainty into our conclusions by classifying some cases as grey. Bacon and Hey have developed this idea further by reclassifying 43% of our cases as ‘undetermined’(3). This ill-defined category derived from unspecified experience is meant to imply uncertainty as to whether maltreatment may have been the cause or a contributory factor in the death. Bacon and Hey accept that cases we classified as unnatural were probably unnatural. They then dismiss the conclusions of standard proceedings and reclassify ALL the cases that we were unable to review as ‘undetermined’.

In 2005 we wrote “Although the infants in these 18 families categorised as having two cases of SIDS were at high risk, in no case was there evidence of abuse at autopsy, and enquiries and case discussions gave no grounds for us to think that any of these deaths were unnatural.” Bacon and Hey3 write with regard to these cases “ We do not suggest that violence in the family, for example, necessarily implies that the baby’s death was unnatural; but we think that when a family has two unexplained deaths this possibility at least has to be considered and may sometimes be true.” This possibility was why Professor Emery, who was one of the first to suggest that some cot deaths might not be natural, initiated the case reviews which included a home interview with the parents and concluded with a case conference. Emery personally conducted at least two thirds of these interviews and trained those, RCC and CDW, who took over from him. To imply that we did not consider that these deaths might have been unnatural contradicts the plain meaning of the text.

Bacon and Hey (3) note that features reported in these 18 cases could be associated with SIDS or covert homicide. Those conducting the case conferences were fully aware of this. Risk factors such as smoking and bed sharing are not diagnostic. Objective assessment of the implications of other factors that we reported, such as violent family relationships and father in prison, requires knowledge of their prevalence in SIDS cases, cases of covert and overt homicide, and in the relevant normal population. Powell (4) found predictors of abuse more prevalent in the community than might be expected. In the absence of such control information we reported the considered conclusions of the cases conferences.

We agree that professionals should keep an open mind in assessing unexplained infant deaths and that such deaths be submitted to full investigation as described in our Lancet report and is now recommended in Working Together to Safeguard Children.(5). However, as noted by the Foundation for the Study of Infant Deaths, the use of a third category ‘undetermined’ goes against the recommendations of the 2004 report of the panel of The Royal College of Pathologists and The Royal College of Paediatrics and Child Health chaired by Baroness Helena Kennedy (6).

R.G. Carpenter,
A. Waite,
R. C. Coombs,
C. Daman Willems
A. McKenzie,
J. Huber.

References

1. Carpenter RG, Waite A, Coombs RC, Daman-Willems C, McKenzie A, .Huber J, Emery JL. Repeat unexpected and unexplained infant deaths: natural or unnatural? Lancet 2005;365:29-35.

2. Bacon C. Repeat sudden unexpected infant deaths. Lancet 2005;365:1137.

3. Bacon CJ, Hey EN. Uncertainty in classification of repeat sudden unexpected infant deaths in Care of the Next Infant programme. BMJ 21 July 2007. 335: 130–133.

4. Powell J. An evaluation of a risk related intervention programme to reduce the rate of possibly preventable postperinatal deaths (including sudden infant deaths) in Portsmouth and South East Hampshire Health District. University of Southampton, Faculty of Medicine. PhD Thesis, 1991.

5. HM Government. Working Together to Safeguard Children: A guide to inter-agency working to safeguard and promote the welfare of children. Chapter 7. TSO (The Stationery Office), 2006, Norwich.

6. Lee JA. Sudden unexpected death in infancy: The report of a working group convened by The Royal College of Pathologists and The Royal College of Paediatrics and Child Health, chaired by The Baroness Helena Kennedy QC. www.rcpath.org and www.rcpch.ac.uk. 2004.

Competing interests: None declared

Adverse reaction to vaccine as a category in cot death 20 August 2007
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Lisa C Blakemore-Brown,
Psychologist
UK

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Re: Adverse reaction to vaccine as a category in cot death

I would like it explained to me why iatrogenic abuse is not considered as part of the differential diagnosis when considering cot deaths?

Given that committees such as the JCVI (Joint Committee on vaccination and Immunisation) and its sub committee the ARVI (Adverse Reactions to Vaccines and Immunisation)have regularly sat to discuss adverse reactions including deaths, in order to advise Government, why do we never see it mentioned when mothers are being accused of murdering their children or when papers are written about how to categorise cot deaths?

Irrespective of how rarely this happens, if it is NEVER mentioned it leaves one suspecting that it is perhaps a serious issue which is being covered up.

Surely honesty is the best policy?

If the data which formed the basis for the original Emery and Carpenter paper could be opened up to proper scrutiny, as Bacon and Hey have based their paper on that paper, perhaps an additional category of adverse reaction to vaccine could then be included to reduce the risks that some mothers (and fathers) are being wrongly accused - and convicted - of killing their infants.

Competing interests: I write about these issues without payment

Uncertainty in classification of repeat sudden unexpected deaths 20 August 2007
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Christopher Bacon,
Retired paediatrician
Yorkshire,
Edmund Hey

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Re: Uncertainty in classification of repeat sudden unexpected deaths

Re: Uncertainty in classification of repeat sudden unexpected deaths in Care of Next Infant programme

Christopher Bacon, Edmund Hey, retired paediatricians, Yorkshire.

Sir,

The reasoned response that our article drew from Robert Carpenter and colleagues clearly deserves a reply. They point out that most of the repeat deaths they classified as natural had been subjected to a case review and that no evidence of maltreatment had emerged. Although case review is rightly recommended as an important part of the investigation of any unexpected infant death, it is an unreliable diagnostic tool: in the absence of hard evidence judgements are ultimately subjective, and therefore potentially variable and fallible. Their variability is shown by the contrast between the findings of the multicentre study of post neonatal mortality conducted in the 1970s, which did not consider any of 293 cases of SIDS to be suspicious [1], and the findings of the CESDI study conducted in the 1990s, which concluded that maltreatment may have played some part in 50 (14.5%) of 346 cases [2]. The potential fallibility of case reviews is illustrated by the family in Carpenter’s paper that had three deaths, the first two of which were classified as natural on initial review, with a later change to unnatural when the father confessed to killing the third baby [3]. It is one thing to say there is no definite evidence of maltreatment. It is quite another to say the death must therefore have been natural. We agree with Carpenter that accurate assessment requires better knowledge of the factors associated with natural and unnatural deaths. This will be hard to establish. Meanwhile sometimes the only tenable conclusion for a case review may be uncertainty.

Carpenter criticises our use of the category “undetermined” on the grounds that it conflicts with the recommendation of the Kennedy committee that coroners should generally avoid use of the term “unascertained” when classifying unexplained infant deaths [4]. However Kennedy conceded that the term “unascertained” might still be appropriate “when no sufficient cause of death has been established but there are gaps in the documentation, or for other reasons the death, whilst not shown to be due to abuse or neglect, does not meet the definition of SIDS.” We think that the deaths we classified as “undetermined” come within these criteria, and that the proportion of repeat deaths we put in this category was not excessive.

We accept, of course, that the large majority of all cot deaths are natural and that parents should not have to suffer unjustified suspicion. On the other hand if all deaths in which there is suspicion but no clear evidence of maltreatment are firmly labelled as natural, it will be harder to intervene where a parent has in fact been in some way responsible for the death. The obligation to protect other children may then go by default. By intervention we do not mean prosecution, which is rarely appropriate, nor necessarily removal of a future baby, but we would include measures such as recognition and treatment of mental disorder, and help, advice and practical support with the care of any other children. Finding the right balance between the need to support bereaved families and the need to protect children is extraordinarily difficult, and we fear that the paper by Carpenter and colleagues may result in insufficient attention to the latter.

Carpenter, as a statistician, questions the experience on which our opinions are based. Like him we have both taken part in surveys of death in infancy. As a basis for this, during our careers as clinicians we were directly involved in the investigation of infant deaths from all causes, in the support of families, and in the identification of abuse. We suggest that lack of clinical experience in child abuse may make it harder to comprehend its full reality.

C.J.Bacon
E.N.Hey

References

1, Knowelden J, Keeling J, Nicoll JP. A multicentre study of post neonatal mortality. Medical Care Research Unit, Sheffield 1984.

2. Fleming P, Blair P, Bacon C, Berry J. Sudden unexpected deaths in infancy: the CESDI SUDI studies 1993-96. Stationery Office, London 2000.

3. Carpenter RG, Waite A, Coombs RC, et al. Repeat unexpected and unexplained infant deaths: natural or unnatural? Lancet 2005; 365: 29-35.

4. Sudden unexpected deaths in infancy: the report of a working group convened by The Royal College of Pathologists and The Royal College of Paediatrics and Child Health, Kennedy H chair. Royal College of Pathologists 2004. www.rcpath.org .

Competing interests: None declared