NSIDRC Journal Article Alert — April 4, 2008
Prepared by the National Sudden Infant Death Resource Center
at Georgetown University.
This journal article alert provides selected items added to
the National Library of Medicine’s PubMed database in
the last week.
Past issues of NSIDRC journal alerts are available at http://www.sidscenter.org.
Availability of full-text journal articles is often limited to
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Sudden Infant Death
1: Sanatani S, Wilson G, Smith CR, Hamilton RM, Williams WG,
Adatia I.
Sudden unexpected death in children with heart disease.
Congenit Heart Dis. 2006 May;1(3):89-97.
Children's Heart Center, British Columbia's Children's Hospital,
Vancouver, BC, Canada.
OBJECTIVE: To review a mortality database, and identify all
sudden unexpected deaths in patients followed by the cardiac
program. DESIGN: Retrospective review of prospectively maintained
database. RESULTS: Over 8 years, we identified 80 sudden unexpected
deaths, among which there were sufficient data in 69 (24 females).
Patients died at a median age of 17.2 months (28 days-18.8
years). Forty-six patients had 2 functional ventricles and
23 had received palliation for a single-functional ventricle.
Patients with a single ventricle died at a younger age (median
120 days; 28 days-17.2 years) and sooner after last assessment
(median 27 days; 1-146 days) than patients in the biventricular
group (median age 2 years; 43 days-18.8 years; median time
since last assessment 49 days, 1 days-1 year) (P < .01;
P = .01). Thrombosis was the most common cause (61%) of death
in the single-ventricle group. Arrhythmia or presumed arrhythmia
was the most common cause (46%) of death in the biventricular
group. Fifty-one patients had undergone surgery. Six patients
had primary electrophysiological disease, and 5 had cardiomyopathy.
Eight deaths occurred in patients with pulmonary vascular disease.
CONCLUSION: Our study demonstrates that sudden unexpected death
occurred at a frequency of at least 10 patients per year over
an 8-year period with 55,730 patient encounters. We were able
to determine a clinical cause of death in most patients. Arrhythmias
(30%) and pulmonary vascular disease (13%) are important causes
of sudden death. Simple aortic valve disease and hypertrophic
cardiomyopathy are rare (4%) causes of sudden death in childhood.
Infants and young children with surgical shunts comprise 23%
of sudden unexpected deaths that occur within a month of the
last evaluation. Close surveillance of these patients is warranted.
2: Mitchell EA, Bajanowski T, Brinkmann B, Jorch G, Stewart
AW, Vennemann MM.
Prone sleeping position increases the risk of SIDS in the day
more than at night.
Acta Paediatr. 2008 Mar 28 [Epub ahead of print]
Department of Paediatrics, University of Auckland, New Zealand.
Background: SIDS mortality is higher during the night than
in the day. Aim: (1) To examine risk factors for SIDS by time
of day and (2) to see if the proportion of deaths at night
has changed from prior to the 'Back to Sleep' campaign, which
recommended infants sleep supine. Methods: A large population-based
SIDS matched case-control (GeSID) study conducted from 1998
to 2001 (when the prevalence of infants placed prone to sleep
was 4.1%). The reference sleep of the controls was matched
for the estimated time of death for the case. Risk factors
for SIDS were examined for night-time and day-time deaths.
The estimated time of death was compared with that from an
earlier study in Germany (1990-1994 when prevalence of prone
sleeping was 32.2%). Results: There were 333 SIDS cases and
998 matched controls. The increased risk with placed prone
to sleep was significantly different during the day [adjusted
OR = 18.15 (95% CI = 5.91-55.69)] compared with during the
night [adjusted OR = 3.49 (95% CI = 1.46-8.39; p-value for
interaction = 0.011)]. There was no significant difference
in the other risk factors examined by time of day in the multivariate
analysis. The mean time found dead was 09:07. In the earlier
study the mean time found dead was 08:54 and the difference
was not significant (p = 0.57). Conclusions: This study confirms
previous observations that prone sleeping position carries
a greater risk during the day than at night. However, the reduction
in infants sleeping prone has not been associated with a reduced
number of deaths in the day in Germany.
Other Infant Death
1: Oehmichen M, Schleiss D, Pedal I, Saternus KS, Gerling
I, Meissner C.
Shaken baby syndrome: re-examination of diffuse axonal injury
as cause of death.
Acta Neuropathol. 2008 Mar 26 [Epub ahead of print]
Institute of Legal Medicine, University Hospital of Schleswig-Holstein,
Campus Lübeck, Lübeck, Germany, moehmichen@gmx.de.
The discussion surrounding shaken baby syndrome (SBS) arose
from the lack of evidence implicating diffuse axonal injury
(DAI) as a cause of death. It was assumed instead that injury
to the cervical cord, medulla, and nerve roots played a causal
role. The present pathomorphological study examines 18 selected
infants (<1-year-old) whose deaths were highly suspicious
for SBS, exhibiting the classical SBS triad of acute subdural
hemorrhage (SDH), retinal bleeding, and encephalopathy. Gross
autopsy and microscopic findings of these infants were compared
with those of 19 victims of sudden infant death syndrome (SIDS;
control group 1) and of 14 infants who died of disease or injuries/violence
not involving the head, neck or eyes (control group 2). Symptoms
of mechanical impact to the head were evident in seven of the
SBS infants, but in none of the control infants. DAI was not
detected in either the SBS or control cases. Localized axonal
injury (AI) was regularly present in the brains of the SBS
infants surviving longer than 1.5-3.0 h, but only occasionally
in the craniocervical junction and within the nerve roots of
the upper cervical cord; it was never present in the medulla.
Epidural hemorrhage of the cervical cord was seen in four of
the ten examined SBS cases, but in none of the control cases.
Based on the absence of DAI in the brain and of signs of generalized
cervical cord or nerve root injuries, we conclude that the
cause of death in the SBS victims was a global cerebral ischemia
secondary to SDH, focal vasospasm, trauma-induced transitory
respiratory and/or circulatory failure.
Bereavement
1: J Palliat Med. 2008 May-Jun;11(3):444-50.
Related Articles, Links
Click here to read
Ethical and logistical considerations of multicenter parental
bereavement research.
Meert KL, Eggly S, Dean JM, Pollack M, Zimmerman J, Anand
KJ, Newth CJ, Willson DF, Nicholson C.
Department of Peditrics, Wayne State University, Children's
Hospital of Michigan, Detroit, Michigan.
ABSTRACT Background: Multicenter research has the potential
to recruit participants with diverse racial, ethnic, and geographic
backgrounds and is essential for understanding heterogeneity
in bereavement. The National Institute of Child Health and
Human Development Collaborative Pediatric Critical Care Research
Network (CPCCRN) is a multicenter network charged with conducting
research on the pathophysiology and management of critical
illness in childhood. Among its research activities, the CPCCRN
has undertaken research in parental bereavement because most
childhood deaths in the United States occur in hospitals, primarily
in critical care units. Objective: The purpose of this paper
is to discuss ethical and logistical issues found by the CPCCRN
to be problematic to multicenter research with bereaved parents
and to explore research strategies that may be practicably
implemented. Results: Ethical and logistical challenges encountered
by the CPCCRN included issues of privacy; confidentiality;
voluntariness; minimizing risks; working with multiple institutional
review boards; researcher qualifications, training and support;
and methods of data collection. Strategies to address these
challenges included local recruitment of participants; flexibility
in consent methods across sites; participant options for methods
of data collection; involvement of local bereavement support
services; central training of researchers with systematic monitoring
and opportunitieas for support; and use of a secure Web-based
collaborative workspace. Conclusions: Multicenter parental
bereavement research has distinct ethical issues that must
be addressed by the logistics of the research plan. Greater
attention to the issues identified may facilitate research
to reduce adverse mental and physical health outcomes in a
diverse population of bereaved individuals.
Miscarriage/Stillbirth/Prenatal Issues
1: Korteweg FJ, Bouman K, Erwich JJ, Timmer A, Veeger NJ,
Ravisé JM, Nijman TH, Holm JP.
Cytogenetic analysis after evaluation of 750 fetal deaths:
proposal for diagnostic workup.
Obstet Gynecol. 2008 Apr;111(4):865-74.
Departments of Obstetrics, Genetics, and Pathology, and the
Trial Coordination Centre, Department of Epidemiology, University
Medical Centre Groningen and University of Groningen, Groningen,
the Netherlands.
OBJECTIVE: To estimate success rates for cytogenetic analysis
in different tissues after intrauterine fetal death, and study
selection criteria and value of cytogenetic testing in determining
cause of death. METHODS: Cytogenetic analyses and the value
of this test in determining cause by a multidisciplinary panel
were studied in 750 fetal deaths. Morphologic abnormalities,
small for gestational age (SGA), advanced maternal age (older
than 35 years) and maceration were studied as selection criteria.
RESULTS: Chromosomal abnormalities were observed in 13% of
fetal deaths. Cytogenetic success rates were significantly
higher for invasive testing (85%) than for postpartum tissue
analysis (28%, P<.001). There were more abnormal chromosomes
(38%) in fetal deaths with morphologic abnormalities than in
those without (5%, P<.001). This was not observed for SGA
(16% compared with 9.2%, P=.22) or for advanced maternal age
(16.7% compared with 12.0%, P=.37). The posterior probability
of a chromosomal abnormality in the absence of morphologic
abnormalities was still 4.6%. Cytogenetic analysis was successful
in 35% of severely macerated fetuses. We do not advise using
these selection criteria, because the failure rate was high
on postpartum tissues. Cytogenetic analysis was valuable in
determining the cause in 19% of the fetal deaths. CONCLUSION:
Parents should be counseled on aspects of cytogenetic analysis
after fetal death. We advise performing nonselective invasive
testing after fetal death and before labor for all fetal deaths.
LEVEL OF EVIDENCE: II.
2: Weselak M, Arbuckle TE, Walker MC, Krewski D.
The influence of the environment and other exogenous agents
on spontaneous abortion risk.
J Toxicol Environ Health B Crit Rev. 2008 Mar;11(3-4):221-41.
McLaughlin Centre for Population Health Risk Assessment,
Institute of Population Health, University of Ottawa, Ottawa,
Ontario, Canada. mandy_weselak@hc-sc.gc.ca
It is estimated that close to 30% of all pregnancies end
in spontaneous abortion. Although about 60% of spontaneous
abortions are thought to be due to genetic, infectious, hormonal,
and immunological factors, the role of the environment remains
poorly understood. Pregnancy involves a delicate balance of
hormonal and immunological functions, which may be affected
by environmental substances. Many toxic substances that are
persistent in the environment and accumulate in the fatty tissues
may disrupt this equilibrium. This overview addresses known
risk factors for spontaneous abortions and examines the role,
if any, that environmental factors (chemical and physical)
may play in the etiology of this adverse health outcome.
3: Tursi A, Giorgetti G, Brandimarte G, Elisei W.
Effect of Gluten-Free Diet on Pregnancy Outcome in Celiac Disease
Patients with Recurrent Miscarriages.
Dig Dis Sci. 2008 Mar 27 [Epub ahead of print]
Digestive Endoscopy Unit, “Lorenzo Bonomo” Hospital,
Via Torino, 49, 70031, Andria, BA, Italy, antotursi@tiscali.it.
Purpose Available literature data show that celiac disease
(CD) is a frequent cause of recurrent miscarriage. However,
data are lacking for pregnancy outcome when the patient is
on a gluten-free diet (GFD). A case-control study about the
effect of GFD on pregnancy was conducted from 1995 to 2006.
A cohort of 13 women (mean age 32 years, range 22-38 years)
affected by CD with recurrent miscarriages was observed. In
all of them several causes of miscarriage (gynecological, endocrine,
hematological, etc.) were excluded. All patients were started
on a gluten-free diet and were reassessed throughout a long-term
follow-up period to evaluate the outcome of pregnancy. Results
Six of 13 became pregnant (46.15%) as follows: 1 patient (7.69%)
1 year after GFD was started, 3 patients (23.07%) 2 years after
GFD was started, 1 patient (7.69%) after 3 years, and finally
1 (7.69%) 4 years after GFD was started. Moreover, two patients
(16.66%) had multiple pregnancies (one had had two childbirths
and another had undergone three births within a 7-year follow-up
period under GFD). Conclusions GFD seems to favor a positive
outcome of pregnancy in most CD patients with recurrent miscarriage.
4: Cherry N, Shaikh K, McDonald C, Chowdhury Z.
Stillbirth in rural Bangladesh: arsenic exposure and other
etiological factors: a report from Gonoshasthaya Kendra.
Bull World Health Organ. 2008 Mar;86(3):172-7.
Community and Occupational Medicine Program, University of
Alberta, Edmonton, AB, Canada.
OBJECTIVE: To use data collected by Gonoshasthaya Kendra,
a large nongovernmental organization providing health care
to some 600 villages, to describe the epidemiological pattern
of stillbirth and any additional contribution made by arsenic
contamination of hand-pump wells in Bangladesh. METHODS: Completed
pregnancies and outcomes (n = 30 984) for two calendar years,
together with existing data on 26 socioeconomic and health
factors were selected for study. The health care in these villages
was administered from 16 geographical centres; information
on the average arsenic concentration in each centre was obtained
from the National Hydrochemical Survey. After univariate analysis,
a multivariate, multilevel, logistic model for stillbirth was
developed. The additional effect of arsenic was calculated
having adjusted for all potential confounders thus identified.
FINDINGS: The overall stillbirth rate was 3.4% (1056/30 984)
and increased with estimated arsenic concentration (2.96% at < 10
microg/l; 3.79% at 10 microg/l to < 50 microg/l; 4.43% at > 50
microg/l). Having adjusted for 17 socioeconomic and health
factors, the odds ratios estimated for arsenic (with < 10
microg/l as reference) remained raised: 1.23 (95% confidence
interval, CI: 0.87A1.74) at 10 microg/l to < 50 microg/l
and 1.80 (95% CI: 1.14A2.86) at 50 microg/l or greater. CONCLUSION:
A increased risk of stillbirth is associated with arsenic contamination.
This risk, substantial enough to be detected by an ecological
approach and not readily attributable to unmeasured confounding,
is essentially preventable and all efforts must be made to
protect women at high risk.
5: Joseph KS.
The fetuses-at-risk approach: clarification of semantic and
conceptual misapprehension.
BMC Pregnancy Childbirth. 2008 Mar 26;8(1):11 [Epub ahead of
print]
ABSTRACT: BACKGROUND: Although proponents of the fetuses-at-risk
approach describe it as a causal model that resolves various
conundrums, several areas of semantic and conceptual misapprehension
remain. Differences in terminology include use of denominators
such as 'ongoing pregnancies' and the need for an ad hoc 'correction
factor' in order to calculate gestational age-specific rates.
Further, there is conceptual disagreement regarding the proper
candidates for neonatal death and related phenomena. Perhaps
the most egregious misconception is the belief that rising
rates of gestational age-specific perinatal mortality observed
under the fetuses-at-risk model automatically imply the need
for indiscriminate increases in iatrogenic preterm delivery.
DISCUSSION: The term 'fetuses at risk' addresses the plurality
of candidates for stillbirth in a multi-fetal pregnancy, while
the use of standard terminology such as 'cumulative incidence'
and 'incidence density' harmonizes the language of perinatal
epidemiology with that used in the general epidemiologic literature.
On the conceptual side, it is necessary to integrate clinical
insights regarding latent periods into models of neonatal morbidity
and mortality. The contention that the fetuses-at-risk approach
implies the need for indiscriminate iatrogenic preterm delivery
is a non-sequitur (just as rising age-specific cancer death
rates do not imply the need for routine chemotherapy and radiation
for all middle aged people). Finally, the traditional and fetuses-at-risk
models are better viewed in terms of function as prognostic
(non-causal) and causal models, respectively. CONCLUSIONS:
A careful examination of terms and concepts helps situate the
traditional perinatal and the fetuses-at-risk approaches within
the broader context of non-causal and causal models within
general epidemiology.
6: Jorge O, Jorge A, Camus G.
Celiac disease associated with antiphospholipid syndrome.
Rev Esp Enferm Dig. 2008 Feb;100(2):102-103.
Introduction: celiac disease may be associated with pathologies
of immune etiology. We present its association with antiphospholipid
syndrome.Case 1: a 26-year-old female was diagnosed with celiac
disease. Six months later she became pregnant, and experienced
fetal death. The following year she became pregnant again.
IgG anticardiolipin antibodies: 20 GPL U/ml (normal value < 11),
and IgM anticardiolipin antibodies: 9 MPL U/ml (n. v. < 10).
Hematological tests were otherwise uneventful. Medicated with
acetylsalicylic acid she had a normal pregnancy.Case 2: a 48-year-old
female diagnosed with celiac disease presented with thrombosis
in her left lower limb and renal infarction. Hematological
tests showed no prothrombotic alterations (antiphospholipid
antibodies were not measured). A year and a half later she
had thrombosis in a finger of her hand. IgG anticardiolipin
antibodies: 10 GPL (n. v. < 13), and IgM anticardiolipin
antibodies: 35 MPL (n. v. < 12).Case 3: a 38-year-old female
was diagnosed with celiac disease. Some time later she experienced
two spontaneous abortions and a transient ischemic cerebral
attack. Nowadays, she is in her sixth month of pregnancy. IgM
anticardiolipin antibodies: 75 MPL/ml (n. v. up to 20), and
IgG anticardiolipin antibodies within normal values. Hematological
tests revealed no other prothrombotic alterations.Discussion:
antiphospholipid syndrome is characterized by arterial and
venous thrombosis, and spontaneous fetal death. Its association
with celiac disease has been described in few cases. Celiac
disease is associated with spontaneous fetal death; consequently,
we hypothesize that antiphospholipid syndrome may be one of
the causes for this event.
7: Vaktskjold A, Talykova LV, Chashchin VP, Odland JØ,
Nieboer E.
Spontaneous abortions among nickel-exposed female refinery
workers.
Int J Environ Health Res. 2008 Apr;18(2):99-115.
Institutt for samfunnsmedisin, Universitetet i Tromsø,
Tromsø, Norway.
A case-control study to investigate whether women employed
in nickel-exposed work areas in early pregnancy are at elevated
risk of spontaneous abortion (SA). Data about pregnancy outcome
and maternal factors were obtained about each delivery and
SA from women in selected work places. Each pregnancy record
was assigned a categorical nickel (Ni) exposure rating according
to the women's occupations at pregnancy onset. The guidelines
were the water-soluble Ni subfraction of the inhalable aerosol
fraction obtained by personal monitoring for nickel- and copper-refinery
workers or/and measured urinary-Ni concentrations. The unadjusted
odds ratio for the association between the maternal exposure
to Ni and an SA for Ni-exposed women was 1.38 (95% confidence
interval: 1.04-1.84), and the adjusted was 1.14 (0.95-1.37).
In conclusion, there was no statistical association between
maternal occupational exposure to water-soluble Ni in early
pregnancy and the risk of self-reported SA. The findings do
not exclude the possibility of a weak excess risk, or a risk
in the first weeks of pregnancy.
8: Taylor MY, Wyatt-Ashmead J, Gray J, Bofill JA, Martin RW,
Morrison JC.
Pregnancy loss after first trimester viability in women with
sickle cell trait: a preliminary report.
South Med J. 2008 Feb;101(2):150-1.
Department of Obstetrics and Gynecology, University of Mississippi
Medical Center, Jackson, Mississippi, USA.
BACKGROUND: Traditionally, sickle cell trait has not been
associated with a higher risk of fetal death, but we noted
several, which led us to assess all such pregnancies. METHODS:
In this retrospective study, 131 patients with sickle cell
trait were analyzed over a two-year period. The Institutional
Review Board approved the collection of deidentified data.
RESULTS: Subjects were African-American with an average age
of 23.9 years, and average gestational age at delivery of 30.1
weeks. There were 10 (8.13%) intrauterine fetal deaths (IUFDs),
and one neonatal death. Ascending amniotic fluid infection
was noted in 50% and 92% meconium histocytes. All placentas
had sickling in the intervillous space and the decidual vessels.
CONCLUSIONS: Sickling in the decidual vessels and poor placental
perfusion may play a role in pregnancy loss in excess of what
has previously been reported. A cohort control study appears
to be in order. NARRATIVE: Pregnant women with sickle cell
trait are thought not to have increased maternal or fetal mortality/morbidity.
Over a two year period, we studied 131 women with this hemoglobinopathy
and found that 10.6% had intrauterine growth retardation (IUGR),
8.4% preterm premature rupture of the membranes, 8.1% intrauterine
fetal demise (n = 10) at most occurring at 16 to 24 weeks,
and one neonatal death. Amniotic fluid infection was noted
in 50%, and meconium histocytes indicating intrauterine hypoxia
were noted, as was unsuspected sickling in the placental vasculature.
Based on this case series, sickle cell trait may not be as
benign for the fetus as was previously thought.
9: Vantyghem MC, Vincent-Desplanques D, Defrance-Faivre F,
Capeau J, Fermon C,
Valat AS, Lascols O, Hecart AC, Pigny P, Delemer B, Vigouroux
C, Wemeau JL.
Fertility and obstetrical complications in women with LMNA-related
familial partial lipodystrophy.
J Clin Endocrinol Metab. 2008 Mar 25 [Epub ahead of print]
Endocrinology and Metabolism Department, Lille University
Hospital, 59037 Lille France; INSERM U859, Lille 59037, France;
UPMC Univ Paris 06, UMR_S 893Eq9, F-75012, Paris, France; AP-HP,
Hôpital Tenon, Service de Biochimie et Hormonologie,
F-75020, Paris, France; INSERM, UMR_S 893Eq9, F-75012, Paris,
France; Diabetology Department, Lille University Hospital,
59037 Lille France; Obstetrics Department, General Hospital,
Lens 62 300, France; AP-HP, Hôpital Saint-Antoine, Département
de Biologie Moléculaire, F-75012, Paris, France; Endocrinology
Department, Reims University Hospital, Reims 51092, France;
Biochemistry and Hormone Department, Lille University Hospital,
59037 Lille, France.
Objective: Familial Partial Lipodystrophy due to LMNA (lamin
A/C) mutations (FPLD2) is a rare disorder characterized by
a selective loss of adipose tissue and insulin resistance.
Dyslipidemia and severe diabetes often occur during its evolution.
Only isolated and contradictory case reports have been published
on the obstetrical prognosis in lipodystrophy. The aim of our
study was to compare the fertility and occurrence of obstetrical
complications of women with FPLD2 with those of non-affected
relatives, women from the general population and women with
polycystic ovary syndrome (PCOS). Material and methods: Data
was obtained from clinical follow-up of seven families with
patients exhibiting mutations in LMNA (5 R482W, 1 R482Q, 1
R439C) (14 affected among 48 women). Results: The mean number
of live children per woman was 1.7 in affected patients vs.
2.8 in non-affected relatives. Fifty-four percent of LMNA-mutated
women exhibited a clinical phenotype of PCOS, 28% suffered
from infertility, 50% experienced at least one miscarriage,
36% developed gestational diabetes and 14% experienced eclampsia
and fetal death. Mean blood leptin level was significantly
lower in LMNA-mutated patients than in non-affected relatives
(5.0+/-3.8 ng/ml vs 14.3+/-3.6; p<0.001) despite similar
BMI (21.0+/-4.2 vs 22.4+/-2.2; p=0.49). Conclusion: In these
LMNA-linked lipodystrophic patients, the prevalence of PCOS,
infertility and gestational diabetes was higher than in the
general population. Moreover, the prevalence of gestational
diabetes and miscarriages was higher in lipodystrophic LMNA-mutated
women than previously reported in PCOS women with similar BMI.
Women with lipodystrophies due to LMNA mutations are at high
risk of infertility, gestational diabetes and obstetrical complications
and require reinforced gynecological and obstetrical care.
Prepared by the
National Sudden Infant Death Resource Center
Georgetown University
2115 Wisconsin Avenue, N.W., Suite 601
Washington, DC 20007
(866) 866-7437 toll free
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