The incidence of gastroschisis
BMJ 2006; 332 doi: https://doi.org/10.1136/bmj.332.7536.250 (Published 02 February 2006) Cite this as: BMJ 2006;332:250
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EDITOR
Kilby says that gastroschisis shows an increasing temporal trend in the number
of affected babies born in United Kingdom (1). This increasing trend has been
observed also in other parts of the world (2).
We have evaluated the data of 25
International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR)
Members Registries with more than seven years of data, a homogeneous
ascertainment and reliable data on termination of pregnancies (3). Fourteen
registries showed a significant increasing temporal trend of gastroschisis (see
table). No similar trend was observed in the 36 malformations analyzed in the
ICBDSR dataset. The possible bias of a simultaneous decreasing trend in
omphalocele indicating changes in reporting nomenclature or coding was excluded.
The increasing trend of gastroschisis is worldwide (namely Japan, Australia,
North-Central-South America, North-Central Europe), but not universal. For
example in Italy four regional birth defects registries (all with a rate ranging
between 0.4 and 0.8 per 10,000) did not experience any increase in the last 25
years.
Until now, no explanation of this
worldwide epidemic has been given. Any possible explanation should consider the
rate variation around the world, the increasing rate experienced worldwide but
not universally and the consistent increased risk in young mothers found in all
studies performed. Etiological research should be concentrated on large enough
material such as only the international collaboration can provide. The trouble
is that almost all birth defects registries are experiencing a chronic lack of
funds and no such study can be planned.
Public health
initiatives in developing and developed countries should include more funds on
research, prevention and treatment of birth defects. When the etiology is
unknown, the only sensible way towards prevention is research.
Prof
Pierpaolo Mastroiacovo MD,
Director
Alessandra
Lisi,Statistician
Centre of the
International Clearinghouse for Birth Defects Surveillance and Research
Via Pilo Albertelli 9,
00195, Roma, Italy. E-mail
icbd{at}icbd.org
Prof
Eduardo E Castilla, MD, Coordinator
ECLAMC,
Departamento de Genetica, Instituto Oswaldo Cruz, Caixa Postal 926, 20001-970,
Rio de Janeiro, and Cemic, Buenos Aires, Argentina. E-mail castilla {at} centroin.com.br
References
- Kilby DM.
The incidence of gastroschisis. BMJ
2006, 332:250-1 (February 4) - Di Tanna
GL, Rosano A, Mastroiacovo P. Prevalence of gastroschisis at birth:
retrospective study. BMJ 2002, 325:1389-90. - ICBDSR
Annual Report 2005. International Clearinghouse for Birth Defects Surveillance
and Research Centre, Roma, Italy www.icbd.org
|
Table. |
|||
|
|
Rate x 10,000 |
|
|
|
First 3 year |
Last 3 year |
P |
|
|
Japan |
0.96 |
2.58 |
*** |
|
|
1.53 |
4.30 |
*** |
|
|
0.71 |
2.44 |
*** |
|
Canada |
1.57 |
3.53 |
*** |
|
USA |
0.85 |
2.48 |
* |
|
Mexico |
1.44 |
5.11 |
*** |
|
South |
0.04 |
2.92 |
*** |
|
Norway |
1.34 |
2.74 |
*** |
|
|
1.70 |
3.73 |
*** |
|
|
0.13 |
2.05 |
*** |
|
|
1.52 |
2.05 |
*** |
|
France |
0.18 |
3.44 |
*** |
|
France: |
0.42 |
1.60 |
*** |
|
Slovak |
0.55 |
1.10 |
* |
|
P of Chi |
|||
Competing interests:
None declared
Competing interests: No competing interests
Is the birth prevalence of Gastroschisis also increasing in Spain among infants of young mothers?
To the Editor –
Recently, Kilby(1) attracts attention to the increasing incidence of
gastroschisis over time in the UK, particularly among babies of young
mothers. This has also been stressed by Donaldson(2), and also by
Mastroiacovo et al.(3) in fourteen registries from 4 continents in their
response to Kilby(1).
We would like to add our experience in the Spanish Collaborative
Study of Congenital Malformations (ECEMC), an ongoing case-control study
and surveillance system(4-6). As a part of the surveillance of congenital
anomalies routinely performed in the ECEMC, we have also observed an
apparent increasing trend in the birth prevalence of gastroschisis in
Spain among mothers aged under 20, not observed among older mothers (see
the graph). Nonetheless, we cannot exclude that the observed increase was
just by chance, since these results are not statistically significant,
although the sample sizes are small. However, it is important to note that
termination of pregnancy (TOP) after the detection of fetal anomalies is
legal in Spain since 1985, and data on TOPs can not be obtained on a
routine basis. This implies that the apparent increasing of the frequency
in young mothers along the time could be more striking, because of the TOP
of some gestations in which fetuses had gastroschisis.
Another important fact is that whereas the increasing frequency in
young mothers is not universal in all of the published studies3, the
relationship between gastroschisis and young maternal age is consistently
found in all of them. Thus, it seems that some maternal or environmental
factors related with young women must be somehow changing with time, at
least in some areas of the world, in the sense of increasing the risk.
This could be associated to multiple also changing factors. So it is clear
that we are facing a new challenge for prevention that cannot be missed
anymore, and we reinforce these authors’ consideration on that the present
way to move forward is collaborative research(1-3), if possible comparing
factors in areas with and without increasing frequency of this rare
congenital anomaly.
Eva Bermejo, PhD, responsible for the epidemiology section. E-mail:
eva.bermejo@isciii.es
Jacobo Mendioroz, collaborator
Lourdes Cuevas, collaborator
María-Luisa Martínez-Frías, PhD, Professor of the department of
pharmacology of the Faculty of Medicine of the Complutense University of
Madrid, and ECEMC and CIAC Director. E-mail: mlmartinez.frias@isciii.es
ECEMC (Spanish Collaborative Study of Congenital Malformations)
CIAC (Centro de Investigación sobre Anomalías Congénitas)
Instituto de Salud Carlos III, Sinesio Delgado 6, 28029 Madrid, Spain
References
1.- Kilby DM. The incidence of gastroschisis. BMJ 2006;332:250-1
(February 4)
2.- Donaldson L. Gastroschisis: a growing concern. London: Department
of Health, 2004. www.dh.gov.uk/assetRoot/04/11/57/82/04115782.pdf
(accessed 4 Jan 2006)
3.- Mastroiacovo P, Lisi A, Castilla E. Gastroschisis is increasing
worldwide. A need for international research. BMJ 2006; 7 February 2006
rapid response to Kilby(1)
4.-Martínez-Frías ML, Salvador J, Prieto L, Zaplana J.
Epidemiological study of gastroschisis and omphalocele in Spain.
Teratology 1984;29:377-382.
5.- Martínez-Frías ML. Gastroschisis: Is the prevalence increasing?
(Letter to the Editor). Am J Med Genet 1994;49:128.
6.- Martínez-Frías ML, Rodríguez-Pinilla E, Prieto L. Prenatal
exposure to salicylates and gastroschisis: A case-control study.
Teratology 1997;56:241-243.
BIRTH PREVALENCE OF GASTROSCHISIS (PER 10,000 NEWBORN INFANTS) IN TWO
MATERNAL AGE GROUPS, BETWEEN 1980 AND 2004 IN SPAIN:
- MATERNAL AGE <_20 xmlns:surveyed="urn:x-prefix:surveyed" xmlns:_95814="urn:x-prefix:_95814" total="total" population="population" surveyed:_="surveyed:_" _95814:_="_95814:_" p="p"/> Years 1980-1985: 1.57 (95% Confidence interval:0.43-4.02)
Years 1986-1999: 2.24 (95% CI:1.16-3.91)
Years 2000-2004: 2.99 (95% CI:0.97-6.98)
- MATERNAL AGE 20+ (Total population surveyed:1,949,593):
Years 1980-1985: 0.48 (95% CI:0.28-0.76)
Years 1986-1999: 0.30 (95% CI:0.20-0.42)
Years 2000-2004: 0.27 (95% CI:0.15-0.46)
Competing interests:
None declared
Competing interests: No competing interests