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Michael Buettcher, Gurli Baer, Jan Bonhoeffer, Urs B. Schaad and Ulrich Heininger
Pediatrics 2007;120;473-480
DOI: 10.1542/peds.2007-0035
The online version of this article, along with updated information and services, is
located on the World Wide Web at:
http://www.pediatrics.org/cgi/content/full/120/3/473
The authors have indicated they have no financial relationships relevant to this article to disclose.
ABSTRACT
OBJECTIVE. We attempted to obtain baseline data on the incidence of intussusception
and its association with gastroenteritis in a cross-sectional observational study in
www.pediatrics.org/cgi/doi/10.1542/
children. peds.2007-0035
METHODS. Admissions to all 38 pediatric units in Switzerland because of intussus- doi:10.1542/peds.2007-0035
ception were reported to the Swiss Pediatric Surveillance Unit from April 2003 to Drs Buettcher and Baer contributed equally
to this study.
March 2006. Patient and disease characteristics were assessed prospectively with
Key Words
the use of a standardized questionnaire based on the case definition for intussus- intussusception, epidemiology,
ception developed by the Brighton Collaboration. Completeness of reporting was Switzerland, rotavirus vaccine
verified through capture-recapture analysis. Abbreviations
SPSU—Swiss Pediatric Surveillance Unit
RESULTS. There were 294 patients with reported intussusception; 35 cases were ICD-10 —International Classification of
Diseases, 10th Revision
excluded for various reasons, and 29 additional patients were identified through SQ—standardized questionnaire
International Classification of Diseases, 10th Revision, codes. After capture-recapture Accepted for publication Apr 3, 2007
analysis, we estimated underreporting to the Swiss Pediatric Surveillance Unit to Address correspondence to Ulrich Heininger,
be 32% and we calculated a true number of 381 intussusception episodes. The MD, Division of Pediatric Infectious Diseases,
University Children’s Hospital Basel, PO Box
highest level of diagnostic certainty was reached by 248 patients, and 20 fulfilled 4005, Basel, Switzerland. E-mail: ulrich.
level 2 criteria; for the remaining 20 patients, available information was insuffi- heininger@ukbb.ch
cient. The mean age of the patients was 2.7 years. The yearly mean incidence of PEDIATRICS (ISSN Numbers: Print, 0031-4005;
Online, 1098-4275). Copyright © 2007 by the
intussusception was 38, 31, and 26 cases per 100 000 live births in the first, second, American Academy of Pediatrics
and third year of life, respectively, with no apparent seasonality. Seventy patients
had a history of coinciding gastroenteritis, and 5 of 61 tested positive for rotavirus.
Spontaneous devagination was observed for 38 patients; enemas reduced intus-
susception successfully in 183 cases, whereas surgical treatment was required in
67. All patients recovered without sequelae.
CONCLUSIONS. This is the first prospective nationwide surveillance of intussusception
in childhood using a standardized case definition. Most cases occurred beyond
infancy, and association with rotavirus gastroenteritis was rare.
474 BUETTCHER et al
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likelihood that cases were managed on an outpatient
basis is very low.
tests were performed, rotavirus was detected in stool (45%) (Table 2). “Red currant jelly” stools were seen in
samples. The distributions of all cases and cases of intus- 36 cases (13%). Infants (n ⫽ 83), being the largest age
susception associated with gastroenteritis were similar group, presented mainly with abdominal pain (n ⫽ 67;
across seasons (Fig 3), with no apparent peak between 81%), vomiting (n ⫽ 56; 66%), and bloody stools (n ⫽
December and April, when most cases of rotavirus gas- 47; 57%).
troenteritis occur in Switzerland.29 An additional 45 chil- Of the cases (n ⫽ 281) for which the onset of symp-
dren (16%) had coinciding illnesses other than gastro- toms before admission to the hospital was known (in
enteritis, with upper respiratory tract infections (n ⫽ 19) hours), 146 (52%) had symptoms for ⬍24 hours, 54
being most frequent. (19%) for ⬎24 hours but ⬍48 hours, and 81 (29%) for
During the study period, there were an average of ⬎48 hours. When we compared patients hospitalized
72 550 live births per year in Switzerland.26 The mean within 24 hours after the onset of illness with those with
yearly incidence rates of intussusception in the first, illness for ⬎24 hours before admission, red currant jelly
second, and third year of life were 38, 31, and 26 cases stools were present in similar proportions (27 of 146
per 100 000, respectively, with some variation from year patients, 18%, versus 20 of 135 patients, 15%; P ⫽ .41),
to year (Fig 4). With the capture-recapture analysis whereas a palpable abdominal mass was observed more
taken into account, however, the true incidence rates frequently for patients with early presentation (77 of 146
can be estimated to be 56, 46, and 38 cases per 100 000, patients, 53%, versus 49 of 135 patients, 36%; P ⫽ .006).
respectively. Ultrasonography was the most frequently used
The most frequently observed symptoms of patients method for diagnosis, and it confirmed intussusception
were abdominal pain (92%), vomiting (53%), and pallor for 236 (85%) of 278 patients. Intermittent intussuscep-
476 BUETTCHER et al
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35
30
25
20
FIGURE 3
Seasonal distribution of all intussusception cases (black 15
bars; n ⫽ 288) and cases associated with gastroenteritis
(gray bars; n ⫽ 70), according to month of occurrence
10
(cumulative for 3 study years).
Au gu st
Ap ri l
Ju ly
Marc h
Oct obe r
May
Ju ne
No ve mb er
Jan ua ry
Fe brua ry
Se ptem ber
Decem ber
FIGURE 4
Incidence (cases per 100 000 live births per
year) of intussusception according to study
year and age group in children up to 3 years
of age.
tion was found for 31 patients (11%), ultrasound ruled 17). Meckel diverticulum (n ⫽ 6) and intestinal lym-
out intussusception for 2 patients (1%), and findings phoma (n ⫽ 3) were discovered as obvious causes of
were inconclusive for 9 patients (3%). Abdominal radio- intussusception during surgery. Intussusception oc-
graphs were performed for 90 patients; 67 (74%) re- curred as a complication of gastrointestinal vasculitis
vealed typical findings of intussusception and another 19 with Henoch-Schönlein purpura in 7 patients; in the
(21%) were consistent with intussusception, as were 3 great majority of the remaining cases (272 of 288 cases,
of 4 abdominal computed tomograms. Of the 188 cases 94%), the underlying cause of intussusception remained
with specific information available, 156 (83%) were il- unknown. Thirty-eight patients (13%) recovered with
eocolic, 31 (16%) were ileoileal, and 1 (0.5%) was colo- spontaneous devagination early during their hospital
colic. stays.
The categorization of intussusception cases according
Outcomes to the level of diagnostic certainty (Brighton Collabora-
Of the 288 patients, 183 (63%) were treated successfully tion case definition) is presented in Table 3. The great
through hydrostatic reduction of intussusception, majority of cases fulfilled the criteria for the highest level
whereas 67 (23%) required surgical treatment (after of diagnostic certainty, and agreement between the 2
unsuccessful conservative treatment for 36 patients, af- independent observers (Drs Buettcher and Baer) who
ter relapse for 14, and as the primary intervention for reviewed and classified the reported cases was 0.96 (
TABLE 3 Categorization of Intussusception Cases According to In our experience, this is most likely explained by coding
Brighton Collaboration Case Definition mistakes in the hospital databases.
n (%) Underreporting is an inherent limitation of any sur-
veillance study. Capture-recapture methods allow esti-
Total Age of
⬍1 y mation of the degree of underreporting (ie, the number
Level 1 diagnostic certainty 248 (86.1) 73 (88.0) of unknown cases) and thus estimation of the true num-
Level 2 diagnostic certainty 20 (6.9) 6 (7.3) ber of cases. Our estimate is likely to be valid, because
Level 3 diagnostic certainty 0 (0) 0 (0) the probability of cases being recorded in one sample
Insufficient information to meet case definition 20 (6.9) 4 (4.8) was independent of the probability of cases being re-
Total 288 (100) 83 (100)
corded in the other, and cases were equally likely to be
captured in either sample.
As members of our group (with the use of data from
value). All patients recovered and were discharged with- the first year of this study) and others showed previ-
out apparent sequelae. ously, the case definition for intussusception developed
recently by the Brighton Collaboration is useful and
reliable,27 and 86% of reported cases reached the highest
DISCUSSION
level of diagnostic certainty. The triad of abdominal pain,
We surveyed prospectively the spectrum of clinical char-
palpable abdominal mass, and red currant jelly stools has
acteristics, management, and outcomes of intussuscep-
been described as the typical presentation of intussus-
tion in children and adolescents in Switzerland over a
ception.5 In our study, abdominal pain, vomiting, and
3-year period, with the use of standardized criteria de-
pallor were the most prominent symptoms at presenta-
fined by the Brighton Collaboration.25 Epidemiologic
and clinical characteristics of intussusception were stud- tion, whereas a palpable abdominal mass (38%) and red
ied previously,2–4,6,30 but none of those studies was per- currant jelly stools (13%) were less common. It could be
formed prospectively and standardized criteria were not argued that the latter would be signs of progressed in-
used to define cases. tussusception. However, the presence of red currant jelly
In contrast, our study involved prospective nation- stools was not dependent on the duration of illness, and
wide surveillance of intussusception in children, and we an abdominal mass was more often palpable in patients
used standardized criteria for data collection. Two re- with early (rather than late) presentation to the hospital.
cently published studies used a similar approach.11,19 Spontaneous devagination of intussusception has
However, those studies included only children ⬍2 years been reported to occur in 4% to 10% of cases.32,33 We
of age, whereas our surveillance included children of observed this phenomenon in 13% of our cases. Possi-
any age. An additional unique strength of our study was bly, comparatively early presentation of children with
the attempt to ascertain the completeness of reporting abdominal pain to hospitals and rapid availability of
through capture-recapture analysis using 2 data sources. imaging techniques allowed sensitive detection of cases.
Surprisingly, the ICD-10 code search captured only 76% Early relapse of intussusception after conservative inter-
of cases reported to the SPSU, which is remarkably sim- vention occurred for 13% of patients, and 23% required
ilar to previous SPSU surveillance of varicella hospital- surgery during the course of their illness, which is in the
izations, for which the corresponding figure was 78%.31 range of previous observations.3,6,32,34
478 BUETTCHER et al
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Only a minority of cases (24%) in this prospective ment of intussusception episodes is feasible, and in-
study had associated gastroenteritis and, despite the high ternationally standardized postlicensure studies would
rate of rotavirus testing (61 of 70 cases), only 5 cases of allow data comparability beyond any single country’s
rotavirus infection were identified. Rotavirus infection pertinent population.
has a distinct seasonal pattern in Switzerland, with
yearly peaks between December and April,29 but no such ACKNOWLEDGMENTS
seasonality for intussusception was observed during the An unrestricted research grant was provided by Glaxo
3-year study period. In accordance with previous re- SmithKline.
ports, this indicates a lack of apparent association be- We thank Dr Terhi Tapiainen for assisting in project
tween these 2 entities.11,16,24,35–37 coordination during the first study year; Daniela Beeli,
We found the highest incidence of intussusception in SPSU secretary, for coordinating the report forms; Esther
infants (ie, 56 cases per 100 000). This is in accordance Schilling, study secretary at the University Children’s
with previous studies,3,4,8,19,30 but the rate is in the lower Hospital Basel, for keeping track of the reports; the SPSU
part of the range from earlier reports that reported spe- representatives of the respective pediatric and pediatric
cifically the incidence in infants, from Venezuela (inci- surgical units in Switzerland (C. Aebi, E. Antonelli, W.
dence: 24 cases per 100 000),5 Chile (incidence: 33 cases Baer, B. Berclaz, M. Bianchetti, M. Bittel, A. Blumberg,
per 100 000),38 Vietnam (incidence: 302 cases per H.-U. Bucher, L. Buetti, E. Bussmann, O. Carrel, P. O.
100 000),11 Australia (incidence: 71–131 cases per Cattin, A. Corboz, G. Délèze, P. Diebold, P. Dolivo, F.
100 000),11,18 United Kingdom (incidence: 66 –100 cases Farron, M. Gehri, T. Gehrke, C.A. Haenggeli, P. S. Hüppi,
per 100 000),4,9 Israel (incidence: 224 cases per P. Imahorn, C. Kind, W. Kistler, B. Knöpfli, O. Lapaire,
100 000),3 and Singapore (incidence: 60 cases per B. Laubscher, U. Lips, N. Lutz, A. Malzacher, J. McDou-
100 000).19 It is also low in the range (incidence: 50 –230 gall, J.-L. Micheli, J. Mayr, M. Mönkhoff, V. Pezzoli,
cases per 100 000) from studies that reported overall B. M. Regazzoni, L. Reinhard, F. Renevey, P. Ri-
incidence but not rates for specific age groups.5–8 Al- mensberger, H. Roten, C. Rudin, J. W. Salomon, V.
though most previous studies found the majority of all Schlumbom, M. Schwöbel, G. Staubli, C. Stüssi, R.
cases in infants,3–5,8,19,30 only 29% of our cases belonged Tabin, B. Wildhaber, J. Wisser, M. Wopman, Z. Za-
to that age group, and incidences in the second and third chariou, U. G. Zeilinger, A. Zemmouri, and U. Zimmer-
years of life were similar to those in infants. The fact that mann) for providing reports; and all of the dedicated
we did not restrict our surveillance to young children physicians for taking care of the patients and helping to
probably explains this difference. Consequently, the complete the questionnaires.
mean age (2.7 years) and median age (1.9 years) of our
patients were considerably higher than those in other
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Three-Year Surveillance of Intussusception in Children in Switzerland
Michael Buettcher, Gurli Baer, Jan Bonhoeffer, Urs B. Schaad and Ulrich Heininger
Pediatrics 2007;120;473-480
DOI: 10.1542/peds.2007-0035
Updated Information including high-resolution figures, can be found at:
& Services http://www.pediatrics.org/cgi/content/full/120/3/473
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