Takeuchi et al.

BMC Pediatrics 2012, 12:36

http://www.biomedcentral.com/1471-2431/12/36

RESEARCH ARTICLE

Open Access

Intussusception among Japanese children: an

epidemiologic study using an administrative
database
Masato Takeuchi1*, Toshio Osamura2, Hideo Yasunaga3, Hiromasa Horiguchi3, Hideki Hashimoto4 and
Shinya Matsuda5

Abstract
Background: The epidemiology of intussusception, including its incidence, can vary between different countries.
The aim of this study was to describe the epidemiology of childhood intussusception in Japan using data from a
nationwide inpatient database.
Methods: We screened the database for eligible cases 18 years of age, who were coded with a discharge
diagnosis of intussusception (International Classification of Diseases, 10th revision: K-561) between July to
December in 2007 and 2008. We then selected cases according to Level 1 of the diagnostic certainty criteria
developed by the Brighton Collaboration Intussusception Working Group. We examined the demographics,
management, and outcomes of cases, and estimated the incidence of intussusception.
Results: We identified 2,427 cases of intussusception. There were an estimated 2,000 cases of infantile
intussusception annually in Japan, an incidence of 180-190 cases per 100,000 infants. The median age at diagnosis
was 17 months, and two-thirds of the patients were male. Treatment with an enema was successful in 93.0% of
cases (2255/2427). The remainder required surgery. Secondary cases accounted for 3.1% (76/2427). Median length
of hospital stay was 3 days. Of the 2,427 cases, we found 2 fatal cases associated with intussusception.
Conclusions: This is currently the largest survey of childhood intussusception in Asia using a standardized case
definition. Our results provide an estimate of the baseline risk of intussusception in Japan, and it is higher than the
risk observed in other countries.

Background
Intussusception is the most common cause of intestinal
obstruction among infants and young children, and can
also affect older children and adolescents [1]. Improvements have been made in the diagnosis and treatment
of intussusception [2]; however, poor outcomes can still
occur, even in developed countries [3].
In 1999, Rotashield (the first-generation rotavirus vaccine licensed in the United States) was withdrawn from
the market because of a potential increased risk of
intussusception [4]. Two recent studies investigated
whether the second-generation Rotavirus vaccine was
also associated with an increased risk of intussusception
* Correspondence: masatotakeuchi@gmail.com
1
Department of Pediatrics, The University of Tokyo Hospital, Tokyo, Japan
Full list of author information is available at the end of the article

[5,6], and revealed conflicting results. One post-marketing survey reported a small but statistically significant
increased risk [5], while the other study found no evidence of an elevated risk of intussusception [6]. The
potential risk of intussusception among vaccinated children has motivated researchers to estimate the baseline
risk of intussusception [7,8]. In the last several years, a
growing number of studies have been published focusing
on the incidence of intussusception [9-11]. Several surveys have also been published regarding the incidence of
intussusception in Eastern Asia including Taiwan, Hong
Kong and Vietnam [12]. However, the results vary, ranging from 70 to 300 cases per 100,000 children, suggesting that there are regional differences in the incidence
of intussusception.

2012 Takeuchi et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative
Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and
reproduction in any medium, provided the original work is properly cited.

Takeuchi et al. BMC Pediatrics 2012, 12:36

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The primary goal of our study was to describe the epidemiology of childhood intussusception in Japan, including patients demographic characteristics, management,
and outcomes, and to estimate the baseline incidence of
infantile intussusception before the introduction of the
Rotavaccine program.

Methods
Data source

Our survey was based on data from the Diagnosis Procedure Combination (DPC) inpatient database in Japan.
The DPC database is a nationwide database of inpatients
that contains administrative claims data and discharge
abstracts. Data are collected for 6-month periods
(between July 1 and December 31) each year. For the
analyses, we used data obtained in 2007 and 2008. In
2008, for example, data were compiled from approximately 2.9 million inpatients at 855 hospitals. This
represented 45% of hospitalized acute care cases in
Japan.
The database includes data on diagnoses, comorbidities at admission and complications after admission
coded using International Classification of Diseases,
10th Revision (ICD-10) codes, surgical procedures,
length of stay, discharge status including in-hospital
death, and costs [13,14].
Because this study was based on a secondary analysis
of the anonymous patient database, the requirement for
informed consent was not applicable. Study approval
was obtained from the institutional review board of the
University of Occupational and Environmental Health.
Criteria

All children aged between 0 and 18 years old admitted

with intussusception (ICD-10 code: K561) were
screened. At this point, patients with various diagnostic
conditions were included. We used criteria developed by
the Brighton Collaboration Intussusception Working
Group to categorize patients into 5 groups (definitive to
not a case), using a mix of major and minor criteria [8].
Although these criteria were primarily developed for
vaccine safety data, they are also highly reliable for epidemiological research (sensitivity: 97%; specificity: 8791%) [15]. Because our database did not contain information about clinical signs and symptoms, we included
only cases meeting the clinical case definition of definite
(level I of diagnostic certainty) intussusception according to surgical, radiological, or autopsy criteria [8], and
excluded all other cases coded as intussusception in the
database. A previous study ensured that this approach
enabled us to capture more than 90% of cases with
intussusception correctly [10].
Readmissions to the same hospital shortly after discharge (e.g., within a week) may involve cases with

Page 2 of 6

either insufficient reduction or with true recurrence. In

this regard, Daneman et al. reported that most of the
recurrence occurred within a few days following enema
reduction [16]. Accordingly, we considered all admissions for intussusception to the same hospital as independent events.
Estimation of population-based incidence of infantile
intussusception

We estimated the population-based incidence of intussusception among infants who are the target population for Rotavirus vaccination (Table 1). The coverage
rate of the DPC database was 45% and it was uncertain whether patient distribution was balanced
between DPC hospitals and non-DPC hospitals; therefore, sampling bias due to the referral pattern may be
present in our dataset. In this regard, we assumed that
patients were equally distributed if the hospital
volumes were similar, regardless of hospital type.
Based on this assumption, we classified DPC hospitals
into four categories according to their hospital bed
volumes; similarly, hospitals throughout Japan were
categorized in this manner. We then calculated the
estimated number of intussusception cases each year
(Yi) and 95% confidence intervals (CI) by applying the
following equation using Wald CIs for the population
proportion:

Yi /Ni = p 1.96 p (1 p) /ni

where i (1 to 4) is the number of each strata, Ni is the
number of beds in all acute care hospitals in Japan, ni
denotes the number of beds in the DPC hospitals and p
= 2Xi/ni (Xi is the observed number of intussusception
cases in DPC hospitals between July and December each
year). In this estimation, we assumed that there was no
seasonality in the incidence of intussusception [17] and
doubled the half-year incidence (X i ) to obtain the
whole-year incidence.
In 2007 and 2008, the number of births was 1,090,000
each year. Therefore, we used this number as the
denominator of the population-based incidence of infantile intussusception.
Statistical analysis

Descriptive statistics were used to summarize the demographic characteristics of patients. To describe the characteristics of the patients, means ( standard deviation)
or medians ( interquartile range) are reported where
appropriate. Fishers exact test was used to calculate
95% CI.
All statistical analyses were performed using SPSS for
Windows 17.0 (SPSS, Chicago, IL, USA) and R 2.10.0
(available at http://www.r-project.org).

Takeuchi et al. BMC Pediatrics 2012, 12:36

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Page 3 of 6

Table 1 Estimation of Population-based Incidence of Infantile Intussusception

Hospital
Volume

No. of
hospitals in
Japan

No. of beds in
Japan (Ni)

No. of beds in DPC

participating hospitals (ni)
2007

2008

2007

2008

2007

2008

< 400 beds

(i = 1)

7,001

566,658

138,979

119,853

86

97

701 (597-806)

917 (788-1046)

400 - < 600

beds (i = 2)

466

175,715

98,050

89,627

176

169

631 (565-697)

663 (592-733)

600 - < 800

beds (i = 3)

161

88,870

54,351

49,740

98

78

320 (276-365)

278 (235-322)

> 800 beds

(i = 4)

95

78,995

50,245

50,245

97

71

305 (262-348)

223 (187-260)

Total

7,723

910,238

341,625

309,465

457

415

1,957 (1743-2216)

2,081 (1802-2361)

Results
Characteristics of cases

A total of 2,427 cases with intussusception were identified; 1,185 in 2007 and 1,242 in 2008. During the same
time period, the number of all-cause admissions of
patients 18 years of age was 626,770. Thus, intussusception accounted for 0.39% of all-cause admissions
(Table 2). The annual number of cases of intussusception among Japanese infants was estimated as 1,957
(95%CI: 1743-2216) in 2007 and 2081 (95%CI: 18022361) in 2008 (Table 1). This corresponded to an incidence of 179 (95%CI: 165-203) per 100,000 infants in
2007 and 191 (95%CI: 165-216) in 2008.
Of the 2,427 patients, 1,610 (66.3%) were male. The
median age at the time of diagnosis was 17 months
(interquartile range: 9.5-31.5) and 92.5% (2244/2427) of
cases were under 5 years of age (Figure 1). Intussusceptions occurred most frequently in the first year of life
(35.9%: 872/2427) with a peak incidence between 8 and
10 months of age, but were rarely found under 3
months of age (n = 9, including 3 neonatal cases).
Among the 2,427 cases with intussusceptions, 93.0%
(2,255) were successfully reduced with an enema, and
the remaining 175 cases required surgical management.
Table 2 All-cause admissions, all intussusceptions and
secondary intussusceptions in each age group
Age
(y)

All-cause
admissions

Intussusceptions
(per 1,000
admissions)

Secondary
cases

157,494

872 (5.5)

14(1.6%)

88,125

657 (7.5)

8 (1.2%)

51,900

415 (8.0)

12 (2.9%)

3-4

74,689

300 (4.0)

7 (2.3%)

5-12

157,099

148 (0.9)

22 (14.9%)

13-18

97,463

35 (0.4)

13 (37.1%)

Total

626,770

2,427 (3.9)

76 (3.1%)

No. of intussusception
from July to December
(Xi)

Estimated No. of cases per year

(Yi)

In the surgical intervention group, bowel resection was

required in 52 cases (29.7% of the surgical cases). The
median length of hospital stay was 3 days (median: 3
days in the non-surgical group and 8 days in the surgical group).
We found that 36 patients (37 episodes) had been
readmitted to the same hospital within a week after
treatment of the initial intussusception (mean: 2.3 days).
At the second admission, 32 cases of intussusception
were successfully reduced by enema and 5 required surgery. In the surgical cases, underlying conditions were
found in 2 children with polyps; none were reported to
have perforation.
Secondary causes of intussusception

We found 76 (3.1%) intussusceptions due to secondary

causes (Table 3). The remaining patients were thought
to be idiopathic cases. Frequencies of secondary cases
were 1-3% under 5 years of age, and increased thereafter. Schnlein-Henoch purpura, Meckels diverticulum
and polyps were the three major causes of the pathological lead points (PLPs) of these 76 cases. Intussusception associated with Schnlein-Henoch purpura mainly
affected young children, while Meckels diverticulum
and polyps were present in children of all age groups.
Complications

We identified 27 (1.1%) patients with complications

related to intussusception. These complications included
perforation and/or peritonitis (n = 9), systemic infections
such as sepsis and meningitis (n = 8), shock (n = 5), seizures (n = 3) and death (n = 2). Intussusception-associated complications occurred irrespective of patient age.
We found 2 fatal cases attributed to intussusception;
thus, the mortality rate in our series was 0.08% (2/2427,
95%CI: 0.01-0.30%). The first patient was a 3-year-old
girl. She was in cardiopulmonary arrest on arriving at
hospital and died within 24 hours after presentation.

Takeuchi et al. BMC Pediatrics 2012, 12:36

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Autopsy revealed invagination of the intestine. The second was a 2-year-old boy. His invagination was reduced
successfully with a non-surgical procedure, but he died
of complicated hemolytic-uremic syndrome on the second hospital day. Autopsy was not performed in the second case.

Figure 1 Age distribution of all intussusception cases (N =

2,427). Inset, Age distribution of intussusception cases in infants (0
to < 12 months; n = 872).

Table 3 Pathological Lead Point (N = 77)

Causes

Number of
Patients

Age
(yrs)

Structural
Meckels diverticulum

13

0-13

Duplication cyst

4-10

Malrotation

0-1

Hirschsprung disease

Mesenteric hernia

Vascular/hematological
Schnlein-Henoch purpura

15

1-7

Hemolytic-uremic syndrome

2-3

Idiopathic thrombocytopenic purpura

Lymphoma

15-16

Nephrotic syndrome

2-7

Leukemia

12

Kawasaki Disease

Polyps

0-15

Benign tumor

6-13

Malignant tumor

0-15

Tumor (details not available)

0-18

Appendicitis/Appendix

1-13

Neoplasms

Others
Postoperative

Foreign Body

Congenital biliary dilation +

Pancreatitis

Endometriosis

15

Discussion
Our study elucidates the nature of childhood intussusception in Japan. To our knowledge, this is the first
study to investigate the incidence of intussusception
among an Asian population using a nationwide database
combined with the Brighton Criteria for diagnostic
accuracy.
Recent analyses of childhood intussusception in Western countries [1,18,19] found that the peak age was
between 4 and 9 months; the male:female ratio was
around 2:1; PLPs were found in 2.6-15% of cases; and
non-surgical enema reductions were successful in 8095% of patients. Our findings are essentially consistent
with these previous reports.
We found that intussusceptions accounted for 3.9
cases per 1,000 all-cause admissions (8.0 cases in 2-yearolds and 0.12 in 18-year-olds). This hospital-based incidence appears to be higher than those observed in Europe (0.66-2.24 per 1,000 children in inpatient
departments) [7]. Similarly, we estimated that the population-based incidence of intussusception was 179-191
per 100,000 infants in our cohort. Only one study is
currently available regarding the population-based incidence of infantile intussusception in Japan [20]. This
earlier study showed that the incidence was 185 cases
per 100,000 infants based on a 25-year survey conducted
in one small region of Japan. This result is in accordance with our estimation and gives further support to
our findings. Our estimated incidence was much higher
than those in other countries (179-191 vs 30-80 cases
per 100,000 population) [12]. Because both the hospital
and population-based incidence rates were higher than
those of other countries, it is very likely that the incidence of intussusception in Japan is high compared with
other countries. Our results also suggest that the incidence of intussusception cannot be easily extrapolated
to other countries, because studies from Eastern Asian
countries outside Japan reported that the incidence of
intussusception was 70-300 cases per 100,000 children
[12]. With respect to the incidence of intussusception,
surveys in each region may be essential.
Our study has several advantages. First, we used a
standardized case definition of intussusception [8]. This
enabled us to compare epidemiologic surveys from
other areas and temporal trends in the same area.
Reported incidence rates of intussusceptions vary among
different populations and times [1,21,22]. Thus,

Takeuchi et al. BMC Pediatrics 2012, 12:36

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standardized case definitions can contribute to maximizing the reliability of epidemiologic data. Second, our
study, including over 2,000 patients, is large enough to
determine the epidemiology of intussusception. Third,
our study determined the case-fatality rate. Little information is available about the mortality rate in developed
countries [23]. The mortality rate in our study was
0.08% (95%CI: 0.01-0.30%), which is comparable to data
available from the United States reporting 18-56 deaths
per 100,000 cases [3]. Furthermore, our data included
epidemiologic data of older children and adolescents,
which is lacking in previous studies. This study thus
provides a deeper insight into intussusception in children of all ages. In addition, our survey included rare
but severe complications such as perforation/peritonitis,
systemic infections (including sepsis, bacterial meningitis), and neurological involvement. Although previous
case reports have sporadically reported these complications [23-25], this study estimates their incidence rates
based on cross-sectional data.
Several limitations of the current study should be
acknowledged. First, because only inpatient data was
included, we may have failed to capture cases treated in
emergency units or outpatient clinics. Outpatient management of children with intussusception is widely practiced in some countries [5,26]. In Japan, however, inhospital observation is recognized as the standard practice even after successful reduction. Thus, it seems unlikely that we failed to capture a number of cases with
intussusception. Second, there is inherent limitation in
observational studies using administrative databases. For
example, some clinical information was not included in
our database (e.g., patients past and present history, site
of intussusception, or types of contrast media used).
Another aspect of this limitation is that our study may
be susceptible to systemic bias; referral bias, for example, may be present in our dataset. Therefore, our
results should be interpreted in the context of the limitations arising from the nature of this study. Finally,
our database collected data only over 6 months because
of the cost to participating hospitals. Therefore, we
could not investigate whether there were seasonal trends
of intussusception in Japan. This limitation could influence the estimation of the population-based incidence
of intussusception. A prospective patient registration
survey may be needed using a standardized case definition such as the criteria set out by the Brighton
Collaboration.

Conclusions
This study describes the epidemiology of childhood
intussusception in Japan, including current information
on incidence data, pathological lead points and complications. We also highlight that the observed incidence

Page 5 of 6

of intussusception among Japanese infants is higher

than those reported from other countries. Until active
surveillance of intussusception is available, the nationwide administrative database has a potential role in
monitoring the incidence of childhood intussusception.
Acknowledgements
This study was funded by Grants-in-Aid for Research on Policy Planning and
Evaluation from the Ministry of Health, Labour and Welfare, Japan.
Author details
1
Department of Pediatrics, The University of Tokyo Hospital, Tokyo, Japan.
2
Department of Pediatrics, Kyoto Second Red Cross Hospital, Kyoto, Japan.
3
Department of Health Management and Policy, Graduate School of
Medicine, The University of Tokyo, Tokyo, Japan. 4Department of Health
Economics and Epidemiology Research, School of Public Health, The
University of Tokyo, Tokyo, Japan. 5Department of Preventive Medicine and
Community Health, University of Occupational and Environmental Health,
Fukuoka, Japan.
Authors contributions
MT performed data analysis and manuscript preparation. TO was involved in
the design of this work. HY, HH (Dr. Horiguchi), HH (Dr. Hashimoto) and SM
were responsible for providing the database. All authors read and approved
the final draft.
Competing interests
The authors declare that they have no competing interests.
Received: 14 June 2011 Accepted: 22 March 2012
Published: 22 March 2012

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Cite this article as: Takeuchi et al.: Intussusception among Japanese
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