Stool Color Card Screening for Early Detection of Biliary Atresia and

Long-Term Native Liver Survival: A 19-Year Cohort Study in Japan

Yan-Hong Gu, MD, MSc, PhD1,2, Koji Yokoyama, MD3, Koichi Mizuta, MD, PhD4, Takashi Tsuchioka, MD, PhD5,
Toyoichiro Kudo, MD, PhD6, Hideyuki Sasaki, MD, PhD7, Masaki Nio, MD, PhD7, Julian Tang, PhD8,
Takayoshi Ohkubo, MD, PhD1, and Akira Matsui, MD, DMSci6

Objective To evaluate the sensitivity and specificity of a stool color card used for a mass screening of biliary
atresia conducted over 19 years. In addition, the age at Kasai procedure and the long-term probabilities of native
liver survival were investigated.
Study design From 1994 to 2011, the stool color card was distributed to all pregnant women in Tochigi Prefec-
ture, Japan. Before or during the postnatal 1-month health checkup, the mothers returned the completed stool color
card to the attending pediatrician or obstetrician. All suspected cases of biliary atresia were referred for further ex-
amination. Diagnosis was confirmed by laparotomy or operative cholangiography for high-risk cases before the
Kasai procedure. Patients with biliary atresia were followed from the date of their Kasai procedure until liver trans-
plantation, death, or October 31, 2013, whichever comes sooner.
Results A total of 313 230 live born infants were screened; 34 patients with biliary atresia were diagnosed. The
sensitivity and specificity of stool color card screening at the 1-month check-up was 76.5% (95% CI 62.2-90.7)
and 99.9% (95% CI 99.9-100.0), respectively. Mean age at the time of Kasai procedure was 59.7 days. According
to Kaplan-Meier analysis, the native liver survival probability at 5, 10, and 15 years was 87.6%, 76.9%, and 48.5%,
respectively.
Conclusions The sensitivity and specificity of the stool color card have been demonstrated by our 19-year cohort
study. We found that the timing of Kasai procedure and long-term native liver survival probabilities were improved,
suggesting the beneficial effect of stool color card screening. (J Pediatr 2015;-:---).

B
iliary atresia is the most frequent hepatic cause of death in early childhood, with an incidence of 0.7 in 10 000, 0.6 in
10 000, and 0.5 in 10 000 live births in the US, UK, and France, respectively.1-3 In Japan, the incidence is greater,
affecting approximately 1.0 in 10 000 live births.4 Biliary atresia is characterized by a complete inability to excrete
bile as a result of sclerosing inflammation of the extra, and possibly intra, hepatic bile ducts.5 Patients with biliary atresia
have 3 main clinical features: pale-pigmented stools, prolonged jaundice, and dark urine. Pale-pigmented stools appears within
the first month after birth for most patients, and 2-5 months for others.4,6 Although there is strong evidence that biliary atresia
develops before birth and progresses after birth, its etiology remains unclear. The Kasai procedure7 commonly is used as a first-
line treatment for all types of biliary atresia.8,9
Prognosis for patients with biliary atresia is primarily related to the patient’s age at the time of Kasai procedure and the anat-
omy of the bile duct remnant.8-10 It is generally acknowledged that a Kasai procedure performed early, especially one that is
performed before the patient reaches 60 days of age, can improve the long-term native liver survival and reduces likelihood
of liver transplantations.10,11 In Japan, 66.1% of living-donor liver transplantations performed for recipients younger than
18 years of age were attributable to biliary atresia.12
Serinet et al10 highlighted the importance of screening for biliary atresia. The
concept of a stool color card for mass screening was introduced for the first time
to the local population in Tochigi Prefecture by Matsui and Dodoriki in early
1994, which resulted in early Kasai procedure (<60 days of age) in 2 of 3 patients 1
From the Department of Hygiene and Public Health,
with biliary atresia.13 Since then, the stool color card had been distributed in the
2
Teikyo University School of Medicine; Graduate School
of Public Health, Teikyo University, Tokyo; Departments
prefecture until March 2011. Subsequently, the concept of stool color card for 3 4
of Pediatrics and Transplant Surgery, Jichi Medical
5
University; Department of Surgery, Dokkyo Medical
mass screening was adopted and used in Taiwan in 2002 and resulted in earlier 6
University, Tochigi; Department of Hepatology, National
referral of patients with biliary atresia nationwide.14 Medical Center for Children and Mothers, National
Center for Child Health and Development, Tokyo;
7
In this present study, we aimed to determine the sensitivity and specificity of Department of Pediatric Surgery, Tohoku University
School of Medicine, Miyagi; and Department of 8

stool color card screening during the 19-year period, as well as its effect on the Education for Clinical Research, National Center for Child
Health and Development, Tokyo, Japan
Supported by the National Center for Child Health and
Development of Japan (25-5 and 26-31). The authors
declare no conflicts of interest.

0022-3476/$ - see front matter. Copyright ª 2015 Elsevier Inc.

JBAR Japanese Biliary Atresia Registry All rights reserved.
http://dx.doi.org/10.1016/j.jpeds.2014.12.063

1
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timing of Kasai procedure and long-term native liver survival respective hospital (across 8 medical centers). Long-term
in the Tochigi cohort. follow-up was possible because all Japanese residents are
covered by at least 1 health insurance plan that allows access
Methods to any necessary procedures post-Kasai procedure.16 In addi-
tion, pediatric patients with any of the 514 intractable
Participants were all infants born to mothers living in Tochigi chronic diseases (including biliary atresia), defined by Minis-
Prefecture, situated about 100 km north of Tokyo (Figure 1; try of Health, Labour and Welfare of Japan, are supported by
available at www.jpeds.com), from August 1994 to March a medical aid program.16 Postsurgical procedures in Tochigi
2011. Infants born in Tochigi Prefecture to mothers who Prefecture are consistent with those in other areas of Japan.
lived outside of the prefecture before giving birth were not To ensure that no patient with biliary atresia in Tochigi Pre-
included. Under the Maternal and Child Health Law in fecture was overlooked, the patient list in our study was
Japan acted since 1965, all children in the country go compared with that of the medical aid program covering
through the same postnatal health management. the 514 intractable chronic diseases.
The stool color card (3rd edition; Figure 2) was placed For the investigation of native liver survival probabilities,
within the Maternal and Child Health Handbook that was patients with biliary atresia in this study were observed
given to all pregnant women by their respective local from the date of Kasai procedure until liver transplantations,
government according to the Maternal and Child Health death, or October 31, 2013, whichever occurred sooner.
Law in Japan. Before or during the infant’s 1-month health
checkup, the mothers were asked to fill in the Statistical Analyses
corresponding number of the image on the stool color card Four reference data sets were used: nationwide data during
(Figure 2) that most resembled the color of her infant’s stool color card screening between 1994 and 2011 from the
stool. The card was then submitted to the attending Japanese Biliary Atresia Registry (JBAR), nationwide data
pediatrician or obstetrician. A positive result was defined as before stool color card screening between 1989 and 1994
a stool color determined by the guardian that matched from JBAR,17 Tochigi Prefecture data before stool color
either image 1, 2, or 3 before or during the infant’s 1- card screening between 1987 and 1992,4 and Tochigi Prefec-
month health checkup. ture data before stool color card screening between 1989 and
The Department of Pediatrics at the Jichi Medical Univer- 199118 (Table I). To quantify uncertainty, 95% CIs were
sity in Tochigi Prefecture (as the stool color card office), used. The records of approximately 80%-90% of
Japan was notified of all positive cases as soon as possible nationwide patients with biliary atresia diagnosed in
by telephone or fax. All stool color cards were collected and hospitals that are part of the Japanese Society of Pediatric
sent to the stool color card office at Jichi Medical University Surgeons were documented in JBAR. All patients with
on a weekly basis. At the office, the cards were rechecked to biliary atresia in our study were registered in JBAR.
confirm whether all corresponding numbers were properly According to the Act on the Protection of Personal
recorded and that positive cases had been properly attended Information, only statistical data and not individual data
to. At the initial phase (first 3 years), all staff was trained on can be used. Student t test or one-sample t test was
how to manage positive cases detected by the stool color card. performed to compare age at Kasai procedure. Kaplan-
Verbal informed consent was obtained from all partici- Meier analysis and the log-rank test were used to estimate
pants. The study protocol was reviewed and approved by the native liver survival probabilities with age (in months)
the Ethics Board of the National Center for Child Health as the time scale. IBM SPSS Statistics 21 (IBM
and Development. Corporation, Armonk, New York) was used for statistical
analysis. P < .05 was considered statistically significant.
Patients with Biliary Atresia and Long-Term For analytical purposes, all 34 patients with biliary atresia
Follow-Up were first considered as a whole (termed “all cases”), and then
For patients with positive stool color card results, the possi- as 2 separate groups: patients identified using stool color
bility of other types of infantile cholestasis was eliminated by chart and referred promptly (Table I).
a pediatric specialist or pediatric hepatologist through clin-
ical, biochemical, radiologic, histologic, and genetic investi- Results
gations when necessary. A final diagnosis for high-risk cases
was determined by laparotomy and/or by operative cholangi- There were 313 230 live births in Tochigi Prefecture from
ography prior to Kasai procedure by a pediatric hepatologist August 1994 to March 2011 (Figure 1). We collected the
or surgeon. None of the false positive cases underwent any stool color cards of 264 071 infants, yielding a return rate
invasive procedures. All patients with biliary atresia received of 84.3% at the 1-month health check-up; 2014 showed a
Kasai procedure at the soonest possibility performed in positive result, and 26 of them were diagnosed with biliary
accordance with the Japanese Society of Pediatric Surgeons atresia. Finally, a total of 34 patients were diagnosed with
classification.15 biliary atresia in Tochigi prefecture during the study
Patients with biliary atresia in Tochigi Prefecture received period. A patient with Alagille syndrome detected by the
Kasai procedure and were followed up regularly by their stool color card (stool color corresponding to image 2) at
2 Gu et al
- 2015 ORIGINAL ARTICLES

Figure 2. The 3rd edition stool color card used in Tochigi Prefecture from August 1994 to March 2011 consisted of 7 photo-
graphic images of stool color taken in both healthy infants and infants with biliary atresia. Images 1-3 denote abnormal stool
color, whereas images 4-7 reflect normal stool color.

1-month health checkup was excluded. At the 1-month procedure at 45 and 88 days of age, respectively. They did
health checkup, the sensitivity, specificity, positive not undergo liver transplantations until October 2013. For 3
predictive value, and negative predictive value were 76.5% patients (Patients 3, 4, and 5), their guardians used the stool
(26/34, 95% CI 62.2-90.7), 99.9% (313 018/313 196, 95% color card and reported pale-pigmented colored stool at the
CI 99.9-100.0), 12.7% (26/204, 95% CI 8.2-17.3), and 1-month health checkup. However, no further examination
99.9% (313 018/313 026, 95% CI 99.9-99.9), respectively. was performed by their respective pediatricians because the
Incidence of biliary atresia was 1.1 in 10 000 infants (34/ infants did not present with visible jaundice. These 3 patients
313 230, 95% CI: 0.7-1.5). eventually underwent Kasai procedure at 62, 77, and
Among the 34 patients with biliary atresia, 8 were missed at 109 days after birth, respectively. Subsequently, Patients 3
the 1-month check-up (Figure 1). Of these patients, 2 (Patients and 5 underwent liver transplantations at 5 and 12 months
1 and 2) were in a neonatal intensive-care unit for more than a of age, respectively. The guardian of 1 of the patients (Patient
month after birth. Because their overall condition was poor, 6) failed to use the stool color card. The patient received
their guardians and the medical staff overlooked the presence Kasai procedure at 97 days of age and underwent liver
of abnormal stool color. These 2 patients received Kasai transplantation at 72 months. One patient (Patient 7) did
Stool Color Card Screening for Early Detection of Biliary Atresia and Long-Term Native Liver Survival: 3
A 19-Year Cohort Study in Japan
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Table I. Age at the time of the Kasai procedure in Tochigi cohort vs reference data, before and during stool color card
screening
During stool
Before stool color color card
card screening screening Tochigi stool color card cohort (August 1994-March 2011)
Patients identified
using stool color Patients with
card and referred type III biliary
Year Tochigi JBAR JBAR All patients (N = 34) promptly (n = 30) atresia (n = 25)
Age at time
of Kasai
procedure, d
Mean or 1987-19924 70.3
mean  SD 1994-2011 67.7 59.7  19.4*,† 56.2  16.5z,x 59.8  19.1x,{
(1994-2002) (67.8)
(2003-2011) (67.6)
Median (range) 1987-19924 65.5
1994-2011 64.0 58.5 (18-109) 56.5 (18-88) 59.0 (18-109)
(1994-2002) (63.0)
(2003-2011) (65.0)
Number, %
(95% CI)
#45 1989-199417 18.9
1994-2011 20.4 8, 23.5 (9.3-37.8) 8, 26.6 (10.8-42.5) 6, 24.0 (7.3-40.7)
#60 1989-199118 34.0
1989-199417 40.5
1994-2011 45.1 19, 55.9 (39.2-72.6) 19, 63.3 (46.1-80.6)** 14, 56.0 (36.5-75.5)
>80 1989-199417 23.1
1994-2011 25.3 4, 11.8 (0.9-22.6)**,†† 1, 3.4 (2.3-15.6)**,†† 2, 8.0 (2.6-18.6)**,††
>90 1989-199118 13.0
1989-199417 15.1
1994-2011 16.2 2, 5.9 (2.0-13.8)**,†† 0, 0.0**,††,zz 1, 4.0 (3.7-11.7)**,††,zz

*P = .023; zP = .001; {P = .000 vs JBAR data during screening (1994-2011), 1-sample t test.
†P = .003; xP = .000 vs Tochigi data before screening (1987-1992), 1-sample t test.
**P < .05 vs JBAR data during screening (1994-2011), ††P < .05 vs JBAR data before screening (1989-1994), and zzP < .05 vs Tochigi data before screening (1987-1992).

not show abnormality at the 1-month health checkup. At greater in patients with biliary atresia who were referred
1.5 months of age, the patient’s guardian noticed pale- promptly after reporting of positive colors. The percentage
pigmented stool and jaundice. The patient received Kasai of Kasai procedure performed after 80 days of age was
procedure at 76 days of age and did not undergo liver significantly lower in the Tochigi cohort (Table I). The
transplantations. One patient (Patient 8) was not on our list mean age  SD of Kasai procedure for the 8 patients with
but was later identified through the medical aid list. The biliary atresia who were missed at the 1-month checkup
patient received Kasai procedure at 66 days of age and did was significantly later compared with the other patients
not undergo liver transplantations until October 2013. with biliary atresia (n = 26; 77.5  20.4 days vs
Therefore, with the exception of Patients 1, 2, 6, and 8, in 54.3  15.8 days; P = .002).
which the usage of stool color card failed, 30 of the total 34
patients showed stool color changes around the time of the Long-Term Native Liver Survival Probabilities of
1-month health checkup. Patients with Biliary Atresia
As of October 2013, 17 patients received liver transplants
Demographic Data of Patients with Biliary Atresia and 17 did not. One female patient died at 13 months
Among the 34 patients with biliary atresia, 11 (32.4%) were without receiving a liver transplant. Kaplan-Meier survival
male and 23 (67.6%) were female. The numbers of patients analysis with the end point defined as liver transplant,
who had type I, II, and III biliary atresia were 5 (14.7%), 1 death, or alive as of October 31, 2013, showed the native
(2.9%), and 25 (73.5%), respectively. The type of biliary liver survival probability at 5, 10, and 15 years to be
atresia in 3 patients was unknown (8.8%). All patients with 87.6%, 76.9%, and 48.5%, respectively (Table II). The
biliary atresia received Kasai procedure (1 patient with type median survival estimated by Kaplan-Meier analysis is the
I biliary atresia received hepaticojejunostomy, and all others earliest time at when the cumulative survival probability
received hepatoportoenterostomy). reached 50% or lower. In this study, the median native
liver survival was 197.2 (95% CI 136.0-258.4), 207.9 (95%
Age at the Time of Kasai Procedure CI 184.6-231.3), and 212.5 (95% CI 146.7-278.4) months
The mean age at the time of Kasai procedure was 59.7 days in in all patients, patients who were referred promptly upon
the 34 patients with biliary atresia (Table I). The percentage reporting of positive color, and patients with type III
of Kasai procedure performed before 60 days of age was biliary atresia, respectively. There was no significant
4 Gu et al
- 2015 ORIGINAL ARTICLES

Table II. Kaplan-Meier analysis of native liver survival at 5, 10, 15, and 20 years in the present study and previous reports
No. teams/ Rate of native liver survival % (SE), y
medical Stool color Study No.
Countries centers card Period design patients 5 10 15 20
19
Yokohama, Japan 1 No 1970-1986 RCR 80 63.0 54.0 - 44.0
Sendai, Japan20 1 No 1975-1980 RCR 60 60.0 60.0 58.0 -
1981-1986 50 68.0 60.0 51.0 -
US21 2 No 1972-1996 RCR 266 49.0 - - -
UK22 3 No 1999-2009 RCR 443 46.0 (95% 40.0 (95% - -
CI 41-51) CI 34-46)
France23 45 No 1986-2009 RCR 1044 40.0 (1.6) 35.8 (1.6) 32.1 (1.7) 29.6 (2.0)
France10 27/45 No 1986-2002 RCR 695 37.9 (2.0) 32.4 (2.0) 28.5 (2.3) -
Present study 8 Yes 1994-2011 Cohort
study
All patients with biliary atresia 34 87.6 (0.06) 76.9 (0.08) 48.5 (0.11) -
Patients identified using the stool color card and referred promptly 30 89.6 (0.06) 77.6 (0.08) 55.5 (11.1) -
Patients with type III biliary atresia 25 86.8 (0.07) 70.5 (0.10) 50.4 (0.12) -

RCR, retrospective chart review in medical center(s).

In this study, the period of native liver survival was from the point of Kasai procedure until liver transplantation, death, or October 31, 2013, whichever occurred sooner.

difference across the 3 aforementioned groups mentioned follow-up. The 10-year native liver survival rate was 52.8%
on the basis of the log-rank test (P > .05). (57/108). The authors also found that when the Kasai proce-
dure was performed at age of <60, 61-90, 91-120, and
Discussion >120 days among patients with type III biliary atresia regis-
tered between 1953 and 2009 whose jaundice disappeared
We have conducted a 19-year Japanese cohort study for after Kasai procedure, the 10-year native liver survival rate
screening of biliary atresia using the stool color card. The was 74.4% (32/43), 74.5% (41/55), 100.0% (6/6), and
high stool color card sensitivity and specificity achieved are 33.3% (1/3), respectively.11
likely to have contributed to more patients with biliary atresia Although the stool color card has been adopted and used
being diagnosed earlier, leading to a timely Kasai procedure. in Taiwan,14 Argentina,25 and Switzerland,26 the outcome
Accordingly, long-term native liver survival probabilities was only reported in Taiwan, where the 5-year native liver
were improved. Serinet et al10 reported that if every patient survival rate (without jaundice) was 64.3% (18/28).27
with biliary atresia were to undergo the Kasai procedure According to JBAR data, the mean and median age at Kasai
before 46 days of age, 5.7% of all liver transplantations per- procedure was not significantly different between the periods
formed annually in France in patients younger than 16 years of 1994-2002 and 2003-2011 (Table I), suggesting that the
could be spared. management of patients with biliary atresia did not change
In our cohort, the 5-, 10-, and 15-year native liver survival drastically over the years. Hence, the improvement of the
probabilities (Table II) were greater compared with studies probability of native liver survival revealed in this study is
conducted in US,21 the UK,22 and France,10,23 where stool likely to be attributable to the younger age at Kasai
color card was not used. Notably, the 5- and 10-year native procedure as a result of stool color card usage.
liver survival probabilities increased by more than 20% Although data of patients who did not use stool color card
during 1994-2011 compared with studies conducted in the were available in JBAR, we could not access them because of
Japanese cities of Yokohama and Sendai where stool color the restriction imposed by the Act on the Protection of Per-
card was not used (Table II).19,20 The 15-year native liver sonal Information. As such, the associations between stool co-
survival probability estimated by Kaplan-Meier analysis in lor card usage/early Kasai procedure and the probability of
the Sendai patients was 51%-58% between 1975 and 198620 long-term native liver survival cannot be statistically analyzed.
(Table II), which is greater than what we found in this On the basis of our results in Tochigi Prefecture, the stool
study. It might be attributable to the data being collected color card was gradually introduced to 16 other autonomous
from a single, highly specialized center, whereas our data administrative divisions in Japan between 1999 and 2010.
were collected from 8 centers. However, only patients from 2 of the regions were followed
There are 2 other reports of long-term native liver survival up. Nonetheless, the mean age of Kasai procedure after the
rates in Japanese patients with biliary atresia. Notably, the introduction of stool color card was found to be significantly
method for the calculation of native liver survival and/or sub- younger in those regions,28,29 demonstrating excellent repro-
jects selected in those studies was different from this study. In ducibility and effectiveness of the stool color card.
our case, we did not consider whether jaundice appeared or In April 2012, a nationwide biliary atresia screening using
not after Kasai procedure. On the basis of JBAR data of an updated edition of the stool color card was initiated. In
1989, Nio et al24 reported the 5-year native liver survival addition, a pilot study was launched in October 2013 in Bei-
rate was 62.0% in 735 patients who did not undergo liver jing, China. The new edition of stool color card consists of
transplantations, and 19 (2.6%) of patients were lost to digital photographic images to ensure quality control and
Stool Color Card Screening for Early Detection of Biliary Atresia and Long-Term Native Liver Survival: 5
A 19-Year Cohort Study in Japan
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greater reproducibility. A modified screening protocol has adolescence: a rational basis for biliary atresia screening. Pediatrics 2009;
also been devised. Instead of a single inspection point, the 123:1280-6.
11. Nio M, Sasaki H, Wada M, Kazama T, Nishi K, Tanaka H. Impact of age
stool color card is now being inspected at 3 intervals; 2 weeks,
at Kasai operation on short- and long-term outcomes of type III biliary
1 month, and 1-4 months after birth, allowing us to identify atresia at a single institution. J Pediatr Surg 2010;45:2361-3.
more patients with biliary atresia. 12. Kasahara M, Umeshita K, Inomata Y, Uemoto S , Japanese Liver Trans-
On the basis of our 19-year experience of stool color card plantation Society. Long-term outcomes of pediatric living donor liver
usage in the Tochigi Prefecture cohort, the effectiveness of transplantation in Japan: an analysis of more than 2200 cases listed in
the registry of the Japanese Liver Transplantation Society. Am J Trans-
the stool color card, a non-invasive technique, was demon-
plant 2013;13:1830-9.
strated. In particular, the stool color card was beneficial for 13. Matsui A, Dodoriki M. Screening for biliary atresia. Lancet 1995;345:
patients with biliary atresia whose jaundice was not obvious. 1181.
However, we are aware that the distribution of the stool color 14. Hsiao CH, Chang MH, Chen HL, Lee HC, Wu TC, Lin CC, et al. Univer-
card in the community alone is not sufficient to achieve sal screening for biliary atresia using an infant stool color card in Taiwan.
Hepatology 2008;47:1233-40.
earlier detection of biliary atresia. Proper usage of the stool
15. Kasai M. Treatment of biliary atresia with special reference to hepatic
color card by guardians coupled with a sound knowledge porto-enterostomy and its modifications. Prog Pediatr Surg 1974;6:5-52.
of biliary atresia among healthcare personnel is essential. n 16. Gu YH, Kato T, Harada S, Sato Y, Kakee N. Medical aid program for
chronic pediatric diseases of specified categories in Japan: current status
and future prospects. Pediatr Int 2008;50:376-87.
We thank all patients and their families for participating in this study,
17. Ibrahim M, Miyano T, Ohi R, Saeki M, Shiraki K, Tanaka K, et al. Jap-
as well as to all pediatricians who participated in our screening pro-
anese biliary atresia registry, 1989 to 1994. Tohoku J Exp Med 1997;181:
gram. We are grateful to Hideki Yamamoto, MD (Gunma Children’s
85-95.
Medical Center), Minoru Kuroiwa, MD (Children Medical Center of
18. Matsui A, Ishikawa T. Identification of infants with biliary atresia in
Toho University), and Wada Hiroki (University of Tsukuba), for their
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cooperation in the follow-up of patients. We would also like to thank
19. Shinkai M, Ohhama Y, Take H, Kitagawa N, Kudo H, Mochizuki K, et al.
Kevin Urayama (Tokyo Medical and Dental University), for his valu-
Long-term outcome of children with biliary atresia who were not trans-
able comments.
planted after the Kasai operation: >20-year experience at a children’s
hospital. J Pediatr Gastroenterol Nutr 2009;48:443-50.
Submitted for publication Jul 16, 2014; last revision received Dec 1, 2014; 20. Howard ER, MacLean G, Nio M, Donaldson N, Singer J, Ohi R. Survival
accepted Dec 19, 2014. patterns in biliary atresia and comparison of quality of life of long-term
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Figure 1. The flowchart for stool color chart screening. The numbers of participants, positive cases, false-negative cases,
patients with biliary atresia in the Tochigi cohort from August 1994 to March 2011. BA, biliary atresia; KP, Kasai procedure;
SCC, stool color card.

Stool Color Card Screening for Early Detection of Biliary Atresia and Long-Term Native Liver Survival: 6.e1
A 19-Year Cohort Study in Japan