A Japanese prospective multicenter study of urinary oxysterols in biliary atresia

Abstract Diagnosis of biliary atresia (BA) can involve uncertainties. In the present prospective multicenter study, we considered whether urinary oxysterols represent a useful marker for diagnosis of BA in Japanese children. Subjects under 6 months old at 7 pediatric centers in Japan were prospectiv...

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Autores principales: Ken-ichiro Konishi, Tatsuki Mizuochi, Hajime Takei, Ryosuke Yasuda, Hirotaka Sakaguchi, Jun Ishihara, Yugo Takaki, Masahiro Kinoshita, Naoki Hashizume, Suguru Fukahori, Hiromichi Shoji, Go Miyano, Koichiro Yoshimaru, Toshiharu Matsuura, Yukihiro Sanada, Takahisa Tainaka, Hiroo Uchida, Yumiko Kubo, Hiromu Tanaka, Hideyuki Sasaki, Tsuyoshi Murai, Jun Fujishiro, Yushiro Yamashita, Masaki Nio, Hiroshi Nittono, Akihiko Kimura
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Publicado: Nature Portfolio 2021
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spelling oai:doaj.org-article:939c855337f941dcb4b91194b34430a72021-12-02T11:35:59ZA Japanese prospective multicenter study of urinary oxysterols in biliary atresia10.1038/s41598-021-84445-w2045-2322https://doaj.org/article/939c855337f941dcb4b91194b34430a72021-03-01T00:00:00Zhttps://doi.org/10.1038/s41598-021-84445-whttps://doaj.org/toc/2045-2322Abstract Diagnosis of biliary atresia (BA) can involve uncertainties. In the present prospective multicenter study, we considered whether urinary oxysterols represent a useful marker for diagnosis of BA in Japanese children. Subjects under 6 months old at 7 pediatric centers in Japan were prospectively enrolled, including patients with cholestasis and healthy controls (HC) without liver disease. Patients with cholestasis constituted 2 groups representing BA patients and others with cholestasis from other causes (non-BA). We quantitatively analyzed 7 oxysterols including 4β-, 20(S)-, 22(S)-, 22(R)-, 24(S)-, 25-, and 27-hydroxycholesterol by liquid chromatography/electrospray ionization-tandem mass spectrometry. Enrolled subjects included 14 with BA (median age 68 days; range 26–170) and 10 non-BA cholestatic controls (59; 14–162), as well as 10 HC (57; 25–120). Total urinary oxysterols were significantly greater in BA (median, 153.0 μmol/mol creatinine; range 24.1–486.7; P < 0.001) and non-BA (36.2; 5.8–411.3; P < 0.05) than in HC (2.7; 0.8–7.6). In patients with BA, urinary 27-hydroxycholesterol (3.61; 0.42–11.09; P < 0.01) was significantly greater than in non-BA (0.71; 0–5.62). In receiver operating characteristic (ROC) curve analysis for distinguishing BA from non-BA, the area under the ROC curve for urinary 27-hydroxycholesterol was 0.83. In conclusion, this first report of urinary oxysterol analysis in patients with BA indicated that 27-hydroxycholesterol may be a useful marker for distinguishing BA from other causes of neonatal cholestasis.Ken-ichiro KonishiTatsuki MizuochiHajime TakeiRyosuke YasudaHirotaka SakaguchiJun IshiharaYugo TakakiMasahiro KinoshitaNaoki HashizumeSuguru FukahoriHiromichi ShojiGo MiyanoKoichiro YoshimaruToshiharu MatsuuraYukihiro SanadaTakahisa TainakaHiroo UchidaYumiko KuboHiromu TanakaHideyuki SasakiTsuyoshi MuraiJun FujishiroYushiro YamashitaMasaki NioHiroshi NittonoAkihiko KimuraNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 11, Iss 1, Pp 1-8 (2021)
institution DOAJ
collection DOAJ
language EN
topic Medicine
R
Science
Q
spellingShingle Medicine
R
Science
Q
Ken-ichiro Konishi
Tatsuki Mizuochi
Hajime Takei
Ryosuke Yasuda
Hirotaka Sakaguchi
Jun Ishihara
Yugo Takaki
Masahiro Kinoshita
Naoki Hashizume
Suguru Fukahori
Hiromichi Shoji
Go Miyano
Koichiro Yoshimaru
Toshiharu Matsuura
Yukihiro Sanada
Takahisa Tainaka
Hiroo Uchida
Yumiko Kubo
Hiromu Tanaka
Hideyuki Sasaki
Tsuyoshi Murai
Jun Fujishiro
Yushiro Yamashita
Masaki Nio
Hiroshi Nittono
Akihiko Kimura
A Japanese prospective multicenter study of urinary oxysterols in biliary atresia
description Abstract Diagnosis of biliary atresia (BA) can involve uncertainties. In the present prospective multicenter study, we considered whether urinary oxysterols represent a useful marker for diagnosis of BA in Japanese children. Subjects under 6 months old at 7 pediatric centers in Japan were prospectively enrolled, including patients with cholestasis and healthy controls (HC) without liver disease. Patients with cholestasis constituted 2 groups representing BA patients and others with cholestasis from other causes (non-BA). We quantitatively analyzed 7 oxysterols including 4β-, 20(S)-, 22(S)-, 22(R)-, 24(S)-, 25-, and 27-hydroxycholesterol by liquid chromatography/electrospray ionization-tandem mass spectrometry. Enrolled subjects included 14 with BA (median age 68 days; range 26–170) and 10 non-BA cholestatic controls (59; 14–162), as well as 10 HC (57; 25–120). Total urinary oxysterols were significantly greater in BA (median, 153.0 μmol/mol creatinine; range 24.1–486.7; P < 0.001) and non-BA (36.2; 5.8–411.3; P < 0.05) than in HC (2.7; 0.8–7.6). In patients with BA, urinary 27-hydroxycholesterol (3.61; 0.42–11.09; P < 0.01) was significantly greater than in non-BA (0.71; 0–5.62). In receiver operating characteristic (ROC) curve analysis for distinguishing BA from non-BA, the area under the ROC curve for urinary 27-hydroxycholesterol was 0.83. In conclusion, this first report of urinary oxysterol analysis in patients with BA indicated that 27-hydroxycholesterol may be a useful marker for distinguishing BA from other causes of neonatal cholestasis.
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author Ken-ichiro Konishi
Tatsuki Mizuochi
Hajime Takei
Ryosuke Yasuda
Hirotaka Sakaguchi
Jun Ishihara
Yugo Takaki
Masahiro Kinoshita
Naoki Hashizume
Suguru Fukahori
Hiromichi Shoji
Go Miyano
Koichiro Yoshimaru
Toshiharu Matsuura
Yukihiro Sanada
Takahisa Tainaka
Hiroo Uchida
Yumiko Kubo
Hiromu Tanaka
Hideyuki Sasaki
Tsuyoshi Murai
Jun Fujishiro
Yushiro Yamashita
Masaki Nio
Hiroshi Nittono
Akihiko Kimura
author_facet Ken-ichiro Konishi
Tatsuki Mizuochi
Hajime Takei
Ryosuke Yasuda
Hirotaka Sakaguchi
Jun Ishihara
Yugo Takaki
Masahiro Kinoshita
Naoki Hashizume
Suguru Fukahori
Hiromichi Shoji
Go Miyano
Koichiro Yoshimaru
Toshiharu Matsuura
Yukihiro Sanada
Takahisa Tainaka
Hiroo Uchida
Yumiko Kubo
Hiromu Tanaka
Hideyuki Sasaki
Tsuyoshi Murai
Jun Fujishiro
Yushiro Yamashita
Masaki Nio
Hiroshi Nittono
Akihiko Kimura
author_sort Ken-ichiro Konishi
title A Japanese prospective multicenter study of urinary oxysterols in biliary atresia
title_short A Japanese prospective multicenter study of urinary oxysterols in biliary atresia
title_full A Japanese prospective multicenter study of urinary oxysterols in biliary atresia
title_fullStr A Japanese prospective multicenter study of urinary oxysterols in biliary atresia
title_full_unstemmed A Japanese prospective multicenter study of urinary oxysterols in biliary atresia
title_sort japanese prospective multicenter study of urinary oxysterols in biliary atresia
publisher Nature Portfolio
publishDate 2021
url https://doaj.org/article/939c855337f941dcb4b91194b34430a7
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