Clinical characteristics of immunoglobulin IgG4-related sclerosing cholangitis: Comparison of cases with and without autoimmune pancreatitis in a large cohort

Itaru Naitoh; Terumi Kamisawa; Atsushi Tanaka; Takahiro Nakazawa; Kensuke Kubota; Hajime Takikawa; Michiaki Unno; Atsushi Masamune; Shigeyuki Kawa; Seiji Nakamura; Kazuichi Okazaki; collaborators

doi:10.1016/j.dld.2021.02.009

Clinical characteristics of immunoglobulin IgG4-related sclerosing cholangitis: Comparison of cases with and without autoimmune pancreatitis in a large cohort

Dig Liver Dis. 2021 Oct;53(10):1308-1314. doi: 10.1016/j.dld.2021.02.009. Epub 2021 Mar 2.

Authors

Collaborators

collaborators:
Keisuke Furumatsu¹², Shigeaki Sawai¹², Takuma Goto¹³, Toshikatsu Okumura¹³, Daisuke Suzuki¹⁴, Masayuki Otsuka¹⁴, Ikuhiro Kobori¹⁵, Masaya Tamano¹⁵, Mitsuhito Koizumi¹⁶, Yoichi Hiasa¹⁷, Naoto Kawabe¹⁷, Yoshiki Hirooka¹⁷, Satoshi Yamamoto¹⁸, Yukio Asano¹⁸, Kazuo Inui¹⁸, Akihiko Horiguchi¹⁸, Hiroyuki Watanabe¹⁹, Daishu Toya¹⁹, Katsuko Hatayama²⁰, Toshiharu Ueki²⁰, Norikatsu Kinoshita²¹, Mitsuru Sugimoto²², Hiromasa Ohira²², Tsuyoshi Mukai²³, Eiichi Tomita²³, Keisuke Iwata²⁴, Shogo Shimizu²⁴, Jun Suetsugu²⁵, Masahito Shimizu²⁵, Keiji Tsuji²⁶, Ryoko Ishida²⁷, Masanori Ito²⁷, Ryutaro Furukawa²⁸, Naoya Sakamoto²⁸, Masahiro Araki²⁹, Satoshi Tanno³⁰, Yasunari Sakamoto³¹, Tetsuhide Ito³², Satoshi Takai³³, Shinichi Ikeya³³, Takanori Yamada³⁴, Norihiko Kudara³⁵, Akinori Shimizu³⁶, Keiji Hanada³⁶, Yasunori Ichiki³⁷, Hideki Kitada³⁸, Michio Hifumi³⁸, Hiroyuki Kimura³⁹, Masayuki Kurosaki⁴⁰, Namiki Izumi⁴⁰, Hajime Sumi⁴¹, Jun-Ichi Haruta⁴¹, Katsumi Hayashi⁴², Ryo Harada⁴³, Masafumi Inoue⁴³, Shinichiro Nakamura⁴⁴, Tetsuya Ito⁴⁵, Ko Tomishima⁴⁶, Hiroyuki Isayama⁴⁶, Kyoko Oura⁴⁷, Tsutomu Masaki⁴⁷, Naoto Shimokawahara⁴⁸, Shirou Tanoue⁴⁹, Kousei Maemura⁴⁹, Akio Ido⁴⁹, Ichiro Mizushima⁵⁰, Mitsuhiro Kawano⁵⁰, Katsunori Yoshida⁵¹, Makoto Naganuma⁵², Miki Murata⁵², Akiyoshi Nishio⁵², Yuji Fujita⁵³, Takuma Teratani⁵³, Shohei Matsubara⁵⁴, Hironao Tamai⁵⁴, Yuu Yoshida⁵⁵, Ryousaku Azemoto⁵⁵, Ken Kamata⁵⁶, Tomohiro Watanabe⁵⁶, Takahiro Kurosu⁵⁷, Wasaburou Koizumi⁵⁷, Jun Fujita⁵⁸, Hideyuki Seki⁵⁸, Yasuhiro Ueda⁵⁸, Takumi Fukumoto⁵⁸, Takuhiro Kousaki⁵⁹, Kazushige Uchida⁵⁹, Toshimasa Ochiai⁶⁰, Takeshi Kawasaki⁶¹, Motohiko Tanaka⁶¹, Etsuji Ishida⁶², Kenji Notohara⁶², Hideaki Mori⁶³, Toshiyuki Mori⁶³, Hideaki Kawabata⁶⁴, Masatoshi Miyata⁶⁴, Junichi Sakagami⁶⁵, Yoshito Itoh⁶⁵, Masahiro Shiokawa⁶⁶, Hiroshi Seno⁶⁶, Noriko Watanabe⁶⁷, Itaru Naitoh⁶⁸, Hiromi Kataoka⁶⁸, Toshinori Aoki⁶⁹, Mitsuhiro Fujishiro⁶⁹, Toru Niihara⁷⁰, Hiroto Nishimata⁷⁰, Akira Mitoro⁷¹, Hitoshi Yoshiji⁷¹, Motoyuki Yoshida⁷², Masafumi Ikeda⁷³, Kengo Tomita⁷⁴, Ryota Hokari⁷⁵, Kenji Hayasaka⁷⁴, Yuji Amano⁷⁴, Kazuhiko Shioji⁷⁶, Kazunao Hayashi⁷⁷, Shuji Terai⁷⁷, Michiko Nakajima⁷⁸, Junya Yamahana⁷⁹, Ryusuke Matsumoto⁸⁰, Hideaki Kikuchi⁸⁰, Akira Kanamori⁸¹, Seiki Kiriyama⁸¹, Shinichi Iwatsu⁸², Yuji Kato⁸², Shigeru Horiguchi⁸³, Takahito Yagi⁸³, Hiroyuki Okada⁸³, Kazuyoshi Ohkawa⁸⁴, Motohiro Hirao⁸⁵, Naoki Hiramatsu⁸⁵, Noriko Oza⁸⁶, Haruo Imamura⁸⁷, Takeshi Baba⁸⁸, Shigeru Nakano⁸⁸, Tetsuya Shinobi⁸⁹, Shomei Ryozawa⁹⁰, Masayo Motoya⁹¹, Hiroshi Nakase⁹¹, Noboru Kinoshita⁹², Kei Ito⁹³, Tatsuya Miyake⁹⁴, Naruaki Kohge⁹⁴, Hiroshi Tobita⁹⁵, Satoru Joshita⁹⁶, Takeji Umemura⁹⁶, Shinya Kawaguchi⁹⁷, Kazuya Ohno⁹⁷, Koichi Sonobe⁹⁸, Akihiko Satoh⁹⁹, Tooru Shimosegawa⁹⁹, Fumihiko Miura¹⁰⁰, Minami Yagi¹⁰⁰, Keiji Sano¹⁰⁰, Atsushi Tanaka¹⁰⁰, Toshifumi Kin¹⁰¹, Akio Katanuma¹⁰¹, Kazuhiko Koike¹⁰², Shin Miura¹⁰³, Atsushi Masamune¹⁰³, Youhei Kawashima¹⁰⁴, Tatehiro Kagawa¹⁰⁴, Seishin Azuma¹⁰⁵, Mamoru Watanabe¹⁰⁵, Mitsuyoshi Honjyo¹⁰⁶, Takao Itoi¹⁰⁶, Akira Honda¹⁰⁷, Katsumasa Kobayashi¹⁰⁸, Toru Asano¹⁰⁹, Terumi Kamisawa¹⁰⁷, Suguru Mizuno¹⁰⁸, Kazuhiko Koike¹⁰⁸, Takayoshi Nishino¹¹⁰, Hideaki Taniguchi¹¹¹, Kazuto Tajiri¹¹¹, Ichiro Yasuda¹¹¹, Yoshiya Tanaka¹¹², Shinji Oe¹¹², Masaru Harada¹¹², Masanao Kurata¹¹³, Mituharu Fukasawa¹¹⁴, Nobuyuki Enomoto¹¹⁴, Yuki Kawaji¹¹⁵, Masayuki Kitano¹¹⁵, Yuko Nishise¹¹⁶, Hidetoshi Hirakawa¹¹⁶, Tetsuya Ishizawa¹¹⁷, Yoshiyuki Ueno¹¹⁷, Miyuki Kaino¹¹⁸, Yuko Fujimoto¹¹⁹, Isao Sakaida¹¹⁹, Kensuke Kubota¹²⁰

Affiliations

¹ Department of Gastroenterology, Nagoya City University Graduate School of Medical Sciences, 1 Kawasumi, Mizuho-cho, Mizuho-ku Nagoya 467-8601, Japan. Electronic address: [email protected].
² Department of Internal Medicine, Tokyo Metropolitan, Komagome Hospital, Tokyo, Japan.
³ Department of Medicine, Teikyo University School of Medicine, Tokyo, Japan.
⁴ Department of Gastroenterology, Nagoya City University Graduate School of Medical Sciences, 1 Kawasumi, Mizuho-cho, Mizuho-ku Nagoya 467-8601, Japan.
⁵ Department of Endoscopy, Yokohama City University Hospital, Yokohama, Japan.
⁶ Faculty of Medical Technology, Teikyo University, Tokyo, Japan.
⁷ Division of Hepato-Biliary Pancreatic Surgery, Tohoku University Graduate School, of Medicine, Sendai, Japan.
⁸ Division of Gastroenterology, Tohoku University Graduate School of Medicine, Sendai, Japan.
⁹ Department of Internal Medicine, Matsumoto Dental University, Matsumoto, Japan.
¹⁰ Section of Oral and Maxillofacial Oncology, Division of Maxillofacial Diagnostic and Surgical Sciences, Faculty of Dental Science, Kyushu University, Fukuoka, Japan.
¹¹ Kansai Medical University Kori Hospital, Kori, Japan.
¹² Akashi Medical Center, Japan.
¹³ Asahikawa Medical College Hospital, Japan.
¹⁴ Chiba University, Japan.
¹⁵ Dokkyo Medical University Saitama Medical Center, Japan.
¹⁶ Ehime University, Japan.
¹⁷ Fujita Health University, Japan.
¹⁸ Fujita Health University Bantane Hospital, Japan.
¹⁹ Fukui-ken Saiseikai Hospital, Japan.
²⁰ Fukuoka University Chikushi Hospital, Japan.
²¹ FukuokaWajiro Hospital, Japan.
²² Fukushima Medical University, Japan.
²³ Gifu Municipal Hospital, Japan.
²⁴ Gifu Prefectural General Medical Center, Japan.
²⁵ Gifu University, Japan.
²⁶ Hiroshima Red Cross Hospital and Atomic-bomb Survivors Hospital, Japan.
²⁷ Hiroshima University, Japan.
²⁸ Hokkaido University, Japan.
²⁹ Ibaraki Prefectural Central Hospital, Ibaraki Cancer Center, Japan.
³⁰ IMS Sapporo Digestive Disease Center General Hospital, Japan.
³¹ IUHW Atami Hospital, Japan.
³² IUHW Fukuoka Sanno Hospital, Japan.
³³ Iwaki City Medical Center, Japan.
³⁴ Iwata City Hospital, Japan.
³⁵ Iwate prefectural Ofunato Hospital, Japan.
³⁶ JA Onomichi General Hospital, Japan.
³⁷ Japan Community Health Care Organization Kyushu Hospital, Japan.
³⁸ Japanese Red Cross Kumamoto Hospital, Japan.
³⁹ Japanese Red Cross Kyoto Daiichi Hospital, Japan.
⁴⁰ Japanese Red Cross Musashino Hospital, Japan.
⁴¹ Japanese Red Cross Nagoya Daiichi Hospital, Japan.
⁴² Japanese Red Cross Nagoya Daini Hospital, Japan.
⁴³ Japanese Red Cross Okayama Hospital, Japan.
⁴⁴ Japanese Red Cross Society Himeji Hospital, Japan.
⁴⁵ Japanese Red Cross Society Nagano Hospital, Japan.
⁴⁶ Juntendo University, Japan.
⁴⁷ Kagawa University, Japan.
⁴⁸ Kagoshima Medical Association Hospital, Japan.
⁴⁹ Kagoshima University, Japan.
⁵⁰ Kanazawa University, Japan.
⁵¹ Kansai Medical University, Japan.
⁵² Kansai Medical University Medical Center, Japan.
⁵³ Kanto Medical Center, NTT East, Japan.
⁵⁴ Kawasaki Municipal Hospital, Japan.
⁵⁵ Kimitsu Chuo Hospital, Japan.
⁵⁶ Kindai University, Japan.
⁵⁷ Kitasato University, Japan.
⁵⁸ KKR Sapporo Medical Center, Japan.
⁵⁹ Kochi University, Japan.
⁶⁰ Koga General Hospital, Japan.
⁶¹ Kumamoto University, Japan.
⁶² Kurashiki Central Hospital, Japan.
⁶³ Kyorin University, Japan.
⁶⁴ Kyoto Okamoto Memorial Hospital, Japan.
⁶⁵ Kyoto prefectural University of Medicine, Japan.
⁶⁶ Kyoto University, Japan.
⁶⁷ Mie Central Medical Center, Japan.
⁶⁸ Nagoya City University, Japan.
⁶⁹ Nagoya University, Japan.
⁷⁰ Nanpuh Hospital, Japan.
⁷¹ Nara Medical University, Japan.
⁷² Nara Prefectural Seiwa Medical Center, Japan.
⁷³ National Cancer Center Hospital East, Japan.
⁷⁴ New Tokyo Hospital, Japan.
⁷⁵ National Defense Medical College, Japan.
⁷⁶ Niigata Cancer Center Hospital, Japan.
⁷⁷ Niigata University, Japan.
⁷⁸ Noheji General Hospital, Japan.
⁷⁹ Noto General Hospital, Japan.
⁸⁰ Obihiro Kousei Hospital, Japan.
⁸¹ Ogaki Municipal Hospital, Japan.
⁸² Oita Prefectural Hospital, Japan.
⁸³ Okayama University, Japan.
⁸⁴ Osaka International Cancer Institute, Japan.
⁸⁵ Osaka Rosai Hospital, Japan.
⁸⁶ Saga-ken Medical Centre Koseikan, Japan.
⁸⁷ Saiseikai Kumamoto Hospital, Japan.
⁸⁸ Saiseikai Yokohamashi Tobu Hospital, Japan.
⁸⁹ Saitama cooperative hospital, Japan.
⁹⁰ Saitama Medical University International Medical Center, Japan.
⁹¹ Sapporo Medical University, Japan.
⁹² Sasebo Chuo Hospital, Japan.
⁹³ Sendai Open Hospital, Japan.
⁹⁴ Shimane Prefectural Central Hospital, Japan.
⁹⁵ Shimane University, Japan.
⁹⁶ Shinshu University, Japan.
⁹⁷ Shizuoka General Hospital, Japan.
⁹⁸ Sonobe Hospital, Japan.
⁹⁹ South Miyagi Medical Center, Japan.
¹⁰⁰ Teikyo University, Japan.
¹⁰¹ Teine-keijinnkai Hospital, Japan.
¹⁰² The Jikei University Daisan Hospital, Japan.
¹⁰³ Tohoku University, Japan.
¹⁰⁴ Tokai University, Japan.
¹⁰⁵ Tokyo Medical and Dental University, Japan.
¹⁰⁶ Tokyo Medical University, Japan.
¹⁰⁷ Tokyo Metropolitan Cancer and Infectious Diseases Center Komagome Hospital, Japan.
¹⁰⁸ Tokyo University, Japan.
¹⁰⁹ Tokyo Metropolitan Bokutoh Hospital, Japan.
¹¹⁰ Tokyo Womens Medical University Yachiyo Medical Center, Japan.
¹¹¹ Toyama University, Japan.
¹¹² University of Occupational and Environmental Health, Japan.
¹¹³ University of Tsukuba, Japan.
¹¹⁴ University of Yamanashi, Japan.
¹¹⁵ Wakayama Medical University, Japan.
¹¹⁶ Yamagata City Hospital Saiseikan, Japan.
¹¹⁷ Yamagata University, Japan.
¹¹⁸ Yamaguchi Rosai Hospital, Japan.
¹¹⁹ Yamaguchi University, Japan.
¹²⁰ Yokohama City University Hospital, Japan.

PMID: 33664004
DOI: 10.1016/j.dld.2021.02.009

Abstract

Background: The clinical characteristics of IgG4-related sclerosing cholangitis (IgG4-SC) especially without autoimmune pancreatitis (AIP) have not been investigated in a large cohort.

Aims: To clarify the clinical characteristics of IgG4-SC and IgG4-SC without AIP.

Methods: We retrospectively reviewed imaging, serology, other organ involvement (OOI) and histology of 872 patients with IgG4-SC who participated in a Japanese nationwide survey in 2019, and compared these items between IgG4-SC with and without AIP.

Results: AIP was present in 83.7% (730/872) of IgG4-SC. In IgG4-SC, bile duct wall thickening was observed on ultrasound (528/650; 81.2%), computed tomography (375/525; 71.4%) and magnetic resonance imaging or cholangiopancreatography (290/440; 65.9%). An elevated serum IgG4 level (≥ 135 mg/dL) was found in 88.0% (322/366). IgG4-related OOI other than AIP was observed in 25.2% (211/836). The proportion of females was significantly higher in IgG4-SC without AIP (28.9% vs. 20.1%; p = 0.025). Hilar stricture was the most common cholangiographic type in IgG4-SC without AIP (39/107; 36.4%).There were no significant differences between IgG4-SC with and without AIP in the rates of bile duct wall thickening, elevated serum IgG4 level, or IgG4-related OOI.

Conclusions: The clinical characteristics of IgG4-SC was similar between IgG4-SC with and without AIP in a large cohort.

Keywords: Autoimmune pancreatitis; Cholangiocarcinoma; IgG4-related disease; IgG4-related sclerosing cholangitis; Isolated IgG4-related sclerosing cholangitis; Primary sclerosing cholangitis.

Publication types

Multicenter Study

MeSH terms

Autoimmune Pancreatitis / epidemiology*
Bile Ducts / diagnostic imaging
Bile Ducts / pathology*
Cholangiography
Cholangitis, Sclerosing / diagnosis
Cholangitis, Sclerosing / epidemiology*
Female
Humans
Immunoglobulin G / blood
Immunoglobulin G4-Related Disease / diagnosis
Immunoglobulin G4-Related Disease / epidemiology*
Japan / epidemiology
Male
Retrospective Studies
Surveys and Questionnaires

Substances

Immunoglobulin G