Hypertrophic pachymeningitis in ANCA-associated vasculitis: a cross-sectional and multi-institutional study in Japan (J-CANVAS)

Abstract Background This study investigated the characteristics of hypertrophic pachymeningitis (HP) in antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV), using information from a multicenter study in Japan. Methods We analyzed the clinical information of 663 Asian patients with...

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Main Authors: Yasuhiro Shimojima, Dai Kishida, Takanori Ichikawa, Takashi Kida, Nobuyuki Yajima, Satoshi Omura, Daiki Nakagomi, Yoshiyuki Abe, Masatoshi Kadoya, Naoho Takizawa, Atsushi Nomura, Yuji Kukida, Naoya Kondo, Yasuhiko Yamano, Takuya Yanagida, Koji Endo, Shintaro Hirata, Kiyoshi Matsui, Tohru Takeuchi, Kunihiro Ichinose, Masaru Kato, Ryo Yanai, Yusuke Matsuo, Ryo Nishioka, Ryota Okazaki, Tomoaki Takata, Takafumi Ito, Mayuko Moriyama, Ayuko Takatani, Yoshia Miyawaki, Toshiko Ito-Ihara, Takashi Kawaguchi, Yutaka Kawahito, Yoshiki Sekijima
Format: Article
Language:English
Published: BMC 2022-08-01
Series:Arthritis Research & Therapy
Subjects:
Online Access:https://doi.org/10.1186/s13075-022-02898-4
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author Yasuhiro Shimojima
Dai Kishida
Takanori Ichikawa
Takashi Kida
Nobuyuki Yajima
Satoshi Omura
Daiki Nakagomi
Yoshiyuki Abe
Masatoshi Kadoya
Naoho Takizawa
Atsushi Nomura
Yuji Kukida
Naoya Kondo
Yasuhiko Yamano
Takuya Yanagida
Koji Endo
Shintaro Hirata
Kiyoshi Matsui
Tohru Takeuchi
Kunihiro Ichinose
Masaru Kato
Ryo Yanai
Yusuke Matsuo
Ryo Nishioka
Ryota Okazaki
Tomoaki Takata
Takafumi Ito
Mayuko Moriyama
Ayuko Takatani
Yoshia Miyawaki
Toshiko Ito-Ihara
Takashi Kawaguchi
Yutaka Kawahito
Yoshiki Sekijima
author_facet Yasuhiro Shimojima
Dai Kishida
Takanori Ichikawa
Takashi Kida
Nobuyuki Yajima
Satoshi Omura
Daiki Nakagomi
Yoshiyuki Abe
Masatoshi Kadoya
Naoho Takizawa
Atsushi Nomura
Yuji Kukida
Naoya Kondo
Yasuhiko Yamano
Takuya Yanagida
Koji Endo
Shintaro Hirata
Kiyoshi Matsui
Tohru Takeuchi
Kunihiro Ichinose
Masaru Kato
Ryo Yanai
Yusuke Matsuo
Ryo Nishioka
Ryota Okazaki
Tomoaki Takata
Takafumi Ito
Mayuko Moriyama
Ayuko Takatani
Yoshia Miyawaki
Toshiko Ito-Ihara
Takashi Kawaguchi
Yutaka Kawahito
Yoshiki Sekijima
author_sort Yasuhiro Shimojima
collection DOAJ
description Abstract Background This study investigated the characteristics of hypertrophic pachymeningitis (HP) in antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV), using information from a multicenter study in Japan. Methods We analyzed the clinical information of 663 Asian patients with AAV (total AAV), including 558 patients with newly diagnosed AAV and 105 with relapsed AAV. Clinical findings were compared between patients with and without HP. To elucidate the relevant manifestations for HP development, multivariable logistic regression analyses were additionally performed. Results Of the patients with AAV (mean age, 70.2 ± 13.5 years), HP was noted in 30 (4.52%), including 20 (3.58%) with newly diagnosed AAV and 10 (9.52%) with relapsed AAV. Granulomatosis with polyangiitis (GPA) was classified in 50% of patients with HP. A higher prevalence of GPA was significantly observed in patients with HP than in those without HP in total AAV and newly diagnosed AAV (p < 0.001). In newly diagnosed AAV, serum proteinase 3 (PR3)-ANCA positivity was significantly higher in patients with HP than in those without HP (p = 0.030). Patients with HP significantly had ear, nose, and throat (ENT) (odds ratio [OR] 1.48, 95% confidence interval [CI] 1.03–2.14, p = 0.033) and mucous membrane/eye manifestations (OR 5.99, 95% CI 2.59–13.86, p < 0.0001) in total AAV. Moreover, they significantly had conductive hearing loss (OR 11.6, 95% CI 4.51–29.57, p < 0.0001) and sudden visual loss (OR 20.9, 95% CI 5.24–85.03, p < 0.0001). Conclusion GPA was predominantly observed in patients with HP. Furthermore, in newly diagnosed AAV, patients with HP showed significantly higher PR3-ANCA positivity than those without HP. The ear and eye manifestations may be implicated in HP development.
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spelling doaj.art-11931efa639a4ce9b88ead58bb14ea4a2022-12-22T03:05:49ZengBMCArthritis Research & Therapy1478-63622022-08-0124111110.1186/s13075-022-02898-4Hypertrophic pachymeningitis in ANCA-associated vasculitis: a cross-sectional and multi-institutional study in Japan (J-CANVAS)Yasuhiro Shimojima0Dai Kishida1Takanori Ichikawa2Takashi Kida3Nobuyuki Yajima4Satoshi Omura5Daiki Nakagomi6Yoshiyuki Abe7Masatoshi Kadoya8Naoho Takizawa9Atsushi Nomura10Yuji Kukida11Naoya Kondo12Yasuhiko Yamano13Takuya Yanagida14Koji Endo15Shintaro Hirata16Kiyoshi Matsui17Tohru Takeuchi18Kunihiro Ichinose19Masaru Kato20Ryo Yanai21Yusuke Matsuo22Ryo Nishioka23Ryota Okazaki24Tomoaki Takata25Takafumi Ito26Mayuko Moriyama27Ayuko Takatani28Yoshia Miyawaki29Toshiko Ito-Ihara30Takashi Kawaguchi31Yutaka Kawahito32Yoshiki Sekijima33Department of Medicine (Neurology and Rheumatology), Shinshu University School of MedicineDepartment of Medicine (Neurology and Rheumatology), Shinshu University School of MedicineDepartment of Medicine (Neurology and Rheumatology), Shinshu University School of MedicineInflammation and Immunology, Graduate School of Medical Science, Kyoto Prefectural University of MedicineDivision of Rheumatology, Department of Medicine, Showa University School of MedicineInflammation and Immunology, Graduate School of Medical Science, Kyoto Prefectural University of MedicineDepartment of Rheumatology, University of Yamanashi HospitalDepartment of Internal Medicine and Rheumatology, Juntendo UniversityCenter for Rheumatic Disease, Japanese Red Cross Society Kyoto Daiichi HospitalDepartment of Rheumatology, Chubu Rosai HospitalImmuno-Rheumatology Center, St. Luke’s International HospitalDepartment of Rheumatology, Japanese Red Cross Society Kyoto Daini HospitalDepartment of Nephrology, Kyoto Katsura HospitalDepartment of Respiratory Medicine and Allergy, Tosei General HospitalDepartment of Hematology and Rheumatology, Kagoshima University HospitalDepartment of Pharmacoepidemiology, Graduate School of Medicine and Public Health, Kyoto UniversityDepartment of Clinical Immunology and Rheumatology, Hiroshima University HospitalDepartment of Diabetes, Endocrinology and Clinical Immunology, Hyogo Medical University School of MedicineDepartment of Internal Medicine (IV), Osaka Medical and Pharmaceutical UniversityDepartment of Immunology and Rheumatology, Division of Advanced Preventive Medical Sciences, Nagasaki University Graduate School of Biomedical SciencesDepartment of Rheumatology, Endocrinology and Nephrology, Graduate School of Medicine, Hokkaido UniversityDivision of Rheumatology, Department of Medicine, Showa University School of MedicineDepartment of Rheumatology, Tokyo Kyosai HospitalDepartment of Rheumatology, Graduate School of Medical Science, Kanazawa UniversityDivision of Respiratory Medicine and Rheumatology, Department of Multidisciplinary Internal Medicine, Faculty of Medicine, Tottori UniversityDivision of Gastroenterology and Nephrology, Tottori UniversityDivision of Nephrology, Shimane University HospitalDepartment of Rheumatology, Shimane University Faculty of MedicineRheumatic Disease Center, Sasebo Chuo HospitalDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical SciencesThe Clinical and Translational Research Center, University Hospital, Kyoto Prefectural University of MedicineDepartment of Practical Pharmacy, Tokyo University of Pharmacy and Life SciencesInflammation and Immunology, Graduate School of Medical Science, Kyoto Prefectural University of MedicineDepartment of Medicine (Neurology and Rheumatology), Shinshu University School of MedicineAbstract Background This study investigated the characteristics of hypertrophic pachymeningitis (HP) in antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV), using information from a multicenter study in Japan. Methods We analyzed the clinical information of 663 Asian patients with AAV (total AAV), including 558 patients with newly diagnosed AAV and 105 with relapsed AAV. Clinical findings were compared between patients with and without HP. To elucidate the relevant manifestations for HP development, multivariable logistic regression analyses were additionally performed. Results Of the patients with AAV (mean age, 70.2 ± 13.5 years), HP was noted in 30 (4.52%), including 20 (3.58%) with newly diagnosed AAV and 10 (9.52%) with relapsed AAV. Granulomatosis with polyangiitis (GPA) was classified in 50% of patients with HP. A higher prevalence of GPA was significantly observed in patients with HP than in those without HP in total AAV and newly diagnosed AAV (p < 0.001). In newly diagnosed AAV, serum proteinase 3 (PR3)-ANCA positivity was significantly higher in patients with HP than in those without HP (p = 0.030). Patients with HP significantly had ear, nose, and throat (ENT) (odds ratio [OR] 1.48, 95% confidence interval [CI] 1.03–2.14, p = 0.033) and mucous membrane/eye manifestations (OR 5.99, 95% CI 2.59–13.86, p < 0.0001) in total AAV. Moreover, they significantly had conductive hearing loss (OR 11.6, 95% CI 4.51–29.57, p < 0.0001) and sudden visual loss (OR 20.9, 95% CI 5.24–85.03, p < 0.0001). Conclusion GPA was predominantly observed in patients with HP. Furthermore, in newly diagnosed AAV, patients with HP showed significantly higher PR3-ANCA positivity than those without HP. The ear and eye manifestations may be implicated in HP development.https://doi.org/10.1186/s13075-022-02898-4Hypertrophic pachymeningitisAntineutrophil cytoplasmic antibodyANCA-associated vasculitisGranulomatosis with polyangiitisEar, nose, and throatMucous membranes/eyes
spellingShingle Yasuhiro Shimojima
Dai Kishida
Takanori Ichikawa
Takashi Kida
Nobuyuki Yajima
Satoshi Omura
Daiki Nakagomi
Yoshiyuki Abe
Masatoshi Kadoya
Naoho Takizawa
Atsushi Nomura
Yuji Kukida
Naoya Kondo
Yasuhiko Yamano
Takuya Yanagida
Koji Endo
Shintaro Hirata
Kiyoshi Matsui
Tohru Takeuchi
Kunihiro Ichinose
Masaru Kato
Ryo Yanai
Yusuke Matsuo
Ryo Nishioka
Ryota Okazaki
Tomoaki Takata
Takafumi Ito
Mayuko Moriyama
Ayuko Takatani
Yoshia Miyawaki
Toshiko Ito-Ihara
Takashi Kawaguchi
Yutaka Kawahito
Yoshiki Sekijima
Hypertrophic pachymeningitis in ANCA-associated vasculitis: a cross-sectional and multi-institutional study in Japan (J-CANVAS)
Arthritis Research & Therapy
Hypertrophic pachymeningitis
Antineutrophil cytoplasmic antibody
ANCA-associated vasculitis
Granulomatosis with polyangiitis
Ear, nose, and throat
Mucous membranes/eyes
title Hypertrophic pachymeningitis in ANCA-associated vasculitis: a cross-sectional and multi-institutional study in Japan (J-CANVAS)
title_full Hypertrophic pachymeningitis in ANCA-associated vasculitis: a cross-sectional and multi-institutional study in Japan (J-CANVAS)
title_fullStr Hypertrophic pachymeningitis in ANCA-associated vasculitis: a cross-sectional and multi-institutional study in Japan (J-CANVAS)
title_full_unstemmed Hypertrophic pachymeningitis in ANCA-associated vasculitis: a cross-sectional and multi-institutional study in Japan (J-CANVAS)
title_short Hypertrophic pachymeningitis in ANCA-associated vasculitis: a cross-sectional and multi-institutional study in Japan (J-CANVAS)
title_sort hypertrophic pachymeningitis in anca associated vasculitis a cross sectional and multi institutional study in japan j canvas
topic Hypertrophic pachymeningitis
Antineutrophil cytoplasmic antibody
ANCA-associated vasculitis
Granulomatosis with polyangiitis
Ear, nose, and throat
Mucous membranes/eyes
url https://doi.org/10.1186/s13075-022-02898-4
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