Literature DB >> 28168662

Multicentric Castleman's disease associated with IgA vasculitis (Henoch-Schönlein purpura) responding well to tocilizumab: a case report.

Yoichi Oshima1, Junichi Hoshino2, Tatsuya Suwabe2, Noriko Hayami2, Masayuki Yamanouchi2, Akinari Sekine2, Toshiharu Ueno2, Hiroki Mizuno2, Junko Yabuuchi2, Aya Imafuku2, Masahiro Kawada2, Rikako Hiramatsu2, Eiko Hasegawa2, Naoki Sawa2, Kenmei Takaichi2,3, Nobukazu Hayashi4, Takeshi Fujii5, Yoshifumi Ubara2,3.   

Abstract

A 41-year-old man was referred to our hospital for the evaluation of hypergammaglobulinemia (IgG 2898 mg/dL and IgA 587 mg/dL), inflammation (CRP 6.7 mg/dL and serum interleukin-6 (IL-6) 15.1 ng/L), and anemia (Hb 10.9 mg/dL). Castleman's disease (CD) was diagnosed by axillary lymph node biopsy. Five months later, painful purpura (multiple palpable 5 mm lesions) developed on his legs, gradually spreading to the upper limbs, thighs, and trunk, accompanied by arthralgia of the wrists, ankles, and knees. Skin biopsy revealed leukocytoclastic vasculitis with IgA deposits in dermal vessels. Accordingly, IgA vasculitis (Henoch-Schönlein purpura) was diagnosed. Tocilizumab (an anti-IL-6 receptor antibody) was administered intravenously at 8 mg/kg and treatment was repeated at monthly intervals. His purpura and clinical findings specific to CD improved rapidly. CD is well known to cause various skin lesions. The findings in this case indicate that overproduction of IL-6 contributes to IgA vasculitis (Henoch-Schönlein purpura) as well as to the pathogenesis of CD.

Entities:  

Keywords:  Castleman’s disease; Henoch-Schönlein purpura; IgA vasculitis; Interleukin-6; Leukocytoclastic vasculitis; Tocilizumab

Mesh:

Substances:

Year:  2017        PMID: 28168662     DOI: 10.1007/s10067-017-3568-y

Source DB:  PubMed          Journal:  Clin Rheumatol        ISSN: 0770-3198            Impact factor:   3.650


  18 in total

1.  CASE records of the Massachusetts General Hospital Weekly Clinicopathological Exercises: Case 40011.

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Journal:  N Engl J Med       Date:  1954-01-07       Impact factor: 91.245

2.  Castleman's tumours and production of autoantibody in paraneoplastic pemphigus.

Authors:  Liangchun Wang; Dingfang Bu; Yong Yang; Xixue Chen; Xuejun Zhu
Journal:  Lancet       Date:  2004-02-14       Impact factor: 79.321

3.  The utility of plasma vascular endothelial growth factor levels in the diagnosis and follow-up of patients with POEMS syndrome.

Authors:  Anita D'Souza; Suzanne R Hayman; Francis Buadi; Michelle Mauermann; Martha Q Lacy; Morie A Gertz; Robert A Kyle; Shaji Kumar; Philip R Greipp; John A Lust; Stephen J Russell; Steven Zeldenrust; David Dingli; Thomas E Witzig; S Vincent Rajkumar; Angela Dispenzieri
Journal:  Blood       Date:  2011-08-31       Impact factor: 22.113

4.  Raised serum levels of interleukins 6 and 8 and antiphospholipid antibodies in an adult patient with Henoch-Schönlein purpura.

Authors:  S Kimura; S Takeuchi; Y Soma; T Kawakami
Journal:  Clin Exp Dermatol       Date:  2013-10       Impact factor: 3.470

Review 5.  What is known about the epidemiology of the vasculitides?

Authors:  Richard A Watts; Suzanne Lane; David G I Scott
Journal:  Best Pract Res Clin Rheumatol       Date:  2005-04       Impact factor: 4.098

6.  Anti-IL-6 receptor antibody increases blood IL-6 level via the blockade of IL-6 clearance, but not via the induction of IL-6 production.

Authors:  Yasushi Uchiyama; Hiroto Yoshida; Nobuo Koike; Naohiko Hayakawa; Atsuko Sugita; Takashi Nishimura; Masahiko Mihara
Journal:  Int Immunopharmacol       Date:  2008-07-26       Impact factor: 4.932

7.  The significance of IgA class of antineutrophil cytoplasmic antibodies (ANCA) in childhood Henoch-Schönlein purpura.

Authors:  Fatih Ozaltin; Aysin Bakkaloglu; Seza Ozen; Rezan Topaloglu; Umut Kavak; Mukaddes Kalyoncu; Nesrin Besbas
Journal:  Clin Rheumatol       Date:  2004-06-18       Impact factor: 2.980

8.  A clinical study of Henoch-Schönlein Purpura associated with malignancy.

Authors:  H Mitsui; N Shibagaki; T Kawamura; H Matsue; S Shimada
Journal:  J Eur Acad Dermatol Venereol       Date:  2009-04       Impact factor: 6.166

9.  The clinical spectrum of Castleman's disease.

Authors:  Angela Dispenzieri; James O Armitage; Matt J Loe; Susan M Geyer; Jake Allred; John K Camoriano; David M Menke; Dennis D Weisenburger; Kay Ristow; Ahmet Dogan; Thomas M Habermann
Journal:  Am J Hematol       Date:  2012-07-13       Impact factor: 10.047

10.  Castleman's Disease with Cutaneous Involvement Manifestating as Multiple Violaceous Plaques on Entire Body.

Authors:  Hyeon-Young Park; Je-Jung Lee; Jee-Bum Lee; Seong-Jin Kim; Seung-Chul Lee; Young Ho Won; Sook Jung Yun
Journal:  Ann Dermatol       Date:  2011-10-31       Impact factor: 1.444

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  3 in total

1.  The efficacy and safety of anti-interleukin-6 receptor monoclonal blockade in a renal transplant patient with Castleman disease: early post-transplant outcome.

Authors:  Masatoshi Matsunami; Yoshifumi Ubara; Keiichi Sumida; Yoichi Oshima; Masahiko Oguro; Kazuya Kinoshita; Kiho Tanaka; Yuki Nakamura; Keiichi Kinowaki; Kenichi Ohashi; Takeshi Fujii; Takuro Igawa; Yasuharu Sato; Yasuo Ishii
Journal:  BMC Nephrol       Date:  2018-10-11       Impact factor: 2.388

2.  Analysis of Clinical Characteristics, Pathological Changes and Changes of Interleukin-6 (IL-6) and C-Reactive Protein (CRP) in Children with Castleman's Disease.

Authors:  Chao Hu; Yan Zou; Jing Pan; Jiliang Yang; Tianyou Yang; Tianbao Tan; Jiahao Li
Journal:  Med Sci Monit       Date:  2020-08-15

3.  Mediterranean fever gene variants modify clinical phenotypes of idiopathic multi-centric Castleman disease.

Authors:  Yushiro Endo; Tomohiro Koga; Yoshihumi Ubara; Remi Sumiyoshi; Kaori Furukawa; Atsushi Kawakami
Journal:  Clin Exp Immunol       Date:  2021-07-26       Impact factor: 4.330

  3 in total

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