Membranous nephropathy associated with multicentric Castleman’s disease that was successfully treated with tocilizumab: a case report and review of the literature

Abstract Background Multicentric Castleman’s disease is a life-threatening disorder involving a systemic inflammatory response and multiple organ failure caused by the overproduction of interleukin-6. Although renal complications of Castleman’s disease include AA amyloidosis, thrombotic microangiopa...

Full description

Bibliographic Details
Main Authors: Ryosuke Saiki, Kan Katayama, Yosuke Hirabayashi, Keiko Oda, Mika Fujimoto, Tomohiro Murata, Ayako Nakajima, Kaoru Dohi
Format: Article
Language:English
Published: BMC 2021-06-01
Series:BMC Nephrology
Subjects:
Online Access:https://doi.org/10.1186/s12882-021-02423-w
_version_ 1818864852986232832
author Ryosuke Saiki
Kan Katayama
Yosuke Hirabayashi
Keiko Oda
Mika Fujimoto
Tomohiro Murata
Ayako Nakajima
Kaoru Dohi
author_facet Ryosuke Saiki
Kan Katayama
Yosuke Hirabayashi
Keiko Oda
Mika Fujimoto
Tomohiro Murata
Ayako Nakajima
Kaoru Dohi
author_sort Ryosuke Saiki
collection DOAJ
description Abstract Background Multicentric Castleman’s disease is a life-threatening disorder involving a systemic inflammatory response and multiple organ failure caused by the overproduction of interleukin-6. Although renal complications of Castleman’s disease include AA amyloidosis, thrombotic microangiopathy, and membranoproliferative glomerulonephritis, membranous nephropathy is relatively rare. We experienced a case of secondary membranous nephropathy associated with Castleman’s disease. Case presentation The patient was a 43-year-old Japanese man who had shown a high zinc sulfate value in turbidity test, polyclonal hypergammaglobulinemia, anemia, and proteinuria. A physical examination revealed diffuse lymphadenopathy, an enlarged spleen and papulae of the body trunk. A skin biopsy of a papule on the patient’s back showed plasma cells in the perivascular area and he was diagnosed with multicentric Castleman’s disease, plasma cell variant. Kidney biopsy showed the appearance of bubbling in the glomerular basement membranes in Periodic acid methenamine silver stain and electron microscopy revealed electron dense deposits within and outside the glomerular basement membranes. Since immunofluorescence study showed predominant granular deposition of IgG1 and IgG2, he was diagnosed with secondary membranous nephropathy associated with Castleman’s disease. He was initially treated with prednisolone alone, however his biochemical abnormalities did not improve. After intravenous tocilizumab (700 mg every 2 weeks) was started, his C-reactive protein elevation, anemia, and polyclonal gammopathy improved. Furthermore, his urinary protein level declined from 1.58 g/gCr to 0.13 g/gCr. The prednisolone dose was gradually tapered, then discontinued. He has been stable without a recurrence of proteinuria for more than 6 months. Conclusions Tocilizumab might be a treatment option for secondary membranous nephropathy associated with Castleman’s disease.
first_indexed 2024-12-19T10:38:15Z
format Article
id doaj.art-01c73b9e96d045208ad6de8923fa2c7f
institution Directory Open Access Journal
issn 1471-2369
language English
last_indexed 2024-12-19T10:38:15Z
publishDate 2021-06-01
publisher BMC
record_format Article
series BMC Nephrology
spelling doaj.art-01c73b9e96d045208ad6de8923fa2c7f2022-12-21T20:25:31ZengBMCBMC Nephrology1471-23692021-06-012211610.1186/s12882-021-02423-wMembranous nephropathy associated with multicentric Castleman’s disease that was successfully treated with tocilizumab: a case report and review of the literatureRyosuke Saiki0Kan Katayama1Yosuke Hirabayashi2Keiko Oda3Mika Fujimoto4Tomohiro Murata5Ayako Nakajima6Kaoru Dohi7Department of Cardiology and Nephrology, Mie University Graduate School of MedicineDepartment of Cardiology and Nephrology, Mie University Graduate School of MedicineDepartment of Cardiology and Nephrology, Mie University Graduate School of MedicineDepartment of Cardiology and Nephrology, Mie University Graduate School of MedicineDepartment of Cardiology and Nephrology, Mie University Graduate School of MedicineDepartment of Cardiology and Nephrology, Mie University Graduate School of MedicineCenter for Rheumatic Diseases, Mie University HospitalDepartment of Cardiology and Nephrology, Mie University Graduate School of MedicineAbstract Background Multicentric Castleman’s disease is a life-threatening disorder involving a systemic inflammatory response and multiple organ failure caused by the overproduction of interleukin-6. Although renal complications of Castleman’s disease include AA amyloidosis, thrombotic microangiopathy, and membranoproliferative glomerulonephritis, membranous nephropathy is relatively rare. We experienced a case of secondary membranous nephropathy associated with Castleman’s disease. Case presentation The patient was a 43-year-old Japanese man who had shown a high zinc sulfate value in turbidity test, polyclonal hypergammaglobulinemia, anemia, and proteinuria. A physical examination revealed diffuse lymphadenopathy, an enlarged spleen and papulae of the body trunk. A skin biopsy of a papule on the patient’s back showed plasma cells in the perivascular area and he was diagnosed with multicentric Castleman’s disease, plasma cell variant. Kidney biopsy showed the appearance of bubbling in the glomerular basement membranes in Periodic acid methenamine silver stain and electron microscopy revealed electron dense deposits within and outside the glomerular basement membranes. Since immunofluorescence study showed predominant granular deposition of IgG1 and IgG2, he was diagnosed with secondary membranous nephropathy associated with Castleman’s disease. He was initially treated with prednisolone alone, however his biochemical abnormalities did not improve. After intravenous tocilizumab (700 mg every 2 weeks) was started, his C-reactive protein elevation, anemia, and polyclonal gammopathy improved. Furthermore, his urinary protein level declined from 1.58 g/gCr to 0.13 g/gCr. The prednisolone dose was gradually tapered, then discontinued. He has been stable without a recurrence of proteinuria for more than 6 months. Conclusions Tocilizumab might be a treatment option for secondary membranous nephropathy associated with Castleman’s disease.https://doi.org/10.1186/s12882-021-02423-wCastleman’s disease - membranous nephropathyProteinuria - tocilizumab
spellingShingle Ryosuke Saiki
Kan Katayama
Yosuke Hirabayashi
Keiko Oda
Mika Fujimoto
Tomohiro Murata
Ayako Nakajima
Kaoru Dohi
Membranous nephropathy associated with multicentric Castleman’s disease that was successfully treated with tocilizumab: a case report and review of the literature
BMC Nephrology
Castleman’s disease - membranous nephropathy
Proteinuria - tocilizumab
title Membranous nephropathy associated with multicentric Castleman’s disease that was successfully treated with tocilizumab: a case report and review of the literature
title_full Membranous nephropathy associated with multicentric Castleman’s disease that was successfully treated with tocilizumab: a case report and review of the literature
title_fullStr Membranous nephropathy associated with multicentric Castleman’s disease that was successfully treated with tocilizumab: a case report and review of the literature
title_full_unstemmed Membranous nephropathy associated with multicentric Castleman’s disease that was successfully treated with tocilizumab: a case report and review of the literature
title_short Membranous nephropathy associated with multicentric Castleman’s disease that was successfully treated with tocilizumab: a case report and review of the literature
title_sort membranous nephropathy associated with multicentric castleman s disease that was successfully treated with tocilizumab a case report and review of the literature
topic Castleman’s disease - membranous nephropathy
Proteinuria - tocilizumab
url https://doi.org/10.1186/s12882-021-02423-w
work_keys_str_mv AT ryosukesaiki membranousnephropathyassociatedwithmulticentriccastlemansdiseasethatwassuccessfullytreatedwithtocilizumabacasereportandreviewoftheliterature
AT kankatayama membranousnephropathyassociatedwithmulticentriccastlemansdiseasethatwassuccessfullytreatedwithtocilizumabacasereportandreviewoftheliterature
AT yosukehirabayashi membranousnephropathyassociatedwithmulticentriccastlemansdiseasethatwassuccessfullytreatedwithtocilizumabacasereportandreviewoftheliterature
AT keikooda membranousnephropathyassociatedwithmulticentriccastlemansdiseasethatwassuccessfullytreatedwithtocilizumabacasereportandreviewoftheliterature
AT mikafujimoto membranousnephropathyassociatedwithmulticentriccastlemansdiseasethatwassuccessfullytreatedwithtocilizumabacasereportandreviewoftheliterature
AT tomohiromurata membranousnephropathyassociatedwithmulticentriccastlemansdiseasethatwassuccessfullytreatedwithtocilizumabacasereportandreviewoftheliterature
AT ayakonakajima membranousnephropathyassociatedwithmulticentriccastlemansdiseasethatwassuccessfullytreatedwithtocilizumabacasereportandreviewoftheliterature
AT kaorudohi membranousnephropathyassociatedwithmulticentriccastlemansdiseasethatwassuccessfullytreatedwithtocilizumabacasereportandreviewoftheliterature