Successful therapy with mycophenolic acid in a membranous glomerulonephritis due to Kimura disease

Gaillard, Julien; Rotman, Samuel; Girardet, Christophe; Spertini, François

doi:10.5414/cn109086

Cited by 8 publications

(4 citation statements)

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“…This was surprising, since Obata et al [8] had previously demonstrated diminishing tumors on prednisolone monotherapy in cases of KD with associated MN. Despite initial remission of the nephrotic syndrome, in line with findings of Gaillard et al [13], sustained remission on prednisolone monotherapy was not possible, and the patient experienced relapse of proteinuria. The patient followed a natural course of relapsing nephrosis with severe proteinuria as typically seen in cases of MN, and hence it was decided to apply common treatment protocols of MN to the patient.…”

Section: Discussion/conclusionsupporting

confidence: 64%

Rituximab Treatment in a Patient with Kimura Disease and Membranous Nephropathy: Case Report

Vissing-Uhre¹,

Hansen²,

Frevert³

et al. 2021

Case Rep Nephrol Dial

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Kimura disease (KD) is a chronic, inflammatory disorder with slowly developing subcutaneous tumor-like swellings, often occurring in the head and neck region. KD is diagnosed based on histology, elevated levels of immunoglobulin type E, and increased peripheral eosinophil granulocytes. KD may coexist with glomerular renal diseases, and this case report is based on a patient with KD-associated membranous nephropathy. Patients with membranous nephropathy without KD have demonstrated responsiveness to treatment with monoclonal anti-CD20 antibodies. This case report is the first to investigate the effect of rituximab treatment in a patient with KD-associated membranous nephropathy. A 30-year-old Italian man living in Denmark was diagnosed with Kimura’s disease based on subcutaneous nodules with eosinophil angiolymphoid hyperplasia. The patient was admitted to the hospital due to nephrotic syndrome. Serology showed eosinophil granulocytosis and negative PLA2-receptor antibody. Renal biopsy showed membranous nephropathy, and the patient was treated with systemic methylprednisolone followed by cyclosporin and then cyclophosphamide with only partial remission. Ultimately, treatment with intravenous rituximab was initiated, which resulted in overall remission and no nephrotic relapses at 30 months of follow-up. Thus, intravenous rituximab effectively decreased proteinuria and prevented nephrotic relapses in a patient with treatment-refractory membranous nephropathy due to KD.

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Section: Discussion/conclusionsupporting

confidence: 64%

Rituximab Treatment in a Patient with Kimura Disease and Membranous Nephropathy: Case Report

Vissing-Uhre¹,

Hansen²,

Frevert³

et al. 2021

Case Rep Nephrol Dial

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“…The pathophysiology of Kimura’s disease remains elusive, although an aberrant immune reaction to a yet unknown antigen has been proposed. Although this is the first known case of comorbidity with confirmed THSD7A-associated MN, several reports have described a possible association of Kimura’s diseases with MN [ 24 – 28 ]. Most such cases were reported from Asian countries, including Japan and China, reflecting the endemic regions of Kimura’s disease.…”

Section: Discussionmentioning

confidence: 98%

Clinicopathological characteristics of thrombospondin type 1 domain-containing 7A-associated membranous nephropathy

et al. 2019

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Thrombospondin type 1 domain-containing 7A (THSD7A) is a recently identified target antigen of idiopathic membranous nephropathy (iMN). The clinicopathological characteristics of THSD7A-associated MN are poorly characterised due to low prevalence among MN patients. Among 469 consecutive cases of pathologically confirmed MN diagnosed at four centres in Japan, 14 cases were confirmed positive for THSD7A by immunohistochemistry (3.0%). The prevalence of THSD7A-associated MN tended to be higher in northern Japan. Most cases demonstrated nephrotic-range proteinuria (12/14 cases, 86%). In two patients, cancer was detected at the time of renal biopsy (small-cell carcinoma of the lung and prostatic adenocarcinoma with neuroendocrine differentiation). Both tumours were negative for THSD7A. Four patients had concurrent or previous incidence of allergic diseases, including one patient with Kimura’s disease. Pathological analysis of kidney biopsy tissue revealed slight mesangial cell proliferation in three cases and spike formation in one case. Immunofluorescence studies demonstrated that IgG subclass was mainly IgG4-dominant/codominant (12/13, 92% cases), while the case with prostatic cancer had an IgG2-dominant distribution. The immunostaining profile for components of the lectin complement pathways was not significant in three cases including two patients with malignancy. One case was dual positive for THSD7A and PLA2R. Of 10 cases with known clinical follow-up data, 6 demonstrated reduced serum creatinine and 8 presented reduced proteinuria. In summary, although the major IgG phenotype was usually IgG4-dominant/codominant, clinical background was otherwise heterogeneous. Further investigation of regional differences in THSD7A-associated MN prevalence may reveal genetic and environmental risk factor and associated pathogenic mechanisms. Electronic supplementary material The online version of this article (10.1007/s00428-019-02558-0) contains supplementary material, which is available to authorized users.

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“…Another concern regarding steroids is that their long-term use of them may lead to complications such as osteoporosis, digestive ulcers and acquired diabetes mellitus. Other systemic immunosuppressive medications, including cyclosporine [34], mycophenolate mofetil, mycophenolic acid [35], leflunomide and tacrolimus [36] were also reported to be effective in some patients. In a recent meta-analysis including 31 studies, the overall recurrence rate of surgical excision was 30.5%, and that of medication alone was 45%, while the combination therapy of surgery and postoperative adjuvant therapy demonstrated the lowest recurrence rate of 26.94% [32].…”

Section: Discussionmentioning

confidence: 99%

A Rare Kimura’s Disease in the Oral Cavity with Severe Sleep Apnea: Case Report and Literature Review

Lei

Feng

et al. 2022

Medicina

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Kimura’s disease (KD) is a rare chronic inflammatory disorder that commonly occurs in Asian males. It mainly presents as painless subcutaneous masses or lymphadenopathy in the head and neck region. The incidence of KD in the oral cavity is quite rare. We reported a rare case of a 53-year-old male who had KD in his soft palate, hard palate and bilateral tonsils associated with severe sleep apnea. This patient underwent radiotherapy and exhibited a good response to the treatment. Throughout the 12-month follow-up period, the patient’s condition remained satisfactory. Of the other 14 reviewed cases of KD in the oral cavity, the lesions can occur in the buccal mucosa, hard and soft palate, and mouth floor with specific clinical features. We further summarized their manifestations and treatments in order to guide the future identification and management of KD with lesions in the oral cavity.

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Successful therapy with mycophenolic acid in a membranous glomerulonephritis due to Kimura disease

Cited by 8 publications

References 0 publications

Rituximab Treatment in a Patient with Kimura Disease and Membranous Nephropathy: Case Report

Rituximab Treatment in a Patient with Kimura Disease and Membranous Nephropathy: Case Report

Clinicopathological characteristics of thrombospondin type 1 domain-containing 7A-associated membranous nephropathy

A Rare Kimura’s Disease in the Oral Cavity with Severe Sleep Apnea: Case Report and Literature Review

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