Proposal of podocytic infolding glomerulopathy as a new disease entity: a review of 25 cases from nationwide research in Japan

Joh, Kensuke; Taguchi, Takashi; Shigematsu, Hidekazu; Kobayashi, Yutaka; Sato, Hiroshi; Nishi, Shinichi; Katafuchi, Ritsuko; Nomura, Shinsuke; Fujigaki, Yoshihide; Utsunomiya, Yasunori; Sugiyama, Hiroshi; Saito, Takao; Makino, Hírofumi

doi:10.1007/s10157-008-0104-z

Cited by 39 publications

(75 citation statements)

References 23 publications

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“…The patient with both TA and SLE reported by Kitazawa et al [6] showed intramembranous microspheres and epithelial cytoplasmic processes on the GBM, the findings of so-called podocytic infolding glomerulopathy (PIG). PIG is a relatively new disease entity proposed in 2008 and sometimes associated with lupus nephritis class V [13]. In the present case, however, the findings of PIG were not observed in the GBM.…”

Section: Discussioncontrasting

confidence: 61%

A Case of Membranous Glomerulonephropathy Associated with Takayasu's Arteritis

Koda¹,

Yoshino²,

Imanishi³

et al. 2014

Case Rep Nephrol Dial

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Glomerulonephropathy is a rare complication of Takayasu's arteritis (TA). To date, most glomerulonephropathies associated with TA show the histological feature of mesangial proliferation. Membranous glomerulonephropathy (MG) is a form of glomerulonephropathy in which the mesangial proliferation is not conspicuous and its association with TA is extremely rare. A 54-year-old man was referred to our hospital due to progressive edema in the lower limbs and nephrotic range proteinuria. Five years previously, he underwent percutaneous angioplasty for left subclavian artery stenosis. Kidney biopsy revealed stage II MG. General examination including enhanced CT scan confirmed the presence of TA. He started oral prednisolone therapy at a dose of 40 mg daily. The C-reactive protein level normalized 7 days after the prednisolone therapy. Three months later, proteinuria had remitted. Though the true relationship between MG and TA was not revealed in present case, considering the fact that complete remission of nephrotic syndrome occurred following the improvement of C-reactive protein level in response to steroid therapy, TA might be the secondary cause of MG. To our best knowledge, only two case reports described the association of MG and TA previously. Those two patients, however, also demonstrated the feature of systemic lupus erythematosus in addition to TA. This is the first case report that describes a patient who presented as MG associated with TA, but not complicated by systemic lupus erythematosus.

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Section: Discussioncontrasting

confidence: 61%

A Case of Membranous Glomerulonephropathy Associated with Takayasu's Arteritis

Koda¹,

Yoshino²,

Imanishi³

et al. 2014

Case Rep Nephrol Dial

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“…The pathological finding of a bubbling appearance with or without immunoglobulin deposition is consistent with a recovery phase of MN or ''podocytic infolding glomerulopathy'' [3], a new term based on 25 cases reported in Japan. Almost 70% of the latter patients have autoimmune diseases such as SLE, Sjögren's syndrome, or mixed connective tissue diseases, and 16% of patients show temporary urinary obstruction, e.g., stone disease.…”

Section: Discussionmentioning

confidence: 94%

“…Joh et al [3] recently reported on a small series with a bubbling appearance, spike formation, or both with or without immunoglobulin deposition, which they termed ''podocytic infolding glomerulopathy.'' It is unclear whether this represents only a recovery phase of MN or a new disease entity.…”

Section: Introductionmentioning

confidence: 97%

A large-sized bubbling appearance of the glomerular basement membrane in a patient with pulmonary limited AL amyloidosis and a past history of lupus nephritis

et al. 2011

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We report an unusual pathological finding, a large-sized bubbling appearance of the glomerular basement membrane (GBM), in a patient with pulmonary limited AL amyloidosis and a past history of lupus nephritis. The first renal biopsy specimen from 10 years ago, when systemic lupus erythematosus was diagnosed, demonstrated mild mesangial proliferation and subepithelial deposits (WHO classification: III + V). Light microscopy of the current biopsy using periodic acid methenamine silver (PAMS) stain demonstrated a large-sized bubbling appearance of the GBM; however, very weak immunoglobulin and complement deposition was observed in immunofluorescence studies. Routine electron microscopy demonstrated partial subendothelial expansion with electron-lucent materials, but no electron-dense deposits or amyloid fibrils. Electron microscopy with PAMS stain revealed electron-lucent endothelial scalloping, including some cellular components and microspheres in the GBM; however, it is not clear if these materials are derived from endothelial cells. One possibility is that these unique findings represent a recovery phase of lupus membranous nephritis; another is that these findings correspond to a new disease entity.

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“…Electron microscopy demonstrated sparse degenerative materials on the epithelial side of the GBM with intervening spikes, which corresponded to the findings on both light and immunofluorescence microscopy. Podocytic infolding glomerulopathy (PIG) [28], a recently proposed disease entity, shows diffuse global bubbling appearance with an occasional spike formation in the GBM on light microscopy. These findings are similar to those of MN, but PIG is characterized by primary podocytic infolding into the GBM and microstructures in the GBM on electron microscopy.…”

Section: Discussionmentioning

confidence: 99%

Angioimmunoblastic T-cell lymphoma and membranous nephropathy: a still unreported association

et al. 2010

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A 21-year-old man with lymphadenopathy and Coombs-positive hemolytic anemia had been treated with steroid maintenance therapy. He developed nephrotic syndrome with size increase of lymphadenopathy. Lymph node examination disclosed angioimmunoblastic T-cell lymphoma (AITL). Light microscopy of a renal biopsy specimen showed typical features of membranous nephropathy (MN), such as bubbling appearance and spike formation. Immunofluorescence studies revealed no significant deposition of immunoglobulins. Electron microscopy showed sparse degenerative materials on the epithelial side of the glomerular basement membranes, with intervening spikes. These unique histological findings suggested secondary MN. High-dose steroid therapy followed by six courses of cyclophosphamide, doxorubicin, vincristine, and prednisolone (CHOP) therapy improved his symptoms. One-year follow-up revealed the patient in good health without any signs of relapse. Glomerular manifestations have rarely been reported in association with AITL. To our knowledge, this is the first reported case of nephrotic syndrome due to MN associated with AITL.

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Proposal of podocytic infolding glomerulopathy as a new disease entity: a review of 25 cases from nationwide research in Japan

Cited by 39 publications

References 23 publications

A Case of Membranous Glomerulonephropathy Associated with Takayasu's Arteritis

A Case of Membranous Glomerulonephropathy Associated with Takayasu's Arteritis

A large-sized bubbling appearance of the glomerular basement membrane in a patient with pulmonary limited AL amyloidosis and a past history of lupus nephritis

Angioimmunoblastic T-cell lymphoma and membranous nephropathy: a still unreported association

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