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Cited by 200IgG4-Related and Inflammatory DiseasesRenal Diseases and GlomerulopathiesVasculitis and related conditions

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Pseudotumors due to IgG4 Immune-Complex Tubulointerstitial Nephritis Associated With Autoimmune Pancreatocentric Disease

Cornell, Lynn D., Chicano, Sonia L., Deshpande, Vikram, Collins, A. Bernard, Selig, Martin K., Lauwers, Gregory Y., Barisoni, Laura, Colvin, Robert B.·2007·American Journal of Surgical Pathology
DOI 10.1097/pas.0b013e318059b87c
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Abstract

Autoimmune pancreatitis (AIP) is a mass-forming chronic fibroinflammatory condition centered on the pancreatobiliary system and characterized by predominant immunoglobulin G4 (IgG4)-positive plasma cells. Recent reports have brought to light the multiorgan involvement of this disease. We describe a series of 5 cases of tubulointerstitial nephritis (TIN) associated with AIP and characterize the clinical, pathologic, ultrastructural, and immunopathologic features of TIN. The specimens consisted of 4 biopsies and 1 nephrectomy. The average patient age was 64 years (range 45 to 78) and the male to female ratio was 4:1. All had histologic and/or clinical and radiographic evidence of AIP, mass-forming sclerosing cholangitis, or both. The clinical impression in 4 patients was a renal mass or vasculitis. Two patients had renal insufficiency. Histologic preparations revealed a dense tubulointerstitial lymphoplasmacytic infiltrate. Eosinophils were often numerous. Tubulitis and tubular injury were present, along with tubular atrophy with focally thickened tubular basement membranes (TBMs). The histologic appearance ranged from a cellular, inflammatory pattern without tubular atrophy to a striking expansive interstitial fibrosis with tubular destruction. The nephrectomy specimen demonstrated a masslike nodular pattern of inflammation with normal renal tissue elsewhere. Glomeruli were uninvolved. By immunohistochemistry or immunofluorescence, numerous plasma cells in the infiltrate were positive for IgG4. TBM granular IgG deposits, predominantly of the IgG4 subclass, were detected in 4 of 5 cases by either immunofluorescence or immunohistochemistry. By electron microscopy, corresponding amorphous electron-dense deposits were present in the TBM in these cases. This type of TIN, typically characterized by a masslike lesion consisting of a lymphoplasmacytic infiltrate with eosinophils and prominent IgG4-positive plasma cells and immune-complex deposits in the TBM, may be part of a systemic IgG4-related disease, which we term "IgG4-associated immune complex Multiorgan Autoimmune Disease" (IMAD).

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Metadata

Authors
Cornell, L. D., Chicano, S. L., Deshpande, V., Collins, A. B., Selig, M. K., Lauwers, G. Y., Barisoni, L., & Colvin, R. B.
Affiliations
  • Massachusetts General Hospital (US)
  • Harvard University (US)
  • New York University (US)
Corresponding author
  • Cornell, Lynn D.
  • Chicano, Sonia L.
  • Deshpande, Vikram
  • Collins, A. Bernard
  • Selig, Martin K.
  • Lauwers, Gregory Y.
  • Colvin, Robert B.
ORCID
Venue
American Journal of Surgical Pathology
Publisher
Ovid Technologies (Wolters Kluwer Health)
Published
2007-10
Volume
31
Issue
10
Pages
1586-1597
ISSN
0147-5185 (ISSN-L)
DOI
10.1097/pas.0b013e318059b87c
OpenAlex
W2067665313
Cited by
200
References
0
Citations by year
2026: 3 · 2025: 3 · 2024: 4 · 2023: 2 · 2022: 4 · 2021: 4 · 2020: 6 · 2019: 9 · 2018: 7 · 2017: 7 · 2016: 15 · 2015: 17 · 2014: 11 · 2013: 19 · 2012: 22
FWCI
9.77
Type
journal article
Language
en
Open access
closed
Locations
  • The American Journal of Surgical Pathology (publishedVersion)
  • PubMed (publishedVersion)
Topics
  • Health Sciences › Medicine › Rheumatology › IgG4-Related and Inflammatory Diseases
  • Health Sciences › Medicine › Nephrology › Renal Diseases and Glomerulopathies
  • Health Sciences › Medicine › Rheumatology › Vasculitis and related conditions
Keywords
IgG4-related disease, autoimmune pancreatitis, interstitial nephritis, immune complex deposition, IgG4-positive plasma cells, sclerosing cholangitis, renal pseudotumor, tubular basement membrane, eosinophils, lymphoplasmacytic infiltration, multisystem involvement, renal failure
MeSH terms
Aged, Antigen-Antibody Complex, Autoimmune Diseases, Basement Membrane, Female, Granuloma, Plasma Cell, Humans, Immunoglobulin G, Immunoenzyme Techniques, Kidney Tubules, Male, Middle Aged, Nephritis, Interstitial, Pancreatitis, Plasma Cells

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