J Korean Soc Radiol.  2020 Jan;81(1):190-196. 10.3348/jksr.2020.81.1.190.

Focal Xanthogranulomatous Pyelonephritis Associated with Xanthogranulomatous Cholecystitis: A Case Report

• Cho SM
• Kim HK
• Lee HK
• Lee B
• Kim KH
• Lee KE
• Shim JC
• Hwang DH
• Lee GJ

• KMID: 2469192
• DOI: http://doi.org/10.3348/jksr.2020.81.1.190

Abstract

Xanthogranulomatous inflammation is a rare inflammatory reaction, characterized by lipid-laden macrophages, known as xanthomas, in histopathologic examination. Aggressive xanthogranulomatous inflammation often manifests as local infiltration but does not affect distant organs unless combined with rare systemic diseases. We report a case of focal xanthogranulomatous pyelonephritis (XGP) associated with severe xanthogranulomatous cholecystitis. Focal XGP was suspected in radiologic examination that showed a cystic lesion with an infiltrative margin, which were surgically resected and confirmed in pathologic examination. To our knowledge, this is the first report of focal xanthogranulomatous pyelonephritis associated with xanthogranulomatous cholecystitis. Moreover, we found peripheral hypointensity around the cystic lesion in the T2-weighted image, probably reflecting hemorrhage and fibrosis of the xanthogranulomatous inflammation.

MeSH Terms

Cholecystitis*
Fibrosis
Hemorrhage
Inflammation
Macrophages
Magnetic Resonance Imaging
Pyelonephritis, Xanthogranulomatous*
Xanthomatosis

Figure

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  Fig. 1 Focal xanthogranulomatous pyelonephritis associated with xanthogranulomatous cholecystitis in a 56-year-old man, presenting with right upper quadrant pain. A. On ultrasonography (left panel), a GB stone is seen as a curvilinear hyper-reflective echo with distal acoustic shadowing. A few anechoic nodules (arrows) are noted within the thickened isoechoic GB walls. Axial CT (right panel) shows diffuse wall thickening of GB with intramural low-attenuation nodules. B. Axial T2- (left panel) and T1-weighted imaging (right panel) show oval GB stones as a dark signal and intramural nodules as a high signal. C. Axial CT shows a cystic lesion in the right kidney with infiltration extending peripherally to the perirenal tissues and hepatorenal recess. Coronal CT better illustrates the infiltrative lesion between the liver and the kidney with focal peritoneal wall thickening. D. Axial T2-weighted imaging (left panel) shows a nodular hyperintense lesion with rim-like low signal intensity (arrow). This lesion is not clearly outlined on the T1-weighted image (right panel). E. The gross specimen of GB shows disfigurement with marked irregular mural fibrous thickening, multifocal extensive mucosal ulcers, transmural old hemorrhages, and multiple yellowish xanthogranulomatous nodules or plaques (left panel). Scanning photomicrograph of GB wall exhibiting mucosal ulcers, transmural hemorrhages, dense mural fibrosis, and nodular aggregates of foamy histiocytes and inflammatory cells (right panel, × 40, H&E stain). F. The gross specimen of the right renal cortex and perirenal fibroadipose tissues (left panel) reveal a few prominent yellowish xanthogranulomatous nodules or plaques, fresh-to-old hemorrhages, and dense fibrous adhesion. Scanning photomicrography shows massive perirenal (a) and subcapsular (c) collection of foamy histiocytes, dense pericapsular fibrosis (b), and inflammatory cell infiltration (middle panel, × 40, H&E stain). Yellowish xanthogranulomatous nodules demonstrate abundant lipid-laden foamy histiocytes and hemosiderin-laden histiocytes, admixed with polymorphic lymphoreticular cells and fibroblasts (right panel, × 200, H&E stain). GB = gallbladder, H&E = hematoxylin and eosin

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