07 September 2026
: Case report
[In Press] Bilateral Xanthogranulomatous Pyelonephritis in a Patient With Spina Bifida and Neurogenic Bladder: A Case Report
Rare disease, Educational Purpose (only if useful for a systematic review or synthesis)
Dagmawi Dereje WaleDOI: 10.12659/AJCR.954146
Am J Case Rep In Press; DOI: 10.12659/AJCR.954146
Available online: 2026-09-07, In Press, Corrected Proof
Publication in the "In-Press" formula aims at speeding up the public availability of the pending manuscript while waiting for the final publication. The assigned DOI number is active and citable. The availability of the article in the Medline, PubMed and PMC databases as well as Web of Science will be obtained after the final publication according to the journal schedule
Abstract
BACKGROUND
Xanthogranulomatous pyelonephritis (XGP) is a rare chronic inflammatory renal infection associated with urinary obstruction, recurrent infections, and progressive renal parenchymal destruction. Bilateral involvement is exceedingly uncommon and associated with substantial morbidity and mortality, particularly in patients with neurogenic bladder and impaired longitudinal urologic follow-up.
CASE REPORT
A 30-year-old man with spina bifida complicated by neurogenic bladder requiring intermittent catheterization presented with respiratory distress, profound anemia, and progressive functional decline. Imaging demonstrated severe bilateral hydronephrosis, diffuse renal parenchymal destruction and calcifications, and right-sided staghorn calculi, consistent with advanced bilateral XGP. The patient rapidly developed septic shock and acute-on-chronic hypercapnic respiratory failure requiring mechanical ventilation and vasopressor support. Percutaneous nephrostomy drainage yielded purulent material, and cultures grew Morganella morganii, Candida parapsilosis, and Candida tropicalis. His hospital course was complicated by dialysis-dependent renal failure, recurrent respiratory failure, and prolonged ventilatory dependence. Definitive surgical intervention was considered but deferred because of severe anatomical deformities, clinical instability, and prohibitive operative risk. After prolonged hospitalization, he was discharged to a long-term acute care facility but later died from complications related to tracheostomy.
CONCLUSIONS
This case illustrates the severe clinical consequences of advanced bilateral XGP in a high-risk patient with neurogenic bladder. The coexistence of recurrent infection, urinary obstruction, and disrupted longitudinal follow-up is consistent with recognized risk factors for upper urinary tract deterioration, although their individual contribution to this patient’s disease progression cannot be established. The case underscores the management challenges that arise when bilateral renal destruction and systemic instability limit definitive source-control options.
Keywords: Dialysis; Hydronephrosis; Pyelonephritis; Pyelonephritis, Xanthogranulomatous; Renal Insufficiency, Chronic; Sepsis; Spinal Cord Diseases; Spinal Dysraphism; Staghorn Calculi
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