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【Introduction】 Lysozyme is a low molecular weight protein of 15 kilodaltons produced by monocytes and macrophages. It is freely filtered by the glomerulus and reabsorbed in the proximal tubule. In monocytic leukemias such as chronic myelomonocytic leukemia (CMML), excessive lysozyme production may occur. This excess lysozyme deposits in the proximal tubules, causing a rare acute kidney injury termed lysozyme-induced nephropathy (LyN).
We report a case of LyN complicated by CMML that improved with hydroxycarbamide therapy, along with our observations.
【Medical history】 The patient is an 80-year-old male with type 2 diabetes. He had been diagnosed with chronic myelomonocytic leukemia by the hematology department three years ago, but due to his advanced age and low disease activity, he was managed conservatively. During follow-up, his renal function showed a deteriorating trend, leading to referral to the nephrology department two years ago. Additionally, one and a half years ago, he was diagnosed with acute uveitis at other ophthalmology clinic and started on steroid eye drops. At the initial visit, serum creatinine was 1.6 mg/dL, but it had worsened to 3.0 mg/dL over the observation of one year, leading to the performance of a renal biopsy.
【Diagnosis】 Renal biopsy findings revealed focal segmental glomerulosclerosis in some glomeruli. No spike formation in the basement membrane or mesangial interposition were observed. Mild mesangial proliferation was noted in the mesangial area, while extensive fibrosis and diverse inflammatory cell infiltration were present in the tubulointerstitial area. Fluorescent antibody staining showed weak positivity for C3c in the mesangial area. Based on these findings, we considered tubulointerstitial nephritis and uveitis syndrome and the patient was admitted for treatment. Prednisolone 25 mg/day was initiated on the second day of hospitalization, but serum creatinine remained at 3.8 mg/dL with no improvement. Additional tests to reevaluate the diagnosis revealed elevated levels of serum lysozyme at 335.0 μg/mL and urinary lysozyme at 1240 μg/mL. Supplementary lysozyme staining on the renal biopsy specimen showed positivity within the tubular cell cytoplasm. Electron microscopy that arrived then revealed high electron-density deposits within the tubular cells, leading to a diagnosis of LyN.
【Clinical course】 Steroid therapy was discontinued immediately, and hydroxycarbamide was initiated instead on day 15 of hospitalization. The white blood cell count decreased, and serum creatinine, which had risen to 4.5 mg/dL, improved to 3.5 mg/dL on the 45th day of hospitalization.
【Conclusion】 LyN is a rare tubular disorder complicating CMML, and treatment of CMML is the only treatment option. LyN should be considered in cases of tubulointerstitial nephritis complicating CMML.
