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Fanconi syndrome with karyomegalic interstitial nephritis after ifosfamide treatment for osteosarcoma: a case report
Yohei Kita1, Sayuri Shirai2, Teppei Koyama1
1Division of Nephrology and Hypertension, Department of Internal Medicine, St. Marianna University Yokohama City Seibu Hospital, Yokohama, Japan.
Abstract:
Patients with ifosfamide-induced renal damage present with Fanconi syndrome. Karyomegalic nephropathy/interstitial nephritis (KNIN) is a rare form of chronic tubulo-interstitial nephritis that was initially considered a type of familial nephropathy. However, several reports of drug-induced KNIN, i.e., KNIN-like nephropathy, have been reported in recent years. We present the case of an 18-year-old man who presented with Fanconi syndrome and progressive renal dysfunction after receiving chemotherapy including ifosfamide and cisplatin for right femoral osteosarcoma. Renal biopsy revealed numerous atrophied tubular epithelial cells with large, polymorphic nuclei, and the definitive diagnosis was KNIN. Most patients with KNIN-like nephropathy who receive ifosfamide are concomitantly treated with cisplatin, indicating that ifosfamide and cisplatin might act synergistically to increase the risk for KNIN-like nephropathy. Further investigation in case series is warranted to reveal potential treatment approaches and to evaluate prognosis.
Insights
Ifosfamide chemotherapy can cause Fanconi syndrome and kidney damage. A case study highlights Karyomegalic nephropathy/interstitial nephritis (KNIN) as a potential drug-induced condition, possibly worsened by cisplatin.
Area of Science:
- Nephrology
- Oncology
- Pharmacology
Background:
- Ifosfamide chemotherapy is known to cause renal damage, often presenting as Fanconi syndrome.
- Karyomegalic nephropathy/interstitial nephritis (KNIN) is a rare chronic tubulo-interstitial nephritis, previously considered familial.
- Recent reports suggest drug-induced KNIN, termed KNIN-like nephropathy, is emerging.
Purpose of the Study:
- To report a case of KNIN-like nephropathy in a patient treated with ifosfamide and cisplatin.
- To investigate the potential synergistic nephrotoxic effects of ifosfamide and cisplatin.
- To highlight the need for further research into KNIN-like nephropathy treatment and prognosis.
Main Methods:
- Case report of an 18-year-old male patient with osteosarcoma.
- Chemotherapy regimen included ifosfamide and cisplatin.
- Renal biopsy for histopathological examination.
Main Results:
- The patient presented with Fanconi syndrome and progressive renal dysfunction.
- Renal biopsy confirmed KNIN, characterized by atrophied tubular cells with large, polymorphic nuclei.
- Concomitant cisplatin use with ifosfamide is common in reported KNIN-like nephropathy cases.
Conclusions:
- Ifosfamide and cisplatin may synergistically induce KNIN-like nephropathy.
- KNIN-like nephropathy is a potential adverse effect of ifosfamide-based chemotherapy.
- Further case series are needed to establish treatment strategies and prognosis for KNIN-like nephropathy.

