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Key Clinical Message Cutaneous oxalosis is a rare manifestation of systemic oxalosis, typically associated with primary or secondary hyperoxaluria. We present a rare case of a 23‐year‐old female diagnosed with primary hyperoxaluria and end‐stage renal disease, who presented with papules on the palms without any vascular complications. The skin can be affected by oxalate deposition, resulting in various manifestations such as vascular complications or calcified nodules. In our case, the patient had primary hyperoxaluria and end‐stage renal disease but exhibited atypical features of cutaneous oxalosis. Histopathology confirmed the presence of oxalate crystals in the dermis, subcutis, and medium‐sized arteries. The mechanism of oxalate deposition in this case remains unclear. This case underscores the importance of considering cutaneous oxalosis in the differential diagnosis of patients with renal failure and skin lesions, and highlights the variability of clinical presentations in primary hyperoxaluria.

Key Clinical Message: Cutaneous oxalosis is a rare manifestation of systemic oxalosis, typically associated with primary or secondary hyperoxaluria. We present a rare case of a 23‐year‐old female diagnosed with primary hyperoxaluria and end‐stage renal disease, who presented with papules on the palms without any vascular complications. The skin can be affected by oxalate deposition, resulting in various manifestations such as vascular complications or calcified nodules. In our case, the patient had primary hyperoxaluria and end‐stage renal disease but exhibited atypical features of cutaneous oxalosis. Histopathology confirmed the presence of oxalate crystals in the dermis, subcutis, and medium‐sized arteries. The mechanism of oxalate deposition in this case remains unclear. This case underscores the importance of considering cutaneous oxalosis in the differential diagnosis of patients with renal failure and skin lesions, and highlights the variability of clinical presentations in primary hyperoxaluria. [ABSTRACT FROM AUTHOR]