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Introduction The development of malignant tumors in patients with hydranencephaly is extremely rare. We describe the first case of testicular cancer that developed in the undescended testes of a long‐term survivor of hydranencephaly. Case presentation A 32‐year‐old man with severe cerebral palsy due to hydranencephaly was referred to our department for the evaluation of a subcutaneous lump in the lower right abdomen. He was a long‐term survivor of hydranencephaly. After confirming the diagnosis of right testicular cancer originating in his undescended testes, surgical resection was performed. Pathological examination revealed a mixed‐type germ cell tumor. Conclusion The decision‐making process for treating malignant tumors, like testicular cancer, in adults with severe cerebral palsy can be challenging. Clinical ethics consultation could be helpful in avoiding treatment delays.