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Von Hippel–Lindau (VHL) disease along with chronic thromboembolic pulmonary hypertension (CTEPH) is a unique and unusual severe complication of ventriculoatrial (VA) shunt implantation in the treatment of hydrocephalus. To the best of our knowledge, this can be the first reported case of an effective treatment with balloon pulmonary angioplasty in a patient with VHL after VA shunt placement. The patient underwent six balloon pulmonary angioplasty procedures. All invasive procedures resulted in haemodynamic and functional improvement.