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Sindrom Dandy Walker termasuk hidrosefalus yang sangat jarang terjadi dengan insiden 1 kasus dari 65 kasus hidrosefalus berdasarkan penelitian profil hidrosefalus di RSUDZA Banda Aceh. Insiden di Indonesia sebanyak 50–60% kasus dari operasi bedah saraf. Kasus berikut seorang bayi laki-laki lahir prematur, umur 1 bulan, berat badan 3,5 kg, ukuran lingkar kepala 45 cm, muntah dan kejang. CT-Scan terdapat kista, kalium 7 mmol/l ditegakkan diagnosa sindrom Dandy Walker dengan hiperkalemia. Manajemen neuroanestesi dengan cara premedikasi dan pemasangan kateter intravena 24G dengan sevofluran via masker O2 100% setelah jalur vena terpasang, diberikan fentanil 10 mcg sebagai analgetik dan induksi propofol 10 mg. Rocuronium 3,5 mg sebagai fasilitas intubasi. Pemeliharaan anestesi dengan sevofluran dan oksigen. Ventilasi frekuensi nafas 30 x/menit dilakukan manual dan kemudian dengan ventilator TV 30 ml, I:E = 1:1,5, RR 30 kali per menit, FiO2 100%. Monitoring hasil frekuensi nadi 100 – 130 kali per menit, SpO2 100%, suhu afebris, CO2 30 mmHg. Simpulan: sindrom Dandy Walker kasus yang sangat jarang terjadi dan hidrosefalus sering bersamaan dengan hiperkalemia terjadi pada bayi prematur karena gangguan reaborbsi kalium dan terjadi perpindahan kompartemen dari intraseluler ke ekstraseluler sehingga kalium banyak di ekstraseluler, maka diperlukan manajemen neuroanestesi pediatrik yang adekuat. Neuroanesthesia Management in Dandy Walker Syndrome with Hyperkalemia Abstract Dandy Walker syndrome including hydrocephalus which is a very rare case with the incidence of 1 case out of 65 cases of hydrocephalus based on a study of hydrocephalus profile at Zainal Abidin Hospital, Banda Aceh. The incidence of Dandy-Walker syndrome in Indonesia are about 50 - 60% cases from all of neurosurgical cases. The following case was a premature baby, 1 month old, weight 3.5 kg, head cicumference 45 cm, vomit and seizure. Cyst was found in the head CT scan and the potassium level of 7 mmol/l. The patient was diagnosed with Dandy-Walker Syndrome with hyperkalemia. Neuroanesthesia management by premedication and infusion using intravenous cathether 24G with sevoflurane and 100% O2 mask After succesful intravenous cathether, given fentanyl 10 mcg as analgesic and propofol 10 mg as induction. Rocuronium 3.5 mg as a facility for intubation. Maintenance of anesthesia with sevoflurane and oxygen. Respiratory rate 30 breaths per minute with ventilation that was done manually and then with TV on ventilator 30 ml, I: E = 1: 1.5, RR 30x/min, FiO2 100%. Monitoring pulse frequency results of 100–130 beats per minute, 100% SpO2, temperature afebrile, CO2 30 mmHg. Conclusions: Dandy-Walker Syndrome was a very rare case and hydrocephalus was often followed with hyperkalemia that occur in premature infants due to potassium reaboration disorder and the displacement of compartment from intracellular to extracellular so that potassium was abundant at extracellular, hence adequate pediatric neuroanesthesia management was needed.