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Premature infants with severe germinal matrix/intraventricular hemorrhage are at high risk of posthemorrhagic ventricular dilation and hydrocephalus (Video 1). We report a 4-month-old premature infant with grade III intraventricular hemorrhage and compartmentalized hydrocephalus with an unusual craniospinal cyst. The cyst extended anteriorly from the mesencephalon to the posterior wall of C6, causing severe compression of the brainstem and spinal cord. An endoscopic procedure was performed first to achieve a unique cranial fenestration, which is detailed in "Endoscopic Transfontanellar Approach of a Cyst Anterior to the Brainstem Crossing the Foramen Magnum-Part I: Failure of Unique Fenestration." One month later the clinical picture recurred. Magnetic resonance imaging revealed hydrocephalus and cyst recurrence. A second procedure with endoscopic fenestration and shunt revision was needed. The same right transfontanellar approach was chosen, using a straight 30° endoscope. The procedure and surgical technique are explained in a step-by-step fashion. Extreme care was taken to align the head and the cervical spine. This allowed access to the extreme caudal cyst membrane posterior to C6 to create multiple transfixing fenestrations of the cyst. At 18-month follow-up, the child demonstrated almost normal neurological and psychomotor development with no cyst recurrence or hydrocephalus. Our report underlines the importance of performing multiple fenestrations of such cysts. We believe that performing a transfixing fenestration through the cyst allows cerebrospinal fluid flow and prevents recurrence, as, for instance, has been shown for suprasellar arachnoid cysts., (Copyright © 2020 Elsevier Inc. All rights reserved.)

Premature infants with severe germinal matrix intraventricular hemorrhage (IVH) are at high risk of posthemorrhagic ventricular dilation and hydrocephalus (Video 1). We report the case of a 3-month-old premature infant referred for posthemorrhagic multilocated hydrocephalus. He presented with somnolence, hypotonia, and a bulging fontanelle. His past medical history included an IVH grade III with hydrocephalus initially treated by temporary ventriculosubgaleal shunting. Magnetic resonance imaging (MRI) showed persistent hydrocephalus associated to a cyst extending anteriorly from the mesencephalon to the posterior wall of the sixth cervical vertebra, causing severe brainstem and spinal cord compression. A two-step surgery was performed, consisting of an endoscopic procedure and a ventriculo peritoneal shunt placement, to achieve hydrocephalus treatment and cyst fenestration. A right transfontanellar approach with the head flexed was chosen, using a straight 30-degree endoscope. The procedure and surgical technique are explained in a step-by-step fashion in the original conditions. A large unique fenestration was performed with no postoperative complications and improvement of the neurological status. One month later the patient presented again with symptomatic intracranial hypertension with compatible clinical and imagery findings. MRI showed cyst recurrence and hydrocephalus. We therefore revised our strategy and performed an additional procedure, which is detailed in the second part of the video (Part II: Success of Multiple Fenestrations). Our report underlines the high risk of recurrence of such cysts in post-hemorrhagic arachnoiditis. Cyst recurrence may be explained by the absence of crossing flow when a unique perforation is made., (Copyright © 2020 Elsevier Inc. All rights reserved.)