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Nasal dermoid sinus cysts (NDSC) are caused by failure of involution of ectodermal and mesectodermal midline remnants. A 13-month-old girl was found to have a NDSC with a tract traversing the nasofrontal suture line and an intracranial component on MRI. Here we present the first case in a pediatric patient of a resection of a NDSC with intracranial extension through an endoscope-assisted open rhinoplasty approach and frontonasal mini-craniotomy without the need for additional skin incisions or traversing the nasal cavity. This approach provided an excellent intraoperative exposure and cosmetic outcome at 1 year.