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We previously reported that an abnormal CSF opening pressure (OP) in children was greater than 28 cm H2O. Since elevated intracranial pressure can cause optic nerve head edema (ONHE), we would expect that most patients with ONHE would have an OP greater than 28 cm H2O. This study describes the range of OP for children with ONHE and compared them to age-matched controls without ONHE.Case subjects were children (1–18 years of age) enrolled in a prospective study of CSF OP that demonstrated ONHE at time of lumbar puncture and that the ONHE later resolved. Patients with ONHE secondary to infectious, inflammatory, or ischemic conditions were excluded. Control subjects from the same study, but without ONHE, were matched to cases.Of the 472 subjects enrolled in the study, 41 OP measurements were obtained from 33 patients with ONHE who did not have any exclusionary criteria and matched to 41 control subjects without ONHE. Case subjects had a significantly higher OP (mean, 41.4 cm H20; range, 22–56) than control subjects (mean, 18.9 cm H2O; range, 9–29; p < 0.01). Forty of 41 (97.6%) case subjects and 2 of 41 (4.8%) control subjects had OP measures >28 cm H2O.Children with ONHE not related to infectious, inflammatory, or ischemic causes typically have an OP >28 cm H2O, significantly higher than age-matched controls without ONHE. This study provides further support to our previously published findings that suggests an abnormal OP in children is typically above 28 cm H2O.