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• W2078189549 abstract "• Severe intracranial hypertension in patients with multisuture craniosynostosis can lead to severe bone deformities including craniolacuna, bone defects and even soft-tissue herniation. • Cranial meningocele and even encephalocele can rarely be expected in multisuture craniosynostosis. Delicate pericranium and dural dissection are needed to prevent more damage to brain tissue during surgery.Beaten-copper appearance originates from the pressure that the brain exerts on the continuously remodeling inner cranium; however, a diffuse severe beaten-copper pattern is considered to be an indicator of chronic intracranial hypertension [1]. Lückenschädel (lacunar skull) is observed in approximately 10% of patients with craniosynostosis [2,3] and is characterized by multiple skull defects sharply separated by dense strips of bone [4,5]. High intracranial pressure can cause brain and dural herniation through these defects [4]. Here we report a very rare presentation of severe neglected craniosynostosis with multiple bone defects in the frontal and frontoorbital areas containing dura mater, arachnoid and gliotoic neural tissue.A 4-year-old girl was admitted due to an abnormal craniofacial shape with concomitant poly/syndactyly in all extremities. Two prominent skin-covered pulsatile nontender masses of heterogenous consistency had been present in her forehead for 2 years, and these had gradually increased in size. Upon examination, there was severe skull deformity, frank asymmetric exophthalmia, midface hypoplasia and retrusion and profound developmental delay with bilateral optic atrophy (fig. 1a). A diagnosis of neglected Apert syndrome was made. A brain computed tomography (CT) scan showed fusion of all skull sutures, a diffuse beaten-copper appearance, hydrocephalus and multiloculated bifrontal masses covered partially with thin bone (fig. 1b, c). The patient underwent monobloc frontofacial advancement with vault expansion and craniofacial deformity correction. Multiple cystic lesions containing dysplastic and gliotic frontal neural tissue and thin dura were evident during the craniotomy, and one of them was fronto-orbital causing asymmetric exophthalmia. There was severe adhesion between the herniated dura mater and the bone which prevented fine dissection causing multiple dural lacerations (fig. 2). Lückenschädel, beaten-copper appearance, skull defects and very rarely cranimeningocele have been observed in patients with craniosynostosis [1,2,3,4]. High intracranial pressure resulting from multisuture synostosis and hydrocephalus is considered to be the cause of the bone abnormalities found in craniofacial syndrome such as this case. Brain and/or dural herniations through these bony defects are not common [4]. Due to high intracranial pressure over a long time, the child reported here had developed optic atrophy and multiple skull defects; the soft tissue was herniated from the intracranial cavity due to these defects. To our knowledge, no previous report in the literature has provided a description of a multiple encephalocele-like lesion in craniosynostosis." @default.
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• W2078189549 date "2012-01-01" @default.
• W2078189549 modified "2023-10-03" @default.
• W2078189549 title "Multiple Acquired Encephaloceles: Dramatic End Point of Chronic Untreated Intracranial Hypertension in Multi-Suture Craniosynostosis" @default.
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• W2078189549 doi "https://doi.org/10.1159/000353615" @default.
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