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Michael O. Kelleher, Dylan J. Murray, Anne McGillivary, Mahmoud H. Kamel, David Allcutt, and Michael J. Earley

Object

The neurobehavioral morbidity of nonsyndromic trigonocephaly is incompletely understood. The purpose of this study was twofold: first, to assess the degree of developmental, educational, and behavioral problems in patients with nonsyndromic trigonocephaly and second, to establish whether patients with mild degrees of trigonocephaly had a lower frequency of such problems.

Methods

The authors performed an observational study of the frequency of developmental, educational, and behavioral problems in 63 children with trigonocephaly at the National Craniofacial Centre in the Republic of Ireland between 1989 and 2004. The parents of the children completed a follow-up questionnaire. Thirty percent of patients had a mild form of trigonocephaly and were treated conservatively. The remainder underwent surgical correction. Speech and/or language delay was reported in 34% of the children. Thirty-three percent of the children needed to be assessed by a school psychologist, and 47% were receiving remedial or resource hours within the school system. Twenty percent of children required a special needs classroom assistant because of behavioral issues, and 37% of parents expressed concerns about their child’s behavior. There were no statistically significant differences between children treated with surgery and those who had a mild deformity and were treated conservatively.

Conclusions

Nonsyndromic trigonocephaly is associated with a high frequency of developmental, educational, and behavioral problems. The frequency of these problems is not related to the severity of the trigonocephaly.

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Ingrid Kieran, Zaitun Zakaria, Chandrasekaran Kaliaperumal, Declan O'Rourke, Alan O'Hare, Eoghan Laffan, John Caird, Mary D. King, and Dylan J. Murray

The authors describe the case of a 3-year-old boy with a giant congenital vertex hemangioma who underwent presurgical embolization with Onyx (ethylene-vinyl alcohol copolymer dissolved in dimethyl sulfoxide) and Glubran (N-butyl-2-cyanoacrylate). This vascular tumor had no intracranial vascular communication as assessed by pre-embolization MRI and catheter angiography. All embolizations were performed by direct percutaneous injection. One week following the last embolization procedure the child presented with a 24-hour history of ataxia and extrapyramidal tremor. He was diagnosed with a possible immune-mediated reaction to Onyx or Glubran, which was treated with an urgent surgical excision of the hemangioma followed by intravenous administration of immunoglobulin and steroids. To the authors' knowledge, this is the first case of possible immune-mediated toxicity secondary to either Onyx or Glubran administration. This case highlights the need for awareness of potential toxic reactions to these embolic agents in the treatment of hemangiomas in the pediatric patient.