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Multisuture craniosynostosis: a case report of unusual presentation of chromosome 14q32 deletion.
Chaisrisawadisuk, Sarut; Vatanavicharn, Nithiwat; Khampalikit, Inthira; Moore, Mark H.
Afiliação
  • Chaisrisawadisuk S; Division of Plastic Surgery, Department of Surgery, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok, Thailand.
  • Vatanavicharn N; Division of Medical Genetics, Department of Pediatrics, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok, Thailand. nithiwat_v@hotmail.com.
  • Khampalikit I; Division of Neurosurgery, Department of Surgery, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok, Thailand.
  • Moore MH; Cleft and Craniofacial South Australia, Women's and Children's Hospital, North Adelaide, South Australia, Australia.
Childs Nerv Syst ; 39(5): 1317-1322, 2023 05.
Article em En | MEDLINE | ID: mdl-36512050
Multisuture craniosynostosis is associated with a number of syndromes and underlying gene mutations. It is rarely caused by chromosome disorders. For the management, multisuture craniosynostosis raises concerns about abnormal head shape and risks of increased intracranial pressure in affected patients. Calvarial reconstruction to reshape the skull shape and expand the intracranial volume plays an essential role in correcting particular problems. Here, we report a 2-month-old female infant presenting with low birth weight, abnormal head shape, dysmorphic facies and pinnae, hypotonia, and feeding difficulty. Three-dimensional computed tomographic scans revealed left unicoronal and sagittal synostoses. Chromosome microarray analysis revealed de novo chromosome 14q32.12-q32.31 deletion. Among the deleted genes, YY1 and BCL11B are the most likely candidate genes causing craniosynostosis. Some clinical features of the patient are similar to Temple syndrome indicating that the deleted region is paternal in origin. In summary, this is a rare case of chromosome 14q32 deletion with multisuture craniosynostosis. We also report the multidisciplinary management and clinical outcomes after early cranial vault remodelling procedures.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Hipertensão Intracraniana / Craniossinostoses Limite: Female / Humans / Infant Idioma: En Revista: Childs Nerv Syst Assunto da revista: NEUROLOGIA / PEDIATRIA Ano de publicação: 2023 Tipo de documento: Article País de afiliação: Tailândia

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Hipertensão Intracraniana / Craniossinostoses Limite: Female / Humans / Infant Idioma: En Revista: Childs Nerv Syst Assunto da revista: NEUROLOGIA / PEDIATRIA Ano de publicação: 2023 Tipo de documento: Article País de afiliação: Tailândia