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Partial craniofacial duplication: a review of the literature and case report.
J Craniomaxillofac Surg. 2014 Jun; 42(4):290-6.JC

Abstract

Diprosopus (Greek; di-, "two" + prosopon, "face"), or craniofacial duplication, is a rare craniofacial anomaly referring to the complete duplication of facial structures. Partial craniofacial duplication describes a broad spectrum of congenital anomalies, including duplications of the oral cavity. This paper describes a 15 month-old female with a duplicated oral cavity, mandible, and maxilla. A Tessier type 7 cleft, midline meningocele, and duplicated hypophysis were also present. The preoperative evaluation, surgical approach, postoperative results, and a review of the literature are presented. The surgical approach was designed to preserve facial nerve innervation to the reconstructed cheek and mouth. The duplicated mandible and maxilla were excised and the remaining left maxilla was bone grafted. Soft tissue repair included closure of the Tessier type VII cleft. Craniofacial duplication remains a rare entity that is more common in females. The pathophysiology remains incompletely characterized, but is postulated to be due to duplication of the notochord, as well as duplication of mandibular growth centres. While diprosopus is a severe deformity often associated with anencephaly, patients with partial duplication typically benefit from surgical treatment. Managing craniofacial duplication requires a detailed preoperative evaluation as well as a comprehensive, staged treatment plan. Long-term follow up is needed appropriately to address ongoing craniofacial deformity.

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Authors+Show Affiliations

Costa MA
Division of Plastic and Maxillofacial Surgery, Children's Hospital Los Angeles (CHLA), Plastic and Reconstructive Surgery, Keck School of Medicine, University of Southern California (USC), Los Angeles, CA, USA.
Borzabadi-Farahani A
Orthodontics, Warwick Dentistry, Warwick Medical School, University of Warwick, Coventry, UK; Formerly, Craniofacial Orthodontics, Division of Dentistry, CHLA, Center for Craniofacial Molecular Biology, USC, Los Angeles, CA, USA.
Lara-Sanchez PA
Department of Genetics, CHLA, Keck School of Medicine, USC, Los Angeles, CA, USA.
Schweitzer D
Department of Genetics, CHLA, Keck School of Medicine, USC, Los Angeles, CA, USA.
Jacobson L
Division of Plastic and Maxillofacial Surgery, Children's Hospital Los Angeles (CHLA), Plastic and Reconstructive Surgery, Keck School of Medicine, University of Southern California (USC), Los Angeles, CA, USA.
Clarke N
Division of Plastic and Maxillofacial Surgery, Children's Hospital Los Angeles (CHLA), Plastic and Reconstructive Surgery, Keck School of Medicine, University of Southern California (USC), Los Angeles, CA, USA.
Hammoudeh J
Division of Plastic and Maxillofacial Surgery, Children's Hospital Los Angeles (CHLA), Plastic and Reconstructive Surgery, Keck School of Medicine, University of Southern California (USC), Los Angeles, CA, USA.
Urata MM
Division of Plastic and Maxillofacial Surgery, Children's Hospital Los Angeles (CHLA), Plastic and Reconstructive Surgery, Keck School of Medicine, University of Southern California (USC), Los Angeles, CA, USA.
Magee WP
Division of Plastic and Maxillofacial Surgery, Children's Hospital Los Angeles (CHLA), Plastic and Reconstructive Surgery, Keck School of Medicine, University of Southern California (USC), Los Angeles, CA, USA. Electronic address: mageewilliam3@hotmail.com.

MeSH

CheekCleft PalateFacial AsymmetryFemaleHumansInfantLipMandibleMaxillaMeningoceleMouth AbnormalitiesNasal BonePituitary Gland

Pub Type(s)

Case Reports
Journal Article
Review

Language

eng

PubMed ID

23969147

Citation

Costa, Melinda A., et al. "Partial Craniofacial Duplication: a Review of the Literature and Case Report." Journal of Cranio-maxillo-facial Surgery : Official Publication of the European Association for Cranio-Maxillo-Facial Surgery, vol. 42, no. 4, 2014, pp. 290-6.
Costa MA, Borzabadi-Farahani A, Lara-Sanchez PA, et al. Partial craniofacial duplication: a review of the literature and case report. J Craniomaxillofac Surg. 2014;42(4):290-6.
Costa, M. A., Borzabadi-Farahani, A., Lara-Sanchez, P. A., Schweitzer, D., Jacobson, L., Clarke, N., Hammoudeh, J., Urata, M. M., & Magee, W. P. (2014). Partial craniofacial duplication: a review of the literature and case report. Journal of Cranio-maxillo-facial Surgery : Official Publication of the European Association for Cranio-Maxillo-Facial Surgery, 42(4), 290-6. https://doi.org/10.1016/j.jcms.2013.05.016
Costa MA, et al. Partial Craniofacial Duplication: a Review of the Literature and Case Report. J Craniomaxillofac Surg. 2014;42(4):290-6. PubMed PMID: 23969147.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Partial craniofacial duplication: a review of the literature and case report. AU - Costa,Melinda A, AU - Borzabadi-Farahani,Ali, AU - Lara-Sanchez,Pedro A, AU - Schweitzer,Daniela, AU - Jacobson,Lia, AU - Clarke,Noreen, AU - Hammoudeh,Jeffery, AU - Urata,Mark M, AU - Magee,William P,3rd Y1 - 2013/08/19/ PY - 2012/08/28/received PY - 2013/05/22/revised PY - 2013/05/23/accepted PY - 2013/8/24/entrez PY - 2013/8/24/pubmed PY - 2015/4/8/medline KW - Accessory mouth KW - Craniofacial duplication KW - Diprosopus KW - Mandibular duplication KW - Maxillary duplication SP - 290 EP - 6 JF - Journal of cranio-maxillo-facial surgery : official publication of the European Association for Cranio-Maxillo-Facial Surgery JO - J Craniomaxillofac Surg VL - 42 IS - 4 N2 - Diprosopus (Greek; di-, "two" + prosopon, "face"), or craniofacial duplication, is a rare craniofacial anomaly referring to the complete duplication of facial structures. Partial craniofacial duplication describes a broad spectrum of congenital anomalies, including duplications of the oral cavity. This paper describes a 15 month-old female with a duplicated oral cavity, mandible, and maxilla. A Tessier type 7 cleft, midline meningocele, and duplicated hypophysis were also present. The preoperative evaluation, surgical approach, postoperative results, and a review of the literature are presented. The surgical approach was designed to preserve facial nerve innervation to the reconstructed cheek and mouth. The duplicated mandible and maxilla were excised and the remaining left maxilla was bone grafted. Soft tissue repair included closure of the Tessier type VII cleft. Craniofacial duplication remains a rare entity that is more common in females. The pathophysiology remains incompletely characterized, but is postulated to be due to duplication of the notochord, as well as duplication of mandibular growth centres. While diprosopus is a severe deformity often associated with anencephaly, patients with partial duplication typically benefit from surgical treatment. Managing craniofacial duplication requires a detailed preoperative evaluation as well as a comprehensive, staged treatment plan. Long-term follow up is needed appropriately to address ongoing craniofacial deformity. SN - 1878-4119 UR - https://www.unboundmedicine.com/medline/citation/23969147/Partial_craniofacial_duplication:_a_review_of_the_literature_and_case_report_ DB - PRIME DP - Unbound Medicine ER -