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• W3127894132 abstract "Facial Plastic Surgery & Aesthetic MedicineVol. 23, No. 3 Invited CommentaryFree AccessCommentary on “Airway Growth in Preoperative Patients with Crouzon Syndrome” by Lu et al.Srinivas M. Susarla and Richard A. HopperSrinivas M. Susarla*Address correspondence to: Srinivas M. Susarla, MD, DMD, MPH, Division of Plastic and Craniofacial Surgery, Seattle Children's Hospital, 4800 Sand Point Way NE, Seattle, WA 98105, USA, E-mail Address: srinivas.susarla@seattlechildrens.orgDivision of Plastic and Craniofacial Surgery and Department of Surgery, Division of Plastic Surgery, Craniofacial Center, Seattle Children's Hospital, University of Washington School of Medicine, Seattle, Washington, USA.Search for more papers by this author and Richard A. HopperDivision of Plastic and Craniofacial Surgery and Department of Surgery, Division of Plastic Surgery, Craniofacial Center, Seattle Children's Hospital, University of Washington School of Medicine, Seattle, Washington, USA.Search for more papers by this authorPublished Online:10 May 2021https://doi.org/10.1089/fpsam.2020.0565AboutSectionsPDF/EPUB ToolsPermissionsDownload CitationsTrack CitationsAdd to favorites Back To Publication ShareShare onFacebookTwitterLinked InRedditEmail Lu et al. provide a comprehensive analysis of airway anatomy in patients with Crouzon syndrome in the context of fourth-dimensional changes with skeletal growth.1 Using data from a shared registry, the authors compared the three-dimensional airway anatomy in patients with Crouzon syndrome to age-matched patients without facial skeletal anomalies. Of note, the Crouzon cohort had not undergone surgery, thus representing a population of patients whose development would be uninfluenced by intervention. Nasal and pharyngeal airways were compared between cases and controls. Across all age groups, patients with Crouzon syndrome had smaller nasal and pharyngeal airway volumes compared with controls, with altered cranial base angulations and reduced subcranial space. Subgroup analyses revealed that infants (age <6 months) with Crouzon syndrome had smaller nasal airway volumes (∼40%), with cross-sectional restriction at the choanae. This difference increased until ∼2 years of age; dimensions were normalized by 6 years of age. In the Crouzon patients, pharyngeal airway anatomy had notable constriction in infancy that did not resolve over time with midfacial growth.The Crouzon midface is a morphologically normal midface that is in an atypical position. In this context, alteration of the skeletal framework through midface advancement, either as a component of frontofacial advancement (e.g., monobloc distraction) or as a solitary subcranial movement (Le Fort III distraction, LF3), makes intuitive sense for airway expansion. What remains to be determined is which patients will benefit from which particular intervention. Compounding this challenge is standard protocols at many centers for cranial vault expansion through fronto-orbital advancement (FOA) in infancy, which will correct the anterior skull base constriction and cranio-orbital morphology, but not reliably address upper airway obstruction. The well-established phenomenon of relapse of the FOA in this population, with regression to the preoperative phenotype, adds to the complexity of decision-making.As mentioned by the authors, the cranial vault and midface dysplasia can be addressed in a single stage with the monobloc procedure, wherein the fronto-orbital framework and the midface are synchronously advanced (i.e., FOA+LF3) using a transcranial approach. This carries the benefit of addressing the cephalocranial disproportion and the potential for airway expansion. This procedure has been less common historically due to concerns over increased risk relative to subcranial procedures. Recent studies have provided mixed data regarding whether transcranial procedures are inherently more risky than subcranial procedures.2,3 This may be an effect of the timing of intervention; monobloc osteotomies in young children may carry a higher risk of dural injury, given the lack of significant ossification of the anterior cranial base before age 5, with dural invaginations and adhesions that are prone to iatrogenic injury during epidural dissection.To the extent that treatment planning is geared toward addressing the primary dysmorphology, there are meaningful observations that can be derived from this study. The predicted magnitude of skeletal movement necessary to effectively normalize the airway anatomy in children <2 years of age may be possible with monobloc distraction, although the relapse rate is unclear; definitive airway management with tracheostomy should not be neglected as an option in severe cases. As the Crouzon facial skeleton develops, continuation of a staged approach established in infancy (e.g., FOA) with subcranial surgery in the mixed dentition is warranted to address both morphology and airway anatomy. Frontofacial advancement may be indicated if there is ICH or substantial relapse of the FOA with associated airway obstruction. Interventions in the late mixed dentition can also include counterclockwise craniofacial distraction osteogenesis (C3DO), which provides coordinate rotation-advancement of the midface and mandible, and has demonstrated efficacy in improving airway obstruction in tracheostomy-dependent children with Treacher Collins syndrome.4 In patients at or near skeletal maturity, one needs to consider whether the morphological deficiencies and their associated functional consequences (malocclusion and upper airway obstruction) can be best treated in a single-stage manner with multilevel procedures (e.g., LF3 osteotomy with simultaneous maxillomandibular advancement) or through a multistaged approach (e.g., LF3 distraction followed by maxillomandibular advancement).5Lu et al. have provided important information about how the Crouzon skeleton changes over time. Although the older unoperated Crouzon patients in the study cohort may not represent the same phenotypes seen in many centers, who typically require some form of skeletal surgery before maturity, the authors have confirmed that airway volume constriction in patients with Crouzon syndrome is multilevel and corresponds to the anatomic deficiencies seen in this population. What remains unclear is whether the observed anatomic differences at a given age will have commensurate correlation to clinically evident upper airway obstruction and if surgical modifications of the skeletal framework will effectively address functionally consequential constriction of a dynamic airway. In this regard, this study has helped develop a robust foundation for further investigation of airway dynamics in the context of anatomic differences, and how various surgical interventions may meaningfully impact upper airway physiology.Ethics StatementBoth authors have contributed to this study in accordance with the ICMJE guidelines for authorship.Author Disclosure StatementNo competing financial interests exist.Funding InformationNo funding was received for this work.References1. Lu X, Forte AJ, Park KE, et al. Airway growth in preoperative patients with Crouzon syndrome. Facial Plastic Surg Aest Med. 2021;23(3):153–159. Google Scholar2. Munabi NCO, Williams M, Nagengast ES, et al. Outcomes of intracranial versus subcranial approaches to the frontofacial skeleton. J Oral Maxillofac Surg. 2020;78(9):1609–1616. Crossref, Medline, Google Scholar3. Zhang RS, Lin LO, Hoppe IC, Swanson JW, Bartlett SP, Taylor JA. Retrospective review of the complication profile associated with 71 subcranial and transcranial midface distraction procedures at a single institution. Plast Reconstr Surg. 2019;143(2):521–530. Crossref, Medline, Google Scholar4. Hopper RA, Kapadia H, Susarla S, Bly R, Johnson K. Counterclockwise craniofacial distraction osteogenesis for tracheostomy-dependent children with Treacher Collins syndrome. Plast Reconstr Surg. 2018;142(2):447–457. Crossref, Medline, Google Scholar5. Hammoudeh JA, Goel P, Wolfswinkel EM, et al. Simultaneous midface advancement and orthognathic surgery: a powerful technique for managing midface hypoplasia and malocclusion. Plast Reconstr Surg. 2020;145(6):1067e. Crossref, Medline, Google ScholarFiguresReferencesRelatedDetails Volume 23Issue 3Jun 2021 InformationCopyright 2021, American Academy of Facial Plastic and Reconstructive Surgery, Inc.To cite this article:Srinivas M. Susarla and Richard A. Hopper.Commentary on “Airway Growth in Preoperative Patients with Crouzon Syndrome” by Lu et al..Facial Plastic Surgery & Aesthetic Medicine.Jun 2021.197-198.http://doi.org/10.1089/fpsam.2020.0565Published in Volume: 23 Issue 3: May 10, 2021Online Ahead of Print:February 12, 2021PDF download" @default.
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