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Christopher R Forrest - Top 30 Publications

Psychometric findings and normative values for the CLEFT-Q based on 2434 children and young adult patients with cleft lip and/or palate from 12 countries.

Patients with cleft lip and/or palate can undergo numerous procedures to improve appearance, speech, dentition and hearing. We developed a cleft-specific patient-reported outcome instrument to facilitate rigorous international measurement and benchmarking.

Assessing technical performance and determining the learning curve in cleft palate surgery using a high fidelity cleft palate simulator.

Technical skills assessment can provide objective measures of surgical performance. This study assessed technical performance in cleft palate repair using a newly developed assessment tool and high fidelity cleft palate simulator through a longitudinal simulation training exercise.

Clinical Significance of Venous Anomalies in Syndromic Craniosynostosis.

The pattern of cranial venous drainage in syndromic craniosynostosis is unpredictable and not adequately understood. Collateral channels substitute for stenotic venous sinuses and pose potential risk for surgical intervention. The purpose of this study was to analyze the patterns of venous drainage in patients with syndromic craniosynostosis and their influence on operative planning and morbidity.

What Matters to Patients With Cleft Lip and/or Palate: An International Qualitative Study Informing the Development of the CLEFT-Q.

The goal of treatment for individuals with cleft lip and/or palate (CL/P) is to improve physical, psychological, and social health. Outcomes of treatment are rarely measured from the patient's perspective. The aim of the study was to develop a conceptual framework for a patient-reported outcome (PRO) instrument for individuals with clefts (CLEFT-Q) by developing an in-depth understanding of issues that individuals consider to be important.

The impact of a career night information session on medical students' perceptions of the scope of practice of plastic surgeons.

Teaching Palatoplasty Using a High-Fidelity Cleft Palate Simulator.

Cleft palate repair is a challenging procedure for cleft surgeons to teach. A novel high-fidelity cleft palate simulator has been described for surgeon training. This study evaluates the simulator's effect on surgeon procedural confidence and palatoplasty knowledge among learners.

Development of a New Module of the FACE-Q for Children and Young Adults with Diverse Conditions Associated with Visible and/or Functional Facial Differences.

Editorial. "Horses for courses".

Establishing Content Validity of the CLEFT-Q: A New Patient-reported Outcome Instrument for Cleft Lip/Palate.

The CLEFT-Q is a new patient-reported outcome instrument designed to measure outcomes that matter to patients. The aim of this qualitative study was to establish content validity of the CLEFT-Q in patients who differ by age and culture.

Infant Robotic Cleft Palate Surgery: A Feasibility Assessment Using a Realistic Cleft Palate Simulator.

A surgical robot offers enhanced precision, visualization, and access and the potential to improve outcomes in cleft palate surgery. The goal of this study was to investigate the feasibility of using the da Vinci robot for cleft palate repair in infants using a cleft palate simulator test bed.

International multiphase mixed methods study protocol to develop a cross-cultural patient-reported outcome instrument for children and young adults with cleft lip and/or palate (CLEFT-Q).

Patient-reported outcome (PRO) instruments should be developed according to rigorous guidelines in order to provide clinically meaningful, scientifically sound measurement. Understanding the methodology behind instrument development informs the selection of the most appropriate tool. This mixed methods protocol describes the development of an internationally applicable PRO instrument, the CLEFT-Q, for evaluating outcomes of treatment for cleft lip and/or palate (CL/P).

Bilateral Malar Reconstruction Using Patient-Specific Polyether Ether Ketone Implants in Treacher-Collins Syndrome Patients With Absent Zygomas.

The authors performed bilateral malar reconstruction using polyether ether ketone implants in 3 patients with Treacher-Collins syndrome with absent, as opposed to hypoplastic, zygomata. These patient-specific implants were fabricated using computed-aided design software reformatted from three-dimensional bony preoperative computed tomography images. The first time the authors performed this procedure the implant compressed the globe resulting in temporary anisocoria that was quickly recognized intraoperatively. The implant was immediately removed and the patient made a full-recovery with no ocular disturbance. The computer-aided design and manufacturing process was adjusted to include periorbital soft-tissue boundaries to aid in contouring the new implants. The same patient, and 2 further patients, subsequently underwent malar reconstruction using this soft tissue periorbital boundary fabrication process with an additional 2 mm relief removed from the implant's orbital surface. These subsequent procedures were performed without complication and with pleasing aesthetic results. The authors describe their experience and the salutary lessons learnt.

Evaluation and Implementation of a High-Fidelity Cleft Palate Simulator.

Cleft palate repair is a challenging procedure to learn because of the delicate tissue handling required and the small confines of the infant oral cavity. As a result, cleft palate simulators have previously been described to augment cleft palate repair training. Although valuable, they lack the fidelity for this complex procedure.

Content Validity of Patient-Reported Outcome Instruments used with Pediatric Patients with Facial Differences: A Systematic Review.

Objective The aim of this systematic review was to identify patient-reported outcome (PRO) instruments used in research with children/youth with conditions associated with facial differences to identify the health concepts measured. Design MEDLINE, EMBASE, CINAHL, and PsycINFO were searched from 2004 to 2016 to identify PRO instruments used in acne vulgaris, birthmarks, burns, ear anomalies, facial asymmetries, and facial paralysis patients. We performed a content analysis whereby the items were coded to identify concepts and categorized as positive or negative content or phrasing. Results A total of 7,835 articles were screened; 6 generic and 11 condition-specific PRO instruments were used in 96 publications. Condition-specific instruments were for acne (four), oral health (two), dermatology (one), facial asymmetries (two), microtia (one), and burns (one). The PRO instruments provided 554 items (295 generic; 259 condition specific) that were sorted into 4 domains, 11 subdomains, and 91 health concepts. The most common domain was psychological (n = 224 items). Of the identified items, 76% had negative content or phrasing (e.g., "Because of the way my face looks I wish I had never been born"). Given the small number of items measuring facial appearance (n = 19) and function (n = 22), the PRO instruments reviewed lacked content validity for patients whose condition impacted facial function and/or appearance. Conclusions Treatments can change facial appearance and function. This review draws attention to a problem with content validity in existing PRO instruments. Our team is now developing a new PRO instrument called FACE-Q Kids to address this problem.

FGFR-associated craniosynostosis syndromes and gastrointestinal defects.

Craniosynostosis is a relatively common birth defect characterized by the premature fusion of one or more cranial sutures. Examples of craniosynostosis syndromes include Crouzon (CS), Pfeiffer (PS), and Apert (AS) syndrome, with clinical characteristics such as midface hypoplasia, hypertelorism, and in some cases, limb defects. Mutations in Fibroblast Growth Factor Receptor-2 comprise the majority of known mutations in syndromic forms of craniosynostosis. A number of clinical reports of FGFR-associated craniosynostosis patients and mouse mutants have been linked to gastrointestinal tract (GIT) disorders, leading to the hypothesis of a direct link between FGFR-associated craniosynostosis syndromes and GIT malformations. We conducted an investigation to determine GIT symptoms in a sample of FGFR-associated craniosynostosis syndrome patients and a mouse model of CS containing a mutation (W290R) in Fgfr2. We found that, compared to the general population, the incidence of intestinal/bowel malrotation (IM) was present at a higher level in our sample population of patients with FGFR-associated craniosynostosis syndromes. We also showed that the mouse model of CS had an increased incidence of cecal displacement, suggestive of IM. These findings suggest a direct relationship between FGFR-related craniosynostosis syndromes and GIT malformations. Our study may shed further light on the potential widespread impact FGFR mutations on different developmental systems. Based on reports of GIT malformations in children with craniosynostosis syndromes and substantiation with our animal model, GIT malformations should be considered in any child with an FGFR2-associated craniosynostosis syndrome. © 2016 Wiley Periodicals, Inc.

Use of the anterolateral thigh flap for reconstruction of the pediatric anophthalmic orbit.

Children with acquired anophthalmia pose unique reconstructive challenges. With implant-based reconstruction, the current standard of care, there is a tendency toward intraorbital and adnexal tissue contraction and an increasing volume discrepancy with orbital growth, resulting in enophthalmos and eyelid retraction, and also difficulty in retaining an ocular prosthesis. We describe a novel technique for secondary reconstruction of the pediatric anophthalmic orbit using a small free anterolateral thigh (ALT) flap. The technique involves release of retracted eyelids and restoration of orbital volume with an ALT flap based on the superficial temporal vessels. The purpose of this study is to detail the results of treatment of a series of four consecutive patients with unilateral acquired anophthalmia managed with ALT flaps. Improved symmetry, successful retention of an ocular prosthesis, and patient satisfaction were achieved in all cases.

Skull base development and craniosynostosis.

Abnormal skull shape resulting in craniofacial deformity is a relatively common clinical finding, with deformity either positional (positional plagiocephaly) or related to premature ossification and fusion of the skull sutures (craniosynostosis). Growth restriction occurring at a stenosed suture is associated with exaggerated growth at the open sutures, resulting in fairly predictable craniofacial phenotypes in single-suture non-syndromic pathologies. Multi-suture syndromic subtypes are not so easy to understand without imaging. Imaging is performed to define the site and extent of craniosynostosis, to determine the presence or absence of underlying brain anomalies, and to evaluate both pre- and postoperative complications of craniosynostosis. Evidence for intracranial hypertension may be seen both pre- and postoperatively, associated with jugular foraminal stenosis, sinovenous occlusion, hydrocephalus and Chiari 1 malformations. Following clinical assessment, imaging evaluation may include radiographs, high-frequency US of the involved sutures, low-dose (20-30 mAs) CT with three-dimensional reformatted images, MRI and nuclear medicine brain imaging. Anomalous or vigorous collateral venous drainage may be mapped preoperatively with CT or MR venography or catheter angiography.

Optimization of Cranio-Orbital Remodeling: Application of a Mathematical Model.

Cranio-orbital remodeling aims to correct the dysmorphic skull associated with craniosynostosis. Traditionally, the skull is reconstructed into a shape that is subjectively normal according to the surgeon's perception. We present a novel technique using a mathematical algorithm to define the optimal location for bony osteotomies and to objectively reshape the fronto-orbital bar into an ideal normal skull contour. Using pre-operative computed tomography images, the abnormal skull contour at the frontal-orbital region was obtained for infants planned to undergo cranio-orbital remodeling. The ideal skull shape was derived from an age- and sex-matched normative skull library. For each patient, the mathematical technique of dynamic programming (DP) was applied to compare the abnormal and ideal skull shapes. The DP algorithm identifies the optimal location of osteotomy sites and calculates the objective difference in surface area remaining between the normative and dysmorphic skull shape for each solution applied. By selecting the optimal solution with minimal objective difference, the surgeon is guided to reproducibly recreate the normal skull contour with defined osteotomies. The DP algorithm was applied in 13 cases of cranio-orbital remodeling. Five female and 8 male infants with a mean age of 11 months were treated for craniosynostosis classified as metopic (n = 7), unicoronal (n = 4), or bicoronal (n = 2). The mean OR time was 190.2  min (SD 33.6), mean estimated blood loss 244  cc (SD 147.6), and 10 infants required blood transfusions. Compared with a historical crania-orbital remodeling group treated without application of the algorithm, there was no significant difference in OR time, estimated blood loss, or transfusion rate. This novel technique enables the craniofacial surgeon to objectively reshape the fronto-orbital bar and reproducibly reconstruct a skull shape resembling that of normal infants.

Psychometrics: Essential for Valid, Reliable, and Responsive Measurement in the Development of Patient-reported Outcome Instruments in Plastic Surgery.

Single versus segmental maxillary osteotomies and long-term stability in unilateral cleft lip and palate related malocclusion.

To investigate the stability of single-piece versus segmental (2-piece) maxillary advancement in patients with unilateral cleft lip and palate (UCLP) treated using conventional Le Fort I orthognathic surgery.

Is Linear Advancement Related to Relapse in Unilateral Cleft Lip and Palate Orthognathic Surgery?

To investigate the stability of major versus minor Le Fort I maxillary advancements in unilateral cleft lip and palate (UCLP) patients.

Cephalometric outcomes of orthognathic surgery in hemifacial microsomia.

Hemifacial microsomia is a hypoplastic disorder of the first and second branchial arches that significantly impacts on the development of the jaws, leading to malocclusion and facial asymmetry. There is little in the literature regarding the application of orthodontic/orthognathic approaches to the correction of these deformities and the stability of the surgical results. To address this, a retrospective chart review of 10 patients with complete orthodontic records and greater than 1 year of follow-up was performed. Posteroanterior cephalograms were assessed by modified Grummons analysis to determine mandibular offset (deviation of the chin point from the skeletal midline) and occlusal cant. These measurements were performed at 3 time points (T1: preoperative, T2: immediate postoperative, T3: follow-up) to elucidate the surgical movement (T2-T1), the postoperative relapse (T3-T2), and the net gain movement (T3-T1). Maxillary movements were quantified, and the occlusal cant was expressed as a ratio between vertical heights of the maxilla at the first molar on each side. One sample t test demonstrated statistically significant surgical movement and net gain. Relapse was statistically insignificant. Repeated-measures analysis of variance demonstrated similar results for chin point position relative to the putative midline. Our results suggest that a combined orthodontic/orthognathic approach at skeletal maturity delivers improved occlusal outcomes in the long term as assessed by chin point deviation and occlusal cant, but secondary surgery rates are higher than those for orthognathic surgery in other patient groups. We advocate limiting surgery to skeletal maturity whenever possible to achieve stable long-term results while limiting morbidity and number of procedures.

Direct genesis of functional rodent and human schwann cells from skin mesenchymal precursors.

Recent reports of directed reprogramming have raised questions about the stability of cell lineages. Here, we have addressed this issue, focusing upon skin-derived precursors (SKPs), a dermally derived precursor cell. We show by lineage tracing that murine SKPs from dorsal skin originate from mesenchymal and not neural crest-derived cells. These mesenchymally derived SKPs can, without genetic manipulation, generate functional Schwann cells, a neural crest cell type, and are highly similar at the transcriptional level to Schwann cells isolated from the peripheral nerve. This is not a mouse-specific phenomenon, since human SKPs that are highly similar at the transcriptome level can be made from neural crest-derived facial and mesodermally derived foreskin dermis and the foreskin SKPs can make myelinating Schwann cells. Thus, nonneural crest-derived mesenchymal precursors can differentiate into bona fide peripheral glia in the absence of genetic manipulation, suggesting that developmentally defined lineage boundaries are more flexible than widely thought.

Outcome analysis after helmet therapy using 3D photogrammetry in patients with deformational plagiocephaly: the role of root mean square.

Deformational plagiocephaly (DP) is a multifactorial non-synostotic cranial deformity with a reported incidence as high as 1 in 7 infants in North America. Treatment options have focused on non-operative interventions including head repositioning and the use of an orthotic helmet device. Previous studies have used linear and two dimensional outcome measures to assess changes in cranial symmetry after helmet therapy. Our objective was to demonstrate improvement in head shape after treatment with a cranial molding helmet by using Root Mean Square (RMS), a measure unique to 3D photogrammetry, which takes into account both changes in volume and shape over time. Three dimensional photographs were obtained before and after molding helmet treatment in 40 infants (4-10 months old) with deformational plagiocephaly. Anatomical reference planes and measurements were recorded using the 3dMD Vultus(®) analysis software. RMS was used to quantify symmetry by superimposing left and right quadrants and calculating the mean value of aggregate distances between surfaces. Over 95% of the patients demonstrated an improvement in symmetry with helmet therapy. Furthermore, when the sample of infants was divided into two treatment subgroups, a statistically significant correlation was found between the age at the beginning of treatment and the change in the RMS value. When helmet therapy was started before 7 months of age a greater improvement in symmetry was seen. This work represents application of the technique of RMS analysis to demonstrate the efficacy of treatment of deformational plagiocephaly with a cranial molding helmet.

Quantitative facial asymmetry: using three-dimensional photogrammetry to measure baseline facial surface symmetry.

Although symmetry is hailed as a fundamental goal of aesthetic and reconstructive surgery, our tools for measuring this outcome have been limited and subjective. With the advent of three-dimensional photogrammetry, surface geometry can be captured, manipulated, and measured quantitatively. Until now, few normative data existed with regard to facial surface symmetry. Here, we present a method for reproducibly calculating overall facial symmetry and present normative data on 100 subjects.

Computed tomography-based morphometric analysis of extended strip craniectomy for sagittal synostosis.

The purpose of this study is 2-fold: (1) to identify computed tomography (CT)-based morphometric parameters that differentiate the normal skull from one with sagittal synostosis and (2) to evaluate correction of sagittal synostosis with extended strip craniectomy and postoperative helmeting based on morphometric parameters.

Surgical outcomes in craniosynostosis reconstruction: the use of prefabricated templates in cranial vault remodelling.

Cranio-orbital reshaping for anterior cranial-vault deformities associated with craniosynostosis traditionally relies on the surgeon's subjective estimate of the shape and appearance of a normal forehead. Computer-aided design/computer-aided manufacture (CAD/CAM) bandeau templates to guide reconstruction were introduced in our centre to eliminate this subjectivity and to effect more reproducible surgical results. The aim of this study was to compare two groups of patients (template, n = 14 vs. no template, n = 23) to measure surgical outcomes. The virtual, computational version of the template was used as an outcome assessment tool. It was used to calculate an intervening area under the curve (AUC) between the normative template and the patient's reconstructed supra-orbital bar on a representative computed tomography (CT) axial section. A comprehensive chart review was conducted of patients in both groups to examine the preoperative and postoperative variables. Based on the analysis performed on the immediate postoperative CT scans, in the template group - as compared to the control, no-template group - the use of the bandeau template led to a greater reduction in AUC (74% vs. 56%, p = 0.016), indicating a better conformity between the reconstructed supra-orbital bar and the ideal, normal bandeau shape. The duration of operation was significantly reduced with the use of the template (212 vs. 258 min, p < 0.001). The application of prefabricated templates in cranio-orbital reshaping is highly useful for accurate preoperative planning; reproducible and efficient intra-operative correction of dysmorphology; and objective surgical outcomes assessment.

Measuring outcomes in craniofacial and pediatric plastic surgery.

This article discusses the measurement of outcomes in craniofacial and pediatric plastic surgery, using examples of craniosynostosis and cleft lip and/or palate (CLP). The challenges in measuring the standard outcomes of function, aesthetics, and health-related quality of life are discussed, along with the importance of developing evidence and studying quality improvement in this specialty. The need to define specific and comprehensive goals is discussed with a focus on patient-reported outcomes (PROs). Examples from the development of the CLEFT-Q, a PRO instrument for patients with CLP, are provided to support the need to seek the patient perspective.

Pharmacologic prophylactic treatment for perioperative protection of skeletal muscle from ischemia-reperfusion injury in reconstructive surgery.

In autogenous muscle transplantation, unpredictable complications can cause prolonged ischemia, resulting in ischemia-reperfusion injury. The authors investigated the efficacy and mechanism of nicorandil, a nitrovasodilator and adenosine triphosphate-sensitive potassium channel opener, in inducing perioperative protection of muscle flaps from ischemia-reperfusion injury.

Craniofacial syndromes and surgery.

After studying this article, the participant should be able to: (1) Understand craniofacial dysmorphology and identify basic pediatric craniofacial syndromes. (2) Understand the functional concerns associated with these syndromes. (3) Achieve familiarity with the management protocols for the treatment of pediatric craniofacial syndromes.