Further Refinements in Otoplasty Surgery: a modified approach to prevent suture extrusion in cartilage-suturing otoplasty using a postauricular dermofascial flap.

BACKGROUND: Suture extrusion has been reported to be the most common complication following cartilage-sparing otoplasty. Several studies have described various designs of postauricular flaps to cover the cartilage sutures and reduce the incidence of suture extrusion. PATIENTS AND METHODS: One hundred consecutive patients with prominent ears were operated between January 2018 and February 2023 using a de-epithelialized postauricular dermofascial flap that is performed as an adjunct to our cartilage-sparing otoplasty technique which is essentially a combined modified Mustardé and Furnas technique. The flap is used to cover the cartilage holding sutures with an extra layer of vascularized tissue to avoid the scar being directly over these sutures. RESULTS: The median age of patients was 12 (IQR 9-15) years. One out of the 100 patients (1%) developed a postauricular skin infection requiring surgical removal of sutures. Primary suture extrusion did not occur, compared to our earlier study of cartilage-sparing technique without the postauricular flap in which 17 out of 200 patients had suture extrusion (p=0.001). No hematoma occurred that necessitated return to the theatre. Skin necrosis and wound dehiscence did not occur in any case. No patients developed cartilage deformities or relapse requiring surgical correction. CONCLUSIONS: Combining cartilage-sparing otoplasty using sutures and the described postauricular dermofascial flap is simple to perform and has significantly reduced the complication rate and improved the outcome compared to cartilage-sparing otoplasty alone. We recommend using this flap for both primary and revisional otoplasty.

Patient factors associated with novel EAR-Q appearance, psychosocial, and social scales: A cross-sectional study and regression analysis.

INTRODUCTION: The EAR-Q is a rigorously validated patient-reported outcome measure, which evaluates ear appearance and health-related quality of life (HRQL) in patients with congenital or acquired ear conditions. The aim of this study was to conduct an exploratory analysis to examine the factors associated with EAR-Q appearance and HRQL scale scores. METHODS: In this study, 862 participants, aged 8-29 years, with congenital or acquired ear conditions, completed the EAR-Q as part of an international field-test study. Patients responded to demographic and clinical questions as well as the EAR-Q. Univariable and multivariable linear regression analyses were used to determine factors that were significant predictors for the scores on the EAR-Q Appearance, Psychological, and Social scales. RESULTS: Most participants were men (57.4%), awaiting treatment (55.0%), and had a microtia diagnosis (70.4%), with a mean age of 13 (±4) years. Worse ear appearance scores (p < 0.02) were associated with male gender, microtia, no history of treatment, ear surgery within 6 months, unilateral involvement, and greater self-reported ear asymmetry. Decreased psychological scores (p < 0.01) were associated with increasing participant age, no treatment history, recent ear surgery, and dissatisfaction with ears matching or overall dissatisfaction. Lower social scores (p ≤ 0.04) were associated with no treatment history, those awaiting surgery, ear surgery within the last 6 months, bilateral involvement, and self-reported ears matching or overall appearance. CONCLUSION: This analysis identified patient factors that may influence ear appearance and HRQL scale scores. These findings provide evidence of patient factors that should be adjusted for when undertaking future observational research designs using the EAR-Q in this patient population.

Management of helical adhesion malformation.

Congenital auricular anomalies are common, however very few articles in the English literature address helical adhesion malformation, whereby the helix is adhered to the scapha. We describe a surgical approach to correct helical adhesion malformation which, unlike previously described methods, is simpler and results in an inconspicuous scar on the back of the ear. We illustrate our technique with an 11-year-old girl with bilateral helical adhesion malformations. Satisfactory ear shape was achieved bilaterally without complication. Meticulous dissection over the helix and exposure beyond the adhesion is of paramount importance in successfully correcting this malformation. Through a posterior auricular incision, this wide exposure and visualisation of the helix can be attained, with the added benefit of a scar hidden on the back of the ear.

Human platelet lysate enhances proliferation but not chondrogenic differentiation of pediatric mesenchymal progenitors.

BACKGROUND AIMS: Cell therapies have the potential to improve reconstructive procedures for congenital craniofacial cartilage anomalies such as microtia. Adipose-derived stem cells (ADSCs) and auricular cartilage stem/progenitor cells (CSPCs) are promising candidates for cartilage reconstruction, but their successful use in the clinic will require the development of xeno-free expansion and differentiation protocols that can maximize their capacity for chondrogenesis. METHODS: We assessed the behavior of human ADSCs and CSPCs grown either in qualified fetal bovine serum (FBS) or human platelet lysate (hPL), a xeno-free alternative, in conventional monolayer and 3-dimensional spheroid cultures. RESULTS: We show that CSPCs and ADSCs display greater proliferation rate in hPL than FBS and express typical mesenchymal stromal cell surface antigens in both media. When expanded in hPL, both cell types, particularly CSPCs, maintain a spindle-like morphology and lower surface area over more passages than in FBS. Both media supplements support chondrogenic differentiation of CSPCs and ADSCs grown either as monolayers or spheroids. However, chondrogenesis appears less ordered in hPL than FBS, with reduced co-localization of aggrecan and collagen type II in spheroids. CONCLUSIONS: hPL may be beneficial for the expansion of cells with chondrogenic potential and maintaining stemness, but not for their chondrogenic differentiation for tissue engineering or disease modeling.

Lop Ear to Conchal Microtia: An Algorithmic Surgical Approach.

BACKGROUND: The lop ear deformity is defined by a deficient helix and scapha, underdeveloped anthelix, and downfolding of the helix. The terminology used is still confusing, and the treatment is not entirely structured. The aim of this study was to provide a new systematic surgical approach of this deformity based on our center's experience. MATERIALS AND METHODS: All patients undergoing surgical correction of lop ears between 2007 and 2019 at Great Ormond Street Hospital were included. Patients' data, surgical techniques, and postoperative complications were recorded. RESULTS: Based on our records, we identified 3 surgical techniques for the correction of lop ears, based on the degree of deformity encountered. In a mild lop ear, correction was achieved with a modified otoplasty technique by improving the definition of the antihelix and superior crus. In a moderate deformity, additional remodeling of the lidded helix was performed (extended otoplasty), whereas for the severe lop ear, the amount of cupping and the deficient cartilage required formal reconstruction using a carved rib cartilage framework. There were a total of 109 patients and 146 lop ears: 58 mild, 27 moderate, and 61 severe lop ears. CONCLUSION: We feel that there is a point in the spectrum of congenital ear deformity when a severe lop ear becomes a conchal microtia and recommend this approach to simplify the management of these cases. This is intended to bring greater clarity to how to deal with lop ears, based on the severity of the deformity and the surgical techniques used.

An international study to develop the EAR-Q patient-reported outcome measure for children and young adults with ear conditions.

BACKGROUND: There is currently a lack of patient-reported outcome measures for ear reconstruction. We developed the EAR-Q to measure ear appearance and post-operative adverse effects from the patient perspective. METHODS: Field-test data were collected from children and young adults in eight countries between 13 May 2016 and 12 December 2019. Rasch measurement theory (RMT) analysis was used to refine the scales and to examine their psychometric properties. RESULTS: Participants had microtia (n = 607), prominent ears (n = 145) or another ear condition (n = 111), and provided 960 assessments for the Appearance scale (e.g., size, shape, photos), and 137 assessments for the Adverse Effects scale (e.g., itchy, painful, numb). RMT analysis led to the reduction of each scale to 10-items. Data fit the Rasch model for the Appearance (X2(80) = 90.9, p = 0.19) and Adverse Effects (X2(20) = 24.5, p = 0.22) scales. All items in each scale had ordered thresholds and good item fit. There was no evidence of differential item function for the Appearance scale by age, gender, language, or type of ear condition. Reliability was high for the Appearance scale, with person separation index (PSI) and Cronbach alpha values with and without extremes ≥0.92. Reliability for the Adverse Effects scale was adequate (i.e., PSI and Cronbach alpha values ≥0.71). Higher scores (liked appearance more) correlated with higher scores (better) on Psychological, Social and School scales. INTERPRETATION: The EAR-Q can be used in those 8-29 years of age to understand the patient perspective in clinical practice and research, and in addition, can be used to benchmark outcomes for ear reconstruction internationally.

Modification of the cartilaginous framework for autologous ear reconstruction: Construction of a stable complete ring framework with grander highs and lows.

BACKGROUND: Successful microtia reconstruction involves fabrication of a framework with well-defined ear subunits. Tragal definition and deep conchal bowl are key elements to produce a natural well-defined and contoured ear. We describe a modification in the cartilage framework with the purpose of increasing framework stability, tragal definition and conchal bowl depth. METHODS: The tragus is placed on two cartilaginous bars (L-shaped), which are fixed to the framework base block creating a complete ring. These increase tragal projection and conchal depth. The tragus is carved angled posteriorly to have a shadowing effect on the absent auditory canal. Aesthetic outcome was assessed, at least 6 months after the second stage, on a 5-point ordinal scale (1-5) using the scoring system published in the UK microtia care standards and agreed on by the International Society for Auricular Reconstruction (ISAR). RESULTS: A total of 406 auricular reconstructions were performed in 363 patients (206 right, 114 left, 43 bilateral). After excluding cases who did not have second stage and those without complete photographs, 290 cases were assessed. The described modification in the framework carving was performed in 255 ears out of these 290 ears. The mean aesthetic score before and after the modification was 2.4 and 3.09 respectively for the tragus (p < 0.001), 2.2 and 2.95 for the intertragal notch (p < 0.001) and 2.77 and 3.49 for the concha (p < 0.001). CONCLUSIONS: This technical refinement has resulted in increased permanence and definition of the tragus and deeper and more aesthetic concha.

Arterio-venous malformations of the ear: Description of distinct anatomical presentation and multidisciplinary management approach.

BACKGROUND: Arterio-venous malformations (AVMs) of the ear are a rare entity and their management should be decided in a dedicated multidisciplinary team (MDT) setting. The aim of this study is to describe the distinct anatomical patterns of the auricular AVMs in our unit and propose a combined interventional radiological and surgical approach. MATERIALS AND METHODS: All consecutive patients presenting with AVMs of the ear and reviewed by the Vascular Anomalies MDT between 2014 and 2019 were included in this study. Signs, symptoms, diagnostic investigations and operative findings were collected prospectively. RESULTS: After reviewing our nine patients, we identified four anatomical patterns of auricular AVMs: I: involves just a component of the ear and should undergo embolization followed by excision and reconstruction without significant loss of form; II: affects the superior two-thirds of the ear, sparing the lobule and part of the conchal bowl; these patients should undergo embolization, excision and monitoring before formal reconstruction of the ear is offered; III: involves the entire ear and should undergo embolization and pinnectomy; if there is no recurrence, the patients can be offered either a carved-rib cartilage reconstruction or a prosthesis, depending on the quality of the surrounding soft-tissues; IV: involves the ear and surrounding tissue, making surgical management and subsequent reconstruction extensive. CONCLUSION: The management of auricular AVMs is based on the extent of the ear involved. We feel that our combined interventional radiological and surgical approach will aid the management of these complex patients.

Modeling Normal and Pathological Ear Cartilage in vitro Using Somatic Stem Cells in Three-Dimensional Culture.

Microtia (underdeveloped ear) is a rare congenital dysmorphology affecting the development of the outer ear. Although human microtic cartilage has not been fully characterized, chondrogenic cells derived from this tissue have been proposed as a suitable source for autologous auricular reconstruction. The aim of this study was to further characterize native microtic cartilage and investigate the properties of cartilage stem/progenitor cells (CSPCs) derived from it. Two-dimensional (2D) systems are most commonly used to assess the chondrogenic potential of somatic stem cells in vitro, but limit cell interactions and differentiation. Hence here we investigated the behavior of microtic CSPCs in three-dimensional spheroid cultures. Remarkable similarities between human microtic cartilages from five patients, as compared to normal cartilage, were observed notwithstanding possibly different etiologies of the disease. Native microtic cartilage displayed poorly defined perichondrium and hyper-cellularity, an immature phenotype that resembled that of the normal developing human auricular cartilage we studied in parallel. Crucially, our analysis of microtic ears revealed for the first time that, unlike normal cartilage, microtic cartilages are vascularized. Importantly, CSPCs isolated from human microtic and normal ear cartilages were found to recapitulate many characteristics of pathological and healthy tissues, respectively, when allowed to differentiate as spheroids, but not in monolayer cultures. Noteworthily, starting from initially homogeneous cell pellets, CSPC spheroids spontaneously underwent a maturation process in culture, and formed two regions (inner and outer region) separated by a boundary, with distinct cell types that differed in chondrogenic commitment as indicated by expression of chondrogenic markers. Compared to normal ear-derived spheroids, microtic spheroids were asymmetric, hyper-cellularized and the inner and outer regions did not develop properly. Hence, their organization resembled that of native microtic cartilage. Together, our results identify novel features of microtic ears and highlight the importance of 3D self-organizing in vitro systems for better understanding somatic stem cell behavior and disease modeling. Our observations of ear-derived chondrogenic stem cell behavior have implications for choice of cells for tissue engineered reconstructive purposes and for modeling the etiopathogenesis of microtia.

Three-dimensional environment and vascularization induce osteogenic maturation of human adipose-derived stem cells comparable to that of bone-derived progenitors.

While human adipose-derived stem cells (hADSCs) are known to possess osteogenic differentiation potential, the bone tissues formed are generally considered rudimentary and immature compared with those made by bone-derived precursor cells such as human bone marrow-derived mesenchymal stem cells (hBMSCs) and less commonly studied human calvarium osteoprogenitor cells (hOPs). Traditional differentiation protocols have tended to focus on osteoinduction of hADSCs through the addition of osteogenic differentiation media or use of stimulatory bioactive scaffolds which have not resulted in mature bone formation. Here, we tested the hypothesis that by reproducing the physical as well as biochemical bone microenvironment through the use of three-dimensional (3D) culture and vascularization we could enhance osteogenic maturation in hADSCs. In addition to biomolecular characterization, we performed structural analysis through extracellular collagen alignment and mineral density in our bone tissue engineered samples to evaluate osteogenic maturation. We further compared bone formed by hADSCs, hBMSCs, and hOPs against mature human pediatric calvarial bone, yet not extensively investigated. Although bone generated by all three cell types was still less mature than native pediatric bone, a fibrin-based 3D microenvironment together with vascularization boosted osteogenic maturation of hADSC making it similar to that of bone-derived osteoprogenitors. This demonstrates the important role of vascularization and 3D culture in driving osteogenic maturation of cells easily available but constitutively less committed to this lineage and suggests a crucial avenue for recreating the bone microenvironment for tissue engineering of mature craniofacial bone tissues from pediatric hADSCs, as well as hBMSCs and hOPs.

Refinements in Otoplasty Surgery: Experience of 200 Consecutive Cases Using Cartilage-Sparing Technique.

BACKGROUND: Many techniques have been described to correct prominent ears, including cartilage-suturing, cartilage-scoring, and cartilage-breaking techniques. Understanding the topography and anatomy of the auricular cartilage is crucial for performing safe otoplasty with consistent results. METHODS: Two hundred consecutive patients with prominent ears were operated on using a modified Mustardé and Furnas technique with some refinements and without performing any cartilage scoring or excision. Adequate dissection and exposure of cartilage and precise repositioning of the tail of helix (cauda helicis) are keys to correction of lobule prominence without the need for any adjunctive procedures such as skin excision from the back of the lobule or suture fixation of the lobular tissues to the concha, mastoid, or scalp. Detailed description of the technique and review of the complications are presented. RESULTS: This suturing-only technique had a low complication rate. Hematoma occurred in two patients only. Skin necrosis and wound dehiscence were not reported in any patient. Suture extrusion was the most common complication and was easily managed, mostly in the clinic. Relapse of deformity needing surgical correction occurred in eight cases. CONCLUSIONS: The series demonstrates that most of the potential complications of otoplasty can be avoided and favorable results can be obtained by paying attention to the anatomical details of the deformity and the auricular cartilage anatomy. The described cartilage-sparing otoplasty procedure with the refinements outlined resulted in a reproducible natural correction, with a low risk profile, that can be applied to almost all prominent ears. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, IV.

Recommendations for the Development and Reform of Microtia and Atresia Services.

BACKGROUND: The initial step in setting up standardized microtia-atresia service is investigating the current status of the service and comparing this to internationally recognized guidelines or care standards. In many countries, documented information about microtia care is lacking. This study is an initiative to guide reform efforts of national microtia service in any country. The UK care standards for microtia-atresia can be a useful model to help set up a comprehensive microtia-atresia service. METHODS: The authors conducted a survey to investigate different aspects of microtia service in Egypt. The major plastic surgery centers (n = 22) were surveyed by a structured questionnaire. The results were compared with the UK care standards for microtia-atresia to identify the aspects that need improvement. Thorough analysis of the main problems in microtia-atresia service is presented. RESULTS: The authors found that microtia service is fragmented between the surveyed centers with 65% of the centers treating less than 10 microtia cases annually. Multiple surgeons are responsible for ear reconstruction in 90% of centers and only 25% of them practise a multidisciplinary team approach. None of the centers uses validated tools of aesthetic or psychological patient-reported outcome measures. RECOMMENDATIONS: These 5 recommendations are the keys to reforming microtia service in any country:(1) Establishing nationally designated centers to concentrate the required expertise.(2) Assigning fewer high-volume surgeons to optimize the surgical outcomes.(3) Providing treatment by experienced multidisciplinary teams.(4) Using validated tools of patient-reported outcome measures.(5) Collecting and keeping standardized records for regular audit and intercenter studies.

A modified reconstructive technique for paediatric congenital alar rim deformity.

Alar subunit retraction or notching whether acquired or congenital, can be challenging to reconstruct. Congenital cysts and benign lumps involving the nasal alar region can also result in acquired alar deformity once resected. Published reports describe different surgical methods, but we demonstrate a simple and highly satisfying technique. We present four paediatric cases with alar rim defects and demonstrate our modified surgical technique. All patients had an unremarkable recovery. Our outcomes demonstrate a minimal yet effective one-stage technique for correction of alar defects in paediatric patients that results in high patient satisfaction and allows for future secondary surgery if required.

Cartilage exposure following autologous microtia reconstruction: An algorithmic treatment approach✰.

BACKGROUND: Successful reconstruction of microtia involves fabrication of a cartilaginous framework and provision of thin, durable, soft tissue cover. Vascular compromise of this skin envelope can lead to exposure of the underlying cartilage, resulting in cartilage resorption and distortion of the final form of the ear construct. We describe our algorithm for management of this complication. METHODS: All patients who underwent autologous ear reconstruction by a single surgeon (NWB) from April 2006 to September 2012 were retrospectively reviewed to identify any that developed exposure of the underlying cartilage framework. Details related to timing, location, size and management of the cartilage exposure were collected. RESULTS: From a total of 230 autologous auricular reconstructions (median age at first stage, 11.4 years), 15 exposures of the cartilage framework were identified. All exposures occurred following the first stage of reconstruction (mean of post-operative day 29, range, 7-86 days). Large areas of exposure (> 10 mm2) required surgical management, with debridement and coverage with either a cutaneous or fascial flap, depending on the location. Areas < 10 mm2 were managed conservatively. All exposures were successfully treated with no adverse effects on the final aesthetic outcome. CONCLUSION: Cartilage exposure following autologous microtia reconstruction can be a devastating complication if not addressed in a prompt and effective manner. The management strategy we propose provides a concise algorithm to guide the treatment of cartilage exposure. LEVEL OF EVIDENCE: Therapeutic, grade III.

Development of a New Patient-reported Outcome Measure for Ear Conditions: The EAR-Q.

BACKGROUND: Patient-reported outcome measures are widely used to improve health services and patient outcomes. The aim of our study was to describe the development of 2 ear-specific scales designed to measure outcomes important to children and young adults with ear conditions, such as microtia and prominent ears. METHODS: We used an interpretive description qualitative approach. Semi-structured qualitative and cognitive interviews were performed with participants with any type of ear condition recruited from plastic surgery clinics in Canada, Australia, United States, and United Kingdom. Participants were interviewed to elicit new concepts. Interviews were audio-recorded, transcribed, and coded using the constant comparison approach. Experts in ear reconstruction were invited to provide input via an online Research Electronic Data Capture survey. RESULTS: Participants included 25 patients aged 8-21 years with prominent ears (n = 9), microtia (n = 14), or another condition that affected ear appearance (n = 2). Analysis of participant qualitative data, followed by cognitive interviews and expert input, led to the development and refinement of an 18-item ear appearance scale (eg, size, shape, look up close, look in photographs) and a 12-item adverse effects scale (eg, itchy, painful, numb). CONCLUSIONS: The EAR-Q in currently being field-tested internationally. Once finalized, we anticipate the EAR-Q will be used in clinical practice and research to understand the patient's perspective of outcomes following ear surgery.

Splinting After Ear Reconstruction: A Stepwise and Inexpensive Workflow Protocol.

BACKGROUND: Long-term postoperative splinting plays a role in the prevention of contracture of the grafted skin after a second-stage ear reconstruction. The scar retraction could lead to an unfavorable aesthetic outcome. Splinting could play a role to overcome or prevent the loss of projection and the obliteration of the sulcus. MATERIAL AND METHODS: We have defined the characteristics of an ideal long-term splint and present a stepwise clinical protocol for the fabrication of an ethylene-vinyl acetate splint. The splint was applied to all patients included in a prospective study on the postoperative splinting regime. Ethylene-vinyl acetate has proved its safety and longevity in dental prosthetics. CONCLUSIONS: Patient compliance was optimal, and no allergic reactions, pressure sores, or skin necrosis were reported. The splint is self-retaining and light weight. Because of its transparent color, it can be easily camouflaged. A stepwise clinical protocol for the fabrication of a low-cost patient-specific ear splint is presented.

An overview of the therapeutic potential of regenerative medicine in cutaneous wound healing.

The global burden of disease associated with wounds is an increasingly significant public health concern. Current treatments are often expensive, time-consuming and limited in their efficacy in chronic wounds. The challenge of overcoming current barriers associated with wound care requires innovative management techniques. Regenerative medicine is an emerging field of research that focuses on the repair, replacement or regeneration of cells, tissues or organs to restore impaired function. This article provides an overview of the pathophysiology of wound healing and reviews the latest evidence on the application of the principal components of regenerative medicine (growth factors, stem cell transplantation, biomaterials and tissue engineering) as therapeutic targets. Improved knowledge and understanding of the pathophysiology of wound healing has pointed to new therapeutic targets. Regenerative medicine has the potential to underpin the design of specific target therapies in acute and chronic wound healing. This personalised approach could eventually reduce the burden of disease associated with wound healing. Further evidence is required in the form of large animal studies and clinical trials to assess long-term efficacy and safety of these new treatments.

Towards reconstruction of epithelialized cartilages from autologous adipose tissue-derived stem cells.

Deformities of the upper airways, including those of the nose and throat, are typically corrected by reconstructive surgery. The use of autologous somatic stem cells for repair of defects could improve quality and outcomes of such operations. The present study explored the ability of paediatric adipose-derived stem cells (pADSCs), a readily available source of autologous stem cells, to generate a cartilage construct with a functional epithelium. Paediatric ADSCs seeded on the biodegradable nanocomposite polymer, polyhedral oligomeric silsesquioxane poly(ϵ-caprolactone-urea) urethane (POSS-PCL), proliferated and differentiated towards mesenchymal lineages. The ADSCs infiltrated three-dimensional POSS-PCL nanoscaffold and chondroid matrix was observed throughout chondrogenically induced samples. In ovo chorioallantoic membrane-grafted ADSC-nanoscaffold composites were enwrapped by host vessels indicating good compatibility in an in vivo system. Furthermore, pADSCs could be induced to transdifferentiate towards barrier-forming epithelial-like cells. By combining differentiation protocols, it was possible to generate epithelial cell lined chondrogenic micromasses from the same pADSC line. This proof-of-concept study appears to be the first to demonstrate that individual pADSC lines can differentiate towards two different germ lines and be successfully co-cultured. This has important implications for bioengineering of paediatric airways and further confirms the plastic nature of ADSCs. Copyright © 2016 John Wiley & Sons, Ltd.

Combined soft and skeletal tissue modelling of normal and dysmorphic midface postnatal development.

BACKGROUND: Midface hypoplasia as exemplified by Treacher Collins Syndrome (TCS) can impair appearance and function. Reconstruction involves multiple invasive surgeries with variable long-term outcomes. This study aims to describe normal and dysmorphic midface postnatal development through combined modelling of skeletal and soft tissues and to develop a surgical evaluation tool. MATERIALS AND METHODS: Midface skeletal and soft tissue surfaces were extracted from computed tomography scans of 52 control and 14 TCS children, then analysed using dense surface modelling. The model was used to describe midface growth, morphology, and asymmetry, then evaluate postoperative outcomes. RESULTS: Parameters responsible for the greatest variation in midface size and shape showed differences between TCS and controls with close alignment between skeletal and soft tissue models. TCS children exhibited midface dysmorphology and hypoplasia when compared with controls. Asymmetry was also significantly higher in TCS midfaces. Combined modelling was used to evaluate the impact of surgery in one TCS individual who showed normalisation immediately after surgery but reversion towards TCS dysmorphology after 1 year. CONCLUSION: This is the first quantitative analysis of postnatal midface development using combined modelling of skeletal and soft tissues. We also provide an approach for evaluation of surgical outcomes, laying the foundations for future development of a preoperative planning tool.