Faculty Bibliography

INTRODUCTION/BACKGROUND:The number of patients who may benefit from evaluation for face transplantation in the United States (US) remains largely unknown. The goal of our study was to better delineate the pool of patients who might benefit from face transplant evaluation based on the characteristics and mechanisms of injury of previously reported face transplant recipients. METHODS:The authors utilized data from the National Electronic Injury Surveillance System-All Injury Program in this study. The US Census Bureau data were used for population estimates. Inclusion and exclusion criteria were determined based on the characteristics of face transplant recipients to date, and the mechanisms of injury they sustained ultimately necessitating face transplantation. Statistical significance was reached if P <0.05. RESULTS:The estimated annual incidence of preventable craniofacial injuries from firearms (44,266-58,299; 31.7% increase), burns (5712-19,433; 240.2% increase), and animal attacks (5355-14,666; 173.9% increase) increased from 2005 to 2014, whereas the estimated annual incidence of craniofacial injuries from machinery (3927-2933; 25.3% decrease) decreased between 2005 and 2014. The authors estimate the annual incidence rate to fall between 32.1 per 100,000 and 58.1 per 100,000 among individuals aged 20 to 64 in the US. CONCLUSION/CONCLUSIONS:In this study, the authors estimate the annual incidence rate of individuals aged 20 to 64 in the US who may benefit from face transplant evaluation and believe that this quantification has the potential to initiate actionable discussions regarding geographical and financial factors affecting access to care in this patient population.

PURPOSE/OBJECTIVE:To highlight the challenges and lessons learned in tooth-bearing maxillomandibular facial allotransplantation. PATIENTS AND METHODS/METHODS:Two patients with ballistic composite facial injury underwent tooth-bearing maxillomandibular facial transplantation (FT) after informed consent and institutional review board approval. Patient 1 had undergone total face, double jaw, teeth, and tongue transplantation in March 2012. Patient 2 had undergone partial face, double jaw, and teeth transplantation in January 2018. Le Fort III and bilateral sagittal split skeletal osteotomies were performed in both transplants. Computerized surgical planning was used in both cases, and the allografts were transferred in intermaxillary fixation (IMF) with prefabricated dental splints before rigid skeletal fixation. RESULTS:Normal class I occlusion was achieved at the conclusion of each surgery. Patient 1 had developed a 2 × 2-mm palatal fistula in the early postoperative period and had also gradually developed class III malocclusion. Orthodontic treatment was started at 5 months after transplantation but failed. A Le Fort III advancement was performed 1 month later with successful restoration of class I occlusion. The palatal fistula was successfully repaired at 9 postoperative months. Patient 2 developed a postoperative palate and floor of mouth dehiscence, requiring palatal repair and hyoid and genioglossus advancement on postoperative day (POD) 11. Orthodontic treatment was initiated for Class II malocclusion. On POD 108, left mandibular nonunion was diagnosed. Left coronoidectomy, open reduction, and internal fixation were performed. IMF was maintained for 2 weeks. Orthodontic treatment was then resumed, with normalization of the occlusion by 10 months after FT. CONCLUSIONS:Maxillomandibular transplantation is a viable reconstructive solution for composite midface defects not amenable to autologous reconstruction. Improvement of functional outcomes and prevention of major complications rely on close attention to occlusal relationships, temporomandibular joint dynamics, dental health, and the intraoral donor-recipient soft tissue interface.

Background/Purpose : APS ACTION Registry was created to study long-term outcomes in persistently antiphospholipid antibody (aPL)-positive patients with and without other systemic autoimmune diseases. Our primary objective was to determine whether clinically significant aPL profiles at baseline remain stable over time. Methods : A web-based data capture system is used to store patient demographics and aPL-related medical history. Inclusion criteria are positive aPL, based on the Updated Sapporo APS Classification Criteria, tested at least twice within one year prior to enrollment. Patients are followed every 12+/-3 months with clinical data and blood collection. For this prospective analysis of available follow-up (f/u) aPL tests, clinically significant aPL profile was defined as positive lupus anticoagulant (LA) test and/or aCL/abeta 2 GPI IgG/M > 40U. Stable aPL profile was defined as a clinically significant aPL profile in at least two-thirds of f/u measurements. Univariate and multivariable generalized linear mixed models with logit link were used to assess the effect of time and other variables of interest on odds of clinically significant aPL profile. Wilcoxon rank-sum and Fisher's exact tests were employed to compare clinical characteristics of patients with stable versus unstable aPL profiles. Results : As of January 2019, 796 patients were enrolled from 26 centers worldwide, 482 had f/u visits with aPL results, and 472 patients had a clinically significant aPL profile at baseline. Based on aPL profiles at f/u visits (median follow up: 5.1 years [interquartile range [IR]: 4.3, 5.8]; median number of f/u visits with aPL profiles: 2 [interquartile range: 1, 3]), 366/472 (78%) patients had stable aPL profiles over time (54 [11%] unstable; 52 [11%] inconclusive). Time did not affect odds of maintaining a clinically significant aPL profile at f/u (p=0.906). In multivariable analysis, time, age, concomitant systemic autoimmune disease (mainly lupus), smoking history, and hydroxychloroquine use did not affect odds of maintaining a clinically significant aPL profile at f/u. Based on crude unadjusted comparisons, patients with stable aPL profiles, compared to those with unstable profiles, were more likely to have baseline positive LA test, aCL IgM > 40U (positive trend for IgG), abeta 2 GPI IgG > 40U (positive trend for IgM), two or more positive aPL tests, and history of arterial events and aspirin use (Table). Conclusion : In approximately 80% of patients with a baseline clinically significant aPL profile (LA test and/or aCL/ abeta 2 GPI IgG/M > 40U), aPL profiles remain consistently significant (stable) during five years of follow-up. Further multivariate analysis will investigate predictors of aPL profile stability over time, and guide future validation studies of stored samples through APS ACTION core laboratories

The technical feasibility and clinical applicability of facial transplantation (FT) have been demonstrated, yet animal models with different technical nuances and allograft compositions continue to be developed. We sought to provide a comprehensive appraisal of the current scope and value of animal models in FT.

Facial transplantation provides a functional and aesthetic solution to severe facial disfigurement previously unresolved by conventional reconstruction. Few facial allografts have been ear containing; hence, there is limited knowledge of the postoperative otologic considerations. We describe the case of a 44-year-old man who underwent transplantation of the total face, eyelids, ears, scalp, and skeletal subunits in 2015 after an extensive thermal injury. We detail the patient's transition from osseointegrated prosthetic ears to an ear-containing facial allograft, and describe the unique surgical approach and challenges encountered. Subsequent bilateral revision meatoplasties were performed, which provided relief from stenosis of the external auditory meatus. Laryngoscope, 2018.

BACKGROUND:Prolonged impairment of protective ocular functions can compromise vision and lead to blindness if uncorrected. Several facial transplants have incorporated periorbital structures with variable eyelid preservation, but objective assessment of post-transplant periorbital function has been limited. MATERIALS AND METHODS/METHODS:Kinematic data were collected from a full-face recipient that included the fist total eyelid transplantation at 5 separate pre-transplant (PRE) and post-transplant time points (T1-T4). Using optical facial tracking, eyelid movements were tracked during involuntary blinking and compared with controls. RESULTS:There was significant improvement in right eye aperture from PRE to T1 (β = 5.54, P < 0.001), with no change between T1 and T4. Aperture fluctuated in the left eye, with a temporary decrease between T2 and T3 corresponding with revision brow lift (β = -4.57, P < 0.001). Although improved from the pre-transplantation, right and left eye apertures remained significantly smaller than controls at T1 and T4 (P < 0.001). Similarly, spatial coupling increased from PRE to T1 (β = 0.63, P < 0.001) and remained high at T4, albeit significantly less than controls (P < 0.001). Temporal coupling improved from PRE to T2 (β = 2.29, P < 0.02) and was sustained at subsequent time points, with no difference relative to controls at T4. Considerable improvement was observed on clinical examination, with full functional status. CONCLUSIONS:Application of a novel method for assessing functional eyelid recovery using facial tracking technology to the first total eyelid transplantation in the setting of a full facial transplant shows clear functional improvement after transplantation and suggests revisions can be performed safely to optimize aesthetic outcomes without permanent negative functional impact.

INTRODUCTION/BACKGROUND:Online resources have become a major source of medical information for the general public. To date, there has not been an assessment of patient-oriented online resources for face and upper extremity transplantation candidates and patients. The goal of this study is to perform a comprehensive assessment of these resources. METHODS:Our analysis relied on 2 dimensions: comprehensiveness and readability. Comprehensiveness was evaluated using 14 predetermined variables. Readability was evaluated using 8 different readability scales through the Readability Studio Professional Edition Software (Oleander Software, Ltd, Vandalia, Ohio). Data were also collected from solid organ transplantation (SOT), specifically kidney and liver, programs for comparison. RESULTS:Face and upper extremity transplantation programs were significantly more likely to list exclusion criteria (73.9% vs 41.2%; P = 0.02), the need for life-long immunosuppression (87.0% vs 58.8%; P = 0.02), and benefits of transplantation (91.3% vs 61.8%; P = 0.01) compared with SOT programs. The average readability level of online resources by all face and upper extremity transplantation programs exceeded the sixth grade reading level recommended by the National Institutes of Health and the American Medical Association. The average reading grade level of online resources by these programs was also significantly higher than those of SOT with both exceeding the recommended reading level (13.95 ± 1.55 vs 12.60 ± 1.65; P = 0.003). CONCLUSIONS:Future efforts in face and upper extremity transplantation should be directed toward developing standardized, comprehensive, and intelligible resources with high-quality content and simple language.

Despite promising short- and long-term results to date in vascularized composite allotransplantation (VCA), acute rejection remains the most common major complication in recipients. Currently, diagnosis of acute rejection relies on clinical inspection correlated with histopathological analysis. However, disagreement exists regarding the value of full-thickness skin and mucosal biopsies and histopathology remains semiquantitative, subject to sampling bias, and prone to intra- and inter-observer variabilities. Additionally, biopsies may cause infection, scarring, and/or potentially incite rejection through immune activation after injury. Noninvasive methods to diagnose rejection represent a critical unmet need for the emerging field of VCA. Here, we propose a novel technique utilizing skin stripping of the epidermis and subsequent molecular analysis to detect known markers of acute rejection. Using a small animal VCA model, we sought to validate our epidermal sampling technique as a noninvasive diagnostic test for acute rejection.

Face transplant (FT) candidates present with unique anatomic and functional defects unsuitable for autologous reconstruction, making the accurate design and transplantation of patient-specific allografts particularly challenging. In this case series, we present our computerized surgical planning (CSP) protocol for FT.