Skeletal and Dental Outcomes after Facial Allotransplantation: The Cleveland Clinic Experience and Systematic Review of the Literature.

BACKGROUND: Most of the literature surrounding face transplantation focuses on technique, immunology, and psychology. Dental and skeletal outcomes remain persistently underreported. This study critically examined the worldwide face transplant experience to evaluate such outcomes. METHODS: A systematic review of all composite allografts containing midface and/or mandible was performed. Dental and skeletal complications were recorded. Formal imaging and photographs available in the literature were analyzed using skeletal measurements, soft-tissue cephalometrics, and the Angle classification. Outcomes of our face transplant patients, including condylar assessment and airway volume measurements, is also presented. RESULTS: Twenty-five patients received allografts containing midface (n = 7) or mandible (n = 2), whereas 16 contained a double-jaw. All midface-only transplants developed skeletal deformity; 57 percent developed a palatal fistula. Both partial and full arch transplantation patients developed skeletal deformity. Among double-jaw transplants, 69 percent developed palatal fistula or floor-of-mouth dehiscence, 66 percent developed malocclusion, 50 percent developed trismus, and 31 percent required corrective orthognathic surgery. In 40 percent of patients, malocclusion recurred after corrective orthognathic surgery. Forty percent of all patients developed dental cavities or periodontal disease. All of our patients received midface and/or mandible. One patient required corrective orthognathic surgery. Midfacial segments showed clockwise rotation. Airway volumes decreased over time. CONCLUSIONS: Skeletal and dental complications remain extremely common after facial allotransplantation involving either single- or double-jaw composites. Corrective orthognathic surgery and dental extraction is often necessitated. These data will aid face transplant teams during surgical planning and preoperative counseling. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, V.

Leukoreduction in ex vivo perfusion circuits: comparison of leukocyte depletion efficiency with leukocyte filters.

BACKGROUND: Leukodepletion of whole blood-based perfusates remains a challenge in experimental models of ex vivo perfusion. This study investigated the leukoreduction efficacy of the commonly used LeukoGuard LG Arterial and BC2 Cardioplegia filters. METHODS: Eleven liters of washed porcine blood was used to evaluate the filtration efficiency of LG (n = 6) and BC2 (n = 5) filters. Filter efficacy was tested by passing 1 L of washed blood through each filter. Complete blood count was performed to detect a reduction of white blood cells, red blood cells, and hemoglobin concentration. RESULTS: The BC2 Cardioplegia filter showed a significant reduction in white blood cell count (13.16 ± 4.2 × 103 cells/µL pre-filtration, 0.62 ± 0.61 cells/µL post-filtration, p = 0.005), red blood cell count (9.18 ± 0.16 × 106 cells/µL pre-filtration, 9.02 ± 0.16 × 106 cells/µL post-filtration, p = 0.012) and hemoglobin concentration (15.89 ± 0.66 g/dL pre-filtration, 15.67 ± 0.83 g/dL post-filtration, p = 0.017). Platelet reduction in the LG filter group was statistically significant (13.23 ± 13.98 × 103 cells/µL pre-filtration, 7.15 ± 3.31 × 103 cells/µL post-filtration, p = 0.029), but no difference was seen in the BC2 group. There was no significant difference in white blood cell count in the LG filter group (10.12 ± 3.0 × 103 cells/µL pre-filtration, 10.32 ± 2.44 × 103 cells/µL post-filtration, p = 0.861). CONCLUSION: Our results suggest that the LG filter should not be used in ex vivo perfusion circuits for the purpose of leukodepletion. The BC2 filter can be used in EVP circuits with flow rates of less than 350 mL/min. Alternatively, perfusate may be leukodepleted before perfusion.

Full Facial Allotransplantation Including the Temporomandibular Joints: A Radiologic and Anatomical Cadaveric Study.

BACKGROUND: Facial allotransplantation including the temporomandibular joints may improve the functional outcomes in face transplant candidates who have lost or damaged this joint. METHODS: Linear and angular measurements were taken in 100 dry skulls and mandibles and in 100 three-dimensionally-reconstructed facial computed tomographic scans to determine the variability of the temporomandibular joint, glenoid fossa, and mandible. A vascular study was performed in six fresh cadaveric heads, followed by harvest of the face allograft in three heads. Next, four heads were used for mock transplantation (two donors and two recipients). The full facial allograft included four different segments: a Le Fort III, a mandibular tooth-bearing, and two condyle and temporomandibular joint-bearing segments. Statistical analysis was performed using SAS software. RESULTS: In only one-third of the skulls, the condylar shape was symmetric between right and left sides. There was a wide variability in the condylar coronal (range, 14.3 to 23.62 mm) and sagittal dimensions (range, 5.64 to 10.96 mm), medial intercondylar distance (range, 66.55 to 89.91 mm), and intercondylar angles (range, 85.27 to 166.94 degrees). This high variability persisted after stratification by sex, ethnicity, and age. The temporomandibular joint was harvested based on the branches of the superficial temporal and maxillary arteries. The design of the allograft allowed fixation of the two condyle and temporomandibular joint-bearing segments to the recipient skull base, preserving the articular disk-condyle-fossa relationship, and differences were adjusted at the bilateral sagittal split osteotomy sites. CONCLUSION: Procurement and transplantation of a temporomandibular joint-containing total face allograft is technically feasible in a cadaveric model.

Extended ex vivo normothermic perfusion for preservation of vascularized composite allografts.

Ischemia and reperfusion injury remains a significant limiting factor for the successful revascularization of amputated extremities. Ex vivo normothermic perfusion is a novel approach to prolong the viability of the amputated limbs by maintaining physiologic cellular metabolism. This study aimed to evaluate the outcomes of extended ex vivo normothermic limb perfusion (EVNLP) in preserving the viability of amputated limbs for over 24 hours. A total of 10 porcine forelimbs underwent EVNLP. Limbs were perfused using an oxygenated colloid solution at 38°C containing washed RBCs. Five forelimbs (Group A) were perfused for 12 hours and the following 5 (Group B) until the vascular resistance increased. Contralateral forelimbs in each group were preserved at 4°C as a cold storage control group. Limb viability was compared between the 2 groups through assessment of muscle contractility, compartment pressure, tissue oxygen saturation, indocyanine green (ICG) angiography and thermography. EVNLP was performed for 12 hours in group A and up to 44 hours (24-44 hours) in group B. The final weight increase (-1.28 ± 8.59% vs. 7.28 ± 15.05%, P = .548) and compartment pressure (16.50 ± 8.60 vs. 24.00 ± 9.10) (P = .151) were not significantly different between the two groups. Final myoglobin and CK mean values in group A and B were: 875.0 ± 325.8 ng/mL (A) versus 1133.8 ± 537.7 ng/mL (B) (P = .056) and 53 344.0 ± 16 603.0 U/L versus 64 333.3 ± 32 481.8 U/L (P = .286). Tissue oxygen saturation was stable until the end in both groups. Infra-red thermography and ICG-angiography detected variations of peripheral limb perfusion. Our results suggest that extended normothermic preservation of amputated limbs is feasible and that the outcomes of prolonged EVNLP (>24 hours) are not significantly different from short EVNLP (12 hours).

Relevant Surgical Anatomy of Pterygomaxillary Dysjunction in Le Fort III Osteotomy.

BACKGROUND: Le Fort III osteotomy represents the foundation of surgical correction for midface hypoplasia. One serious complication of Le Fort III osteotomy is severing the internal maxillary artery or its branches during osteotome advancement for pterygomaxillary dysjunction. This study sought to characterize the relevant surgical anatomy of the infratemporal fossa and of the internal maxillary artery as it enters the pterygomaxillary fissure. METHODS: Bilateral midface dissections were performed on 15 fresh, normocephalic adult cadavers (30 hemifaces). Four superficial anatomical measurements were performed on the surface of the face, followed by 10 deep measurements of the internal maxillary artery and its branches relative to the infratemporal fossa and its surrounding bony landmarks. RESULTS: The distance from the anterosuperior aspect of the zygomatic arch to the sphenopalatine artery entering the pterygomaxillary fissure was 38.9 ± 3.2 mm. The distance from the alveolar process of the maxillary bone to the sphenopalatine artery entry into the pterygomaxillary fissure was 30.3 ± 6.4 mm. The zygomaticofrontal suture was 43.4 ± 8.5 mm from the sphenopalatine artery entry into the pterygomaxillary fissure, 58.8 ± 8.0 mm from the pterygomaxillary junction, and 74.9 ± 6.5 mm from the maxillary alveolar process. The distance from the sphenopalatine artery to the posterior superior alveolar artery was 14.4 ± 4.1 mm. Elevation of the internal maxillary artery from the lateral pterygoid plate was 5.8 ± 2.5 mm. CONCLUSION: This study characterizes the surgical anatomy of the infratemporal fossa in the context of Le Fort III osteotomies and their associated pterygomaxillary dysjunctions.

Classification and Management of Metopic Craniosynostosis.

BACKGROUND: In the craniofacial surgery literature, there is a wide disparity of opinions regarding the management of nonsyndromic metopic synostosis. With the lack of level I evidence to support a particular regimen, we aimed to elucidate the current state of practice among craniofacial surgeons with the hope of establishing a standard of care. METHODS: A survey was sent to 102 craniofacial surgeons. The survey featured 2 parts: clinical scenarios and questions regarding the following: primary indication for surgery, preference of timing, and choice of operative intervention for patients presenting with nonsyndromic isolated metopic synostosis. Surgeons were also queried regarding preoperative, intraoperative, and postoperative protocols. RESULTS: The total response rate was 72% (73/102) for the clinical scenarios and 63% (64/102) for the complete survey. There was a large discrepancy when classifying and managing mild metopic synostosis, with between 16% and 35% of surgeons electing to operate on a mild case. All surgeons agreed to operate on moderate and severe cases. For 95% of respondents, skull deformity was the primary indication for treatment of craniosynostosis. Open surgical management was most commonly performed at 6 months (29%) of age. Open frontal orbital advancement was the most commonly performed procedure in mild (27%), moderate (77%), and severe (89%) cases. Endoscopic approaches were more likely to be used in milder cases by 19% of surgeons. CONCLUSION: Our survey demonstrates that there is a wide disparity of opinion among craniofacial surgeons regarding the diagnosis and management of mild nonsyndromic metopic synostosis. LEVEL OF EVIDENCE: Diagnostic, level 5.

Novel techniques for the surgical management of abdominopelvic constriction rings in amniotic band syndrome.

Constriction rings are associated with amniotic band syndrome and most often present in the extremities. Constriction bands of the trunk are rare, and a standard of surgical care remains elusive. Traditional methods of constriction ring excision rely on soft-tissue rearrangement with multiple Z-plasties, but renewed interest in linear closure and limited Z-plasty has emerged. The authors review contemporary literature and report two cases of abdominopelvic constriction ring reconstruction with long-term follow-up. Novel techniques including anterior sheath Y-V plasty, pteruges release of the Scarpa fascia, and limited Z-plasty closure may minimize the need for serrated scar patterns.

The expansile properties of kryptonite relating to cranioplasty.

BACKGROUND AND PURPOSE: Since 2009, a synthetic material known as kryptonite has become increasingly utilized during cranioplasty to repair bony defects. It provides bone-like strength and adhesive properties that make it a suitable replacement for bone. However, applications have been observed in the immediate postoperative period that demonstrates an increase in its original volume, giving rise to irregularities in the cranial surface. METHODS: Ten kryptonite samples were reconstituted and allowed to polymerize according to the manufacturer's directions. The kryptonite samples were molded into a cylindrical shape, and they were immersed in 10 graduated cylinders filled with normal saline. Measurements of the rise in saline relative to baseline were taken at 0, 10, 20, 30, 40, 50, and 60 minutes, and then hourly through 5 hours, with the final measurement recorded at 24 hours. RESULTS: The mean expansion of kryptonite was approximately 49% with an SD of 22%. The bulk of the expansion occurred within the first 2 hours, after which the rate tended to plateau for the remaining 22 hours. CONCLUSIONS: Kryptonite has been touted as an excellent alternative for repairing contour abnormalities manifested in cranioplasty. Given the unpredictability of its expansile properties, the surgeon must take this variability into careful consideration when planning the desired surgical outcome. The results of the current study were communicated with the manufacture. Immediately thereafter, the manufacturer withdrew the product from the US market and is no longer Food and Drug Administration approved for cranioplasty.

Priorities for the education of members of the American Society of Maxillofacial Surgery.

We present the complete results of our 2013 survey of the members of the American Society of Maxillofacial Surgery (ASMS). We surveyed all 799 members of the Society from around the world regarding educational themes and modalities they wish to add to future ASMS educational programs. We also asked our members about surgical modalities and care paths in which they have the most expertise. The objective of the survey was to provide data that can be used to improve the education and training of the ASMS members. The results suggest that some widely taught topics perhaps should be taught less, whereas coverage of other topics should be increased.

Squamosal suture craniosynostosis in Muenke syndrome.

Muenke syndrome caused by point mutation (C749G) in the FGFR3 gene affects 1 in 30,000 newborns and accounts for 25% to 30% of genetic causes of craniosynostosis. Anomalies in patients with Muenke syndrome include craniosynostosis, hypertelorism, sensorineural hearing loss, and developmental delay, among others. Most craniosynostoses in patients with Muenke syndrome involve bicoronal suture fusion. This article reports, for the first time, the existence of squamosal craniosynostosis in patients with Muenke syndrome.

Management of sagittal synostosis: a solution to equipoise.

BACKGROUND: In the craniofacial surgery literature, there is a wide disparity of opinions regarding the appropriate treatment of nonsyndromic sagittal synostosis. With the lack of level 1 evidence to support a particular regimen, our study aims to elucidate the current state of practice among craniofacial surgeons with the hope of establishing a standard of care. METHODS: An internet-based survey was sent to 102 craniofacial surgeons in 14 countries on 4 continents. Data were collected regarding the following parameters: primary indication for surgery, preference of timing, and choice of operative intervention for patients presenting with nonsyndromic isolated sagittal synostosis with normative intracranial pressure values. Surgeons were also queried regarding preoperative, intraoperative, and postoperative protocols. RESULTS: After 2 mailings, the response rate was 58% (59/102). For 63% of respondents, skull deformity was the primary indication for treatment of craniosynostosis. Open surgical management of sagittal craniosynostosis was most commonly performed at 6 months (35%) of age. Total cranial vault remodeling was the most commonly performed procedure (37%). Thirty-five percent of craniofacial surgeons chose an endoscopic surgical approach for patients presenting at younger than 4 months. Only 10% of craniofacial surgeons selected spring-assisted strip craniectomy. Seventy-one percent of polled surgeons performed computed tomographic scans of the skull in all cases, irrespective of presentation. CONCLUSION: Our survey demonstrates that there exists a wide disparity of opinion regarding diagnosis and treatment of nonsyndromic sagittal synostosis. When current practice is compared to findings in the literature, significant discrepancies exist.

Opinion leaders and evidence-based medicine in craniofacial surgery.

BACKGROUND: In health care, it is widely known that evidence-based medicine (EBM) has a significant impact on clinical practice, and opinion leaders can enhance the clinician's application of EBM in various disciplines. In this article, we examine the existence and impact of opinion leaders in craniofacial surgery as well as barriers to evidence-based practice. METHODS: We compiled the answers of an Internet questionnaire, which was sent to 102 craniofacial surgeons. RESULTS: Our results demonstrate that opinion leaders most definitely can be identified in craniofacial surgery. They are tightly connected to their field's social network and promote EBM. In this survey, 44% of craniofacial surgeons reported that their greatest obstacle to clinical decision making in the management of nonsyndromic synostosis was lack of surgical consensus. In addition, craniofacial surgeons stated that EBM and opinion leaders are the most influential factors that caused them to change their management of craniosynostosis. CONCLUSIONS: We expect that the use of opinion leaders can further enhance the uptake of EBM in craniofacial surgery.

Long-Term Follow-up of a Tessier Number 5 Facial Cleft.

The Tessier number 5 cleft is a rare craniofacial anomaly. Although there are case reports outlining various methods of primary surgical repair and outcomes, few cases reveal long-term follow-up or discuss secondary reconstruction. This article presents the case of a man born with a unilateral Tessier number 5 cleft who presented to our institution at the age of 41 after more than 25 previous surgeries. Over the course of 11 years, he had several surgeries including an orbital repositioning through a combined intra- and extracranial approach, an orbital enucleation, and an anterolateral thigh flap. The patient was fitted with an orbital prosthesis and a satisfactory aesthetic result was achieved.

Anatomic survey of arachnoid foveolae and the clinical correlation to cranial bone grafting.

INTRODUCTION: When performing in situ harvesting of cranial bone grafts, there is a risk of entering the pericranial-intracranial venous system, either directly or indirectly through the arachnoid foveolae. The aims of this study were to investigate the size and location of arachnoid foveolae and to provide an anatomic road map to prevent penetrating these structures. METHODS: Three hundred dry skulls were selected from the Hamann-Todd osteological collection (Cleveland, OH); skulls were collected between 1912 and 1938. Our study skulls were limited to whites or African American adults. Exclusion criteria included children (<18 y), ethnic groups other than African Americans and whites, skulls demonstrating fracture or craniofacial abnormalities, or any skull whose age, ethnicity, and sex could not be confirmed. From the 300 skulls in the collection, 200 met the criteria and were included in our review. The mean age of these 200 individuals was 43.86 years, with a male-to-female proportion of 100:100, and a white-to-African American proportion of 144:56. A 500-W candescent light was used to transilluminate the arachnoid foveola, and digital photographs with scale were obtained. The location and diameters of foveolae for arachnoid granulations relative to the coronal and sagittal suture were measured. CONCLUSIONS: Approximately 90% of major arachnoid foveolae are located within 2.5 cm of the coronal and 1.5 cm of the sagittal suture for the left and right parietal bones. Major arachnoid foveolae are located at closer distances to the superior sagittal suture and the coronal suture in the right and left parietal bone than minor foveolae. The results of this study imply that potential complications can be minimized by avoiding these areas and by harvesting in situ bone grafts from the absolute and relative safe zones described in this study.

Calcium phosphate cements in skull reconstruction: a meta-analysis.

BACKGROUND: This work addresses the controversy regarding the indications and results of calcium phosphate cements in skull reconstruction through a meta-analysis of the published literature. METHODS: A PubMed search for articles reporting the use of calcium phosphate cements for skull reconstruction was performed. Data collected included age, volume of cement, defect size, material used, length of follow-up, placement in communication with paranasal sinuses or in irradiated fields, and complications. RESULTS: Nineteen articles met the authors' inclusion criteria. The mean rates of complications were as follows: total complications, 13 percent (range, 0 to 62 percent); major complications, 9 percent (range, 0 to 62 percent); minor complications, 2 percent (range, 0 to 5 percent); infection, 5 percent (range, 0 to 22 percent); reoperation, 14 percent (range, 0 to 62 percent); and secondary surgery for contour correction, 1 percent (range, 0 to 12 percent). There was significant heterogeneity in the estimated rate of total and major complications, infection, and reoperation (p < 0.001), but minor complications and secondary contour correction had less heterogeneity (p = 0.58 and p = 0.78, respectively). Radiotherapy and communication with the paranasal sinuses significantly increased the complication rate (p < 0.05). Duration between surgery and complications averaged 17.5 months (range, 1 to 89 months). CONCLUSIONS: When mean complication rate and complication range of calcium phosphate cements in our meta-analysis were compared with previous large cranioplasty studies using methylmethacrylate or autogenous bone, calcium phosphate fared no better, and sometimes fared worse, than these other modalities. Calcium phosphate, therefore, should only be used selectively, and prospective long-term studies are needed to further refine its role in skull reconstruction.

Lessons learned reconstructing complex scalp defects using free flaps and a cranioplasty in one stage.

BACKGROUND: The purpose of this study was to review our experience in single-stage reconstruction of skull and scalp defects, aiming to highlight pitfalls in our management. METHODS: We performed a retrospective chart review of all patients who had a single-stage cranioplasty and free-tissue transfer at our institution over the last 10 years. Thirteen patients (9 men and 4 women) with an average age of 66.5 years (range, 34-83 years) were identified. Etiology of the defects included malignancy (n = 7), osteoradionecrosis (n = 3), and infection (n = 3). The size of the bony defect averaged 103.1 cm(2) (range, 12-300 cm(2)). Cranioplasty procedures included reconstruction by methylmethacrylate and titanium mesh (n = 10), methylmethacrylate only (n = 1), or mesh only (n = 2). Free flaps used were anterolateral thigh (n = 10), latissimus dorsi (n = 2), or a rectus flap (n = 1). RESULTS: Five patients (38%) developed at least 1 complication. These included 2 anastomotic problems that were successfully salvaged, 5 cases of wound dehiscence, and 1 mortality due to a respiratory event. Four patients developed a recurrence, and 2 patients required flap contouring at a second stage. Two patients had further reconstruction using 1 (1 patient) or 2 (1 patient) additional free flaps. CONCLUSIONS: Given the complexity of these procedures, the high recurrence rate, and the likelihood of complications, methylmethacrylate is contraindicated in 1-stage cranioplasty and soft-tissue reconstruction in high-risk patients. For unfavorable local conditions (eg previous infection, radiotherapy), the surgeon can either postpone the cranioplasty until the soft-tissue reconstruction has healed, or use a nonanatomical titanium mesh alone. The soft-tissue flap should be harvested of larger dimensions than anticipated.

Use of calcium-based bone cements in the repair of large, full-thickness cranial defects: a caution.

BACKGROUND: Calcium-based bone cements have increased in popularity for the correction of craniofacial contour defects. The authors' experience with them in more than 120 patients has resulted in the establishment of strict criteria for their use. Although the authors' overall complication rate with these cements has been low, certain patient groups have an unacceptably high complication rate. The authors describe their experience with the repair of large, full-thickness cranial defects using calcium-based bone cements. METHODS: The study group comprised 16 patients who underwent correction of large, full-thickness (>25 cm2) skull defects. The surgical technique included reconstruction of the floor of the defect with rigid fixation to the surrounding native bone, interposition of the cement to ideal contour, and closure of the defect. RESULTS: The mean patient age was 35 years (range, 1 to 69 years). The mean defect area was 66.4 cm2 (range, 30 to 150 cm2). Cases were equally divided between BoneSource and Norian CRS. The mean amount of bone cement used was 80 g. Follow-up varied between 1 and 6 years (mean, 3 years). Major complications occurred in eight of 16 patients, with one occurring as late as 6 years postoperatively. Complications occurred throughout the course of review, indicating that they were not caused by a learning curve. CONCLUSION: Because of the unacceptably high complication rate with the use of calcium-based bone cements in large skull defects, the authors believe that their use is contraindicated and have returned to using autogenous split skull cranial bone reconstruction for these patients.

A pneumatic orthotic cranial molding helmet for correcting positional plagiocephaly.

The objective of this study was o determine the efficacy of a newly developed pneumatic orthotic cranial molding helmet for correcting positional plagiocephaly. The design was retrospective and the setting was a tertiary care center. Subjects were all patients in whom positional plagiocephaly has been diagnosed and who have been fitted by the Orthotics and Prosthetics Department for the helmet. Diagonal cranial lengths and widths were measured at each visit. Analysis included the calculation of the ratio change in oblique diameters compared with time, patient's age, and head circumference. Seventy-five patients met inclusion criteria (50 boys, 25 girls). Patients with pneumatic orthotic cranial molding helmet therapy had significantly improved outcomes as compared with pretreatment measurements (P < or = 0.0001). The helmet did not limit cranial growth as evidenced by significant normalization of the oblique measurement ratio when compared with increasing cranial circumference and age (P = 0.0003, P < or = 0.0001, respectively). The pneumatic orthotic cranial molding helmet successfully corrects positional plagiocephaly and does not hinder cranial growth.