BACKGROUND:Complications post-cranial vault reconstruction surgery can be associated with significant morbidity. Our institution previously reported a 12.1% (22 of 182) wound complication rate, requiring readmission to the hospital with a 9.9% (18 of 182) reoperation rate following the use of resorbable PLGA plates. Following this, our clinical practice changes were implemented, including greater consideration given to preoperative sterilization of the surgical field, including attention to the amount of hair shaved preoperatively; double skin preparation in theatre using chlorhexidine and betadine. Intraoperatively, the minimum quantity and size of resorbable plates are used. Plate placement close to the wound closure site is avoided. This study examined whether these changes in clinical practice resulted in lower resorbable plate-associated wound complications. METHODS:A retrospective comparative study of patients before and after the clinical practice changes was conducted. Patient demographics, operative, and postoperative details were compared. Continuous data were described using means and standard deviations (SD) for normally distributed data and compared using independent samples t tests. Non-normal data were described using medians and interquartile ranges and compared using Mann-Whitney U tests. Differences in categorical data were described using counts, percentages, and χ 2 or Fisher's exact test as appropriate. Data were analyzed using SPSS Version 28. RESULTS:A total of 75 patients underwent surgery between 2018 and 2021, with 5.3% (4 of 75) of patients experiencing resorbable plate-associated wound complications. There were no significant differences between the 2 cohorts regarding gender, age, or weight at the time of surgery. The proportion of patients requiring readmission was significantly lower following the introduction of clinical practice changes [4.0% (3/75) versus 12.1% (22/182), P =0.033]. CONCLUSIONS:In conclusion, 4 changes in our clinical practice introduced at our institution in 2018 with the aim of reducing our wound complication rates post-CVR have resulted in a significant reduction in wound complications requiring readmission among patients with craniosynostosis.
Craniosynostosis is a congenital bone developmental condition characterized by the premature ossification of calvarial sutures, leading to restricted skull expansion and potential neurological complications. Although little is known about the signaling that governs this accelerated fusion, our research group has previously identified a stiffness-dependent upregulation of osteogenic genes in cells derived from fused sutures, highlighting the role of mechanotransduction in disease progression. Building on these findings, the present study describes the development of a unique patient-derived three-dimensional (3D) tissue-engineering (TE) model of non-syndromic craniosynostosis (NS-CS) to investigate how extracellular matrix (ECM) composition and biochemical cues regulate ossification timing and patterns. Cells isolated from clinically relevant tissues, surgically obtained from patent and prematurely fused calvarial sutures of pediatric NS-CS patients, were characterized and cultured under both two-dimensional (2D) and 3D suture-mimicking conditions. Comparative analysis revealed differences in cellular responsiveness between cells isolated from fused and patent sutures across the different experimental conditions, with cells from fused sutures consistently exhibiting higher expression of osteogenic markers. Notably, the elevated expression of osteogenic and chondrogenic markers suggested the possible involvement of endochondral-like ossification mechanisms during the pathological process of suture fusion. This patient-derived model was designed to recapitulate biophysical and biochemical features of the extracellular matrix of healthy and pathological sutures, serving as a tool for future research, helping us to understand the underlying mechanisms behind the pathophysiology of craniosynostosis.
Cranioplasty in the pediatric population presents unique challenges due to ongoing skull growth, variable bone quality, and material-specific risks. This study reviews an 18-year experience of pediatric cranioplasty at a national referral center, evaluating complication and revision rates across autologous and synthetic implant types. A retrospective chart review was performed for all patients aged younger than or equal to 16 years who underwent cranioplasty between 2006 and 2023. Data collected included demographics, implant material, storage method for autologous bone, defect characteristics, and postoperative outcomes. A total of 103 cranioplasties were performed in 94 patients, with a mean age of 10.3 years and mean defect size of 77.7 cm2. Autologous bone was used in 52.4% of cases, hydroxyapatite in 29.1%, titanium in 10.7%, and polyetheretherketone (PEEK)/patient‑specific implants (PSI) in 7.8%. Overall complication and revision rates were 11.7% and 9.7%, respectively. Autologous bone showed the highest resorption rate (11.1%), particularly in cryopreserved flaps. Hydroxyapatite demonstrated the lowest complication rate (6.7%) but was associated with pseudomeningocele formation. PEEK/PSI implants had favorable outcomes, with one infection in a patient with extensive prior surgical intervention. Titanium implants had low complication rates but were discontinued due to patient-reported morbidity, including cold sensitivity and soft-tissue discomfort. This large single-center series highlights the importance of tailored material selection in pediatric cranial reconstruction. Although autologous bone remains widely used, synthetic materials offer favorable complication profiles. Long-term, multicentre outcomes are required to determine optimal implant choice across age groups and defect sizes.
Craniosynostosis is a congenital disorder marked by the early fusion of one or more cranial sutures, potentially leading to functional impairments, including airway obstruction. This is particularly common in syndromic craniosynostosis, such as in Apert, Crouzon, Pfeiffer, Saethre-Chotzen, and Muenke syndromes, where midface hypoplasia and retrusion are often present. Airway obstruction, including obstructive sleep apnea, affects up to 68% of individuals with syndromic craniosynostosis. Understanding the severity and variability of airway compromise across these syndromes is critical for guiding interventions, such as Le Fort advancements. This study retrospectively reviewed preoperative computed tomography scans from patients diagnosed with Apert, Crouzon, Pfeiffer, Saethre-Chotzen, Muenke, and craniofrontonasal dysplasia syndromes. Key airway metrics, including nasopharyngeal and oropharyngeal anteroposterior diameter, volume, and minimal cross-sectional area, were measured and compared across syndromes. The analysis also explored age-related changes in airway parameters to assess how obstruction evolves over time. Significant variability in airway obstruction was observed across the syndromes. Pfeiffer syndrome exhibited the most severe obstruction, with a 50% to 70% reduction in airway dimensions. Apert syndrome showed milder obstruction, with a 30% to 40% reduction in younger individuals, improving to 15% to 20% with age. Saethre-Chotzen and Muenke syndromes demonstrated the mildest obstruction, with reductions of 10% to 25%. These findings highlight the diverse severity of airway obstruction across craniosynostosis syndromes. Pfeiffer syndrome shows the most significant early childhood obstruction, while Saethre-Chotzen and Muenke syndromes experience milder airway compromise. Surgical interventions, like Le Fort advancements, are essential in managing severe cases, emphasizing the importance of individualized surgical and airway management strategies.
Eye-tracking Technology (ETT) measures an individual's eye movements to reflect their attentional behavior. Medicine and surgery are highly visual disciplines, with health care professionals (HCPs) processing visual information in the diagnosis of many conditions, including craniosynostosis. ETT has been applied to the clinical setting in many ways, including in the training and assessment of HCPs. The authors' objectives are to design an education session for HCPs to identify abnormal head shapes associated with non-syndromic craniosynostosis, while utilizing ETT as a learning assessment tool. HCPs and student HCPs were recruited. The Tobii Pro Nano eye tracker was used with iMotions software. Photographs of patients with non-syndromic craniosynostosis were shown, and participants were asked to analyze each photograph for features of craniosynostosis while being monitored by ETT. Participants then attended an education session delivered by a consultant craniofacial surgeon. After the session, participants were shown a different set of photographs of patients with non-syndromic craniosynostosis while being monitored by ETT. Their performance before and after the education session was compared by performing Wilcoxon Signed-Rank tests for several area of interest (AOI) metrics. Seven participants took part in our eye-tracking study. Statistical analysis of AOI metrics revealed that participants improved in several metrics after attending the educational session, including mean dwell count and mean revisit count. The authors' results demonstrate that ETT was a useful adjunct in objectively assessing the effectiveness of their education session. The authors' study is the first to demonstrate the effectiveness of ETT as an educational adjunct in craniofacial conditions.
Progressive hemifacial atrophy (PHA) is a rare idiopathic disorder that can have a significant impact on quality of life. Herein, a patient describes his experience of the condition.
We present the case of a pregnant 32-year-old woman who presented with a giant fetal facial tumour at 22 weeks. The mass, initially 4 × 3.5 × 3 cm in size, was largely cystic with a small solid component. It subsequently increased to 9 × 9 × 10 cm. Significant compression effects on the fetal orbit, temple and infratemporal fossa, with potential compression of the optic nerve, were noted on ultrasound and MRI. The cyst required drainage twice in the pregnancy: firstly to reduce the compression effects and secondly to facilitate caesarean delivery. Postnatally, the baby had significant compression and displacement of the craniofacial skeleton from the mass effect. Postnatal histology revealed a diagnosis of a teratoma. This case highlights the complexities and challenges surrounding the diagnosis and management of a giant fetal facial tumour.
A multitude of operations exists for the treatment of patients with sagittal craniosynostosis presenting after 6 months of age, of which total cranial vault remodeling now provides the most reliable aesthetic outcome. As our national craniofacial center evolved and began to develop a comprehensive multi-disciplinary team to manage complex surgical cases, we offered cranial vault distraction as an alternative to more invasive surgery for late presentation nonsyndromic scaphocephaly. The authors conducted a retrospective review of all patients undergoing this procedure. An average distraction distance of 39.4mm was achieved in 15 patients aged 1 to 9 years. As a result, the cephalic index changed an average of 4%. The mean transfusion volume in the perioperative period was 32.47% of estimated blood volume. There were 2 complications requiring further operative intervention and distraction was stopped early in 1 patient. The authors propose that internal calvarial distraction is a viable alternative to total cranial vault remodeling for the management of late presentations of sagittal craniosynostosis.
The European Society for Plastic, Reconstructive and Aesthetic Surgery (ESPRAS) is the overarching organization for Plastic Surgery which pursues the interests of all the affiliated national societies for plastic surgery throughout Europe. ESPRAS is dedicated to providing patients with the best possible treatment by highly trained and skilled board-certified Plastic Surgeons in all the subspecialty areas of Plastic Surgery, including reconstructive, aesthetic, hand and burns surgery.
Background: Systematic reviews have identified the need for a patient-reported outcome measure for facial nerve paralysis (FNP). The aim of this study was to determine the psychometric properties of FACE-Q Craniofacial module scales when used in a combined sample of children and older adults with FNP. Methods: Data were collected between December 2016 and December 2019. We conducted qualitative interviews with children and adults with FNP. FACE-Q data were collected from patients aged 8 years and older with FNP. Rasch measurement theory analysis was used to examine the reliability and validity of the relevant scales in the FNP sample. Results: Twenty-five patients provided 2052 qualitative codes related to appearance, physical, psychological, and social function. Many patient concerns were common across age. The field-test sample included 235 patients aged 8-81 years. Of the 13 scales examined, all 122 items had ordered thresholds and good item fit to the Rasch model. For 12 scales, person separation index values were ≥0.79 and Cronbach's alpha values were ≥0.82. The 13th scale's reliability values were ≥0.71. Conclusion: The FACE-Q Craniofacial module scales described in this study can be used to collect and compare evidence-based outcome data from children and adults with FNP.
Background: Blood loss and subsequent transfusion are key concerns in the surgical management of craniosynostosis, and have been associated with increased morbidity, requirement for intensive care admission and increased length of hospital stay. Patient blood management guidelines advocate treatment of anemia before elective surgical procedures where significant blood loss is anticipated. At present there is little evidence in the literature investigating the clinical value of this practice in pediatric craniofacial surgery. Aims: The authors examined the effect of preoperative oral iron supplementation on blood loss and transfusion rates in a national pediatric craniofacial unit. Methods: A total of 157 patients were included in a retrospective and prospective observational cohort study conducted between July 2011 and November 2016. Eighty-five (85) patients included in the preoperative iron supplementation group were prescribed oral ferrous fumarate before total cranial vault reconstruction, frontal-orbital advancement or extended strip cranial vault remodeling procedures. This cohort was retrospectively compared to seventy-two (72) consecutive patients who did not receive iron supplementation. Results: Calculated blood loss was 51.3 mL/kg in the intervention group, and 56.65 mL/kg in the control group. Transfusion rate and mean volumes for the intervention group were 85.9% and 25 mL/kg. The control group had transfusion rate of 86.1% with mean transfused volume of 24.7 mL/kg. These differences were not statistically significant. Intraoperative tranexamic acid was associated with significantly reduced transfusion volumes overall. Conclusions: This study did not show a statistically significant benefit to preoperative iron supplementation. Secondary outcomes of this study showed a statistically significant difference in estimated versus calculated intraoperative blood loss. Further research in to specific iron supplementation protocols is indicated.
This study examined the consensus between the primary care radiological diagnosis and specialist clinical diagnosis of abnormal skull shapes in children. We performed a retrospective review of children treated at the National Paediatric Craniofacial Centre at Children's Health Ireland, Dublin, Ireland. Group 1 were referred by primary care colleagues concerned about suspected abnormal skull shapes from 1 January 2015 to 30 May 2017. These included cases where they sought specialist confirmation that the skull shape was normal. Group 2 underwent surgery for craniosynostosis from 1 January 2011 to 25 October 2017. The primary care skull X-ray reports were examined for both groups to see whether they matched the specialist diagnosis. Group 1 comprised 300 children, and 59 (20%) had pre-referral skull X-rays. The primary care X-ray reports and specialist diagnoses agreed in 44 (75%) cases, including 19 (43%) who had a normal skull shape. Group 2 comprised 274 children, and 63 (23%) had pre-referral skull X-rays. In this group, there was agreement in 41 (65%) diagnoses; however, the primary care X-ray reports did not diagnose craniosynostosis for the remaining 22 (35%) children. X-rays were of little value in diagnosing abnormal skull shapes, especially craniosynostosis, and primary care clinicians should refer concerns to specialist teams.
Background: Resorbable plates are commonly used in cranial vault reconstruction surgery. There are few published papers examining their safety profile. The authors examined the prevalence of wound complications associated with the use of resorbable plates (Inion CPS Fixation System) in pediatric patients undergoing cranial vault reconstruction. Methods: A retrospective review of patients (n = 182) who underwent cranial vault reconstruction using resorbable plate fixation was undertaken. All procedures were performed by a single Craniofacial Surgeon at the National Pediatric Craniofacial Center from 2008 to 2016. Wound complications were identified from a prospectively maintained database and medical note review. Several key patient characteristics and surgical variables were also recorded and tested for associations with wound complications. Results: A total of 58.8% (107 of 182) of patients were male with a median age at surgery of 16.2 months. Overall, 12.1% (22 of 182) experienced a postoperative wound complication requiring hospital admission. A total of 2.73% (5 of 182) of the patients that returned to theatre had remnants of plates removed. The authors had a mean time from primary operation to secondary reoperation of 103 days. In univariate statistical analysis, females were more likely to develop a wound complication. However, in stratified analyses excluding patients with an underlying genetic syndrome, increasing age, and lower weight but not gender were associated with wound complications. Conclusions: A 12.1% (22 of 182) wound complication rate with the use of the Inion CPS Fixation System was observed. Inion plates appear to have an equivalent safety profile to other fixation devices. Increasing age and lower weight were associated with an increased risk of wound complications in nonsyndromic patients.
We have recently demonstrated that c-Jun N-terminal kinase 3 (JNK3) is a key modulator of the enhanced osteogenic potential of stem cells derived from children when compared to those derived from adults. In this study, we formulated a JNK3-activator nanoparticle (JNK3*) that recapitulates the immense osteogenic potential of juvenile cells in adult stem cells by facilitating JNK3 activation. Moreover, we aimed to functionalize a collagen-based scaffold by incorporating the JNK3* in order to develop an advanced platform capable of accelerating bone healing by recruitment of host stem cells. Our data, in vitro and in vivo, demonstrated that the immense osteogenic potential of juvenile cells could be recapitulated in adult stem cells by facilitating JNK3 activation. Moreover, our results revealed that the JNK3* functionalized 3D scaffold induced the fastest bone healing and greatest blood vessel infiltration when implanted in critical-size rat calvarial defects in vivo. JNK3*scaffold fastest bone healing in vivo was associated with its capacity to recruit host stem cells to the site of injury and promote angiogenic-osteogenic coupling (e.g. Vegfa, Tie1, Runx2, Alp and Igf2 upregulation). In summary, this study has demonstrated the potential of harnessing knowledge of age-altered stem cell mechanobiology in order to develop a materials-based functionalization approach for the repair of large tissue defects.
BACKGROUND:The CLEFT-Q includes 12 independently functioning scales that measure appearance (face, nose, nostrils, teeth, lips, jaws), health-related quality of life (psychological, social, school, speech distress), and speech function, and an eating/drinking checklist. Previous qualitative research revealed that the CLEFT-Q has content validity in noncleft craniofacial conditions. This study aimed to examine the psychometric performance of the CLEFT-Q in an international sample of patients with a broad range of facial conditions. METHODS:Data were collected between October 2016 and December 2019 from 2132 patients aged 8 to 29 years with noncleft facial conditions. Rasch measurement theory (RMT) analysis was used to examine Differential Item Function (DIF) by comparing the original CLEFT-Q sample and the new FACE-Q craniofacial sample. Reliability and validity of the scales in a combined cleft and craniofacial sample (n=4743) were examined. RESULTS:DIF was found for 23 CLEFT-Q items when the datasets for the two samples were compared. When items with DIF were split by sample, correlations between the original and split person locations showed that DIF had negligible impact on scale scoring (correlations ≥0.995). In the combined sample, RMT analysis led to the retention of original content for ten CLEFT-Q scales, modification of the Teeth scale, and the addition of an Eating/Drinking scale. Data obtained fit with the Rasch model for 11 scales (exception School, p=0.04). Person Separation Index and Cronbach alpha values met the criteria. CONCLUSION:The scales described in this study can be used to measure outcomes in children and young adults with cleft and noncleft craniofacial conditions.
Background The European Leadership Forum (ELF) of the European Society of Plastic, Reconstructive and Aesthetic Surgery (ESPRAS) previously identified the need for harmonisation of breast reconstruction standards in Europe, in order to strengthen the role of plastic surgeons. This study aims to survey the status, current trends and potential regional differences in the practice of breast reconstruction in Europe, with emphasis on equity and access. Materials and Methods A largescale web-based questionnaire was sent to consultant plastic and reconstructive surgeons, who are experienced in breast reconstruction and with understanding of the national situation in their country. Suitable participants were identified via the Executive Committee (ExCo) of ESPRAS and national delegates of ESPRAS. The results were evaluated and related to evidence-based literature. Results A total of 33 participants from 29 European countries participated in this study. Overall, the incidence of breast reconstruction was reported to be relatively low across Europe, comparable to other large geographic regions, such as North America. Equity of provision and access to breast reconstruction was distributed evenly within Europe, with geographic regions potentially affecting the type of reconstruction offered. Standard practices with regard to radiotherapy differed between countries and a clear demand for European guidelines on breast reconstruction was reported. Conclusion This study identified distinct lack of consistency in international practice patterns across European countries and a strong demand for consistent European guidance. Large-scale and multi-centre European clinical trials are required to further elucidate the presented areas of interest and to define European standard operating procedures.
A patient presented to our unit with a long history of a discharging skin infection on his left cheek, which came and went. He had been seen by numerous healthcare practitioners including his general practitioner, general dental practitioner and dermatologist, with no resolution. He was eventually diagnosed with an odontogenic cutaneous fistula (OCF), for which he underwent surgical management. The purpose of the study is to describe the diagnosis and surgical management of an OCF, from initial assessment through to postoperative review and discharge. Following surgical management of the OCF and treatment of the source of infection by dental extraction, the patient is no longer experiencing purulent discharge through his left cheek. The extraoral skin site of drainage at his left cheek has resolved completely, with minimal residual scarring. OCF can be managed by a number of different treatment modalities. The treatment of an OCF by surgical excision is presented.
BackgroundThe FACE-Q Craniofacial Module is a patient-reported outcome measure designed for patients aged 8 to 29 years with conditions associated with a facial difference. In part 1, we describe the psychometric findings for the original CLEFT-Q scales tested in patients with cleft and noncleft facial conditions. The aim of this study was to examine psychometric performance of new FACE-Q Craniofacial Module scales.MethodsData were collected between December 2016 and December 2019 from patients aged 8 to 29 years with conditions associated with a visible or functional facial difference. Rasch measurement theory (RMT) analysis was used to examine psychometric properties of each scale. Scores were transformed from 0 (worst) to 100 (best) for tests of construct validity.Results1495 participants were recruited with a broad range of conditions (e.g., birthmarks, facial paralysis, craniosynostosis, craniofacial microsomia, etc.) RMT analysis resulted in the refinement of 7 appearance scales (Birthmark, Cheeks, Chin, Eyes, Forehead, Head Shape, Smile), two function scales (Breathing, Facial), and an Appearance Distress scale. Person separation index and Cronbach alpha values met criteria. Three checklists were also formed (Eye Function, and Eye and Face Adverse Effects). Significantly lower scores on eight of nine scales were reported by participants whose appearance or functional difference was rated as a major rather than minor or no difference. Higher appearance distress correlated with lower appearance scale scores.ConclusionThe FACE-Q Craniofacial Module scales can be used to collect and compare patient reported outcomes data in children and young adults with a facial condition.
Alexander Kanapin合作论文数SwissProt group at EBI3