Jeffrey A. Fearon

Single sutural craniosynostoses: surgical outcomes and long-term growth.

Background: This study was designed to examine long-term skull growth following single sutural synostosis corrections and to evaluate surgical outcomes. Methods: A retrospective outcome assessment of all children treated with a single-stage, open-remodeling procedure for sagittal, metopic, unilateral coronal, and unilateral lambdoid synostosis was performed. Long-term growth was assessed from serial anthropometric measurements taken up to 11 years postoperatively (mean, 4 years). Results: Of 296 consecutive patients, complete records were available for 248 operative procedures. The mean surgical age was 12.3 months (range, 2.5 months to 8 years), with 36 patients older than 12 months. Transfusion rates decreased from 81 to 19 percent following the institution of blood conservation strategies. The average hospitalization was 2.5 days. There was one nonsurgically treated infection (0.4 percent), and no major complications or deaths. Five patients underwent secondary remodeling procedures (2 percent). Serial anthropometric measurements, obtained in 75 patients, showed normalization of cranial indices 6 weeks postoperatively, but subsequent measurements revealed statistically significant diminished growth. Earlier surgical treatment of metopic synostosis (4 months) was associated with significantly more growth inhibition than seen in those treated at an older age (12 months), with the other synostoses showing similar tendencies. Conclusions: Treatment of single sutural synostosis was extremely safe with very low reoperative rates, but subsequent calvarial growth was abnormal, with a tendency toward recapitulation of the primary deformity. Growth was less diminished in procedures performed in older infants. Surgeons treating single sutural craniosynostosis should consider expanding treatment goals beyond normalization to an overcorrection of the abnormal skull shape.

Halo distraction of the Le Fort III in syndromic craniosynostosis: a long-term assessment.

Background: Little is known about long-term outcomes after Le Fort III halo distraction, such as indications for distraction, amount of relapse, and long-term maxillary growth. Methods: During a 5-year period, 51 Le Fort III osteotomies were performed; 41 of these were by halo distraction for the treatment of craniofacial dysostosis (Apert syndrome, 25; Crouzon syndrome, nine; Pfeiffer syndrome, four; other, three). Patient records were reviewed and 104 cephalograms were analyzed. Results: The average treatment age was 8 years, and the average hospitalization was 5.3 days (3.6 days for the second half of this series). The average distraction was 26 mm (range, 14 to 44 mm), the sella-nasion-subspinale angle increased from 70 degrees to 87 degrees, the upper incisal edge advanced 18 mm, and the vertical distance between nasion and point A increased by 7 mm. There was no maxillary relapse at 1 year and no measurable forward maxillary growth for up to 5 years, although the maxilla did grow vertically (p ≤ 0.001). Three patients underwent tracheostomy removal; sleep studies showed the average respiratory disturbance index improved from 24 to 11 (p = 0.004) and the minimum oxygen saturation increased from 81 to 89 (p = 0.006). Three early patients had incomplete advancements. Conclusions: The maxilla remained stable after Le Fort III halo distraction without any relapse, and no anterior growth was observed up to 5 years postoperatively. This procedure effectively eliminated sleep apnea in most children, and had a low overall complication rate that declined with experience. Most distracted patients will likely avoid the traditional second Le Fort III.

Observations on the Use of Rigid Fixation for Craniofacial Deformities in Infants and Young Children

With the introduction of smaller plating systems, many surgeons have adopted their use for craniofacial procedures in infants and small children. We have encountered some previously undescribed problems associated with the use of plates and screws in infants and children. These problems can be classified into three general areas: 1) Plate displacement. This occurs after placement of the metal plates on the outside of the infant calvaria. With subsequent growth, these plates and screws may be found along the inner cortex of the skull with screw points embedded through dura. 2) Plate and screw isolation. In certain locations in young children, plates placed on the outer cortex of bone may later be found to be more prominent, isolated on a peninsula of bone. 3) Plate placement. We have also encountered plates that have been placed along the inner cortex of the skull during a previous operative procedure. This makes subsequent surgery extremely difficult and may increase the risk for secondary surgery. We propose that a previously described model for growth, involving areas of bony resorption and deposition, may be responsible for the observed plate displacement.

The Le Fort III osteotomy: to distract or not to distract?

Treatment of the craniofacial dysostoses (e.g., Crouzon, Apert, Pfeiffer, Saethre‐Chotzen syndromes) is critically dependent on the successful advancement of the midface with a Le Fort III procedure. The purpose of this retrospective clinical outcome study was to evaluate a new technique for distracting the Le Fort III procedure and to compare its results in growing children with those of the standard Le Fort III osteotomy. The records of 22 children were reviewed; 10 patients (mean age, 6.5 years) underwent a standard Le Fort III procedure, and 12 patients (mean age, 7.5 years) underwent a Le Fort III distraction procedure. The distraction group included two separate techniques, bilateral buried distraction (n = 2) and halo distraction (n = 10). Preoperative and 2‐ to 3‐month postoperative cephalograms were analyzed. The average horizontal advancement achieved in the standard Le Fort III group was 6 mm, compared with 19 mm of advancement in the distraction group (p ≤ 0.005). Complications were evenly distributed between the two groups (one infection and one tracheostomy in each group), and the lengths of hospitalization were similar. No documented improvement in sleep apnea was identified in the standard Le Fort III group. However, in the distraction group two patients experienced normalization of sleep studies postoperatively as measured by respiratory disturbance index, and two patients underwent successful decannulation of tracheotomies. For aesthetic reasons, halo distraction was preferred over bilateral buried distraction. With halo distraction the vector of traction is focused in the facial midline, which helps to reposition the concave midface and to provide a more convex facial profile. In growing children, the ideal vector for distraction is determined by the malar position and not by dental occlusion. The amount of overcorrection can be calculated from tables of normal anthropologic data. On this preliminary review, it was concluded that the use of halo distraction, in combination with a modified Le Fort III osteotomy, provided a significantly further forward‐positioned midface and seemed to offer a better correction of sleep apnea than did the standard Le Fort III osteotomy. (Plast. Reconstr. Surg. 107: 1091, 2001.)

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Sagittal craniosynostosis: surgical outcomes and long-term growth.

Background: Numerous publications have examined technique-specific results in treating sagittal synostosis, but there are scant data examining long-term outcomes. This retrospective study was designed to evaluate a large series of surgically repaired children with sagittal craniosynostosis and to examine long-term cranial growth following correction. Methods: From 1990 to 2003, 132 children with sagittal synostosis were evaluated and 89 surgically treated (single-stage posterior remodeling) patients were retrospectively reviewed (43 were excluded for multisutural involvement, incomplete records, or nonsurgical treatment). Long-term growth was assessed from anthropologic measurements taken from 3 to 11 years postoperatively (mean, 4.7 years). Results: The surgical age ranged from 2 months to 5 years (mean, 8 months), and the hospital stay was less than 3 days. There were no deaths, major complications, infections, or repeated remodeling procedures performed. Postoperatively, the cephalic index was overcorrected. Subsequent growth of the cranial breadth and length was less than predicted. Moreover, growth was more deficient in cranial breadth than length, resulting in a regression of the cephalic index over time (p < 0.01). Frontal breadth improved immediately postoperatively but did not subsequently narrow over time. Head circumference also showed a statistically significant diminished capacity for growth (p < 0.001). Conclusions: This study demonstrated abnormal skull growth in children following the surgical correction of scaphocephaly. On the basis of these findings, surgeons treating sagittal synostosis may wish to expand their goals of treatment beyond normalization to an overcorrection of the abnormal skull shape.

Complications with facial advancement: a comparison between the Le Fort III and monobloc advancements.

Certain procedures for facial advancement may carry greater risk than others. While many believe that separating the cranial base by monobloc advancement leads to a higher complication rate, no comparative series between the Le Fort III and monobloc advancements has ever been reported. We reviewed our series of these different techniques. Over a 15-year period, 29 patients underwent 30 surgical procedures, with either a midfacial or frontofacial advancement. The average age of patients at the time of surgery was 12 years, with a range from 3 to 26 years. There were 20 Le Fort III and 10 monobloc advancements. Follow-up averaged 4 years, with a range from 10 weeks to 13 years. There were no deaths in this series. The infectious complications differed significantly between the two groups, with all major infections occurring in the monobloc group. The noninfectious complications (2 major and 20 minor) were proportionately distributed between the Le Fort III and monobloc groups. Aesthetic results of the midface, judged by the percentage of revisions necessary, were found to be the same between the two procedures. Aesthetic results were noted to correlate strongly with age at the time of surgery, with the older patients beingjudged as having a better aesthetic result and most of the younger patients requiring a repeat of the facial advancement. We conclude that while we were unable to determine any definitive aesthetic advantage of one procedure over the other in our series, there was a significantly higher infection rate with the monobloc advancement. On the basis of these results, we recommend a staging of the forehead and midfacial advancements.

The use of recombinant erythropoietin in the reduction of blood transfusion rates in craniosynostosis repair in infants and children.

&NA; The vast majority of infants and children undergoing craniosynostosis correction receive a blood transfusion. The risks of blood transfusion include, but are not limited to, acute hemolytic reactions (≈1 of 250,000), human immunodeficiency virus (≈1 of 200,000), hepatitis B and C (≈1 of 30,000 each), and transfusion‐related lung injuries (≈1 of 5000). This prospective, single‐blinded, randomized study was undertaken to examine the safety and efficacy of preoperative single weekly dosing with erythropoietin (epoetin alfa) in reducing the rate of transfusion in infants and small children undergoing craniosynostosis repair. A total of 29 patients (<8 years) undergoing craniosynostosis repair were randomized into two groups: one received preoperative erythropoietin (600 U/kg) weekly for 3 weeks, and the other served as a control. All caregivers responsible for blood transfusions were blinded, and strict criteria for transfusion were established. A pediatric hematologist monitored both groups, and all patients received supplemental iron (4 mg/kg). Fourteen patients were randomized to receive erythropoietin, and eight of these 14 patients (57 percent) required transfusion (mean age, 17 months; mean weight, 10.1 kg). Of the six patients not requiring transfusion, three were younger than 12 months old (mean, 6 months). Fourteen of 15 patients (93 percent) in the control group (mean age, 13 months; mean weight, 9.3 kg) required a blood transfusion during the study. The only control patient not requiring transfusion was the eldest (5 years old). The difference between the two groups was statistically significant (Fishers exact test = 0.03). The control group showed no change in hemoglobin levels from baseline to preoperative levels, but the erythropoietin group increased their average hemoglobin levels from 12.1 to 13.1 g/dl. There were no adverse effects noted among children receiving erythropoietin, nor were there any surgical complications. The authors conclude that the preoperative administration of erythropoietin significantly raised hemoglobin levels and reduced the need for a blood transfusion with craniosynostosis correction. More suggestions are made that may further reduce the need for blood transfusions, and a cost‐benefit analysis is discussed. (Plast. Reconstr. Surg. 109: 2190, 2002.)

The diagnosis and treatment of single-sutural synostoses : Are computed tomographic scans necessary?

Background: Computed tomographic scan evaluation is the current standard of care for diagnosing craniosynostosis. Recent publications, and the National Cancer Institute, have raised concerns about ionizing radiation associated with computed tomographic scans in children (e.g., developmental delays, tumor induction). The authors sought to ascertain the diagnostic accuracy of the physical examination in evaluating single-sutural craniosynostosis and assess the need for computed tomographic scans in surgical correction. Methods: This prospective, multicenter, outcome assessment included children clinically diagnosed with a single-sutural synostosis by craniofacial surgeons (with 1 to 18 years’ experience) at four centers over a 1-year period. Clinical diagnoses were compared with computed tomographic scan evaluations. All surgeons scored the utility of computed tomographic scans during surgical repair. Results: Sixty-seven patients were clinically diagnosed with single-sutural craniosynostosis (mean age, 7 months; range, 1 week to 48 months). Sixty-six of 67 patients were diagnosed with craniosynostosis by computed tomographic scan (sagittal, 40 percent; metopic, 31 percent; right unilateral coronal, 16 percent; left unilateral coronal, 6 percent; and right lambdoid, 6 percent), for a diagnostic accuracy exceeding 98 percent. One patient with suspected lambdoid synostosis was radiologically diagnosed with positional plagiocephaly. Three of four craniofacial surgeons scored computed tomographic scans as “unnecessary” for surgical correction; one scored scans as “sometimes helpful.” Conclusions: Craniofacial surgeons with various experience levels were able to accurately diagnose single-sutural synostosis by physical examination alone. Considering potential side effects from ionizing radiation, risks of sedation, and costs, surgeons may wish to reserve computed tomographic scans only for infants with suspected single-sutural craniosynostosis in whom the physical examination is not clearly diagnostic.

Major morbidity and mortality rates in craniofacial surgery: an analysis of 8101 major procedures.

Background: The first combined evaluation of morbidity and mortality rates in craniofacial surgery was published 30 years ago; many surgeons believe these procedures have since become safer. The authors performed a contemporary evaluation of craniofacial morbidity and mortality rates to help surgeons more accurately counsel families about current risks, and to gain insight into reducing future incidences. Methods: This study used two methodologies to capture all serious morbidities and mortalities associated with major craniofacial procedures between 1990 and 2008: a comprehensive two-center retrospective review (Dallas and Seattle), and an Internet-based survey sent to all North American craniofacial centers. Results: Combining the two-center data with the survey results yielded a database of 7328 intracranial and 773 subcranial procedures, for a total of 8101 major craniofacial procedures. The combined intracranial major morbidity rate was less than 0.1 percent, and the combined mortality rate was 0.1 percent. Of the eight perioperative deaths following intracranial procedures, four (50 percent) intracranial mortalities were directly attributed to blood loss. The combined subcranial procedure major morbidity rate was 0.1 percent and the mortality rate was 0.3 percent (airway related). Comparing the earliest published intracranial mortality rate to our current review revealed a statistically significant reduction in incidence (p < 0.001). Conclusions: The incidence rates for serious morbidities and mortalities among major craniofacial procedures have significantly fallen since first published. On the basis of these analyses, the authors believe that a greater focus on protocols for airway management, blood salvage and replacement, age-appropriate deep venous thrombosis prophylaxis, and timing of subcranial midfacial advancements might result in further reductions in craniofacial mortality rates.