| 1. |
- Bernhardt, Peter, 1966, et al.
(author)
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A novel quantitative image-based method for evaluating cranial symmetry and its usefulness in patients undergoing surgery for unicoronal synostosis.
- 2013
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In: The Journal of craniofacial surgery. - 1536-3732. ; 24:1, s. 166-9
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Journal article (peer-reviewed)abstract
- Background: Unicoronal synostosis presents with cranial asymmetry. Fixed points are difficult to identify; surgical results are therefore difficult to evaluate. The aim of this study was to develop a computer-based method for evaluation of forehead symmetry to enable evaluation of surgical results in unicoronal synostosis. Methods: The MATLAB tool was programmed to segment computed tomographic images, leaving the outermost contour. Cephalometric images were segmented manually due to lower contrast. A center-point (O) and an end-point were manually defined in the midline of the forehead and at the nonfused coronal suture, respectively. The program then found a point (p) on the fused side, at the same distance from the O as the end-point. The contours of the left and right side of the forehead were thereafter superimposed, and the position of minimal area mismatch of the sides was identified. To correct for growth between preoperative images and follow-up, the number of mismatching pixels was related to the area outlined by the contour of the forehead, the end-point and p. Two quantities, the relative symmetry change and the absolute symmetry change, were defined and evaluated by repeated measurements on spherical and elliptical phantoms and 15 patients. Results: Measurements with the MATLAB program were reliable with an SD of 0.26% to 5.39% for the expected range of differences. The SD was lower for measurements on computed tomographic images than for measurements on cephalometric images. The SD was also lower in patients with large surgical improvement than in patients with little improvement. The results support the use of relative symmetry change to evaluate surgical results. Conclusions: Our new computer-based method is capable of measuring forehead symmetry with good precision. This method can be used for systematic evaluation of surgical outcome for unicoronal synostosis and other asymmetric skull deformities.
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| 2. |
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| 3. |
- Fischer, Sara, et al.
(author)
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Intracranial volume is normal in infants with sagittal synostosis.
- 2015
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In: Journal of plastic surgery and hand surgery. - 2000-6764. ; 49:1, s. 62-64
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Journal article (peer-reviewed)abstract
- Abstract Premature sagittal synostosis results in an elongated, narrow skull shape, scaphocephaly. It has been unclear whether the intracranial volume (ICV) of these children is different from that of normal children. The aim of the present study was to precisely determine the ICV in a large cohort of children with premature sagittal synostosis and to compare it to the ICV of a sex- and age-matched control group. All patients (n = 143) with isolated sagittal synostosis registered in the Göteborg Craniofacial Registry until the end of 2012 with a preoperative CT examination were identified. For each case, a sex- and age- (±30 days) matched control was identified from children who had undergone CT for post-traumatic or neurological reasons. The ICV was measured in a semi-automatic MATLAB program with functions such as region growing, watershed, and thresholding in axial CT slices. The ICV was calculated using the Cavalieri principle. The mean (± SEM) values of ICV for children with sagittal synostosis and for corresponding controls were 866 ± 13 ml and 870 ± 15 ml, respectively. The mean ages of these groups were 173 ± 8 days and 172 ± 8 days, respectively. Subgroup analysis of sex and age at CT (≤180 days and >180 days) did not reveal any differences in ICV between cases and controls. Precise determination of ICV in addition to the use of adequate controls has made it possible to conclude that children with premature isolated sagittal synostosis have a normal ICV.
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| 4. |
- Hagmarker, Linn, et al.
(author)
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A new quantitative image-based method for evaluation of bony temporal hollowing in metopic synostosis
- 2016
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In: Journal of Plastic Surgery and Hand Surgery. - : Medical Journals Sweden AB. - 2000-656X .- 2000-6764. ; 50:6, s. 343-348
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Journal article (peer-reviewed)abstract
- Objective: Premature craniosynostosis is a congenital disorder causing a skull deformity. For both functional and cosmetic reasons, the deformity is surgically treated with a cranioplasty before the age of 1 year. Temporal hollowing is a common and undesirable remaining deformity after cranioplasty for metopic synostosis. The most common method to determine the degree of temporal hollowing is subjective judgement of the temporal region. The aim of the present project was to develop a quantitative semi-automatic computer tool for objective measurement of bony temporal hollowing. Methods: Using MATLAB, a tool was developed to segment computed tomography images, defining the outermost contour. The images were dorsally limited to the widest point of the head. In each case, a sex-and age-matched control was identified and the contours compared. The bony temporal hollowing of the cases was calculated. Results: The intra-user coefficient of variation (CV) was 5.0% (95% CI = 4.2%-6.2%) and the inter-user CV was 3.0% (95% CI = 2.1%-8.6%). For clinical testing purposes, the tool was used in 14 patients, seven of whom had been operated on with a spring-assisted cranioplasty and seven with a cranioplasty using a bone graft. Conclusions: In summary, this study presents a new tool for objective measurement of the surgical result after cranioplasty for metopic synostosis.
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| 5. |
- Kljajić, Marizela, et al.
(author)
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Children Treated for Nonsyndromic Craniosynostosis Exhibit Average Adaptive Behavior Skills with Only Minor Shortcomings
- 2021
-
In: Plastic and reconstructive surgery. - 1529-4242. ; 147:2, s. 453-464
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Journal article (peer-reviewed)abstract
- Copyright © 2021 by the American Society of Plastic Surgeons. BACKGROUND: Adaptive behavior skills are important when assessing cognitive functions related to daily life; however, few studies have assessed these skills in patients treated for nonsyndromic craniosynostosis. In this study, the authors assessed the adaptive behavior skills of children treated for craniosynostosis and examined whether their outcomes are related to surgical technique. METHODS: The Adaptive Behavior Assessment System, 2nd Edition, parent report was used for children (age, 7 to 16 years) treated for sagittal (n = 41), metopic (n = 24), and other rare synostoses (n = 8). Background data, including intelligence quotient, were controlled for confounders. RESULTS: All evaluated children treated for craniosynostosis were estimated as lower in all aspects of adaptive behavior skills (full-scale, conceptual, social, and practical composites; effect size, 0.36 to 0.44) as compared with norms. The sagittal group showed shortcomings in social composite (effect size, 0.48) and subscales measuring self-care and self-direction, although no difference was observed between spring-assisted surgery and pi-plasty regarding outcomes of adaptive behavior skills. In addition, children treated for metopic synostosis showed results indicating shortcomings with adaptive behavior according to the full-scale, conceptual, and social composites (effect size, 0.53 to 0.61) relative to norms. Furthermore, attrition analysis revealed no significant differences between responders (rate, 80.2 percent) and nonresponders. CONCLUSION: These results found that children treated for craniosynostosis display average adaptive behavior skills, and that the two surgical techniques used to treat sagittal synostosis did not differ in their behavioral outcomes. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.
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| 6. |
- Kljajić, Marizela, et al.
(author)
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Sustained attention and vigilance of children treated for sagittal and metopic craniosynostosis.
- 2020
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In: Child neuropsychology : a journal on normal and abnormal development in childhood and adolescence. - : Informa UK Limited. - 1744-4136. ; 26:4, s. 475-488
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Journal article (peer-reviewed)abstract
- Attention problems are common in patients with craniosynostosis. Craniosynostosis is a rare condition, studies face challenges of selection bias, small sample sizes, and wide age ranges. The aim of the study was to assess the sustained attention and vigilance of children treated for sagittal and metopic craniosynostosis. To reduce selection bias, we included children that had previously undergone surgery for craniosynostosis, were between 8 and 16years, and lived close to the craniofacial centre. The Connors Continuous Performance Test (3rd edition) was used to measure sustained attention and vigilance (n =61; response rate: 76.3%). Attrition analysis revealed no differences between responding and non-responding groups regarding background variables. One identified difference between the SS (n =28) and MS (n =23) groups involved significantly better performance by the SS group in the hit-reaction time (HRT) test relative to the MS group (p<0.05). Compared with the norms, the SS group showed significantly worse response style, detectability, omissions, commissions, perseverations, HRT response speed (HRT-SD), HRT inter-stimulus interval change (HRT-iC) (p<0.01 for all), and variability (p<0.05). The MS group showed significantly worse detectability, HRT-SD, variability (p<0.01 for all), commissions, perseverations and HRT-iC (p<0.05 for all) as compared with norms. No differences regarding attention was detected for the two different surgical techniques used for correction of sagittal synostosis. There were shortcomings in sustained attention and vigilance as compared with the norms in the SS and MS groups, although the deviations were small.
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| 7. |
- Kljajić, Marizela, et al.
(author)
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The Cognitive Profile of Children with Non-syndromic Craniosynostosis
- 2019
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In: Plastic and reconstructive surgery. - 1529-4242. ; 143:5
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Journal article (peer-reviewed)abstract
- Long-term neuropsychological and cognitive outcomes in patients with non-syndromic craniosynostosis have proven difficult to evaluate objectively due to methodological problems with published studies based on their small and biased samples of patients, wide age ranges, and testing with unacceptable psychometric properties. This study evaluated full-scale intelligence quotient (FSIQ) and its subscales in a cohort with a small selection bias.Patients (aged 7-16 years) born with non-syndromic craniosynostosis and surgically treated were tested using the Wechsler Intelligence Scale for Children, Fourth Edition (WISC-IV). Ninety-one patients were invited, and 73 patients were tested.There was no difference in FSIQ between patients having undergone operations for sagittal synostosis or metopic synostosis and norms provided by the test. Patients operated on for sagittal synostosis showed a significantly higher perceptual reasoning IQ, but also significantly lower working-memory IQ and processing-speed IQ as compared with the norms. Patients operated on for metopic synostosis showed no differences in any IQ index as compared with the norm. Additionally, attrition analysis showed no differences in background factors between responders and non-responders.These results derived from a group of patients with uniform age range, and tested using an established tool revealed that non-syndromic children having undergone surgery for craniosynostosis exhibited average intellectual ability. However, the analysis indicated possible issues with working memory and processing speed in patients operated on for sagittal synostosis, highlighting impairments potentially associated with neuropsychological problems and that might contribute to learning disabilities.
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| 8. |
- Maltese, Giovanni, 1974, et al.
(author)
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Intracranial volume before and after surgical treatment for isolated metopic synostosis.
- 2014
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In: The Journal of craniofacial surgery. - 1536-3732. ; 25:1, s. 262-266
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Journal article (peer-reviewed)abstract
- Metopic synostosis results in a keel-shaped forehead, hypotelorism, and an increased interparietal width. This study aimed to measure the frontal and total intracranial volume in patients with metopic synostosis before and after surgery and to compare the effect of 2 different operation methods. All patients operated for isolated metopic synostosis between 2002 and 2008 at Sahlgrenska University Hospital who had undergone preoperative and/or postoperative computed tomographic examination (at 3 y of age) were included. The patients were grouped according to operation method: (1) forehead remodeling in combination with a bone graft or (2) forehead remodeling in combination with a spring. Sex- and age-matched controls were identified. A previously developed MATLAB computer program was used to measure the frontal and total intracranial volumes. Sixty patients and 198 controls were included. Preoperatively, the patients with metopic synostosis had significantly lower frontal volumes than those of the controls (P < 0.001) but equal total intracranial volumes. The operations redistributed the intracranial volume and resulted in an improved, frontal-total intracranial volume ratio. However, at 3 years of age, the frontal volume (P < 0.001), total intracranial volume (P ≤ 0.002), and ratio between the 2 (P < 0.001) were significantly lower in the patients than in the controls. The 2 operation methods were equally efficient in creating an improved frontal-total ratio. Surgery for metopic synostosis improves the distribution of the intracranial volume but does not result in normal total intracranial volume or frontal volume at 3 years of age.
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| 9. |
- Maltese, Giovanni, 1974, et al.
(author)
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New objective measurement of forehead symmetry in unicoronal craniosynostosis - comparison between fronto-orbital advancement and forehead remodelling with a bone graft.
- 2014
-
In: Journal of plastic surgery and hand surgery. - 2000-6764. ; 48:1, s. 59-62
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Journal article (peer-reviewed)abstract
- Abstract Patients with unicoronal synostosis (UCS) present with ipsilateral forehead flattening, contralateral frontal bossing, and rotation of the facial midline. Uni- or bilateral fronto-orbital advancement (FOA) techniques are the most common surgical approaches for correction of UCS. The purpose of this study was to objectively evaluate the surgical outcome in patients for UCS, using a new MATLAB computer tool programmed to measure the symmetry of the two halves of the forehead.Files were reviewed from a consecutive series of patients treated for UCS at the unit, from 1979-2008. The patients were grouped according to the method of operation used. The computer tool evaluated preoperative and postoperative cephalograms and CT scans. Eighty-eight patients were included. The male-to-female ratio was 1:2.4. Forty-six patients had been operated on with FOA and 42 with forehead remodelling using a calvarial bone graft. Forehead symmetry was significantly improved by both techniques (p < 0.001 for both), but the postoperative forehead symmetry was significantly better after forehead remodelling (p = 0.025). The reoperation rate was much lower for the second group (6.5 vs 37.2%, p < 0.001). It is concluded that forehead remodelling with a calvarial bone graft creates a more symmetrical forehead than FOA and may, therefore, be a better alternative for treatment of unicoronal synostosis.
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| 10. |
- Söfteland, Madiha Bhatti, 1977, et al.
(author)
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Temporal Deformity Objectively Measured before and after Surgery for Metopic Synostosis: Retrusion Rather than Hollowing
- 2017
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In: Journal of Craniofacial Surgery. - 1049-2275. ; 28:7, s. 1757-1760
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Journal article (peer-reviewed)abstract
- The temporal contour deformity typical of metopic synostosis is often referred to as temporal hollowing, but has not been quantitatively defined. This deformity is present before surgery and remains to a varying extent at long-term follow-up. The present study aimed to objectively evaluate the degree of this contour deformity in metopic synostosis before and after surgical correction. All children surgically treated for metopic synostosis at Sahlgrenska University Hospital between 2002 and 2014 (n = 120) with appropriate computed tomography scans (n = 160) performed preoperatively and/or at follow-up at 3 years of age were included. Depending on age, 1 of 2 surgical techniques was used. Children presenting before the age of 6 months were treated with frontal remodeling in combination with a spring (S group), whereas children older than 6 months were treated with a bone transplant (BT group). The bony temporal deformity was measured with a semiautomatic MATLAB program and patients were compared to sex-and age-matched controls. The deformity was significantly reduced in both groups (P < 0.001). In the S group, it was reduced from a mean ± standard deviation of 3.6 ± 1.9% to 1.0 ± 1.2% and in the BT group, it was reduced from 3.3% ± 1.4% to 1.1% ± 0.8%. The contour deformity in metopic synostosis is present both before and after surgery and should therefore be termed temporal retrusion (TR). This assessment method enables objective comparison of TR before and after surgical correction and is a potential tool to evaluate TR in metopic synostosis. © 2017 by Mutaz B. Habal, MD.
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| 11. |
- Söfteland, Madiha Bhatti, 1977, et al.
(author)
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The Degree of Surgical Frontal Volume Correction in Metopic Synostosis Determines Long-Term Outcomes
- 2017
-
In: Journal of Craniofacial Surgery. - : Ovid Technologies (Wolters Kluwer Health). - 1049-2275. ; 28:5, s. 1161-1163
-
Journal article (peer-reviewed)abstract
- Metopic synostosis results in a keel-shaped forehead, reduced frontal intracranial volume (ICV), and lower frontal to total volume ratio. The ratio improves with cranioplasty, but at 3 years of age, the ratio is still not normalized when compared to that in normal children. The aim of the present study was to investigate whether a low frontal to total ICV ratio at 3 years of age was due to relapse or insufficient correction.All children surgically treated for metopic synostosis in combination with a spring at Sahlgrenska University Hospital with subsequent spring extraction between 2002 and 2008 (n=20) were included. A MATLAB program was used to measure frontal and total ICV.Preoperatively, the frontal to total ICV ratio was 9.81.3% (mean +/- standard deviation). At spring removal, 6 months after cranioplasty, the ratio had increased to 11.8 +/- 2.4%. At 3 years of age, the ratio was 11.6 +/- 1.9%. In age-matched normal children, the ratio was 14.4 +/- 1.9% preoperatively, 15.3 +/- 2.2% at time of spring extraction, and 13.4 +/- 1.4% at 3 years of age.Cranioplasty thus improved the frontal to total ICV ratio, but did not normalize it. The ratio did not change from 6 months after the cranioplasty to 3 years of age. These results indicate that a more pronounced frontal volume correction during cranioplasty is necessary to achieve a normalized distribution of ICV in metopic synostosis.
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| 12. |
- Tovetjärn, Robert, 1982, et al.
(author)
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Intracranial volume in 15 children with bilateral coronal craniosynostosis.
- 2014
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In: Plastic and reconstructive surgery. Global open. - 2169-7574. ; 2:11
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Journal article (peer-reviewed)abstract
- Intracranial volume (ICV) growth in patients with bilateral coronal craniosynostosis (BCS) is not well described. It is therefore important to evaluate the consequences of cranial surgery in children with this condition. The aim of the present study was to evaluate ICVs in patients operated on for BCS.
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| 13. |
- Wikberg, Emma, et al.
(author)
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A new computer tool for systematic evaluation of intracranial volume and its capacity to evaluate the result of the operation for metopic synostosis.
- 2012
-
In: Journal of plastic surgery and hand surgery. - 2000-6764. ; 46:6, s. 393-8
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Journal article (peer-reviewed)abstract
- Abstract The aim of this project was to develop a tool for systematic evaluation of volumetric changes after surgery for craniosynostosis. A computer program using MATLAB was developed to measure total intracranial volume and frontal volume, anterior to the coronary sutures, by multiplying the area with slice thickness of each slice from just above foramen magnum to just beneath the vertex in CT examinations. The ratio between frontal volume and total volume was used for evaluation of the clinical result in 12 patients operated on for metopic synostosis. In 0.625 and 5 millimetre slices the coefficients of variation were 0.00049 and 0.00058, respectively, for measurements of total volume. The highest coefficient of variation was found in postoperative measurements of the frontal volume and was 0.014 in 0.625 millimetre slices. Measurements in 5 millimetre slices resulted in 3.8% ± 1.5% (mean ± SD) lower total volumes and 5.8% ± 5.3% lower frontal volumes than measurements in 0.625 millimetre slices. In patients operated on for metopic synostosis the ratio between frontal volume and total volume increased 25% ± 16% for patients operated on with cranioplasty in combination with a spring (n = 6) and 20% ± 13% for patients operated on with cranioplasty in combination with a bone transplant (n = 6). In summary, this study has developed a tool that can determine frontal and total intracranial volume with little variation. This tool can be used for systematic evaluation of the result of the operation for metopic synostosis.
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| 14. |
- Bevilacqua, Ruggero, et al.
(author)
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Spring-Assisted Surgery in the Treatment of Complex Craniosynostosis
- 2018
-
In: Journal of Craniofacial Surgery. - 1049-2275. ; 29:4, s. 920-924
-
Journal article (peer-reviewed)abstract
- Multisutural nonsyndromic craniosynostosis is a rare group of malformations, whose frequency has been reported between 3% and 7% of all craniosynostosis. The clinical diagnosis can be difficult and computed tomography is usually required. Surgical treatment is challenging and staged procedures are performed in up to 80% of patients. The aim of the present study was to determine the reoperation rate and to evaluate the surgical outcomes by measuring intracranial volume (ICV) preoperatively and at follow-up, and comparing it to a control group. Perioperative variables and reoperation rate were recorded. Fifty-one patients presented with a complex pattern of synostosis without a recognizable syndrome (5% of cases of total patients evaluated). Fifteen patients have been treated with spring-assisted surgery, either alone or in combination with a foreheadplasty. The mean follow-up was 6.2 years. The mean preoperative ICV of the patients was smaller, but not significantly, than in the normal population (P = 0.13). Postoperatively, the mean ICV was similar to that of the control group at 1 year (P = 0.92), while at 3 years it was appreciably smaller, although not significantly different (P = 0.06). Five patients (33%) went through a secondary skull expansion for either raised intracranial pressure or cosmetic reasons. Spring-assisted surgery seems to temporarily expand ICV in children with complex synostosis and lower the reoperation rate, thus reducing the need for a second procedure. A longer follow-up would be necessary to further investigate the effects of springs over time.
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| 15. |
- Fischer, Sara, et al.
(author)
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Comparisons of Intracranial Volume and Cephalic Index After Correction of Sagittal Craniosynostosis With Either Two or Three Springs
- 2021
-
In: The Journal of craniofacial surgery. - 1536-3732. ; 32:8, s. 2636-2640
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Journal article (peer-reviewed)abstract
- ABSTRACT: In this retrospective study, the authors determined changes in intracranial volume (ICV) and cephalic index (CI) in patients with sagittal craniosynostosis and operated with craniotomy combined with either 2 or 3 springs. The authors included patients (n=112) with complete follow-up that had undergone surgical correction for isolated sagittal craniosynostosis with craniotomy combined with springs between 2008 and 2017. All patients underwent computed tomography examination preoperative, at the time of spring extraction, and at 3years of age. Intracranial volume was measured using a semiautomatic MATLAB program, and CI was calculated as the width/length of the skull. The authors found that craniotomy combined with 2 springs increased the ICV from a preoperative value of 792±113mL (mean±standard deviation) to 1298±181mL at 3years of age and increased the CI from 72.1±4.1 to 74.6±4.3, whereas craniotomy combined with 3 springs increased the ICV from 779±128mL to 1283±136mL and the CI from 70.7±4.3 to 74.8±3.7. The relative increase in ICV was 65±21% in the two-spring group and 68±34% in the three-spring group (P value=0.559), and the relative increase in CI was 3.6±3.3% in the two-spring group as compared with 6.0±5.0% in the three-spring group (P=0.004). These findings demonstrated that use of 3 springs resulted in additional absolute and relative CI-specific effects as compared with 2 springs during the time when the springs were in place, with this effect maintained at 3years of age.
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| 16. |
- Fischer, Sara, et al.
(author)
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Psychosocial conditions in adults with Crouzon syndrome: a follow-up study of 31 Swedish patients.
- 2014
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In: Journal of plastic surgery and hand surgery. - 2000-6764. ; 48:4, s. 244-247
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Journal article (peer-reviewed)abstract
- Abstract Crouzon syndrome presents with craniosynostosis, maxillary hypoplasia, exophtalmus, and sometimes hampered neuropsychological development. The aim of the present study was to evaluate the quality of life for adult patients with Crouzon syndrome. Forty patients with Crouzon syndrome born before 1990 could be identified. A questionnaire addressing education, employment, social relations, and quality-of-life was used. A matched control group was created for comparison. Logistic regression, correcting for the influence of age and sex, was used to compare patients and controls. Thirty-one patients and 285 controls answered the questionnaire. The level of education was lower in patients than in controls (p < 0.015). Patients were less often married or had a partner (p = 0.059), had fewer children of their own (p = 0.004), and had less experience of a sexual relationship (p < 0.001). The difference in housing was not significant, and only one patient lived in a care centre and three patients required a personal assistant to manage activities of daily living. The patients' estimation of their somatic health was equal to that of the controls, but the patients more often used anti-epileptic medication (p = 0.003). Periods of depressive mood were more common in patients (p = 0.001), but there was no difference between the groups regarding a general positive attitude to life. In conclusion, patients with Crouzon syndrome often have intellectual and social shortcomings that negatively affect their lives. However, the range of abilities is wide in this group.
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| 17. |
- Fröjd, Victoria, 1986, et al.
(author)
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Conservative Healing of an 11×9-cm Aplasia Cutis Congenita of the Scalp with Bone Defect.
- 2014
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In: Journal of neurological surgery reports. - : Georg Thieme Verlag KG. - 2193-6358 .- 2193-6366. ; 75:2
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Journal article (peer-reviewed)abstract
- ObjectivesAplasia cutis congenita is a rare congenital condition, and it is difficult to find scientific support for optimal treatment strategies. In addition, these may vary due to defect size, tissue layers involved, contemporary malformations, and the physiologic status of the affected child. Clinical PresentationThis case report describes complete skin coverage in 20 weeks and uneventful healing of a large 11×9-cm defect of the vertex, involving both skin and skull bone, using conservative treatment. To prevent infection and promote healing, the defect was kept moist and covered at all times, and it was treated with surgical debridement when necessary. For infection control, ionized silver-coated dressings were used in addition to prophylactic antibiotics over the first 3.5 weeks. Follow-up was 2 years. ConclusionSurgical treatment is usually preferred for larger aplasia cutis congenita defects, but it is accompanied with potential risks and will exacerbate secondary reconstruction of alopecia or skull bone defects. This case shows that even very complex defects may be treated conservatively.
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| 18. |
- Hallen, T., et al.
(author)
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Cranioplasty without Periosteal Dissection Reduces Blood Loss in Pi-Plasty Surgery for Sagittal Synostosis
- 2017
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In: Pediatric Neurosurgery. - : S. Karger AG. - 1016-2291 .- 1423-0305. ; 52:4, s. 284-287
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Journal article (peer-reviewed)abstract
- Background/Aims: Cranioplasty is often accompanied by a substantial oozing bleeding from the bone surface and bone edges. Our aim was to measure if strict subgaleal dissection without any periosteal release reduces blood loss during pi-plasty surgery for sagittal synostosis. Method: A group of 32 children who underwent pi-plasty surgery at the Sahlgrenska University Hospital between 2010 and 2014 for premature sagittal synostosis with traditional subgaleal dissection combined with incision and release of the periosteum adjacent to the osteotomy lines was compared to a group of 7 children who underwent pi-plasty with strict subgaleal dissection and osteotomy through the bone with the periosteum attached. Information about blood loss and body weight was extracted from medical records. Results: The blood loss in the group of 7 children with strict subgaleal dissection was 102 +/- 86 mL (mean +/- SD) (10 +/- 7 mL/kg) compared to 320 +/- 119 mL (32 +/- 12 mL/kg) in the control group with traditional periosteal release (p < 0.001). Conclusion: Intact periosteum at the osteotomy lines significantly reduces blood loss in pi-plasty surgery for sagittal synostosis. The mechanism is likely because of preserved veins between the bone surface and periosteum.
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| 19. |
- Knudsen, Elisabet, 1944, et al.
(author)
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Parental estimation of early psychological development in children operated on for single suture synostosis.
- 2012
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In: Journal of plastic surgery and hand surgery. - 2000-6764. ; 46:3-4, s. 152-154
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Journal article (peer-reviewed)abstract
- Abstract Impaired psychological development in children with single suture craniosynostosis is often described in the literature. The authors' own experience was that these children appeared to have normal psychological development during their first years of life. The aim of the present study was, therefore, to evaluate if parental estimation would reveal any shortcomings in psychological development. All consecutive patients operated on for non-syndromal single suture synostosis between October 2002 and June 2006 were included (n = 66). A questionnaire was filled out by the parents when the child was 3 years old. The questions concerned development of speech, motor control, personal abilities, and emotional development. The results were compared with randomly selected controls of the same age (n = 180) whose parents answered the same questionnaire. The results showed that children operated on for single suture synostosis did not show any signs of lasting disadvantages due to craniofacial surgery. The children did not in any respect suffer from a hampered psychological development up to 3 years of age. In summary the psychological development of patients operated on for single suture synostosis seems to be normal up to 3 years of age as far as parental estimation can reveal.
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| 20. |
- Kölby, David, 1993, et al.
(author)
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Craniotomy of the Fused Sagittal Suture Over the Superior Sagittal Sinus Is a Safe Procedure
- 2017
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In: Journal of Craniofacial Surgery. - : Ovid Technologies (Wolters Kluwer Health). - 1049-2275. ; 28:3, s. 666-669
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Journal article (peer-reviewed)abstract
- Introduction: Spring-assisted cranioplasty to correct sagittal synostosis is based on midline craniotomy through the closed sagittal suture, over the superior sagittal sinus (SSS). The aim of the present study was to evaluate the perioperative safety of this technique. Materials and Methods: This is a retrospective study of all patients operated with median craniotomy and springs from 1998 to the end of 2015. For comparison, all Pi-plasties performed during the same time interval were also evaluated. The safety measures were evaluated based on incidence of damage to SSS, incidence of dural tears, perioperative blood loss, operative time, and hospital stay. Results: In the group that had undergone midline craniotomy combined with springs (n = 225), 4 perioperative damages to SSS and 1 dural tear were seen. The perioperative blood loss was 62.8 +/- 65.3mL (mean +/- standard deviation). The operative time was 67.9 +/- 21.5 minutes and the hospital stay was 4.8 +/- 1.1 days. In the group that had undergone pi-plasty (n = 105), no damages to SSS but 3 dural tears were seen. The perioperative blood loss was 352.8 +/- 174.4 mL. The operative time was 126.0 +/- 31.7 minutes and the hospital stay was 7.1 +/- 1.4 days. Conclusion: Craniotomy SSS in sagittal synostosis is a safe procedure with low morbidity in terms of damage to the SSS. Midline craniotomy combined with springs has significantly lower preoperative blood loss, operative time, and hospital stay (P < 0.001 for all) compared to pi-plasty.
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| 21. |
- Malmqvist, Carina, et al.
(author)
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Simplified Postoperative Care After Spring-assisted Strip Craniotomy for Sagittal Synostosis: A Prospective Before-and-After Study.
- 2021
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In: The Journal of craniofacial surgery. - 1536-3732. ; 32:4, s. 1507-1510
-
Journal article (peer-reviewed)abstract
- Since the development of spring-assisted techniques for corrective craniofacial surgery, routine postoperative admission to intensive care units (ICUs) has been questioned. However, close monitoring is necessary if the continuous infusion of morphine is used as recommended for better pain relief. In this study, the authors evaluated a simplified postoperative protocol without continuous morphine infusion and no indwelling urinary catheter following spring-assisted surgery (SAS) for sagittal synostosis. Ten children were cared for according to a standard protocol with postoperative intravenous (i.v.) infusion of morphine and an indwelling urinary catheter, and 11 consecutive children were treated according to a simplified protocol with pain relief based on intermittent injections of morphine and clonidine [according to Face, Legs, Activity, Cry, Consolability (FLACC) scores >4] without the indwelling catheter. A Mann-Whitney U test was used for comparison of distributions between the two groups. The results revealed no differences between groups regarding the proportion of FLACC scores >4, total amount of administered i.v. morphine and clonidine, total volume of buffered glucose infused, time to first feeding on breast milk or substitute, or the length of stay. Despite the inherent limitations of our small observational study, the authors concluded that at our institution, it was possible to exclude a standard continuous i.v. infusion of morphine and an indwelling urinary catheter from our postoperative care protocol without decreasing the quality of pain relief in children submitted to SAS for sagittal synostosis. This finding supports downgrading the level of care from the ICU to a regular ward after limited immediate postoperative observation.
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| 22. |
- Maltese, Giovanni, 1974, et al.
(author)
-
Correction of hypotelorism in isolated metopic synostosis.
- 2014
-
In: Journal of plastic surgery and hand surgery. - 2000-6764. ; 48:1, s. 63-66
-
Journal article (peer-reviewed)abstract
- Abstract Surgical correction of trigonocephaly is performed by fronto-orbital reshaping and advancement. Some authors supplement the fronto-orbital remodelling with direct surgical correction of the hypotelorism, but the role of this step of the procedure has been questioned. At Sahlgrenska University Hospital, hypotelorism in metopic synostosis is treated with spring-assisted surgery (S) in children below 6 months of age and with fronto-orbital advancement with the interposition of a bone graft (BG) in the fronto-orbital region at higher age. The aim of the present study was to evaluate the anterior bony interorbital distance (BIOD) preoperatively and at follow-up in patients operated on with the two techniques and to compare the results with adequate control groups. Preoperatively, the patients affected by metopic synostosis had a significantly reduced BIOD compared to their respective controls (S group: 13.8 ± 1.6 (mean ± SD) mm vs 18.6 ± 1.4 mm, p < 0.001, BG group: 14.7 ± 1.0 mm vs 18.8 ± 1.4 mm, p < 0.001). At 3 years follow-up, BIOD was improved in both groups, but only in the S group the mean BIOD did not differ from the control group (19.7 ± 2.9 mm vs 20.2 mm ± 1.3 mm, p = 0.3). In the BG group the BIOD was still significantly different between patients and controls (17.6 ± 1.8 vs 20.0 ± 1.1 mm, p < 0.001). Spring-assisted surgery performed before 6 months of age can normalise hypotelorism in metopic synostosis. Full correction in a large series of patients has now been achieved for the first time.
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| 23. |
- Maltese, Giovanni, 1974, et al.
(author)
-
Spring-assisted surgery in the treatment of sagittal synostosis: A systematic review.
- 2015
-
In: Journal of plastic surgery and hand surgery. - 2000-6764. ; 49:3, s. 177-182
-
Journal article (peer-reviewed)abstract
- Abstract Background: Premature sagittal synostosis (SS) can be surgically corrected using extensive cranioplasties or using less invasive methods, e.g. spring-assisted surgery (SAS). Aim: The aim of the present study was to perform a proper systematic review of springs in the treatment of SS. Methods: A literature search was performed with the assistance of a professional librarian in the databases PubMed, EMBASE, and The Cochrane Library between 1997 and September 2013. Studies that fulfilled the PICO (patients, interventions, controls, outcome) criteria were included. All studies were graded for methodological quality according to MINORS, and all retrospective studies were assessed according to a scale developed for retrospective studies in paediatric surgery. The quality of evidence was rated according to GRADE. Results: A total of 241 abstracts were extracted in the literature search. Five studies met the PICO criteria. Two of these five were considered as preliminary reports and excluded. Assessment according to MINORS showed a mean score of 21, i.e. fair quality. The clinical outcome regarding cephalic index did not differ between the surgical techniques, but the quality of evidence, according to GRADE, that SAS was equally efficient, was very low. Clinical outcome regarding operation time, blood loss, ICU stay and hospital stay was in favour for SAS, but the quality of evidence was low. Conclusions: This systematic review has revealed that the level of evidence for SAS being an equally efficient surgical method as more extensive cranioplasties for SS is low or very low. The results point out the need for well-designed prospective studies within craniofacial surgery.
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| 24. |
- Mellgren, Jonas, 1997, et al.
(author)
-
Improved Facial and Skull-base Symmetry Following Osteotomy and Distraction of Unilateral Coronal Synostosis.
- 2024
-
In: Plastic and reconstructive surgery. - 1529-4242. ; 153:2, s. 447-456
-
Journal article (peer-reviewed)abstract
- Unilateral coronal synostosis (UCS) results in a surgically demanding deformation, as the deformity is asymmetric in the calvarium but also presents with facial scoliosis and orbital dystopia. Traditional cranioplasties correct the forehead but have little effect on the face and orbits. Here, we describe a consecutive series of patients operated for UCS with osteotomy of the fused suture combined with distraction osteogenesis (FOD).Fourteen patients [mean age: 8.0 months (range: 4.3-16.6 months)] were included in this study. We measured and compared the orbital dystopia angle (ODA), anterior cranial fossa deviation (ACFD), and anterior cranial fossa cant (ACFC) between results from preoperative computed tomography and those at distractor removal.Blood loss was 6.1 mL/kg (range: 2.0-15.2 mL/kg), and length of stay was 4.4 days (range: 3.0-6.0 days). We observed significant improvements in the ODA from [median (95% confidence interval)] -9.8° (-12.6° to -7.0°) to -1.1° (-3.7° to -1.5°) (p<0.001), ACFD from 12.9° (9.2-16.6°) to 4.7° (1.5-7.9°) (p<0.001), and ACFC from 2.5° (1.5-3.5°) to 1.7° (0.0-3.4) (p=0.003).The results showed that osteotomy combined with a distractor for UCS straightened the face and relieved orbital dystopia by affecting the nose angle relative to the orbits, correcting the deviation of the cranial base in the anterior fossa, and lowering the orbit on the affected side. Furthermore, this technique demonstrated a favorable morbidity profile with low perioperative bleeding and a short inpatient period, suggesting its potential to improve the surgical treatment of UCS.
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| 25. |
- Michaëlsson, Isak, et al.
(author)
-
Circulating Brain-Injury Markers After Surgery for Craniosynostosis
- 2023
-
In: World Neurosurgery. - : Elsevier BV. - 1878-8750 .- 1878-8769. ; 173
-
Journal article (peer-reviewed)abstract
- Objective: Historically, there have been few quantitative methods for effectively evaluating outcomes after surgery for craniosynostosis. In this prospective study, we assessed a novel approach for detecting possible postsurgery brain injury in patients with craniosynostosis. Methods: We included consecutive patients operated on for sagittal (pi-plasty or craniotomy combined with springs) or metopic (frontal remodeling) synostosis at the Craniofacial Unit at Sahlgrenska University Hospital, Gothenburg, Sweden, from January 2019 to September 2020. Plasma concentrations of the brain-injury biomarkers neurofilament light (NfL), glial fibrillary acidic protein (GFAP), and tau were measured immediately before induction of anesthesia, immediately before and after surgery, and on the first and the third postoperative days using single-molecule array assays. Results: Of the 74 patients included, 44 underwent craniotomy combined with springs for sagittal synostosis, 10 underwent pi-plasty for sagittal synostosis, and 20 underwent frontal remodeling for metopic synostosis. Compared with baseline, GFAP level showed a maximal significant increase at day 1 after frontal remodeling for metopic synostosis and pi-plasty (P = 0.0004 and P = 0.003, respectively). By contrast, craniotomy combined with springs for sagittal synostosis showed no increase in GFAP. For neurofilament light, we found a maximal significant increase at day 3 after surgery for all procedures, with significantly higher levels observed after frontal remodeling and pi-plasty compared with craniotomy combined with springs (P < 0.001). Conclusions: These represent the first results showing significantly increased plasma levels of brain-injury biomarkers after surgery for craniosynostosis. Furthermore, we found that more extensive cranial vault procedures resulted in higher levels of these biomarkers relative to less extensive procedures.
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| 26. |
- Paganini, Anna, 1979, et al.
(author)
-
In search of a single standardised system for reporting complications in craniofacial surgery: a comparison of three different classifications.
- 2019
-
In: Journal of plastic surgery and hand surgery. - 2000-6764. ; 59:6, s. 321-7
-
Journal article (peer-reviewed)abstract
- Comparing complication rates between centres is difficult due to the lack of unanimous criteria regarding what adverse events should be defined as complications and how these events should be compiled. This study analysed all adverse events in a cohort of craniofacial (CF) operations over a 10-year period and applied three different scales (Clavien-Dindo, Leeds and Oxford) for systematic comparison. A total of 1023 consecutive CF procedures in 641 patients was identified. The Clavien-Dindo scale captured 74 complications in 74 procedures (7.2%), whereas the Leeds and Oxford scales captured 163 complications in 134 procedures (13.1%) and 85 complications in 83 procedures (8.1%), respectively. The Clavien-Dindo scale appeared less suitable for CF surgery, because it is predominantly adapted to severe complications and also regards blood transfusion as a complication. The Leeds scale provided a detailed picture of all complications, as well as minor events, whereas the Oxford scale captured all major complications well but applied less accurate definitions for the minor events. Our findings contribute to the benchmarking of complications between CF centres and suggest that both the Leeds and the Oxford scale appear relevant, depending on the emphasis required for major and minor complications and inter-centre audits, respectively.
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| 27. |
- Selvaggi, Gennaro, 1973, et al.
(author)
-
Ethical Considerations in Surgery for Single-suture Craniosynostosis.
- 2023
-
In: The Journal of craniofacial surgery. - 1049-2275 .- 1536-3732. ; 34:7, s. 1922-1926
-
Journal article (peer-reviewed)abstract
- Single-suture craniosynostosis (SSC) describes the premature fusion of one cranial suture, which restricts cranial growth and consequently results in unaffected regions presenting a compensatory expansion. Surgery can redistribute intracranial volume, reduce the risk of elevated intracranial pressure, and improve head shape, potentially leading to improved neurocognitive function and social acceptance. However, there is limited evidence that surgery for SSC improves neurocognitive function and social acceptance. Given the inherent surgical risks and uncertainty of outcomes, the conditions under which this surgery should be allowed remain uncertain. Here, we discuss ethical questions regarding the permissibility of surgery, value of neurocognitive function and social acceptance, research ethics associated with SSC, patient autonomy and parental roles, and the process of recommending surgery and obtaining consent. Because surgery for SSC has become a routine procedure, its practice now presents a relatively low risk of complications. Furthermore, having acquired an understanding of the risks associated with this surgery, such knowledge fulfils the principle of non-maleficence although not beneficence. Thus, we advocate that surgery should only be offered within Institutional Review Board-approved research projects. In these situations, decisions concerning enrollment in scientific research involves health care providers and parents or guardians of the child, with the former acting as gate-keepers upon recognition of a lack of coping skills on the part of the parent or guardian in dealing with unforeseen outcomes. To minimize associated surgical risks and maximize its benefits, there exists a moral obligation to refer patients only to highly specialized centers.
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| 28. |
- Säljö, Karin, 1981, et al.
(author)
-
Craniosynostosis: A Reversible Pathology?
- 2019
-
In: The Journal of craniofacial surgery. - 1536-3732. ; 30:6, s. 1628-1630
-
Journal article (peer-reviewed)abstract
- The formation of the cranial sutures, in utero, occurs when the ossification of the skull bones reaches predestined positions around gestational week 15 to 20. Craniosynostosis, and the consequent skull shape deformities, is treated with surgery including osteotomies of the fused sutures. The occasional appearance of a new suture in the osteotomy lines has previously been described as sporadic events. In this retrospective study, a 4-year consecutive series of osteotomies combined with springs for craniosynostosis were systematically analysed regarding the appearance of neosutures. In total, 84 patients were included and in 16 patients (19%) a new radiologically normal suture appeared in a part of the suture that was completely closed preoperatively. Additionally, in 7 patients (8%) a new suture appeared in a part of the suture that had a discernible suture prior to surgery.In conclusion, in this consecutive and well-defined patient cohort operated for craniosynostosis, the formation of a neosuture is not a rare, and speculatively not a random, event. The appearance of a new suture long after the normal time period for suture formation in utero indicates that the craniosynostosis may just as well be caused by disturbed formation of the suture as actual premature closure.
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| 29. |
- Söfteland, Madiha Bhatti, 1977, et al.
(author)
-
Correction of Unicoronal Synostosis With Springs: Two Patients With Improved Facial Symmetry
- 2024
-
In: JOURNAL OF CRANIOFACIAL SURGERY. - 1049-2275 .- 1536-3732. ; 35:1, s. 10-12
-
Journal article (peer-reviewed)abstract
- Objective: Surgical correction of unicoronal synostosis (UCS) entails extensive cranioplasties which do not address facial scoliosis. This paper presents the first results with springs that motivated the shift from extensive cranioplasties to dynamic techniques for surgical correction of UCS.Methods: Two cases of UCS were operated with a linear osteotomy combined with springs. The deviation in facial symmetry (orbital dystopia angle) and skull base angles were measured on pre and postoperative computed tomography scans until 3 years of age.Results: The facial scoliosis was corrected. At spring removal, the orbital dystopia angle had gone from a 9.2 to 13.2-degree deviation preoperatively to a 0.5 to 0.9-degree overcorrection compared with the ideal 0-degree deviation. Also, the skull base deviation improved.Conclusion: Linear osteotomy combined with springs corrects the facial scoliosis in UCS. These cases indicate that dynamic methods may be beneficial for improving the results of surgical correction of UCS.
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| 30. |
- Tarnow, Peter, 1963, et al.
(author)
-
Incidence of Non-Syndromic and Syndromic Craniosynostosis in Sweden
- 2022
-
In: Journal of Craniofacial Surgery. - : Ovid Technologies (Wolters Kluwer Health). - 1049-2275. ; 33:5, s. 1517-1520
-
Journal article (peer-reviewed)abstract
- Premature craniosynostosis is a rare condition, with a wide range of incidence estimations in the literature. The aim of this study was to establish the current incidence among the Swedish population. Since the surgical care for these children is centralized to the 2 centers of Sahlgrenska University Hospital and Uppsala University Hospital, the 2 craniofacial hospital registries were examined for surgically treated children, all having a computed tomography verified diagnosis. Results show an incidence of 7.7 cases per 10,000 live births, including 0.60/10,000 syndromic craniosynostosis. Due to information programs among health care staff and a system for early diagnosis through rapid communication, these results seem to mirror the true incidence of craniosynostosis in the Swedish population. The updated incidence data will facilitate healthcare planning and make future studies of possible changes in craniosynostosis incidence more accurate.
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| 31. |
- Tengström, Emin, et al.
(author)
-
Minnen från studenttiden vid Göteborgs universitet
- 2018
-
Book (other academic/artistic)abstract
- Boken ger några självbiografiska exempel på hur livet som student vid GU kunde te sig under senare halvan av 1900-talet (från 40-tal till 90-tal). Bland de teman som tas upp kan nämnas den studiesociala situationen, studentlivet i allmänhet, studierna inom ett antal ämnesområden, erfarenheterna och upplevelserna av den utbildning om erbjöds samt de dåtida ekonomiska förutsättningarna för studierna. Författare är Emin Tengström, Britta Olinder, Martin Fritz, Allan Svensson, Mats Oscarson, Britt-Mari Näsström, Peter Sjögren, Peter Tarnow och Henrik Zetterberg.
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| 32. |
- Topa, Alexandra, 1978, et al.
(author)
-
The value of genome-wide analysis in craniosynostosis
- 2024
-
In: FRONTIERS IN GENETICS. - 1664-8021. ; 14
-
Journal article (peer-reviewed)abstract
- Background: This study assessed the diagnostic yield of high-throughput sequencing methods in a cohort of craniosynostosis (CS) patients not presenting causal variants identified through previous targeted analysis.Methods: Whole-genome or whole-exome sequencing (WGS/WES) was performed in a cohort of 59 patients (from 57 families) assessed by retrospective phenotyping as having syndromic or nonsyndromic CS.Results: A syndromic form was identified in 51% of the unrelated cases. A genetic cause was identified in 38% of syndromic cases, with novel variants detected in FGFR2 (a rare Alu insertion), TWIST1, TCF12, KIAA0586, HDAC9, FOXP1, and NSD2. Additionally, we report two patients with rare recurrent variants in KAT6A and YY1 as well as two patients with structural genomic aberrations: one with a 22q13 duplication and one with a complex rearrangement involving chromosome 2 (2p25 duplication including SOX11 and deletion of 2q22). Moreover, we identified potentially relevant variants in 87% of the remaining families with no previously detected causal variants, including novel variants in ADAMTSL4, ASH1L, ATRX, C2CD3, CHD5, ERF, H4C5, IFT122, IFT140, KDM6B, KMT2D, LTBP1, MAP3K7, NOTCH2, NSD1, SOS1, SPRY1, POLR2A, PRRX1, RECQL4, TAB2, TAOK1, TET3, TGFBR1, TCF20, and ZBTB20.Conclusion: These results confirm WGS/WES as a powerful diagnostic tool capable of either targeted in silico or broad genomic analysis depending on phenotypic presentation (e.g., classical or unusual forms of syndromic CS).
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| 33. |
- Tovetjärn, Robert, 1982, et al.
(author)
-
Children with apert syndrome as adults: a follow-up study of 28 scandinavian patients.
- 2012
-
In: Plastic and reconstructive surgery. - 1529-4242. ; 130:4
-
Journal article (peer-reviewed)abstract
- : Apert syndrome (acrocephalosyndactyly type 1) includes craniofacial deformities, malformations of the extremities and the central nervous system, and often mental retardation. The aim of this study was to investigate the life situation of adult patients with Apert syndrome.
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| 34. |
- Tovetjärn, Robert, 1982, et al.
(author)
-
Spring-assisted cranioplasty for bicoronal synostosis.
- 2012
-
In: The Journal of craniofacial surgery. - 1536-3732. ; 23:4, s. 977-81
-
Journal article (peer-reviewed)abstract
- Background: Numerous surgical techniques for cranial reconstruction of patients with bicoronal synostosis have been suggested. The outcome is, however, still often suboptimal. Methods: Since 2005, we have, at the Craniofacial Unit, Sahlgrenska University Hospital, Gothenburg, used a standardized surgical technique with advancement and remodeling of the forehead combined with spring distraction of the occipital area. The aim of the current study was to evaluate this operative technique. Eighteen consecutive patients (9 boys and 9 girls) with bicoronal synostosis operated on using this technique were identified. Sixteen patients had syndromic bicoronal synostosis, and 2 had nonsyndromic bicoronal synostosis. Cephalic index was obtained from three-dimensional computed tomography scans, and photographs were analyzed for aesthetic evaluation. Results: The preoperative calvarial shape was hyperbrachycephalic in all subjects. Postoperatively, the calvarial shape was, in general, much closer to the norm. The reduction in the mean cephalic index from the preoperative stage (94) to the 3-year follow-up (82) was statistically significant (P < 0.0001). The mean duration of surgery was 155 (SD, 32) minutes, with a mean perioperative bleeding of 237 (SD, 95) mL. The mean hospital stay was 6.3 (SD, 1.5) days, of which the mean intensive care unit stay was 1.6 (SD, 1.2) days. In 2 patients, one of the springs had to be reinserted because of postoperative dislocation. No other major complications were observed. Conclusions: Spring-assisted cranioplasty for bicoronal synostosis is a safe technique, is less invasive than many other cranioplasties, and results in marked improvement in the calvarial shape.
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| 35. |
- Andine, A., et al.
(author)
-
Cleft Palate in Apert Syndrome: A Descriptive Study of Incidence and Surgical Outcome
- 2023
-
In: The Cleft Palate-Craniofacial Journal. - 1055-6656.
-
Journal article (peer-reviewed)abstract
- Objective Apert syndrome (AS) is a rare congenital craniofacial disorder that requires a multidisciplinary approach to treatment and multiple surgeries. Given that cleft palate (CP) is presented in some of these cases, this poses an additional risk of aggravating obstructed airways after closure. The timing and outcome of CP repair in these patients remains disputed and requires additional attention. Design This retrospective analysis included patients diagnosed with CP and AS, born between 1950 and 2020, and treated at our institution. Data were collected from medical records and evaluated using descriptive statistics. Setting Data analyses were conducted at Sahlgrenska University Hospital in Gothenburg, Sweden. Patients/Participants A registry of 83 patients with AS resulted in a cohort of 26 patients also presenting with CP. Main Outcome Measures Postoperative complications, requirement for intensive care, and reoperations following CP repair. Results CP incidence among all registered patients was 31%. Patients undergoing CP repair at low age (mean: 22.5 months) tended to experience more frequent postoperative complications and requirements for intensive care. Among the evaluated cohort with medical records describing CP repair (n = 14), 29% experienced postoperative complications, all of which involved aggravation of obstructed airways. Conclusions This study highlights the importance of airway assessment before and after CP repair in AS. The findings suggest that surgical outcomes might benefit from postponing CP repair, avoiding combined surgeries, and operating in two stages when indicated. However, additional and larger studies are required.
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| 36. |
- Bergman, Annika, et al.
(author)
-
Germline mutation screening of the Saethre-Chotzen-associated genes TWIST1 and FGFR3 in families with BRCA1/2-negative breast cancer
- 2009
-
In: Scandinavian Journal of Plastic and Reconstructive Surgery and Hand Surgery. - : Taylor & Francis. - 0284-4311 .- 1651-2073. ; 43:5, s. 251-255
-
Journal article (peer-reviewed)abstract
- Saethre-Chotzen syndrome is one of the most common craniosynostosis syndromes. It is an autosomal dominantly inherited disorder with variable expression that is caused by germline mutations in the TWIST1 gene or more rarely in the FGFR2 or FGFR3 genes. We have previously reported that patients with Saethre-Chotzen syndrome have an increased risk of developing breast cancer. Here we have analysed a cohort of 26 women with BRCA1/2-negative hereditary breast cancer to study whether a proportion of these families might have mutations in Saethre-Chotzen-associated genes. DNA sequence analysis of TWIST1 showed no pathogenic mutations in the coding sequence in any of the 26 patients. MLPA (multiplex ligation-dependent probe amplification)-analysis also showed no alterations in copy numbers in any of the craniofacial disorder genes MSX2, ALX4, RUNX2, EFNB1, TWIST1, FGFR1, FGFR2,FGFR3, or FGFR4. Taken together, our findings indicate that mutations in Saethre-Chotzen-associated genes are uncommon or absent in BRCA1/2-negative patients with hereditary breast cancer.
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| 37. |
- Cassuto, Jean, et al.
(author)
-
Adrenoceptor subtypes in the control of burn-induced plasma extravasation.
- 2005
-
In: Burns : journal of the International Society for Burn Injuries. - : Elsevier BV. - 0305-4179. ; 31:2, s. 123-9
-
Journal article (peer-reviewed)abstract
- Burn trauma is known to induce a significant rise in circulating catecholamine levels and despite catecholamines being potent endogenous vasoactive agents with known actions on microvascular permeability, their effect on burn edema has been poorly investigated. The present study in rats investigated the role and importance of adrenergic receptor subtypes in the regulation of basal capillary permeability in normal skin and hyperpermeability in partial- and full-thickness skin burns. Edema was quantified by spectrophotometric analysis of extravasated Evans blue-albumin. Evaluation was based on intravenous administration of the following adrenergic agonists and antagonists: l-phenylephrine (alpha(1)-receptor agonist), prazosin (alpha(1)-receptor antagonist), clonidine (alpha(2)-receptor agonist), yohimbine (alpha(2)-receptor antagonist), prenalterol (beta(1)-receptor agonist), terbutaline (beta(2)-receptor agonist), or propranolol (beta(1)- and beta(2)-receptor antagonist). Results showed increased capillary permeability in normal skin following administration of terbutaline (p<0.01) and yohimbine (p<0.01). In partial-thickness burns, clonidine significantly (p<0.05) reduced edema formation, whereas in full-thickness burns edema was significantly reduced by clonidine (p<0.05) and l-phenylephrine (p<0.01). In conclusion, the inhibition of postburn edema induced by stimulation of alpha(1)-receptors (l-phylephrine) and alpha(2)-receptors (clonidine) could be secondary to increased vascular resistance and reduced tissue perfusion pressure and/or suppressed inflammatory reaction in the burn injury. In the treatment of burn patients, clonidine is particularly interesting since the agent has previously been proven to induce potent analgesia in thermally injured.
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| 38. |
|
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| 39. |
- Cassuto, Jean, et al.
(author)
-
Regulation of postburn ischemia by alpha- and beta-adrenoceptor subtypes.
- 2005
-
In: Burns : journal of the International Society for Burn Injuries. - : Elsevier BV. - 0305-4179. ; 31:2, s. 131-7
-
Journal article (peer-reviewed)abstract
- Deep skin burns are characterised by progressive ischemia secondary to vasoconstriction and thrombosis formation. Burn trauma elicits increased sympathetic activity and elevation of circulating catecholamines acting on adrenoceptors in vascular tissue playing an important role in the regulation of organ blood flow. The present study in rats investigated the role of alpha- and beta-adrenoceptors in the circulatory changes taking place in normal skin and in partial- and full-thickness skin burns using laser Doppler flowmetry. Evaluation was based on intravenous administration of the following adrenergic agonists and antagonists: l-phenylephrine (alpha(1)-agonist), prazosin (alpha(1)-antagonist), clonidine (alpha(2)-agonist), yohimbine (alpha(2)-antagonist), prenalterol (beta(1)-agonist), terbutaline (beta(2)-agonist), and propranolol (beta(1)- and beta(2)-antagonist). Blood flow in normal skin was reduced by phenylephrine (p<0.001), clonidine (p<0.001) and propranolol (p<0.01), and increased by prazosin (p<0.05), yohimbine (p<0.05), prenalterol (p<0.05) and terbutaline (p<0.01). In partial-thickness burns, blood flow was reduced by phenylephrine (p<0.01), clonidine (p<0.01) and propranolol (p<0.05). In full-thickness burns, only clonidine reduced perfusion (p<0.05). In conclusion, beta(1)- and beta(2)-adrenoceptors play important role in the physiological regulation of skin perfusion but are of lesser importance for postburn skin perfusion. Vasoconstrictive alpha(1)- and alpha(2)-adrenoceptors were shown to be tonically active in normal skin and in partial-thickness burns, exerting a negative effect on skin perfusion which was further potentiated by exogenous administration of alpha(1)- and alpha(2)-agonists and reversed by selective alpha-blockers. In full-thickness burns, activation of alpha(2)-receptors was shown to significantly impair skin circulation, raising a flag of warning for the use of clonidine to treat pain in burn patients.
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| 40. |
- Cassuto, Jean, et al.
(author)
-
The discotheque fire in Gothenburg 1998. A tragedy among teenagers.
- 2003
-
In: Burns : journal of the International Society for Burn Injuries. - 0305-4179. ; 29:5, s. 405-16
-
Journal article (peer-reviewed)abstract
- The fire disaster in Gothenburg, Sweden, 1998 killing 63 and wounding 213 teenagers was caused by arson committed by a youth from the same community. The fire was started in the basement of an overcrowded discotheque and made, due to unfortunate circumstances, devastating progress. The ensuing rescue work performed by other youth, fire fighters, police and medical staff was prompt and must be seen in the light of a very difficult situation. As a result of these orchestrated efforts and the fact that this disaster occurred in a major city with substantial resources, all the injured were able to be hospitalized within 2h. The load on four local hospitals was initially severe due to the large number of injured and the limited number of staff on night duty. The situation was contained by relocating patients from the intensive care units to ordinary wards and by transporting several of the most severe burn injuries by helicopter to burn units in other parts of Sweden and to Norway. Hundreds of relatives and friends gathered at the local hospitals. This was a new experience for the hospitals and staff, involving many positive aspects as well as some negative aspects such as violence, threats and rumors. As a result of the large number of injuries vast psychosocial rehabilitation program was initiated by health care staff, religious communions, schools and the community, has continued over the past years. Such a disaster emphasises a requirement for extensive preparation not only in the rescue and medical services, but also in the ways and areas to rehabilitate patients in society.
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| 41. |
- Fogdestam, Ingemar, 1941, et al.
(author)
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Extended free lateral arm flap with preservation of the posterior cutaneous nerve of the forearm.
- 1996
-
In: Scandinavian journal of plastic and reconstructive surgery and hand surgery / Nordisk plastikkirurgisk forening [and] Nordisk klubb for handkirurgi. - 0284-4311. ; 30:1, s. 49-55
-
Journal article (peer-reviewed)abstract
- The free lateral arm flap has become a well-defined and reliable flap for various reconstructive purposes. Little attention has been paid, however, to the possibility of preservation of the posterior cutaneous nerve of the forearm (nervus cutaneus antebrachii posterior) sacrifice of which results in numbness of the dorsal part of the forearm. In this study, an anatomical dissection showed that in many cases it would be possible to preserve the nerve. We did 23 free lateral arm flaps in 22 patients during the period 1989-1994. The maximum flap length was 40 cm. Standard maximum width in most cases was 6 cm, and by using a new expansion technique it reached 10 cm in one case. Furthermore, with meticulous dissection the posterior cutaneous nerve of the forearm was either preserved or cut and rejoined in 21 patients, so minimising sensory loss at the donor site.
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| 42. |
- Jönsson, Anders, 1959, et al.
(author)
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Effects of amide local anaesthetics on eicosanoid formation in burned skin.
- 1999
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In: Acta anaesthesiologica Scandinavica. - 0001-5172. ; 43:6, s. 618-22
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Journal article (peer-reviewed)abstract
- Previous studies have demonstrated potent inhibition of burn oedema and progressive ischaemia by local anaesthetics. Since eicosanoids have been suggested to play an important role in the pathophysiology of burns, we compared in the present ex vivo study the effects of topical lidocaine/prilocaine cream (EMLA, ASTRA, Sweden) and intravenous lidocaine with that of saline on eicosanoid formation by normal and burned rat skin.
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| 43. |
- Jönsson, Anders, 1959, et al.
(author)
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Local anesthetics improve dermal perfusion after burn injury.
- 1998
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In: The Journal of burn care & rehabilitation. - 0273-8481. ; 19:1 Pt 1, s. 50-6
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Journal article (peer-reviewed)abstract
- Deep partial-thickness burn injury was induced in the abdominal skin of anesthetized rats. Dermal perfusion was assessed by laser Doppler flowmetry. In the first set of experiments, one group of rats (n = 15) was topically treated with a lidocaine-prilocaine cream 5% (25 mg of each in 1 g) for 6 hours, starting 5 minutes after inducing the burn injury. In one control group (n = 14), the thermal injury was treated with placebo cream. Results showed a markedly reduced perfusion in the skin of the control animals within the first hour after burn injury, with further decrease during the following 5 hours of observation. In animals treated with the lidocaine-prilocaine cream, skin perfusion in the burned area was significantly increased during the first 30 minutes after the burn injury compared to before the burn (p < 0.01), followed by a decrease to a level below the preburn stage but significantly higher than that of control animals during the first hour after burn injury (p < 0.05). As opposed to burned control animals, skin perfusion gradually recovered toward preburn levels at the end of the experiment in local anesthetic-treated animals. In the second experimental set, four groups of animals were burned and subsequently treated with a bolus dose of lidocaine intravenously (2 mg/kg), followed by continuous intravenous lidocaine infusions at a rate of 50 (n = 10), 100 (n = 11), or 150 (n = 10) micrograms.kg-1.min-1. The infusions were started 5 minutes after the burn injury and lasted for 6 hours. Corresponding volumes of saline solution were given to burned control animals (n = 10). Results showed a significantly improved skin perfusion in the lidocaine-treated group in a dose-response fashion as compared to control animals. A maximum improvement of dermal perfusion in the burned area was induced by intravenous lidocaine at an infusion rate of 150 micrograms.kg-1.min-1 as compared to burned controls treated with isotonic saline solution infusions (p < 0.01). Results showed that topical or systemic administration of local anesthetics can prevent progressive dermal ischemia after thermal injury.
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| 44. |
- Jönsson, Anders, 1959, et al.
(author)
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Topical local anaesthetics (EMLA) inhibit burn-induced plasma extravasation as measured by digital image colour analysis.
- 1998
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In: Burns : journal of the International Society for Burn Injuries. - 0305-4179. ; 24:4, s. 313-8
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Journal article (peer-reviewed)abstract
- Amide local anaesthetics have previously been shown to reduce oedema and improve dermal perfusion following experimental burns. Previous studies have used invasive techniques for burn oedema quantification which do not allow continuous monitoring in the same animal. The present study used digital image colour analysis to investigate the effect of topical local anaesthetics on burn-induced extravasation of Evans blue albumin. A standardised full-thickness burn injury (1 x 1 cm) was induced in the abdominal skin of anaesthetised rats. The burn area was subsequently covered with 0.5 g of lidocaine-prilocaine cream 5% (25 mg of each in 1 g; EMLA, ASTRA, Sweden) or placebo cream during the first hour post-burn. One hour after the burn trauma, animals received Evans blue dye intravenously. Skin colour appearances were recorded by macrophotography before the burn and 5, 60. 65, 90, 120, 150, and 180 min post-burn. Colour slides were digitised and colour changes were analysed using the normalised red-green-blue (n-rgb) colour system. Results showed a significant inhibition of Evans blue extravasation between 60 and 180 min post-burn in EMLA-treated animals versus controls. Topical local anaesthetics are potent inhibitors of burn-induced plasma albumin extravasation, probably by direct action on vascular permeability and by inhibition of various steps of the pathophysiological response after burn injury.
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| 45. |
- Lahti, A-M, 1959, et al.
(author)
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Effect of alpha-trinositol on secretion induced by Escherichia coli ST-toxin in rat jejunum.
- 2003
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In: Acta physiologica Scandinavica. - : Wiley. - 0001-6772. ; 179:4, s. 373-9
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Journal article (peer-reviewed)abstract
- d-myo-inositol-1,2,6-trisphosphate (alpha-trinositol, PP56), is a synthetic isomer of the intracellular second messenger, d-myo-inositol-1,4,5-trisphospahate. The pharmacological actions of alpha-trinositol include potent anti-inflammatory properties and inhibition of the secretion induced by cholera toxin and obstructive ileus. In the present study, we investigated whether alpha-trinositol was able to influence the secretion induced by heat-stable ST-toxin from Escherichia coli in the rat jejunum.
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| 46. |
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| 47. |
- Lauritzen, Claes G, et al.
(author)
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Craniofacial surgery over 30 years in Göteborg.
- 2003
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In: Scandinavian journal of surgery : SJS : official organ for the Finnish Surgical Society and the Scandinavian Surgical Society. - 1457-4969. ; 92:4, s. 274-80
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Journal article (peer-reviewed)abstract
- Craniofacial surgery is a new sub-specialty in the field of plastic reconstructive surgery and is dedicated to the treatment of severe cranial and facial malformations. Craniofacial surgery gradually started in Göteborg in the late 1970's and has been acting as the Scandinavian center since 1983. Over these 30 years an almost complete change in surgical techniques has evolved. Also profound changes in timing of surgery have followed. Results have been dramatically improved based on critical evaluation of standardized registration of long-term results. One of the most dramatic developments has been the introduction of implantable stainless steel springs. This has changed the treatment of Craniosynostosis completely and has made midfacial advancement procedures possible without relapse.
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| 48. |
- Lauritzen, Claes G, et al.
(author)
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Reconstruction of eyelid defects: a prefabricated multilayer sandwich graft.
- 1999
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In: Scandinavian journal of plastic and reconstructive surgery and hand surgery / Nordisk plastikkirurgisk forening [and] Nordisk klubb for handkirurgi. - 0284-4311. ; 33:1, s. 99-104
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Journal article (peer-reviewed)abstract
- We have developed a new technique for reconstruction of full thickness eyelid defects. A composite three-layer skin-cartilage-mucosal unit is manufactured and tailored three-dimensionally. Use of this sandwich graft in our patients proved to be simple and safe for eyelid reconstruction and has given good results without complications in four patients.
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| 49. |
- Lindblom, Lucky, 1947, et al.
(author)
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Importance of nitric oxide in the regulation of burn oedema, proteinuria and urine output.
- 2000
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In: Burns : journal of the International Society for Burn Injuries. - 0305-4179. ; 26:1, s. 13-7
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Journal article (peer-reviewed)abstract
- Burn injuries trigger a pronounced inflammatory response in the burned skin, resulting in oedema formation and impaired circulation. This response involves activation of the nitric oxide (NO) synthetic pathway, which could play a key role in the complex hemodynamic and hemostatic changes occurring as a result of a burn trauma. The results presented in full-thickness skin burns of rats show that the NO-precursor, L-arginine (n = 10), inhibit burn-induced plasma extravasation as compared to saline-treated burned controls (n = 10) (p<0.001) to a level not significantly different from nonburned animals. Administration of the NO-synthase inhibitor. NG-nitro-L-arginine (L-NNA) (n = 10), did not significantly influence burn extravasation compared to burned controls. Accumulated urine volume 90 min post-burn increased ten-fold in burned animals treated with L-arginine compared to saline-treated burned controls (p<0.001) and nonburned animals (p<0.001), while L-NNA had no significant effect on diuresis. A significantly increased proteinuria occurred in L-arginine treated burned animals as compared to burned controls and nonburned controls (p<0.001), whereas L-NNA did not significantly influence the leakage of protein in the urine. Activation of NO synthesis significantly suppresses burn edema and strongly increases diuresis along with increased proteinuria.
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| 50. |
- Lindblom, Lucky, 1947, et al.
(author)
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Importance of vasoactive intestinal polypeptide in the regulation of burn perfusion.
- 2000
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In: Burns : journal of the International Society for Burn Injuries. - 0305-4179. ; 26:5, s. 435-42
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Journal article (peer-reviewed)abstract
- Vasoactive intestinal polypeptide is one of the body's most potent vasodilators and has been shown to increase blood flow in a number of tissues. Its effects on postburn skin perfusion and progressive ischemia was investigated in rats exposed to partial- and full-thickness experimental skin burns. Systemic administration of VIP elicited a significant drop in mean arterial blood pressure versus saline (p<0.001) and VIP antiserum (p<0.001) both in burned and nonburned animals. VIP also decreased heart rate versus saline (p<0.05) and anti-VIP (p<0.001) in nonburned and burned animals. In contrast, VIP antiserum significantly increased blood pressure (p<0. 001) and heart rate (p<0.001) versus saline in all the groups. Skin perfusion in normal skin was significantly impaired by VIP infusions as compared to saline (p<0.01) while VIP-antiserum did not differ significantly from saline. Similarly, VIP significantly reduced blood flow versus saline-treatment in partial-thickness (p<0.01) and full-thickness burns (p<0.05) while VIP-antiserum had no significant effect on skin perfusion in any of the burned groups as compared to saline treatment. The present results show that VIP is directly involved in general cardiovascular control but plays a minor role in the maintenance of skin perfusion following a thermal injury as suggested by the lack of effect of VIP-antiserum. In contrast, exogenous administration of VIP significantly and dramatically impaired skin perfusion in normal and burned skin probably by increasing blood flow in organs of higher priority such as the brain and heart and concomitantly inducing a pronounced vasoconstriction in the skin, probably as a result of increased sympathetic effect on peripheral organs in order to maintain blood pressure.
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