Pub Date : 2024-06-21Print Date: 2024-09-01DOI: 10.3171/2024.5.PEDS24115
Andrew T Timberlake, Kshipra Hemal, Jonas A Gustafson, Le Thi Hao, Irene Valenzuela, Anne Slavotinek, Michael L Cunningham, Kristopher T Kahle, Richard P Lifton, John A Persing
Objective: Occurring once in every 2000 live births, craniosynostosis (CS) is the most frequent cranial birth defect. Although the genetic etiologies of syndromic CS cases are well defined, the genetic cause of most nonsyndromic cases remains unknown.
Methods: The authors analyzed exome or RNA sequencing data from 876 children with nonsyndromic CS, including 291 case-parent trios and 585 additional probands. The authors also utilized the GeneMatcher platform and the Gabriella Miller Kids First genome sequencing project to identify additional CS patients with AXIN1 mutations.
Results: The authors describe 11 patients with nonsyndromic CS harboring rare, damaging mutations in AXIN1, an inhibitor of Wnt signaling. AXIN1 regulates signaling upstream of key mediators of osteoblast differentiation. Three of the 6 mutations identified in trios occurred de novo in the proband, while 3 were transmitted from unaffected parents. Patients with nonsyndromic CS were highly enriched for mutations in AXIN1 compared to both expectation (p = 0.0008) and exome sequencing data from > 76,000 healthy controls (p = 2.3 × 10-6), surpassing the thresholds for genome-wide significance.
Conclusions: These findings describe the first phenotype associated with mutations in AXIN1, with mutations identified in approximately 1% of nonsyndromic CS cases. The results strengthen the existing link between Wnt signaling and maintenance of cranial suture patency and have implications for genetic testing in families with CS.
{"title":"AXIN1 mutations in nonsyndromic craniosynostosis.","authors":"Andrew T Timberlake, Kshipra Hemal, Jonas A Gustafson, Le Thi Hao, Irene Valenzuela, Anne Slavotinek, Michael L Cunningham, Kristopher T Kahle, Richard P Lifton, John A Persing","doi":"10.3171/2024.5.PEDS24115","DOIUrl":"10.3171/2024.5.PEDS24115","url":null,"abstract":"<p><strong>Objective: </strong>Occurring once in every 2000 live births, craniosynostosis (CS) is the most frequent cranial birth defect. Although the genetic etiologies of syndromic CS cases are well defined, the genetic cause of most nonsyndromic cases remains unknown.</p><p><strong>Methods: </strong>The authors analyzed exome or RNA sequencing data from 876 children with nonsyndromic CS, including 291 case-parent trios and 585 additional probands. The authors also utilized the GeneMatcher platform and the Gabriella Miller Kids First genome sequencing project to identify additional CS patients with AXIN1 mutations.</p><p><strong>Results: </strong>The authors describe 11 patients with nonsyndromic CS harboring rare, damaging mutations in AXIN1, an inhibitor of Wnt signaling. AXIN1 regulates signaling upstream of key mediators of osteoblast differentiation. Three of the 6 mutations identified in trios occurred de novo in the proband, while 3 were transmitted from unaffected parents. Patients with nonsyndromic CS were highly enriched for mutations in AXIN1 compared to both expectation (p = 0.0008) and exome sequencing data from > 76,000 healthy controls (p = 2.3 × 10-6), surpassing the thresholds for genome-wide significance.</p><p><strong>Conclusions: </strong>These findings describe the first phenotype associated with mutations in AXIN1, with mutations identified in approximately 1% of nonsyndromic CS cases. The results strengthen the existing link between Wnt signaling and maintenance of cranial suture patency and have implications for genetic testing in families with CS.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-06-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11200303/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141437031","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-06-21Print Date: 2024-09-01DOI: 10.3171/2024.4.PEDS23310
Nallammai Muthiah, Hope M Reecher, Taylor J Abel
Objective: Epilepsy affects approximately 470,000 children in the United States. The estimated median incidence is 50.4 cases per 100,000 persons per year. There are approximately 3.1 million seizure-related emergency department (ED) visits per year among children. Vagus nerve stimulation (VNS) is a treatment option for drug-resistant epilepsy (DRE). While its primary goal is to decrease seizure burden, VNS may decrease seizure intensity and improve quality of life. The authors assessed whether VNS decreased the number of seizure-related ED visits in a cohort of children with DRE.
Methods: The authors performed a retrospective chart review of pediatric patients (aged 0-21 years) who underwent implantation of a vagus nerve stimulator between January 2009 and January 2020 at the University of Pittsburgh Medical Center Children's Hospital of Pittsburgh. They used paired t-tests to assess differences in the number of ED visits 2 years before versus 2 years after VNS device implantation. Univariable linear regression analyses were used to test associations of preoperative characteristics with change in the number of ED visits following vagus nerve stimulator insertion.
Results: This study included 240 patients. Compared with patients without seizure-related ED visits before VNS, patients with ≥ 1 ED visits were younger in age at first VNS surgery (9.5 vs 10.8 years), had a shorter epilepsy duration before VNS surgery (5.8 vs 7.4 years), had a later year of device implantation (2014 vs 2012), and on average took more antiseizure medications (ASMs; 2.4 vs 2.1). There was no significant difference between the total number of seizure-related ED visits pre- versus post-VNS surgery (1.72 vs 1.59, p = 0.50), and no difference in status epilepticus-related visits (0.59 vs 0.46, p = 0.17). Univariable linear regression analyses revealed a mean change in ED visits of +0.3 for each year prior to 2022 and -0.5 for each additional ASM that patients took before vagus nerve stimulator insertion.
Conclusions: This single-institution analysis demonstrated no significant change in the number of seizure-related ED visits within 2 years following VNS device implantation. Earlier VNS surgery was associated with more seizure-related ED visits after device insertion, suggesting that medical management and center experience may play a role in decreasing seizure-related ED visits. A greater number of ASMs was associated with fewer seizure-related ED visits after VNS device insertion, suggesting the role of medical management, patient baseline seizure threshold, and caregiver comfort with at-home seizure management.
{"title":"Effect of vagus nerve stimulation on emergency department utilization in children with drug-resistant epilepsy: a retrospective cohort study.","authors":"Nallammai Muthiah, Hope M Reecher, Taylor J Abel","doi":"10.3171/2024.4.PEDS23310","DOIUrl":"10.3171/2024.4.PEDS23310","url":null,"abstract":"<p><strong>Objective: </strong>Epilepsy affects approximately 470,000 children in the United States. The estimated median incidence is 50.4 cases per 100,000 persons per year. There are approximately 3.1 million seizure-related emergency department (ED) visits per year among children. Vagus nerve stimulation (VNS) is a treatment option for drug-resistant epilepsy (DRE). While its primary goal is to decrease seizure burden, VNS may decrease seizure intensity and improve quality of life. The authors assessed whether VNS decreased the number of seizure-related ED visits in a cohort of children with DRE.</p><p><strong>Methods: </strong>The authors performed a retrospective chart review of pediatric patients (aged 0-21 years) who underwent implantation of a vagus nerve stimulator between January 2009 and January 2020 at the University of Pittsburgh Medical Center Children's Hospital of Pittsburgh. They used paired t-tests to assess differences in the number of ED visits 2 years before versus 2 years after VNS device implantation. Univariable linear regression analyses were used to test associations of preoperative characteristics with change in the number of ED visits following vagus nerve stimulator insertion.</p><p><strong>Results: </strong>This study included 240 patients. Compared with patients without seizure-related ED visits before VNS, patients with ≥ 1 ED visits were younger in age at first VNS surgery (9.5 vs 10.8 years), had a shorter epilepsy duration before VNS surgery (5.8 vs 7.4 years), had a later year of device implantation (2014 vs 2012), and on average took more antiseizure medications (ASMs; 2.4 vs 2.1). There was no significant difference between the total number of seizure-related ED visits pre- versus post-VNS surgery (1.72 vs 1.59, p = 0.50), and no difference in status epilepticus-related visits (0.59 vs 0.46, p = 0.17). Univariable linear regression analyses revealed a mean change in ED visits of +0.3 for each year prior to 2022 and -0.5 for each additional ASM that patients took before vagus nerve stimulator insertion.</p><p><strong>Conclusions: </strong>This single-institution analysis demonstrated no significant change in the number of seizure-related ED visits within 2 years following VNS device implantation. Earlier VNS surgery was associated with more seizure-related ED visits after device insertion, suggesting that medical management and center experience may play a role in decreasing seizure-related ED visits. A greater number of ASMs was associated with fewer seizure-related ED visits after VNS device insertion, suggesting the role of medical management, patient baseline seizure threshold, and caregiver comfort with at-home seizure management.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-06-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141437032","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-06-21Print Date: 2024-09-01DOI: 10.3171/2024.5.PEDS24171
Justine Izah, Joseline Haizel-Cobbina, Shilin Zhao, E Haley Vance, Michelle Dunlap, Stephen R Gannon, Campbell Liles, Aaron M Yengo-Kahn, Matthew E Pontell, Robert P Naftel, John C Wellons, Michael C Dewan
Objective: The aim of this study was to compare clinical and craniometric outcomes of patients treated for hydrocephalus following fetal myelomeningocele repair (fMMR) via a ventriculoperitoneal shunt (VPS) or endoscopic third ventriculostomy with choroid plexus cauterization (ETV/CPC).
Methods: This was a retrospective cohort study of children who were treated for hydrocephalus following fMMR via VPS or ETV with or without CPC (ETV ± CPC) at Vanderbilt between 2012 and 2021. The primary outcomes were treatment failure and time to failure (TTF). Secondary outcomes included changes in hydrocephalus metrics (fronto-occipital horn ratio [FOHR] and head circumference measurements) and healthcare resource utilization (number of hospital admissions, clinic visits, and neuroimaging findings).
Results: Among 88 patients who underwent fMMR, 37 (42%) required permanent CSF diversion, of whom 19 received treatment at the authors' institution. Twelve patients underwent ETV ± CPC, and 7 underwent VPS placement at a median corrected age of 23 weeks versus 1 week (p = 0.002). The preoperative median head circumference percentiles and z-scores for patients in the ETV ± CPC cohort were similar to those of the VPS cohort (percentiles: 98.5 vs 94.0, p = 0.064; z-scores: 2.32 vs 1.60, p = 0.111). There was no difference in preoperative median FOHR measurements between the two cohorts (0.57 vs 0.59, p = 0.53). At 6 months postoperatively, the median head circumference percentile and z-score for the ETV ± CPC cohort remained similar between the two cohorts (percentiles: 98.0 vs 67.5, p = 0.315; z-scores: 2.12 vs 0.52, p = 0.307). There was no difference in the change in FOHR (-0.06 vs -0.09, p = 0.37) and change in head circumference percentile (-1.33 vs -28.6, p = 0.058) between the cohorts 6 months after the index CSF diversion procedure. One patient in the ETV ± CPC cohort experienced a seizure and a nonoperative subdural hemorrhage postoperatively; no other complications were observed. Six of the 7 patients in the VPS cohort required shunt revision with a median TTF of 9.8 months while 2 of the 12 ETV ± CPC patients required a repeat ETV at a median of 17.5 months (86% vs 17%, p = 0.013). The median number of hydrocephalus-related hospital readmissions was significantly lower in the ETV ± CPC cohort than in the VPS cohort (0 vs 1, p = 0.006). The ETV ± CPC cohort had fewer CT scans (0 vs 2, p = 0.004) and radiographs (0 vs 2, p < 0.001) than the VPS cohort.
Conclusions: In a single-center cohort, hydrocephalic fMMR patients treated via ETV ± CPC remained shunt free, while a majority of patients receiving an upfront shunt required revision. This is the first study comparing ETV ± CPC with VPS in the fMMR hydrocephalus population. While larger, multicenter studies are needed, these results suggest that ETV/CPC may be a preferred means of CSF diversion following fMMR.
研究目的本研究旨在比较通过脑室腹腔分流术(VPS)或内镜下第三脑室造口术联合脉络丛烧灼术(ETV/CPC)进行胎儿脊髓膜膨出修补术(fMMR)后脑积水患者的临床和头颅测量结果:这是一项回顾性队列研究,研究对象是2012年至2021年期间在范德比尔特接受FMMR治疗后,通过VPS或ETV(带或不带CPC)(ETV ± CPC)进行脑积水治疗的儿童。主要结果是治疗失败和治疗失败时间 (TTF)。次要结果包括脑积水指标的变化(前枕骨角比 [FOHR] 和头围测量值)和医疗资源利用率(入院次数、门诊次数和神经影像学检查结果):在 88 名接受 fMMR 的患者中,37 人(42%)需要永久性 CSF 转移,其中 19 人在作者所在机构接受了治疗。12名患者接受了ETV±CPC治疗,7名患者接受了VPS置入治疗,中位矫正年龄分别为23周和1周(P = 0.002)。ETV ± CPC队列患者的术前中位头围百分位数和z-分数与VPS队列相似(百分位数:98.5 vs 94.0,p = 0.064;z-分数:2.32 vs 1.60,p = 0.111)。两组患者术前的中位 FOHR 测量值没有差异(0.57 vs 0.59,p = 0.53)。术后 6 个月,ETV ± CPC 组群的中位头部周长百分位数和 Z 值在两组之间保持相似(百分位数:98.0 vs 67.5,p = 0.315;Z 值:2.12 vs 0.52,p = 0.307)。在指数 CSF 转移术后 6 个月,两组患者的 FOHR 变化(-0.06 vs -0.09,p = 0.37)和头围百分位数变化(-1.33 vs -28.6,p = 0.058)没有差异。ETV ± CPC组别中有一名患者术后出现癫痫发作和非手术硬膜下出血,未观察到其他并发症。VPS 组群的 7 名患者中有 6 名需要进行分流术翻修,中位 TTF 为 9.8 个月,而 12 名 ETV ± CPC 患者中有 2 名需要再次进行 ETV,中位时间为 17.5 个月(86% vs 17%,P = 0.013)。ETV ± CPC队列中与脑积水相关的再住院中位数明显低于VPS队列(0 vs 1,p = 0.006)。ETV ± CPC队列的CT扫描次数(0 vs 2,p = 0.004)和X光检查次数(0 vs 2,p < 0.001)均少于VPS队列:结论:在单中心队列中,通过 ETV ± CPC 治疗的颅脑积水 fMMR 患者仍无分流,而大多数接受前期分流的患者需要进行翻修。这是第一项在 fMMR 脑积水患者中比较 ETV ± CPC 与 VPS 的研究。虽然还需要更大规模的多中心研究,但这些结果表明,ETV/CPC 可能是 fMMR 后 CSF 分流的首选方法。
{"title":"Treatment of hydrocephalus following fetal repair of myelomeningocele: comparing endoscopic third ventriculostomy with choroid plexus cauterization to ventricular shunting.","authors":"Justine Izah, Joseline Haizel-Cobbina, Shilin Zhao, E Haley Vance, Michelle Dunlap, Stephen R Gannon, Campbell Liles, Aaron M Yengo-Kahn, Matthew E Pontell, Robert P Naftel, John C Wellons, Michael C Dewan","doi":"10.3171/2024.5.PEDS24171","DOIUrl":"10.3171/2024.5.PEDS24171","url":null,"abstract":"<p><strong>Objective: </strong>The aim of this study was to compare clinical and craniometric outcomes of patients treated for hydrocephalus following fetal myelomeningocele repair (fMMR) via a ventriculoperitoneal shunt (VPS) or endoscopic third ventriculostomy with choroid plexus cauterization (ETV/CPC).</p><p><strong>Methods: </strong>This was a retrospective cohort study of children who were treated for hydrocephalus following fMMR via VPS or ETV with or without CPC (ETV ± CPC) at Vanderbilt between 2012 and 2021. The primary outcomes were treatment failure and time to failure (TTF). Secondary outcomes included changes in hydrocephalus metrics (fronto-occipital horn ratio [FOHR] and head circumference measurements) and healthcare resource utilization (number of hospital admissions, clinic visits, and neuroimaging findings).</p><p><strong>Results: </strong>Among 88 patients who underwent fMMR, 37 (42%) required permanent CSF diversion, of whom 19 received treatment at the authors' institution. Twelve patients underwent ETV ± CPC, and 7 underwent VPS placement at a median corrected age of 23 weeks versus 1 week (p = 0.002). The preoperative median head circumference percentiles and z-scores for patients in the ETV ± CPC cohort were similar to those of the VPS cohort (percentiles: 98.5 vs 94.0, p = 0.064; z-scores: 2.32 vs 1.60, p = 0.111). There was no difference in preoperative median FOHR measurements between the two cohorts (0.57 vs 0.59, p = 0.53). At 6 months postoperatively, the median head circumference percentile and z-score for the ETV ± CPC cohort remained similar between the two cohorts (percentiles: 98.0 vs 67.5, p = 0.315; z-scores: 2.12 vs 0.52, p = 0.307). There was no difference in the change in FOHR (-0.06 vs -0.09, p = 0.37) and change in head circumference percentile (-1.33 vs -28.6, p = 0.058) between the cohorts 6 months after the index CSF diversion procedure. One patient in the ETV ± CPC cohort experienced a seizure and a nonoperative subdural hemorrhage postoperatively; no other complications were observed. Six of the 7 patients in the VPS cohort required shunt revision with a median TTF of 9.8 months while 2 of the 12 ETV ± CPC patients required a repeat ETV at a median of 17.5 months (86% vs 17%, p = 0.013). The median number of hydrocephalus-related hospital readmissions was significantly lower in the ETV ± CPC cohort than in the VPS cohort (0 vs 1, p = 0.006). The ETV ± CPC cohort had fewer CT scans (0 vs 2, p = 0.004) and radiographs (0 vs 2, p < 0.001) than the VPS cohort.</p><p><strong>Conclusions: </strong>In a single-center cohort, hydrocephalic fMMR patients treated via ETV ± CPC remained shunt free, while a majority of patients receiving an upfront shunt required revision. This is the first study comparing ETV ± CPC with VPS in the fMMR hydrocephalus population. While larger, multicenter studies are needed, these results suggest that ETV/CPC may be a preferred means of CSF diversion following fMMR.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-06-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141437034","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-06-14Print Date: 2024-09-01DOI: 10.3171/2024.4.PEDS2432
Nisha Gadgil, Aloysia L Schwabe, Edward Wright, Amy Barbuto, Eric L Dugan, Sruthi P Thomas, Jeffrey S Shilt, Dorothy Beauvais, Yushane Shih, Brian G Smith, David F Bauer, Daniel J Curry
Objective: Selective dorsal rhizotomy (SDR) is a neurosurgical procedure to reduce spasticity in children with cerebral palsy and spastic diplegia. The authors developed a procedure called focal SDR for children with spasticity predominantly in the L5 or S1 motor distribution, which can be combined with orthopedic correction of fixed soft-tissue or bony deformity. The authors describe in detail the technique of minimally invasive focal SDR and propose selection criteria.
Methods: The authors conducted a retrospective study of patients who underwent focal SDR at their institution and underwent baseline and 1-year postoperative 3D gait analysis. Modified Ashworth scale (MAS) and Gait Deviation Index (GDI) scores were the primary outcome measures.
Results: Ten patients met the study criteria, all with an underlying diagnosis of cerebral palsy. All underwent focal SDR at the unilateral or bilateral S1 level, and 4 additionally underwent focal SDR at the L5 level unilaterally or bilaterally. All but 1 patient underwent concurrent orthopedic surgery. The improvement in spasticity of the plantar flexors, as measured by the MAS score, was 2.2 (p < 0.001). In the patients who underwent L5 focal SDR, there was an improvement in the hamstring MAS score of 1.4 (p = 0.004). The mean improvement in the GDI score following focal SDR was 11 (range -6 to 29, p < 0.001).
Conclusions: Focally impairing spasticity in the gastrocsoleus complex and/or hamstrings muscle group in the setting of less functionally impactful proximal tone is extremely common in cerebral palsy. The novel technique of focal SDR, combined with orthopedic intervention, improves spasticity scores and overall gait mechanics. Further investigation is warranted to define the ideal candidacy and outcomes.
{"title":"Focal selective dorsal rhizotomy and concurrent deformity correction: a combined approach.","authors":"Nisha Gadgil, Aloysia L Schwabe, Edward Wright, Amy Barbuto, Eric L Dugan, Sruthi P Thomas, Jeffrey S Shilt, Dorothy Beauvais, Yushane Shih, Brian G Smith, David F Bauer, Daniel J Curry","doi":"10.3171/2024.4.PEDS2432","DOIUrl":"10.3171/2024.4.PEDS2432","url":null,"abstract":"<p><strong>Objective: </strong>Selective dorsal rhizotomy (SDR) is a neurosurgical procedure to reduce spasticity in children with cerebral palsy and spastic diplegia. The authors developed a procedure called focal SDR for children with spasticity predominantly in the L5 or S1 motor distribution, which can be combined with orthopedic correction of fixed soft-tissue or bony deformity. The authors describe in detail the technique of minimally invasive focal SDR and propose selection criteria.</p><p><strong>Methods: </strong>The authors conducted a retrospective study of patients who underwent focal SDR at their institution and underwent baseline and 1-year postoperative 3D gait analysis. Modified Ashworth scale (MAS) and Gait Deviation Index (GDI) scores were the primary outcome measures.</p><p><strong>Results: </strong>Ten patients met the study criteria, all with an underlying diagnosis of cerebral palsy. All underwent focal SDR at the unilateral or bilateral S1 level, and 4 additionally underwent focal SDR at the L5 level unilaterally or bilaterally. All but 1 patient underwent concurrent orthopedic surgery. The improvement in spasticity of the plantar flexors, as measured by the MAS score, was 2.2 (p < 0.001). In the patients who underwent L5 focal SDR, there was an improvement in the hamstring MAS score of 1.4 (p = 0.004). The mean improvement in the GDI score following focal SDR was 11 (range -6 to 29, p < 0.001).</p><p><strong>Conclusions: </strong>Focally impairing spasticity in the gastrocsoleus complex and/or hamstrings muscle group in the setting of less functionally impactful proximal tone is extremely common in cerebral palsy. The novel technique of focal SDR, combined with orthopedic intervention, improves spasticity scores and overall gait mechanics. Further investigation is warranted to define the ideal candidacy and outcomes.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141321014","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-06-14Print Date: 2024-09-01DOI: 10.3171/2024.4.PEDS23517
Uma V Mahajan, Dana Defta, David C Kaelber, Sanjay P Ahuja, Brian D Rothstein, Krystal L Tomei
Objective: During the COVID-19 pandemic, the authors' institution managed ventriculoatrial (VA) shunt complications in 2 teenage patients in close proximity to a symptomatic COVID-19 infection. Systemic thrombotic events are an established complication of COVID-19 infection due to a hypercoagulable state. Thrombotic complications, particularly elevated central venous pressure, can cause VA shunt failure. The true effect of COVID-19 on patients with intravascular devices is currently unknown. In this study, the authors aimed to determine if there was an association between COVID-19 infection and VA shunt failure.
Methods: TriNetX, an aggregated electronic health record platform, was used to analyze data of more than 13 million US pediatric patients. Two matched cohorts of patients < 18 years of age with a VA shunt were defined. Group 1 (n = 311) had a positive laboratory test for COVID-19 from March 1, 2020, to March 31, 2022. Group 2 (n = 311), a control group, had any medical appointment from March 1, 2020, to March 31, 2022, and never had a positive laboratory test for COVID-19. The authors analyzed outcomes 1 year after testing positive for COVID-19 in group 1, and after the medical appointment in group 2. Outcomes included shunt complications, shunt revisions or replacements, and thromboembolic complications. To protect patient privacy, individual results of fewer than 10 patients are not specified in TriNetX.
Results: Group 1 had a greater odds of mechanical shunt complication than group 2 (20% vs 4%, OR 5.71, 95% CI 3.07-10.62). Group 1 had a greater odds of shunt reoperation than group 2 (11% vs < 3%, OR > 3.7, 95% CI 1.72-7.62). There were 1-10 patients in group 1 (≤ 3% of group 1) who experienced a thromboembolism due to the shunt, compared with no patients in group 2 who had a thromboembolism due to the shunt.
Conclusions: This analysis demonstrates an association of shunt complications, reoperations, and thromboembolic events in patients with VA shunts following COVID-19 infection.
{"title":"The association of COVID-19 with increased pediatric ventriculoatrial shunt failures: a national retrospective cohort.","authors":"Uma V Mahajan, Dana Defta, David C Kaelber, Sanjay P Ahuja, Brian D Rothstein, Krystal L Tomei","doi":"10.3171/2024.4.PEDS23517","DOIUrl":"10.3171/2024.4.PEDS23517","url":null,"abstract":"<p><strong>Objective: </strong>During the COVID-19 pandemic, the authors' institution managed ventriculoatrial (VA) shunt complications in 2 teenage patients in close proximity to a symptomatic COVID-19 infection. Systemic thrombotic events are an established complication of COVID-19 infection due to a hypercoagulable state. Thrombotic complications, particularly elevated central venous pressure, can cause VA shunt failure. The true effect of COVID-19 on patients with intravascular devices is currently unknown. In this study, the authors aimed to determine if there was an association between COVID-19 infection and VA shunt failure.</p><p><strong>Methods: </strong>TriNetX, an aggregated electronic health record platform, was used to analyze data of more than 13 million US pediatric patients. Two matched cohorts of patients < 18 years of age with a VA shunt were defined. Group 1 (n = 311) had a positive laboratory test for COVID-19 from March 1, 2020, to March 31, 2022. Group 2 (n = 311), a control group, had any medical appointment from March 1, 2020, to March 31, 2022, and never had a positive laboratory test for COVID-19. The authors analyzed outcomes 1 year after testing positive for COVID-19 in group 1, and after the medical appointment in group 2. Outcomes included shunt complications, shunt revisions or replacements, and thromboembolic complications. To protect patient privacy, individual results of fewer than 10 patients are not specified in TriNetX.</p><p><strong>Results: </strong>Group 1 had a greater odds of mechanical shunt complication than group 2 (20% vs 4%, OR 5.71, 95% CI 3.07-10.62). Group 1 had a greater odds of shunt reoperation than group 2 (11% vs < 3%, OR > 3.7, 95% CI 1.72-7.62). There were 1-10 patients in group 1 (≤ 3% of group 1) who experienced a thromboembolism due to the shunt, compared with no patients in group 2 who had a thromboembolism due to the shunt.</p><p><strong>Conclusions: </strong>This analysis demonstrates an association of shunt complications, reoperations, and thromboembolic events in patients with VA shunts following COVID-19 infection.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141321035","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-06-14Print Date: 2024-09-01DOI: 10.3171/2024.4.PEDS23510
David P Alper, Mariana N Almeida, Kevin G Hu, Jenny F Yang, Andrew T Timberlake, Jinesh Shah, John A Persing, Michael Alperovich
Objective: Previous work identified an association between genetics and neurodevelopmental delays in patients with nonsyndromic craniosynostosis. The authors investigated the role of genetic mutations on behavioral outcomes of patients with treated sagittal synostosis.
Methods: Parents of children aged 6-18 years with surgically corrected sagittal synostosis were recruited to complete the Child Behavioral Checklist (overall behavioral problems), Conners 3rd Edition-Parent (attention-deficit/hyperactivity disorder), Social Responsiveness Scale 2nd Edition (autism spectrum disorder [ASD]), and Behavior Rating Inventory of Executive Function 2nd Edition (executive function). Genomic analysis was completed, and patients were identified if they had mutations in high probability of loss of function intolerant (pLI) genes (high pLI vs nonhigh pLI). Genetic burden was assessed relative to controls. Multivariate linear regression determined the association of mutations in high pLI genes with behavioral scores, while controlling for sociodemographic factors, age at surgery, surgery type, and IQ.
Results: Sixteen of 45 patients were in the high pLI group. There were no differences between the groups in terms of sociodemographic factors. A greater proportion of children in the high pLI group scored at or above borderline clinical levels for aggression (18.8% vs 0.0%, p = 0.05) and externalizing problems (31.3% vs 3.7%, p = 0.02). Among children in the nonhigh pLI group, older age at surgery was associated with worse scores on the rule-breaking, aggression, and externalizing problems domains and four out of five ASD domains.
Conclusions: Children with treated nonsyndromic sagittal synostosis and mutations in high pLI genes had worse behavioral problems in externalizing behaviors and aggression, whereas older age at surgery was a significant predictor of worse behavioral outcomes in patients without mutations in high pLI genes.
目的:以前的研究发现,遗传与非综合征颅突症患者的神经发育迟缓存在关联。作者研究了基因突变对接受治疗的矢状突眼患者行为结果的影响:招募了 6-18 岁矢状突触症手术矫正患儿的家长,让他们填写儿童行为检查表(总体行为问题)、康纳斯第三版-家长(注意力缺陷/多动症)、社交反应量表第二版(自闭症谱系障碍 [ASD])和执行功能行为评级量表第二版(执行功能)。基因组分析已经完成,如果患者的不耐受功能缺失(pLI)基因发生突变(高 pLI 与非高 pLI),则会被识别出来。评估了相对于对照组的基因负担。多变量线性回归确定了高pLI基因突变与行为评分的关系,同时控制了社会人口学因素、手术年龄、手术类型和智商:45名患者中有16名属于高pLI组。结果:45 名患者中有 16 名属于高 pLI 组,两组在社会人口因素方面没有差异。高pLI组中有更多儿童的攻击行为(18.8% vs 0.0%,p = 0.05)和外化问题(31.3% vs 3.7%,p = 0.02)达到或超过临床边缘水平。在非高pLI组的儿童中,手术年龄越大,在破坏规则、攻击性和外化问题领域以及五个ASD领域中的四个领域的得分越低:结论:接受过治疗的非综合征矢状突节和高pLI基因突变的儿童在外化行为和攻击性方面的行为问题更严重,而在没有高pLI基因突变的患者中,手术年龄越大,其行为结果越差,这是一个重要的预测因素。
{"title":"The role of genetics on behavioral outcomes in nonsyndromic sagittal synostosis.","authors":"David P Alper, Mariana N Almeida, Kevin G Hu, Jenny F Yang, Andrew T Timberlake, Jinesh Shah, John A Persing, Michael Alperovich","doi":"10.3171/2024.4.PEDS23510","DOIUrl":"10.3171/2024.4.PEDS23510","url":null,"abstract":"<p><strong>Objective: </strong>Previous work identified an association between genetics and neurodevelopmental delays in patients with nonsyndromic craniosynostosis. The authors investigated the role of genetic mutations on behavioral outcomes of patients with treated sagittal synostosis.</p><p><strong>Methods: </strong>Parents of children aged 6-18 years with surgically corrected sagittal synostosis were recruited to complete the Child Behavioral Checklist (overall behavioral problems), Conners 3rd Edition-Parent (attention-deficit/hyperactivity disorder), Social Responsiveness Scale 2nd Edition (autism spectrum disorder [ASD]), and Behavior Rating Inventory of Executive Function 2nd Edition (executive function). Genomic analysis was completed, and patients were identified if they had mutations in high probability of loss of function intolerant (pLI) genes (high pLI vs nonhigh pLI). Genetic burden was assessed relative to controls. Multivariate linear regression determined the association of mutations in high pLI genes with behavioral scores, while controlling for sociodemographic factors, age at surgery, surgery type, and IQ.</p><p><strong>Results: </strong>Sixteen of 45 patients were in the high pLI group. There were no differences between the groups in terms of sociodemographic factors. A greater proportion of children in the high pLI group scored at or above borderline clinical levels for aggression (18.8% vs 0.0%, p = 0.05) and externalizing problems (31.3% vs 3.7%, p = 0.02). Among children in the nonhigh pLI group, older age at surgery was associated with worse scores on the rule-breaking, aggression, and externalizing problems domains and four out of five ASD domains.</p><p><strong>Conclusions: </strong>Children with treated nonsyndromic sagittal synostosis and mutations in high pLI genes had worse behavioral problems in externalizing behaviors and aggression, whereas older age at surgery was a significant predictor of worse behavioral outcomes in patients without mutations in high pLI genes.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-06-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141321036","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-06-07Print Date: 2024-09-01DOI: 10.3171/2024.4.PEDS23589
Weihong Yuan, Charles B Stevenson, Paolo Moretti, Francesco T Mangano, Marco Pavanello, Armando Cama, Domenico Tortora, Chiara Tacchino, Amy F Bailes, Kelly R Greve, Jilda N Vargus-Adams, Jonathan A Dudley, Karen Harpster, Brad G Kurowski, Alexis Mitelpunkt, Bruce Aronow
Objective: The aims of this study were to 1) assess and quantify white matter (WM) microstructural characteristics derived from diffusion tensor imaging (DTI) in children with cerebral palsy (CP) prior to selective dorsal rhizotomy (SDR), and 2) investigate potential associations between WM diffusion properties and gross motor function and spasticity in children with spastic CP who underwent SDR.
Methods: This study is a multisite study based on DT images acquired prior to SDR as well as postoperative outcome data. DTI data collected from two sites were harmonized using the ComBat approach to minimize intersite scanner difference. The DTI abnormalities between children with spastic CP and controls were analyzed and correlated with the severity of impaired mobility based on the Gross Motor Function Classification System (GMFCS). The improvement in gross motor function and spasticity after SDR surgery was assessed utilizing the Gross Motor Function Measure-66 (GMFM-66), the Modified Tardieu Scale (MTS), and the modified Ashworth scale (MAS). Alterations in these outcome measures were quantified in association with DTI abnormalities.
Results: Significant DTI alterations, including lower fractional anisotropy (FA) in the genu of the corpus callosum (gCC) and higher mean diffusivity (MD) in the gCC and posterior limb of the internal capsule (PLIC), were found in children in the SDR group when compared with the age-matched control group (all p < 0.05). Greater DTI alterations (FA in gCC and MD in gCC and PLIC) were associated with lower mobility levels as determined based on GMFCS level (p < 0.05). The pre- to post-SDR improvement in motor function based on GMFM-66 was statistically significant (p = 0.006 and 0.002 at 6-month and 12-month follow-ups, respectively). The SDR efficacy was also identified as improving spasticity in lower-extremity muscle groups assessed with the MTS and MAS. Partial correlation analysis presented a significant association between pre- to post-SDR MTS alteration and DTI abnormalities.
Conclusions: The findings in the present study provided initial quantitative evidence to establish the WM microstructural characteristics in children with spastic CP prior to SDR surgery. The study generated data for the association between baseline DTI characteristics and mobility in children with CP prior to SDR surgery. The study also demonstrated SDR efficacy in improving motor function and spasticity based on the GMFM-66, MTS, and MAS, respectively, in association with DTI data.
{"title":"White matter characteristics in children with cerebral palsy prior to selective dorsal rhizotomy: a multicenter diffusion tensor imaging study.","authors":"Weihong Yuan, Charles B Stevenson, Paolo Moretti, Francesco T Mangano, Marco Pavanello, Armando Cama, Domenico Tortora, Chiara Tacchino, Amy F Bailes, Kelly R Greve, Jilda N Vargus-Adams, Jonathan A Dudley, Karen Harpster, Brad G Kurowski, Alexis Mitelpunkt, Bruce Aronow","doi":"10.3171/2024.4.PEDS23589","DOIUrl":"10.3171/2024.4.PEDS23589","url":null,"abstract":"<p><strong>Objective: </strong>The aims of this study were to 1) assess and quantify white matter (WM) microstructural characteristics derived from diffusion tensor imaging (DTI) in children with cerebral palsy (CP) prior to selective dorsal rhizotomy (SDR), and 2) investigate potential associations between WM diffusion properties and gross motor function and spasticity in children with spastic CP who underwent SDR.</p><p><strong>Methods: </strong>This study is a multisite study based on DT images acquired prior to SDR as well as postoperative outcome data. DTI data collected from two sites were harmonized using the ComBat approach to minimize intersite scanner difference. The DTI abnormalities between children with spastic CP and controls were analyzed and correlated with the severity of impaired mobility based on the Gross Motor Function Classification System (GMFCS). The improvement in gross motor function and spasticity after SDR surgery was assessed utilizing the Gross Motor Function Measure-66 (GMFM-66), the Modified Tardieu Scale (MTS), and the modified Ashworth scale (MAS). Alterations in these outcome measures were quantified in association with DTI abnormalities.</p><p><strong>Results: </strong>Significant DTI alterations, including lower fractional anisotropy (FA) in the genu of the corpus callosum (gCC) and higher mean diffusivity (MD) in the gCC and posterior limb of the internal capsule (PLIC), were found in children in the SDR group when compared with the age-matched control group (all p < 0.05). Greater DTI alterations (FA in gCC and MD in gCC and PLIC) were associated with lower mobility levels as determined based on GMFCS level (p < 0.05). The pre- to post-SDR improvement in motor function based on GMFM-66 was statistically significant (p = 0.006 and 0.002 at 6-month and 12-month follow-ups, respectively). The SDR efficacy was also identified as improving spasticity in lower-extremity muscle groups assessed with the MTS and MAS. Partial correlation analysis presented a significant association between pre- to post-SDR MTS alteration and DTI abnormalities.</p><p><strong>Conclusions: </strong>The findings in the present study provided initial quantitative evidence to establish the WM microstructural characteristics in children with spastic CP prior to SDR surgery. The study generated data for the association between baseline DTI characteristics and mobility in children with CP prior to SDR surgery. The study also demonstrated SDR efficacy in improving motor function and spasticity based on the GMFM-66, MTS, and MAS, respectively, in association with DTI data.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-06-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141288202","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-06-07DOI: 10.3171/2024.3.PEDS24114
Hector E James
{"title":"Letter to the Editor. Ventriculogallbladder shunts as treatment alternative for infants with necrotizing enterocolitis.","authors":"Hector E James","doi":"10.3171/2024.3.PEDS24114","DOIUrl":"10.3171/2024.3.PEDS24114","url":null,"abstract":"","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-06-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141288201","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-05-31Print Date: 2024-08-01DOI: 10.3171/2024.4.PEDS2413
Tej D Azad, Kelly Jiang, Carly Weber-Levine, Ryan P Lee, Amit Jain, Paul Sponseller, Mari Groves, Nicholas Theodore, Isam W Nasr, Eric M Jackson
Objective: The PEDSPINE I and PEDSPINE II scores were developed to determine when patients require advanced imaging to rule out cervical spine injury (CSI) in children younger than 3 years of age with blunt trauma. This study aimed to evaluate these scores in an institutional cohort.
Methods: The authors identified patients younger than 3 years with blunt trauma who received cervical spine MRI from their institution's prospective database from 2012 to 2015. Patient demographics, injury characteristics, and imaging were compared between patients with and without CSI using chi-square and Wilcoxon rank-sum tests.
Results: Eighty-eight patients were identified, 8 (9%) of whom had CSI on MRI. The PEDSPINE I system had a higher sensitivity (50% vs 25%) and negative predictive value (93% vs 92%), whereas PEDSPINE II had a higher specificity (91% vs 65%) and positive predictive value (22% vs 13%). Patients with CSI missed by the scores had mild, radiologically significant ligamentous injuries detected on MRI. Both models would have recommended advanced imaging for the patient who required halo-vest fixation (risk profile: no CSI, 81.9%; ligamentous, 10.1%; osseous, 8.0%). PEDSPINE I would have prevented 52 (65%) of 80 uninjured patients from receiving advanced imaging, whereas PEDSPINE II would have prevented 73 (91%). Using PEDSPINE I, 10 uninjured patients (13%) could have avoided intubation for imaging. PEDSPINE II would not have spared any patients intubation.
Conclusions: Current cervical spine clearance algorithms are not sensitive or specific enough to determine the need for advanced imaging in children. However, these scores can be used as a reference in conjunction with physicians' clinical impressions to reduce unnecessary imaging.
{"title":"Evaluation of cervical spine clearance scores in children younger than 3 years with blunt trauma.","authors":"Tej D Azad, Kelly Jiang, Carly Weber-Levine, Ryan P Lee, Amit Jain, Paul Sponseller, Mari Groves, Nicholas Theodore, Isam W Nasr, Eric M Jackson","doi":"10.3171/2024.4.PEDS2413","DOIUrl":"10.3171/2024.4.PEDS2413","url":null,"abstract":"<p><strong>Objective: </strong>The PEDSPINE I and PEDSPINE II scores were developed to determine when patients require advanced imaging to rule out cervical spine injury (CSI) in children younger than 3 years of age with blunt trauma. This study aimed to evaluate these scores in an institutional cohort.</p><p><strong>Methods: </strong>The authors identified patients younger than 3 years with blunt trauma who received cervical spine MRI from their institution's prospective database from 2012 to 2015. Patient demographics, injury characteristics, and imaging were compared between patients with and without CSI using chi-square and Wilcoxon rank-sum tests.</p><p><strong>Results: </strong>Eighty-eight patients were identified, 8 (9%) of whom had CSI on MRI. The PEDSPINE I system had a higher sensitivity (50% vs 25%) and negative predictive value (93% vs 92%), whereas PEDSPINE II had a higher specificity (91% vs 65%) and positive predictive value (22% vs 13%). Patients with CSI missed by the scores had mild, radiologically significant ligamentous injuries detected on MRI. Both models would have recommended advanced imaging for the patient who required halo-vest fixation (risk profile: no CSI, 81.9%; ligamentous, 10.1%; osseous, 8.0%). PEDSPINE I would have prevented 52 (65%) of 80 uninjured patients from receiving advanced imaging, whereas PEDSPINE II would have prevented 73 (91%). Using PEDSPINE I, 10 uninjured patients (13%) could have avoided intubation for imaging. PEDSPINE II would not have spared any patients intubation.</p><p><strong>Conclusions: </strong>Current cervical spine clearance algorithms are not sensitive or specific enough to determine the need for advanced imaging in children. However, these scores can be used as a reference in conjunction with physicians' clinical impressions to reduce unnecessary imaging.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-05-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141183793","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-05-31Print Date: 2024-08-01DOI: 10.3171/2024.3.PEDS2424
Hudin Jackson, Rita Snyder, Jacob R Lepard, David F Bauer
Objective: The objective was to describe the indications, technique, and initial outcomes of autologous rib graft with recombinant human bone morphogenetic protein (rhBMP) in pediatric patients undergoing posterior cervical fusion.
Methods: A retrospective study was performed of all pediatric patients who underwent autologous rib grafting with extra-small rhBMP-2 for posterior craniocervical or cervical arthrodesis at a single institution between May 2020 and July 2023. Patients with less than 3 months of postoperative follow-up and no postoperative CT data were excluded. Primary outcomes included presence of fusion on CT, 30-day perioperative complications, and rib harvest complications.
Results: Twenty-eight sequential patients met inclusion criteria. Thirteen were male, 15 were female, and the average age was 9 years. There were no surgical site infections or instances of postoperative seroma or unplanned return to the operating room. All patients had solid fusion on postoperative CT at 3 months. The average follow-up was 14.5 months, with a range of 4 months to 3 years. There were no complications associated with the rib harvest, including no instances of harvest site pain, and all patient incisions healed well.
Conclusions: The authors' preliminary results demonstrate that autologous rib graft with extra-small rhBMP-2 is an effective strategy to achieve a high rate of fusion in pediatric patients undergoing posterior instrumented craniocervical or cervical fusion. In this series, the authors found an acceptable safety profile, without seroma, surgical site infection, unplanned return to the operating room, or rib harvest complications.
{"title":"Autologous rib graft for posterior cervical fusion in pediatric patients: efficacy and safety in the early postoperative period.","authors":"Hudin Jackson, Rita Snyder, Jacob R Lepard, David F Bauer","doi":"10.3171/2024.3.PEDS2424","DOIUrl":"10.3171/2024.3.PEDS2424","url":null,"abstract":"<p><strong>Objective: </strong>The objective was to describe the indications, technique, and initial outcomes of autologous rib graft with recombinant human bone morphogenetic protein (rhBMP) in pediatric patients undergoing posterior cervical fusion.</p><p><strong>Methods: </strong>A retrospective study was performed of all pediatric patients who underwent autologous rib grafting with extra-small rhBMP-2 for posterior craniocervical or cervical arthrodesis at a single institution between May 2020 and July 2023. Patients with less than 3 months of postoperative follow-up and no postoperative CT data were excluded. Primary outcomes included presence of fusion on CT, 30-day perioperative complications, and rib harvest complications.</p><p><strong>Results: </strong>Twenty-eight sequential patients met inclusion criteria. Thirteen were male, 15 were female, and the average age was 9 years. There were no surgical site infections or instances of postoperative seroma or unplanned return to the operating room. All patients had solid fusion on postoperative CT at 3 months. The average follow-up was 14.5 months, with a range of 4 months to 3 years. There were no complications associated with the rib harvest, including no instances of harvest site pain, and all patient incisions healed well.</p><p><strong>Conclusions: </strong>The authors' preliminary results demonstrate that autologous rib graft with extra-small rhBMP-2 is an effective strategy to achieve a high rate of fusion in pediatric patients undergoing posterior instrumented craniocervical or cervical fusion. In this series, the authors found an acceptable safety profile, without seroma, surgical site infection, unplanned return to the operating room, or rib harvest complications.</p>","PeriodicalId":16549,"journal":{"name":"Journal of neurosurgery. Pediatrics","volume":null,"pages":null},"PeriodicalIF":2.1,"publicationDate":"2024-05-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141183751","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}