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INTRODUCTION: Congenital hydrocephalus often results in irreversible and severe damage to the brain despite postnatal interventions. The potential for prenatal intervention to mitigate these deleterious effects underscores the importance of a suitable animal model. We aimed assess the results of an ultrasound guided transuterine approach to replicate the BioGlue injection fetal hydrocephalus model. METHODS: Pregnant ewes were anesthetized at 95 days of gestation and BioGlue was injected into the fetal cisterna magna under ultrasound guidance through the uterus. Ventriculomegaly was assessed by MRI and histology. RESULTS: Nine pregnant ewes were included in the study, and their fetuses were divided into the the BioGlue intervention group (n=9 fetuses) or the control group (n=7 fetuses) who were not injected. Although hydrocephalus was noted in 5 of 9 fetuses in the intervention group, the ability to induce hydrocephalus went from 0% to 100% in the last 3 fetuses following technical modifications. None of the controls developed hydrocephalus. Fetal brains with hydrocephalus demonstrated increased IBA1+ compared to control animals. CONCLUSIONS: While technical challenges were noted, the ultrasound guided transuterine approach to replicate the BioGlue fetal hydrocephalus model in sheep showed consistent and reproducible results. This model offers the advantage of directly visualizing the location of the needle tip and injection of the BioGlue. This technique offers an alternative for testing novel approaches for prenatal congenital hydrocephalus treatment.
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OBJECTIVE: Chiari malformation type I (CM-I) is a congenital disorder occurring in 0.1% of the population. In symptomatic cases, surgery with posterior fossa decompression (PFD) is the treatment of choice. Surgery is, however, associated with peri- and postoperative complications that may require readmission or renewed surgical intervention. Given the associated financial costs and the impact on patients' well-being, there is a need for predictive tools that can assess the likelihood of such adverse events. The aim of this study was therefore to leverage machine learning algorithms to develop a predictive model for 30-day readmissions and reoperations after PFD in pediatric patients with CM-I. METHODS: This was a retrospective study based on data from the National Surgical Quality Improvement Program-Pediatric database. Eligible patients were those undergoing PFD (Current Procedural Terminology code 61343) for CM-I between 2012 and 2021. Patients undergoing surgery for tumors or vascular lesions were excluded. Unplanned 30-day readmission and unplanned 30-day reoperation were the main study outcomes. Additional outcome data considered included the length of hospital stay, 30-day complications, discharge disposition, and 30-day mortality. Training and testing samples were randomly generated (80:20) to study the 30-day readmission and reoperation using logistic regression, decision tree, random forest (RF), K-nearest neighbors, and Gaussian naive Bayes algorithms. RESULTS: A total of 7106 pediatric patients undergoing PFD were included. The median age was 9.2 years (IQR 4.7, 14.2 years). Most of the patients were female (56%). The 30-day readmission and reoperation rates were 7.5% and 3.4%, respectively. Headaches (32%) and wound-related complications (30%) were the most common reasons for 30-day readmission, while wound revisions and evacuation of fluid or blood (62%), followed by CSF diversion-related procedures (28%), were the most common reasons for 30-day reoperation. RF classifiers had the highest predictive accuracy for both 30-day readmissions (area under the curve [AUC] 0.960) and reoperations (AUC 0.990) compared with the other models. On feature importance analysis, sex, developmental delay, ethnicity, respiratory disease, premature birth, hydrocephalus, and congenital/genetic anomaly were some of the variables contributing the most to both RF models. CONCLUSIONS: Using a large-scale nationwide dataset, machine learning models for the prediction of both 30-day readmissions and reoperations were developed and achieved high accuracy. This highlights the utility of machine learning in risk stratification and surgical decision-making for pediatric CM-I.
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Malformación de Arnold-Chiari , Fosa Craneal Posterior , Descompresión Quirúrgica , Aprendizaje Automático , Readmisión del Paciente , Humanos , Malformación de Arnold-Chiari/cirugía , Femenino , Masculino , Descompresión Quirúrgica/métodos , Niño , Estudios Retrospectivos , Adolescente , Readmisión del Paciente/estadística & datos numéricos , Fosa Craneal Posterior/cirugía , Resultado del Tratamiento , Preescolar , Pronóstico , Reoperación/estadística & datos numéricos , Complicaciones Posoperatorias/epidemiología , Complicaciones Posoperatorias/etiología , Estudios de CohortesRESUMEN
We present the case of a 3-week-old girl with LUMBAR syndrome, a rare condition involving segmental infantile hemangiomas (IH) in the lumbosacral region, myelopathy, and other congenital anomalies. The patient developed severe ulceration of a left buttock IH. Treatment included broad-spectrum antibiotics, debridement, and a laparoscopic temporary colostomy. Propranolol therapy, wound care, and fecal diversion led to successful healing of the ulceration. The report highlights the challenges of ulcerated IH in the perineal area due to exposure to urine and stool. The comprehensive approach resulted in positive outcomes, including the successful surgical treatment of spinal dysraphism, successful colostomy reversal, and developmental progress. This case contributes insight into the surgical management of severe perianal ulcerated IH associated with LUMBAR syndrome, highlighting the importance of a tailored surgical approach with a multidisciplinary framework.
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OBJECTIVE: Sagittal craniosynostosis is the most common form of craniosynostosis and typically results in scaphocephaly, which is characterized by biparietal narrowing, compensatory frontal bossing, and an occipital prominence. The cephalic index (CI) is a simple metric for quantifying the degree of cranial narrowing and is often used to diagnose sagittal craniosynostosis. However, patients with variant forms of sagittal craniosynostosis may present with a "normal" CI, depending on the part of the suture that is closed. As machine learning (ML) algorithms are developed to assist in the diagnosis of cranial deformities, metrics that reflect the other phenotypic features of sagittal craniosynostosis are needed. In this study the authors sought to describe the posterior arc angle (PAA), a measurement of biparietal narrowing that is obtained with 2D photographs, and elucidate the role of PAA as an adjuvant to the CI in characterizing scaphocephaly and the potential relevance of PAA in new ML model development. METHODS: The authors retrospectively reviewed 1013 craniofacial patients treated during the period from 2006 to 2021. Orthogonal top-down photographs were used to calculate the CI and PAA. Distribution densities, receiver operating characteristic (ROC) curves, and chi-square analyses were used to describe the relative predictive utility of each method for sagittal craniosynostosis. RESULTS: In total, 1001 patients underwent paired CI and PAA measurements and a clinical head shape diagnosis (sagittal craniosynostosis, n = 122; other cranial deformity, n = 565; normocephalic, n = 314). The area under the ROC curve (AUC) for the CI was 98.5% (95% confidence interval 97.8%-99.2%, p < 0.001), with an optimum specificity of 92.6% and sensitivity of 93.4%. The PAA had an AUC of 97.4% (95% confidence interval 96.0%-98.8%, p < 0.001) with an optimum specificity of 94.9% and sensitivity of 90.2%. In 6 of 122 (4.9%) cases of sagittal craniosynostosis, the PAA was abnormal while the CI was normal. This means that adding a PAA cutoff branch to a partition model increases the detection of sagittal craniosynostosis. CONCLUSIONS: Both CI and PAA are excellent discriminators for sagittal craniosynostosis. Using an accuracy-optimized partition model, the addition of the PAA to the CI increased model sensitivity compared to using the CI alone. Using a model that incorporates both CI and PAA could assist in the early identification and treatment of sagittal craniosynostosis via automated and semiautomated algorithms that utilize tree-based ML models.
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Craneosinostosis , Humanos , Lactante , Estudios Retrospectivos , Craneosinostosis/diagnóstico por imagen , Craneosinostosis/cirugía , Cráneo/cirugía , Procedimientos Neuroquirúrgicos , AlgoritmosRESUMEN
OBJECTIVE: Quantitative measurements of trigonocephaly can be used to characterize and track this phenotype, which is associated with metopic craniosynostosis. Traditionally, trigonocephaly metrics were extracted from CT scans; however, this method exposes patients to ionizing radiation. Three-dimensional optical scans are another option but are not routinely available in most outpatient settings. Recently, the authors developed semiautomated artificial intelligence algorithms that extract craniometric data from orthogonal 2D photographs. Although 2D photographs are safe, inexpensive, and straightforward to obtain, the accuracy of photograph-based craniometrics in comparison to CT and 3D optical scan correlates has not been established. In this study the authors compared the classification power of 2D photograph-based metrics of trigonocephaly with four CT-based metrics and one 3D optical scan-based metric in a heterogeneous series of patients who presented to an outpatient craniofacial clinic. METHODS: In this study the authors performed retrospective craniometric analyses of patient 2D photographs, 3D optical scans, and CT scans. Imaging-derived craniometrics include the 2D photograph-based anterior arc angle (AAA2D-photo), anterior-posterior ratio (APR2D-photo), and anterior-middle ratio (AMR2D-photo); the CT-based anterior arc angle (AAACT), metopic index (MICT), endocranial-bifrontal angle (eBFACT), and interfrontal angle (IFACT); and the 3D optical scan-based anterior arc angle (AAA3D-optical). Receiver operating characteristics (ROCs) were used to identify craniometrics strongly descriptive of trigonocephaly. Interrater comparisons were made between paired trigonocephaly measurements obtained from photographs and either CT scans or 3D optical scans. RESULTS: There were 13 photograph-based and CT-based pairs and 22 paired measurements from 2D photographs and 3D optical scans. AAA displayed the strongest classification capacity across all three imaging modalities. Significant agreement was observed between AAACT and AAA2D-photo (intraclass correlation coefficient [ICC] = 0.68 [95% CI 0.24-0.89], p = 0.0035), and AAA3D-optical and AAA2D-photo (ICC = 0.70 [95% CI 0.41-0.87], p < 0.0001). There was no significant correlation between APR2D-photo or AMR2D-photo and conventional CT-based metrics describing longitudinal width ratios (MICT). CONCLUSIONS: Photograph-based craniometrics are powerful tools that can be used to quantify the severity of trigonocephaly and exhibit high concordance with standard measurements derived from CT scans and 3D optical scans. The authors developed and freely share a research-use application to calculate trigonocephaly metrics from 2D photographs. Given the availability of digital photography, lack of ionizing radiation, and low cost of photograph-based craniometric derivation, this technique may be useful to supplement routine ambulatory care and objectively track outcomes following treatment.
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Inteligencia Artificial , Craneosinostosis , Humanos , Estudios Retrospectivos , Benchmarking , Craneosinostosis/diagnóstico por imagen , Cefalometría/métodos , Imagenología Tridimensional/métodosRESUMEN
PURPOSE: To compare the outcomes of conducting left and right hemisphere surgical revascularization on the same day versus different days for bilateral pediatric moyamoya arteriopathy patients. METHODS: We retrospectively analyzed mortality, stroke, and transient neurologic event (TNE) rates in North American bilateral pediatric moyamoya arteriopathy patients who underwent bilateral cerebral revascularization. RESULTS: A total of 38 pediatric (≤ 18 years old) patients at our institution underwent bilateral cerebral revascularization for moyamoya arteriopathy. Of these patients, 24 (63.2%) had both operations on the same day and 14 (36.8%) had the two operations on different days. The average length of stay for patients who underwent same-day bilateral revascularization was 6.9 ± 2.0 days and the average length of stay for each operation for patients who underwent staged bilateral revascularization was 4.5 ± 1.4 days, p = 0.001. While there were 7 (14.6%) postoperative strokes in patients who had both hemispheres revascularized on the same day, 0 (0%) strokes occurred in hemispheres after they had been operated on in the staged cohort, p = 0.042. Additionally, the postoperative stroke-free survival time in the ipsilateral hemisphere and TNE-free survival time were significantly longer in patients in the staged revascularization cohort. CONCLUSION: Same-day bilateral revascularization was associated with longer length of stay per operation, higher rate of ipsilateral stroke, and shorter postoperative TNE-free and stroke-free survival time in the revascularized hemisphere.
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Revascularización Cerebral , Enfermedad de Moyamoya , Accidente Cerebrovascular , Humanos , Niño , Adolescente , Estudios Retrospectivos , Enfermedad de Moyamoya/diagnóstico por imagen , Enfermedad de Moyamoya/cirugía , Accidente Cerebrovascular/etiología , Accidente Cerebrovascular/cirugía , Procedimientos Quirúrgicos Vasculares , Resultado del TratamientoRESUMEN
BACKGROUND: Hirayama disease or juvenile-onset monomelic amyotrophy is a clinical syndrome that disproportionately affects young males. Standard of care revolves around conservative management, but some patients experience disease progression that may benefit from surgical intervention. METHODS: Using Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines, a systematic review of previous reports of surgical treatment for Hirayama disease was performed. Studies were included if they provided individual patient-level data, described the clinical presentation and surgical intervention, and reported neurological improvement at last follow-up. Comparison between those who improved and those with stable symptoms at last follow-up was performed. Decision-tree analysis was used to identify the best predictors of neurological improvement by last follow-up. RESULTS: Of 624 unique articles, 30 were included in the qualitative review and 23 in the meta-analysis. Among the 70 patients in the meta-analysis, mean age was 21.2 ± 6.3 years, 91% were male, and mean symptom duration at presentation was 43.3 ± 61.8 months. Fifty-nine patients (84.3%) had improvement in their neurological symptoms by last follow-up. Univariable analysis showed the only significant predictor of improvement in neurological symptoms by last follow-up was the use of stabilization-alone versus decompression with or without stabilization. Baseline clinical symptoms nor radiographic features predicted outcome. Decision-tree analysis showed surgical strategy (stabilization-alone vs. decompression ± stabilization), age (<20 vs. ≥20), and surgical approach (anterior-only vs. posterior-only or anterior-posterior) predicted a higher likelihood of neurological improvement by last follow-up. CONCLUSIONS: Nearly 85% of patients experienced improvement in neurological symptoms. Improvement was best for those who underwent stabilization-alone, and decision-tree analysis suggested that the likelihood of improvement was also superior for patients under 20 years of age and those treated with an anterior versus posterior or staged approach.
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Imagen por Resonancia Magnética , Atrofias Musculares Espinales de la Infancia , Masculino , Humanos , Adolescente , Adulto Joven , Adulto , Femenino , Atrofias Musculares Espinales de la Infancia/cirugía , Atrofias Musculares Espinales de la Infancia/diagnóstico , Descompresión Quirúrgica , Progresión de la Enfermedad , Resultado del Tratamiento , Vértebras Cervicales/cirugíaRESUMEN
BACKGROUND: Chiari I malformation is a common pediatric neurosurgical disorder with an established treatment paradigm. Posterior fossa decompression and duraplasty (PFDD) is associated with symptom improvement but it carries postoperative risk, particularly cerebrospinal fluid (CSF) leak and wound complications. In addition, the cosmetic outcomes of PFDD have been overlooked in the literature. OBJECTIVE: To describe a novel approach for PFDD in which the transverse surgical incision is completely hidden above the hairline and to report early outcomes in a prospective patient cohort. METHODS: Clinical and cosmetic outcomes were recorded for 15 consecutive pediatric patients who underwent PFDD for Chiari I malformation via the above-the-hairline transverse suboccipital approach. RESULTS: The median clinical follow-up time was 6 months (range 1-12 months), and the majority of patients experienced significant improvement of their preoperative symptoms. Three patients (20%) experienced complications associated with surgery, which included injury to the greater occipital nerve, CSF hypotension and subfascial pseudomeningocele, and superficial wound dehiscence that resolved spontaneously with oral antibiotics. Zero patients (0%) returned to the operating room for persistent CSF leak, deep wound infection, or revision decompression. An excellent cosmetic outcome was achieved in 12 patients (80%). No patient had a poor cosmetic outcome. CONCLUSION: The above-the-hairline transverse suboccipital approach for PFDD in patients with Chiari I malformation offers favorable cosmetic outcomes and fascial closure while permitting adequate decompression.
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Malformación de Arnold-Chiari , Descompresión Quirúrgica , Humanos , Niño , Estudios Prospectivos , Duramadre/cirugía , Malformación de Arnold-Chiari/diagnóstico por imagen , Malformación de Arnold-Chiari/cirugía , Complicaciones Posoperatorias/cirugía , Pérdida de Líquido Cefalorraquídeo/cirugía , Pérdida de Líquido Cefalorraquídeo/complicacionesRESUMEN
OBJECTIVE: Complex tethered spinal cord (cTSC) release in children is often complicated by surgical site infection (SSI). Children undergoing this surgery share many similarities with patients undergoing correction for neuromuscular scoliosis, where high rates of gram-negative and polymicrobial infections have been reported. Similar organisms isolated from SSIs after cTSC release were recently demonstrated in a single-center pilot study. The purpose of this investigation was to determine if these findings are reproducible across a larger, multicenter study. METHODS: A multicenter, retrospective chart review including 7 centers was conducted to identify all cases of SSI following cTSC release during a 10-year study period from 2007 to 2017. Demographic information along with specific microbial culture data and antibiotic sensitivities for each cultured organism were collected. RESULTS: A total of 44 SSIs were identified from a total of 655 cases, with 78 individual organisms isolated. There was an overall SSI rate of 6.7%, with 43% polymicrobial and 66% containing at least one gram-negative organism. Half of SSIs included an organism that was resistant to cefazolin, whereas only 32% of SSIs were completely susceptible to cefazolin. CONCLUSIONS: In this study, gram-negative and polymicrobial infections were responsible for the majority of SSIs following cTSC surgery, with approximately half resistant to cefazolin. Broader gram-negative antibiotic prophylaxis should be considered for this patient population.
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OBJECTIVE: Chiari I malformation is treated with suboccipital craniectomy with cervical laminectomy, a procedure which has been associated with the possibility of pre-existing or iatrogenic occipitocervical instability. The long-term risk of subsequent spinal deformity and need for occipito cervical fusion after standard Chiari decompression in pediatric patients has not yet been characterized. METHODS: We queried our institutional electronic database for patients aged 18 and under, with at least 5 years of follow-up, that underwent surgical decompression for Chiari I malformation. Occurrence of subsequent occipitocervical fusion at follow-up comprised the primary endpoint. Cases with myelomeningocele, Chiari II, or fusion at time of decompression were excluded. RESULTS: A total of 30 patients (median age 5.5 years, 60% males) were analyzed. Age distribution was as follows: n = 3 for 0-1 years, n = 11 for 1-5 years, n = 4 for 5-10 years, and n = 12 for 10-18 years. Median tonsillar descent below the foramen magnum was 12.5 mm (interquartile range [IQR]: 10.8-19.5 mm). Syringomyelia was observed in 43%, retroflexion of the dens in 55%, basilar invagination in 6.7%, and medullary kinking in 27%. The median clivo-axial angle was 142° (132-150°). The majority of patients underwent C1 laminectomy (n = 24, 80%), followed by C1-C2 laminectomy (n = 4, 13%), while one patient had C1-upper C2 and C1-C3 laminectomy each, respectively. At a median follow-up of 6.3 years, there was only one patient (3.3% of overall cohort) that underwent subsequent occipitocervical fusion. The patient (4-year-old male) initially had a suboccipital craniectomy with C1 laminectomy and duraplasty and presented with recurrence of posterior headaches and neck pain 4 months after original surgery. We proceeded with occiput-C2 fusion with subsequent resolution of his symptoms. CONCLUSION: Current analysis shows that in the absence of clinical or imaging features suggestive of craniocervical instability, Chiari I decompressive surgery is associated with very low long-term risk of requiring occipitocervical fusion. This observance can be used to guide surgical treatment decisions, especially in young children with Chiari I malformations.
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Malformación de Arnold-Chiari , Siringomielia , Malformación de Arnold-Chiari/complicaciones , Malformación de Arnold-Chiari/diagnóstico por imagen , Malformación de Arnold-Chiari/cirugía , Niño , Preescolar , Descompresión Quirúrgica/efectos adversos , Descompresión Quirúrgica/métodos , Femenino , Foramen Magno/cirugía , Humanos , Laminectomía/efectos adversos , Imagen por Resonancia Magnética , Masculino , Siringomielia/cirugía , Resultado del TratamientoRESUMEN
OBJECTIVE: Arteriovenous malformations (AVMs) are a major cause of intracerebral hemorrhage in children, resulting in significant morbidity and mortality. Moreover, the rate of AVM recurrence in children is significantly higher than in adults. The aim of this study was to define the risk of delayed pediatric AVM (pAVM) recurrence following confirmed radiological obliteration. Further understanding of this risk could inform the role of long-term radiological surveillance. METHODS: The authors conducted a retrospective review of ruptured and unruptured pAVM cases treated at a single tertiary care referral center between 1994 and 2019. Demographics, clinical characteristics, treatment modalities, and AVM recurrence were analyzed. RESULTS: A total of 102 pediatric patients with intracranial AVMs, including 52 (51%) ruptured cases, were identified. The mean patient age at presentation was 11.2 ± 4.4 years, and 51 (50%) patients were female. The mean nidus size was 2.66 ± 1.44 cm. The most common Spetzler-Martin grades were III (32%) and II (31%). Stereotactic radiosurgery was performed in 69.6% of patients. AVM obliteration was radiologically confirmed in 68 (72.3%) of 94 patients with follow-up imaging, on angiography in 50 (73.5%) patients and on magnetic resonance imaging in 18 (26.5%). AVM recurrence was identified in 1 (2.3%) of 43 patients with long-term surveillance imaging over a mean follow-up of 54.7 ± 38.9 months (range 2-153 months). This recurrence was identified in a boy who had presented with a ruptured AVM and had been surgically treated at 5 years of age. The AVM recurred 54 months after confirmed obliteration on surveillance digital subtraction angiography. Two other cases of presumed AVM recurrence following resection in young children were excluded from recurrence analysis because of incomplete sets of imaging available for review. CONCLUSIONS: AVM recurrence following confirmed obliteration on imaging is a rare phenomenon, though it occurs more frequently in the pediatric population. Regular long-term follow-up with dedicated surveillance angiography is recommended even after obliteration following resection.
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OBJECTIVE: CSF shunt insertion is the most commonly performed neurosurgical procedure for pediatric patients with hydrocephalus, and complications including infections and catheter obstruction are common. The rate of readmission in the first 30 days after surgery has been used across surgical disciplines to determine healthcare quality. In the current study, the authors sought to assess factors associated with early shunt revision within 30 days using real-world data. METHODS: Targeted shunt data set participant user files of the National Surgical Quality Improvement Program (NSQIP) from 2016 to 2019 were queried for patients undergoing a shunt procedure. A multivariable logistic regression model was performed to assess the impact of demographics, etiologies, comorbidities, congenital malformations, and shunt adjuncts on shunt revision within 30 days, as well as shunt revision due to infection within 30 days. RESULTS: A total of 3919 primary pediatric shunt insertions were identified in the NSQIP database, with a mean (± SD) patient age of 26.3 ± 51.6 months. There were a total of 285 (7.3%) unplanned shunt revisions within 30 days, with a mean duration of 14.9 ± 8.5 days to first intervention. The most common reason for intervention was mechanical shunt failure (32.6% of revision, 2.4% overall, n = 93), followed by infection (31.2% of all interventions, 2.3% overall, n = 89) and wound disruption or CSF leak (22.1% of all interventions, 1.6% overall, n = 63). Patients younger than 6 months of age had the highest overall unplanned 30-day revision rate (8.5%, 203/2402) as well as the highest 30-day shunt infection rate (3%, 72/2402). Patients who required a revision were also more likely to have a cardiac risk factor (34.7%, n = 99, vs 29.2%, n = 1061; p = 0.048). Multivariable logistic regression revealed that compared to patients 9-18 years old, those aged 2-9 years had significantly lower odds of repeat shunt intervention (p = 0.047), while certain etiologies including congenital hydrocephalus (p = 0.0127), intraventricular hemorrhage (IVH) of prematurity (p = 0.0173), neoplasm (p = 0.0005), infection (p = 0.0004), and syndromic etiology (p = 0.0136), as well as presence of ostomy (p = 0.0095), were associated with higher odds of repeat intervention. For shunt infection, IVH of prematurity was found to be associated with significantly higher odds (p = 0.0427) of shunt infection within 30 days, while use of intraventricular antibiotics was associated with significantly lower odds (p = 0.0085). CONCLUSIONS: In this study of outcomes after pediatric shunt placement using a nationally derived cohort, early shunt failure and infection within 30 days were found to remain as considerable risks. The analysis of this national surgical quality registry confirms that, in accordance with other multicenter studies, hydrocephalus etiology, age, and presence of ostomy are important predictors of the need for early shunt revision. IVH of prematurity is associated with early infections while intraventricular antibiotics may be protective. These findings could be used for benchmarking in hospital efforts to improve quality of care for pediatric patients with hydrocephalus.
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Derivaciones del Líquido Cefalorraquídeo/efectos adversos , Derivaciones del Líquido Cefalorraquídeo/métodos , Hidrocefalia/cirugía , Mejoramiento de la Calidad , Reoperación , Adolescente , Niño , Preescolar , Estudios de Cohortes , Falla de Equipo , Femenino , Humanos , Lactante , Masculino , Readmisión del Paciente , Complicaciones Posoperatorias/epidemiología , Complicaciones Posoperatorias/etiología , Factores de RiesgoRESUMEN
INTRODUCTION: Prenatal closure of open spina bifida via open fetal surgery improves neurologic outcomes for infants in selected pregnancies. Fetoscopic techniques that are minimally invasive to the uterus aim to provide equivalent fetal benefits while minimizing maternal morbidities, but the optimal technique is undetermined. We describe the development, evolution, and feasibility of the laparotomy-assisted 2-port fetoscopic technique for prenatal closure of fetal spina bifida in a newly established program. METHODS: We conducted a retrospective cohort study of women consented for laparotomy-assisted fetoscopic closure of isolated fetal spina bifida. Inclusion and exclusion criteria followed the Management of Myelomeningocele Study (MOMS). Team preparation involved observation at the originating center, protocol development, ancillary staff training, and surgical rehearsal using patient-matched models through simulation prior to program implementation. The primary outcome was the ability to complete the repair fetoscopically. Secondary maternal and fetal outcomes to assess performance of the technique were collected prospectively. RESULTS: Of 57 women screened, 19 (33%) consented for laparotomy-assisted 2-port fetoscopy between February 2017 and December 2019. Fetoscopic closure was completed in 84% (16/19) cases. Over time, the technique was modified from a single- to a multilayer closure. In utero hindbrain herniation improved in 86% (12/14) of undelivered patients at 6 weeks postoperatively. Spontaneous rupture of membranes occurred in 31% (5/16) of fetoscopic cases. For completed cases, median gestational age at birth was 37 (range 27-39.6) weeks and 50% (8/16) of women delivered at term. Vaginal birth was achieved in 56% (9/16) of patients. One newborn had a cerebrospinal fluid leak that required postnatal surgical repair. CONCLUSION: Implementation of a laparotomy-assisted 2-port fetoscopic spina bifida closure program through rigorous preparation and multispecialty team training may accelerate the learning curve and demonstrates favorable obstetric and perinatal outcomes.
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Meningomielocele , Disrafia Espinal , Femenino , Fetoscopía/efectos adversos , Humanos , Lactante , Recién Nacido , Laparotomía , Meningomielocele/cirugía , Embarazo , Estudios Retrospectivos , Disrafia Espinal/diagnóstico por imagen , Disrafia Espinal/cirugíaRESUMEN
OBJECTIVE: The authors sought to evaluate the accuracy of a novel telehealth-compatible diagnostic software system for identifying craniosynostosis within a newborn (< 1 year old) population. Agreement with gold standard craniometric diagnostics was also assessed. METHODS: Cranial shape classification software accuracy was compared to that of blinded craniofacial specialists using a data set of open-source (n = 40) and retrospectively collected newborn orthogonal top-down cranial images, with or without additional facial views (n = 339), culled between April 1, 2008, and February 29, 2020. Based on image quality, midface visibility, and visibility of the cranial equator, 351 image sets were deemed acceptable. Accuracy, sensitivity, and specificity were calculated for the software versus specialist classification. Software agreement with optical craniometrics was assessed with intraclass correlation coefficients. RESULTS: The cranial shape classification software had an accuracy of 93.3% (95% CI 86.8-98.8; p < 0.001), with a sensitivity of 92.0% and specificity of 94.3%. Intraclass correlation coefficients for measurements of the cephalic index and cranial vault asymmetry index compared to optical measurements were 0.95 (95% CI 0.84-0.98; p < 0.001) and 0.67 (95% CI 0.24-0.88; p = 0.003), respectively. CONCLUSIONS: These results support the use of image processing-based neonatal cranial deformity classification software for remote screening of nonsyndromic craniosynostosis in a newborn population and as a substitute for optical scanner- or CT-based craniometrics. This work has implications that suggest the potential for the development of software for a mobile platform that would allow for screening by telemedicine or in a primary care setting.
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Cefalometría/métodos , Craneosinostosis/diagnóstico , Interpretación de Imagen Asistida por Computador/métodos , Aprendizaje Automático , Tamizaje Masivo/métodos , Telemedicina/métodos , Femenino , Humanos , Recién Nacido , Masculino , Programas InformáticosRESUMEN
The authors describe an endoscopic strip craniectomy through a single incision for the treatment of sagittal craniosynostosis in a young infant. The endoscopic strip craniectomy was first introduced with the use of two incisions on either end of the fused suture. This single-incision technique offers several advantages. There is a cosmetic advantage and a reduced risk of wound complications. This technique also allows for early control of emissary veins and an inside-out identification of the lambdoid sutures. Endoscopic visualization is optimized to reduce the risk of blood loss, especially because circulating blood volume is very limited in these young infants. The video can be found here: https://vimeo.com/514366415.
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OBJECTIVES: We sought to characterize patterns of in utero dilation in isolated severe fetal ventriculomegaly (ISVM) and investigate their value in predicting obstetrical and postnatal outcomes. METHODS: This is a retrospective cohort study. ISVM was defined as a sonographic cerebral ventricle atrial with width ≥15 mm in the absence of additional cerebral or other anatomic anomalies. The aim of this study was to characterize two ISVM groups using a receiver operator curve to evaluate the rate of ventricular progression versus need for ventriculoperitoneal (VP) shunt postnatally. Outcomes were compared between the groups using Pearson's chi-squared test, Student t-test, and descriptive statistics. RESULTS: Based on the ROC analysis, ventricular growth of ≥3 mm/week versus <3 mm/week distinguished fetuses likely to require a postnatal VP shunt. Fetuses were characterized as accelerators if ventricle growth was ≥3 mm/week at any point and plateaus if <3 mm/week. Accelerators showed a greater average rate of ventricle progression than plateaus (4.1 vs. 1.0 mm/week, respectively, p = .031) and were more likely to be delivered at earlier gestational ages (34.7 vs. 37.1 weeks respectively, p = .02). Ninety percent of accelerators demonstrated a need for shunt placement compared with 18.8% of plateaus (p < .001). Significantly more plateaus (87.5%) underwent a trial of labor while accelerators were more likely to have planned cesareans (70%, p = .009). CONCLUSIONS: This study characterizes ISVM into two distinct populations based upon the rate of ventricle expansion, differentiated by the need for postnatal shunting. Once a ventricular growth pattern is determined, these distinctions should prove useful in prenatal management and delivery planning.
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Hidrocefalia , Derivación Ventriculoperitoneal , Aceleración , Ventrículos Cerebrales/diagnóstico por imagen , Dilatación , Femenino , Feto , Humanos , Embarazo , Estudios RetrospectivosRESUMEN
OBJECTIVE: Pediatric Chiari I malformation decompression is a common neurosurgical procedure. Liposomal bupivacaine (LB) is a novel formulation that can have an impact on postoperative recovery for particular procedures, but its potential role in pediatric neurosurgery is largely unexplored. The authors sought to describe and assess their initial experience with LB in pediatric Chiari I malformation decompression to better define its potential role as an analgesic agent in a procedure for which the postoperative course is often remarkably painful. METHODS: A retrospective review of all pediatric Chiari procedures performed at the authors' institution between 2018 and 2020 was conducted. Patients were divided into those who were treated with a single intraoperative dose of LB (LB group) and those who were not (control group). Comparisons of total opioid use and pain control were made using chi-square and Wilcoxon rank-sum tests. RESULTS: A total of 18 patients were identified, 9 (50%) in the LB group and 9 (50%) in the control group. Overall, there were 13 (72%) female and 5 (28%) male patients with a mean age of 15.9 years. No surgical complications were observed over a mean length of stay of 2.7 days. Within the first 24 hours after surgery, the LB group had significantly lower total opioid use than the control group (17.5 vs 47.9 morphine milligram equivalents, respectively; p = 0.03) as well as lower mean pain scores reported by patients using a 10-point visual analog scale (3.6 vs 5.5 for the LB vs control groups, p = 0.04). However, from the first 24 postoperative hours to discharge, total opioid use (p = 0.51) and mean pain scores (p = 0.09) were statistically comparable between the two groups. There were 2/9 (22%) LB patients versus 0/9 (0%) control patients who did not require opioid analgesia at any point during hospitalization. CONCLUSIONS: The use of a single intraoperative dose of LB in pediatric Chiari I malformation surgery appears to be safe and has the potential to reduce pain scores and opioid use when administered during the first 24 postoperative hours. From that time period to discharge, however, there may be no significant difference in total opioid use or pain scores.
Asunto(s)
Analgésicos Opioides/administración & dosificación , Anestésicos Locales/administración & dosificación , Malformación de Arnold-Chiari/cirugía , Bupivacaína/administración & dosificación , Cuidados Intraoperatorios/métodos , Dolor Postoperatorio/prevención & control , Adolescente , Malformación de Arnold-Chiari/tratamiento farmacológico , Niño , Estudios de Cohortes , Femenino , Humanos , Liposomas , Masculino , Manejo del Dolor/métodos , Dimensión del Dolor/efectos de los fármacos , Dimensión del Dolor/métodos , Dolor Postoperatorio/diagnóstico , Estudios Retrospectivos , Resultado del TratamientoRESUMEN
OBJECTIVE: To cope with the changing health care services in the era of SARS-CoV-2 pandemic. We share the institutional framework for the management of anomalous fetuses requiring fetal intervention at Mayo Clinic, Rochester, Minnesota. To assess the success of our program during this time, we compare intraoperative outcomes of fetal interventions performed during the pandemic with the previous year. PATIENTS: We implemented our testing protocol on patients undergoing fetal intervention at our institution between March 1, and May 15, 2020, and we compared it with same period a year before. A total of 17 pregnant patients with anomalous fetuses who met criteria for fetal intervention were included: 8 from 2019 and 9 from 2020. METHODS: Our testing protocol was designed based on our institutional perinatal guidelines, surgical requirements from the infection prevention and control (IPAC) committee, and input from our fetal surgery team, with focus on urgency of procedure and maternal SARS-CoV-2 screening status. We compared the indications, types of procedures, maternal age, gestational age at procedure, type of anesthesia used, and duration of procedure for cases performed at our institution between March 1, 2020, and May 15, 2020, and for the same period in 2019. RESULTS: There were no statistically significant differences among the number of cases, indications, types of procedures, maternal age, gestational age, types of anesthesia, and duration of procedures (P values were all >.05) between the pre-SARS-CoV-2 pandemic in 2019 and the SARS-CoV-2 pandemic in 2020. CONCLUSIONS: Adoption of new institutional protocols during SARS-CoV-2 pandemic, with appropriate screening and case selection, allows provision of necessary fetal intervention with maximal benefit to mother, fetus, and health care provider.