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OBJECTIVE: Women can develop posttraumatic stress disorder in response to experienced or perceived traumatic, often medically complicated, childbirth; the prevalence of these events remains high in the United States. Currently, no recommended treatment exists in routine care to prevent or mitigate maternal childbirth-related posttraumatic stress disorder. We conducted a systematic review and meta-analysis of clinical trials that evaluated any therapy to prevent or treat childbirth-related posttraumatic stress disorder. DATA SOURCES: PsycInfo, PsycArticles, PubMed (MEDLINE), ClinicalTrials.gov, CINAHL, ProQuest, Sociological Abstracts, Google Scholar, Embase, Web of Science, ScienceDirect, Scopus, and the Cochrane Central Register of Controlled Trials (CENTRAL) were searched for eligible trials published through September 2023. STUDY ELIGIBILITY CRITERIA: Trials were included if they were interventional, if they evaluated any therapy for childbirth-related posttraumatic stress disorder for the indication of symptoms or before posttraumatic stress disorder onset, and if they were written in English. METHODS: Independent coders extracted the sample characteristics and intervention information of the eligible studies and evaluated the trials using the Downs and Black's quality checklist and Cochrane's method for risk of bias evaluation. Meta-analysis was conducted to evaluate pooled effect sizes of secondary and tertiary prevention trials. RESULTS: A total of 41 studies (32 randomized controlled trials, 9 nonrandomized trials) were reviewed. They evaluated brief psychological therapies including debriefing, trauma-focused therapies (including cognitive behavioral therapy and expressive writing), memory consolidation and reconsolidation blockage, mother-infant-focused therapies, and educational interventions. The trials targeted secondary preventions aimed at buffering childbirth-related posttraumatic stress disorder usually after traumatic childbirth (n=24), tertiary preventions among women with probable childbirth-related posttraumatic stress disorder (n=14), and primary prevention during pregnancy (n=3). A meta-analysis of the combined randomized secondary preventions showed moderate effects in reducing childbirth-related posttraumatic stress disorder symptoms when compared with usual treatment (standardized mean difference, -0.67; 95% confidence interval, -0.92 to -0.42). Single-session therapy within 96 hours of birth was helpful (standardized mean difference, -0.55). Brief, structured, trauma-focused therapies and semi-structured, midwife-led, dialogue-based psychological counseling showed the largest effects (standardized mean difference, -0.95 and -0.91, respectively). Other treatment approaches (eg, the Tetris game, mindfulness, mother-infant-focused treatment) warrant more research. Tertiary preventions produced smaller effects than secondary prevention but are potentially clinically meaningful (standardized mean difference, -0.37; -0.60 to -0.14). Antepartum educational approaches may help, but insufficient empirical evidence exists. CONCLUSION: Brief trauma-focused and non-trauma-focused psychological therapies delivered early in the period following traumatic childbirth offer a critical and feasible opportunity to buffer the symptoms of childbirth-related posttraumatic stress disorder. Future research that integrates diagnostic and biological measures can inform treatment use and the mechanisms at work.
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Parto , Trastornos por Estrés Postraumático , Humanos , Trastornos por Estrés Postraumático/prevención & control , Trastornos por Estrés Postraumático/terapia , Trastornos por Estrés Postraumático/psicología , Femenino , Embarazo , Parto/psicología , Terapia Cognitivo-Conductual/métodosRESUMEN
OBJECTIVE: Intrathecal baclofen (ITB) pumps are commonly used in pediatric patients with cerebral palsy (CP) and medically refractory spasticity. However, catheter malfunction and associated risk factors are not well understood. The aim of this study was to examine potential risk factors for spinal catheter malfunction and characterize postoperative follow-up to understand the clinical consequences. METHODS: Patients who received ITB pump replacement or revision at Boston Children's Hospital between 2010 and 2023 were retrospectively reviewed. The spinal catheter revision cohort (SCRC) included patients whose spinal catheter was occluded requiring lumbar catheter revision. The second cohort included abdominal pump replacements only (APRC). Between-group comparisons and multivariable regression identified factors associated with catheter revision and postoperative outcomes. RESULTS: Forty-one (33.6%) patients underwent spinal catheter revision and were compared with 81 patients (66.4%) who underwent abdominal pump replacement only. Younger age at surgery and an elevated preoperative lower-extremity modified Ashworth scale grade were associated with spinal catheter revision (p < 0.05). Catheter model type, tip location, and history of spinal fusion were not associated with obstruction. Postoperatively, SCRC patients experienced a higher rate of infection (17.1%) relative to APRC patients (0%) within 30 days from their ITB pump replacement procedure (p < 0.05) and greater likelihood of subsequent ITB system removal compared with the APRC (24.4% vs 7.4%, p < 0.05). Although not differing preoperatively, SCRC patients had lower postoperative ITB doses when compared with the APRC group (median dose 143 vs 350 µg/day, p < 0.05) at hospital discharge and remained statistically different at the 6-month and 1-year follow-ups (p < 0.05). There were no postoperative differences in baclofen overdose, withdrawal, or median number of hospital readmissions within 30 days. Overall, 31.7% of spinal catheter revisions were unanticipated by the clinical team at time of surgery. CONCLUSIONS: Younger age at surgery and increased preoperative lower-extremity tone may be risk factors for catheter obstruction, resulting in a higher rate of postoperative infection and subsequent ITB pump removal compared with pump replacement alone. Spinal catheter occlusion can complicate revision or replacement procedures, especially when unanticipated. Routine clinical assessment may be inadequate for diagnosing insidious catheter malfunction. Catheter occlusion deserves further study, and routine assessment of catheter patency may be warranted to prevent suboptimal tone therapy.
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Baclofeno , Parálisis Cerebral , Bombas de Infusión Implantables , Relajantes Musculares Centrales , Humanos , Baclofeno/administración & dosificación , Baclofeno/efectos adversos , Masculino , Femenino , Niño , Bombas de Infusión Implantables/efectos adversos , Factores de Riesgo , Relajantes Musculares Centrales/administración & dosificación , Estudios Retrospectivos , Adolescente , Parálisis Cerebral/cirugía , Parálisis Cerebral/complicaciones , Preescolar , Espasticidad Muscular/tratamiento farmacológico , Espasticidad Muscular/etiología , Espasticidad Muscular/cirugía , Reoperación/métodos , Inyecciones Espinales/métodos , Resultado del Tratamiento , Complicaciones Posoperatorias/etiología , Falla de Equipo , Estudios de CohortesRESUMEN
OBJECTIVE: Optimal choroid plexus tumor (CPT) treatment involves gross total resection; however, intraoperative hemorrhage risk remains significant given tumor vascularity. This study describes pediatric CPT management and identifies patients most likely to benefit from preoperative embolization. METHODS: CPTs resected from 1997 to 2021 were included. The characteristics of embolized patients were compared to nonembolized patients; nonembolized patients were further stratified based on open vascular control-pedicle feeder ligation versus no pedicle ligation prior to tumor debulking. Statistical analyses identified factors associated with estimated blood loss (EBL), transfusion, length of stay, and complications. RESULTS: Among the 46 CPT cases identified, 98% achieved gross total resection, and 15% received embolization. Embolized patients were younger, smaller, and had larger tumors compared to nonembolized patients (median: 0.8 vs. 2.1 years; 9.3 vs. 14.4 kg; 91.08 vs. 5.5 cm3). Transfused patients were similarly younger and smaller (P < 0.05) than nontransfused patients. Among nonembolized patients, open vascular control was achieved in smaller tumors (<13 cm3) with significantly lower EBL (P = 0.002). Higher EBL was observed in patients with larger tumors, hydrocephalus, transependymal edema, vomiting, lethargy, and developmental regression (all P < 0.05). Patients with lethargy had longer hospital stays and a higher likelihood of postoperative complications (P < 0.05). There were no significant differences in complication rates between the embolization and nonembolization groups. CONCLUSIONS: Despite higher surgical risk profiles, embolized patients had similar complication rates and postoperative hydrocephalus management as nonembolized patients. Embolization was particularly beneficial in patients at high risk for surgical morbidity, such as those <2 years, weighing <10 kg, and with a tumor volume >15 cm3.
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Neoplasias del Plexo Coroideo , Embolización Terapéutica , Hidrocefalia , Papiloma del Plexo Coroideo , Niño , Humanos , Letargia/complicaciones , Neoplasias del Plexo Coroideo/cirugía , Neoplasias del Plexo Coroideo/complicaciones , Hidrocefalia/cirugía , Hidrocefalia/complicaciones , Pérdida de Sangre Quirúrgica , Embolización Terapéutica/efectos adversos , Estudios Retrospectivos , Papiloma del Plexo Coroideo/complicacionesRESUMEN
BACKGROUND: Middle fossa arachnoid cysts (MFACs) are rare, congenital lesions that may rupture and cause symptoms of elevated intracranial pressure. We sought to describe the presence of and factors associated with optic nerve edema in MFACs, focusing on the utility of ophthalmologic evaluations for guiding cyst management. METHODS: We reviewed clinical and radiographic information for all patients with MFACs with ophthalmologic evaluations at our institution. Headache, cranial nerve palsy, emesis, altered mental status, fatigue, and seizures were considered MFAC-related symptoms. Univariate and multivariable analyses evaluated factors associated with optic edema. RESULTS: Fifty-one patients between 2003 and 2022 were included. Cysts were a median volume of 169.9 cm3 (interquartile range: 70.5, 647.7). Evidence of rupture with subdural hematoma/hygroma occurred in 19 (37.3%) patients. Eighteen (35.3%) patients underwent surgery for their cyst and/or rupture-associated intracranial bleed. Eleven (21.6%) patients had optic edema; all were symptomatic and experienced cyst rupture. Ten of these patients received surgery. Postoperatively, optic edema resolved in 80% of cases. Cyst volume and symptoms were not associated with optic edema; however, patients with ruptured cysts, particularly those with traumatic rupture, were more likely to have optic edema and receive surgery (P < 0.001). CONCLUSIONS: We found optic edema in 21.6% of evaluated MFACs, and this comprised of 57.9% of ruptured cases. Optic edema was not found in unruptured cysts. Cyst fenestration improved optic edema and patient symptoms. In conjunction with clinical history and neuroimaging, optic edema may help guide MFAC management, particularly in patients with cyst rupture.
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Quistes Aracnoideos , Fosa Craneal Media , Humanos , Quistes Aracnoideos/complicaciones , Quistes Aracnoideos/diagnóstico por imagen , Masculino , Femenino , Fosa Craneal Media/diagnóstico por imagen , Niño , Preescolar , Adolescente , Estudios Retrospectivos , Lactante , Papiledema/etiología , Enfermedades del Nervio Óptico/etiología , Enfermedades del Nervio Óptico/diagnóstico por imagenRESUMEN
BACKGROUND: Focal epilepsy caused by a posterior fossa lesion is a rare phenomenon. In these cases, seizure onset typically occurs during the first few months of life, with episodes of epileptic hemifacial spasms and abnormal eye movements. Patients often present with drug-resistant epilepsy and often require resection for the best chance of seizure freedom. OBSERVATIONS: The authors present the case of a 19-month-old male with intractable epileptic hemifacial spasms and a dorsally exophytic right brainstem and middle cerebellar peduncle hamartoma, following 2 prior subtotal resections. The authors recommended a third suboccipital craniotomy with intraoperative electrocorticography, which revealed interictal spiking from an intralesional depth electrode. Near-total resection led to durable seizure freedom. LESSONS: Although posterior fossa lesions are rarely associated with epileptiform activity, this case demonstrates that pediatric patients with epileptic hemifacial spasms associated with a posterior fossa lesion may respond favorably to resection. Furthermore, this case demonstrates that intralesional electrocorticography can detect epileptic activity in posterior fossa lesions, which may predict postoperative seizure outcomes. https://thejns.org/doi/10.3171/CASE2452.
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OBJECTIVE: Reports on the management and survival of children with myelomeningocele defects in Bangladesh are limited. This study describes the characteristics and outcomes of these children, focusing on the timing of surgical repair and factors affecting survival. METHODS: We enrolled patients with myelomeningoceles in a case-control study on arsenic exposure and spina bifida in Bangladesh. Cases were subsequently followed at regular intervals to assess survival. Demographic, clinical, and surgical characteristics were reviewed. Univariate tests identified factors affecting survival. RESULTS: Between 2016 and 2022, we enrolled 272 patients with myelomeningocele. Postnatal surgical repair was performed in 63% of cases. However, surgery within 5 days after birth was infrequent (<10%) due to delayed presentation, and there was a high rate (29%) of preoperative deaths. Surgical repair significantly improved patient survival (P < 0.0001). Older age at time of surgery was also associated with improved survival rates, which most likely represents that those who survived to older ages prior to surgery accommodated better with their lesions. Patients who presented with ruptured lesions had lower survival rates. CONCLUSIONS: Timely neurosurgical repair of myelomeningoceles in Bangladesh is hindered by late patient presentation, resulting in a high preoperative patient death rate. Neurosurgical intervention remains a significant predictor of survival. Increased access to neurosurgical care and education of families and non-neurosurgical providers on the need for timely surgical intervention are important for improving the survival of infants with myelomeningoceles.
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Meningomielocele , Procedimientos Neuroquirúrgicos , Humanos , Bangladesh/epidemiología , Meningomielocele/cirugía , Masculino , Femenino , Procedimientos Neuroquirúrgicos/métodos , Lactante , Estudios de Casos y Controles , Recién Nacido , Preescolar , Tiempo de Tratamiento , Niño , Resultado del Tratamiento , Tasa de SupervivenciaRESUMEN
Objectives: Brain-limited pathogenic somatic variants are associated with focal pediatric epilepsy, but reliance on resected brain tissue samples has limited our ability to correlate epileptiform activity with abnormal molecular pathology. We aimed to identify the pathogenic variant and map variant allele fractions (VAFs) across an abnormal region of epileptogenic brain in a patient who underwent stereoelectroencephalography (sEEG) and subsequent motor-sparing left frontal disconnection. Methods: We extracted genomic DNA from peripheral blood, brain tissue resected from peri-sEEG electrode regions, and microbulk brain tissue adherent to sEEG electrodes. Samples were mapped based on an anatomic relationship with the presumed seizure onset zone (SOZ). We performed deep panel sequencing of amplified and unamplified DNA to identify pathogenic variants with subsequent orthogonal validation. Results: We detect a pathogenic somatic PIK3CA variant, c.1624G>A (p.E542K), in the brain tissue samples, with VAF inversely correlated with distance from the SOZ. In addition, we identify this variant in amplified electrode-derived samples, albeit with lower VAFs. Discussion: We demonstrate regional mosaicism across epileptogenic tissue, suggesting a correlation between variant burden and SOZ. We also validate a pathogenic variant from individual amplified sEEG electrode-derived brain specimens, although further optimization of techniques is required.
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Objective: Postpartum women can develop post-traumatic stress disorder (PTSD) in response to complicated, traumatic childbirth; prevalence of these events remains high in the U.S. Currently, there is no recommended treatment approach in routine peripartum care for preventing maternal childbirth-related PTSD (CB-PTSD) and lessening its severity. Here, we provide a systematic review of available clinical trials testing interventions for the prevention and indication of CB-PTSD. Data Sources: We conducted a systematic review of PsycInfo, PsycArticles, PubMed (MEDLINE), ClinicalTrials.gov, CINAHL, ProQuest, Sociological Abstracts, Google Scholar, Embase, Web of Science, ScienceDirect, and Scopus through December 2022 to identify clinical trials involving CB-PTSD prevention and treatment. Study Eligibility Criteria: Trials were included if they were interventional, evaluated CB-PTSD preventive strategies or treatments, and reported outcomes assessing CB-PTSD symptoms. Duplicate studies, case reports, protocols, active clinical trials, and studies of CB-PTSD following stillbirth were excluded. Study Appraisal and Synthesis Methods: Two independent coders evaluated trials using a modified Downs and Black methodological quality assessment checklist. Sample characteristics and related intervention information were extracted via an Excel-based form. Results: A total of 33 studies, including 25 randomized controlled trials (RCTs) and 8 non-RCTs, were included. Trial quality ranged from Poor to Excellent. Trials tested psychological therapies most often delivered as secondary prevention against CB-PTSD onset (n=21); some examined primary (n=3) and tertiary (n=9) therapies. Positive treatment effects were found for early interventions employing conventional trauma-focused therapies, psychological counseling, and mother-infant dyadic focused strategies. Therapies' utility to aid women with severe acute traumatic stress symptoms or reduce incidence of CB-PTSD diagnosis is unclear, as is whether they are effective as tertiary intervention. Educational birth plan-focused interventions during pregnancy may improve maternal health outcomes, but studies remain scarce. Conclusions: An array of early psychological therapies delivered in response to traumatic childbirth, rather than universally, in the first postpartum days and weeks, may potentially buffer CB-PTSD development. Rather than one treatment being suitable for all, effective therapy should consider individual-specific factors. As additional RCTs generate critical information and guide recommendations for first-line preventive treatments for CB-PTSD, the psychiatric consequences associated with traumatic childbirth could be lessened.
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OBJECTIVE: The assessment of hemorrhagic risk is of central importance in the management of pediatric brain arteriovenous malformations (AVMs). A recently published scoring system, the R2eD AVM (race, exclusive deep location or not, AVM size < 3 or ≥ 3, venous drainage exclusively deep or not, and monoarterial feeding or >1 arterial feeders) considers patient race and AVM location, size, venous drainage, and arterial supply and was successfully externally validated for use in adults to predict the risk of AVM hemorrhagic presentation. In this study the authors sought to externally validate the R2eD AVM score for prediction of hemorrhagic risk in the pediatric AVM population. METHODS: A pediatric database at Boston Children's Hospital was retrospectively reviewed for all patients with a diagnosis of intracranial AVM. Exclusion criteria included age > 21 years, multiple AVMs, and incomplete clinical/radiographic data. Demographic data and R2eD AVM score calculations were collected for all patients. Univariate binary logistic regression and multivariate stepwise backward elimination models were used to determine factors associated with hemorrhagic presentation. RESULTS: A total of 212 AVMs were identified in 212 patients with vascular anomalies from 1995 to 2021, and 144 patients met the inclusion criteria (74 [51.4%] male, 111 [77.1%] White), of whom 87 (60.4%) patients presented with rupture and 122 (84.7%) underwent resection. The mean age at surgery was 12 years (range 3 months to 20 years). The R2eD AVM scoring system was applied to each patient. The score components of size < 3 cm and exclusive deep venous drainage were significantly associated with rupture risk (p < 0.05). The complete model for the R2eD AVM score had an area under the curve of 0.671 (95% CI 0.586-0.757). CONCLUSIONS: This study demonstrated poor external validity of the R2eD AVM score in predicting pediatric AVM rupture risk. The results suggest that future studies are warranted to determine a better scoring method to capture pediatric rupture risk, given the significant differences in clinical presentation in pediatric compared with adult AVM patients.
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Malformaciones Arteriovenosas Intracraneales , Adulto , Humanos , Niño , Masculino , Lactante , Adulto Joven , Femenino , Estudios Retrospectivos , Malformaciones Arteriovenosas Intracraneales/complicaciones , Predicción , Modelos Logísticos , EncéfaloRESUMEN
BACKGROUND: The occurrence of both an intracranial aneurysm and epilepsy, especially drug-resistant epilepsy (DRE), is rare. Although the overall incidence of aneurysms associated with DRE is unclear, it is thought to be particularly infrequent in the pediatric population. Surgical ligation of the offending aneurysm has been reported in conjunction with resolving seizure activity, although few cases have cited a combined approach of aneurysm ligation and resection of an epileptogenic focus. OBSERVATIONS: We present the case of a 14-year-old female patient with drug-resistant temporal lobe epilepsy and an ipsilateral supraclinoid internal carotid artery aneurysm. Seizure semiology, electroencephalography monitoring, and magnetic resonance imaging all indicated a left temporal epileptogenic focus, in addition to an incidental aneurysm. The authors recommended a combined surgery involving resection of the temporal lesion and surgical clip ligation of the aneurysm. Near-total resection and successful ligation were achieved, and the patient has remained seizure free since surgery at 1 year postoperatively. LESSONS: In patients with focal DRE and an adjacent intracranial aneurysm, a combined surgical approach involving both resection and surgical ligation can be used. Several surgical timing and neuroanesthetic considerations should be made to ensure the overall safety and efficacy of this procedure.