Search Results

You are looking at 1 - 7 of 7 items for

  • Author or Editor: Laura M. Prolo x
  • Refine by Access: all x
Clear All Modify Search
Free access

Letter to the Editor. Posterior transdural approach for cervical stenosis caused by retroflexion of anterior elements in a child with Pfeiffer syndrome

Laura M. Prolo, David I. Bass, Jennifer M. Bauer, and Samuel R. Browd

Free access

Increased utilization of healthcare services in children with craniosynostosis

Gabriela D. Ruiz Colón, Michael C. Jin, Gerald A. Grant, and Laura M. Prolo

OBJECTIVE

Craniosynostosis is characterized by the premature fusion of at least one cranial suture. Although evidence suggests that patients with both syndromic and nonsyndromic craniosynostosis may benefit from developmental, behavioral, and mental health support, data on utilization of healthcare services are lacking. In this study the authors compared utilization of mental health care, rehabilitation therapies, and other specialty medical services among children with craniosynostosis, children with plagiocephaly, and healthy controls.

METHODS

The Optum Clinformatics Data Mart database was queried to identify 1340 patients with craniosynostosis, of whom 200 had syndromic craniosynostosis. Long-term utilization of mental health care, rehabilitation therapies, and other medical services up to the age of 6 years was calculated. Rates of utilization were compared to healthy controls (n = 1577) and children with plagiocephaly (n = 1249).

RESULTS

Patients with syndromic and nonsyndromic craniosynostosis used mental health care, occupational therapy, speech–language pathology, and other medical services at similar rates (p = 0.1198, p > 0.9999, p = 0.1097, and p = 0.8119, respectively). Mental health services were used more frequently by patients with craniosynostosis (11.0% in patients with syndromic craniosynostosis and 7.5% in those with nonsyndromic craniosynostosis) compared to patients in the plagiocephaly (5.0%, p = 0.0020) and healthy control (2.9%, p < 0.0001) cohorts. Rehabilitation therapies were more frequently used by patients with syndromic craniosynostosis and plagiocephaly (16.0% and 14.1%, respectively), which was significantly higher than use by healthy controls (p < 0.0001). Other medical subspecialty services (developmental pediatrics, ophthalmology, optometry, and audiology) were used by 37.0% of patients with craniosynostosis, compared with 20.9% (p < 0.0001) and 15.1% (p < 0.0001) of patients with plagiocephaly and healthy controls, respectively. Among patients with craniosynostosis, utilization did not differ by race or household income, but it was not uniform by age. Whereas ophthalmology utilization did not differ by age (p = 0.1003), mental health care was most commonly used among older children (p = 0.0107).

CONCLUSIONS

In this study, the authors demonstrate that rates of utilization of mental health care, rehabilitation therapies, and other medical subspecialty services are similar between patients with syndromic and those with nonsyndromic craniosynostosis, but higher than in healthy controls. Although surgical correction may be considered an isolated event, providers and parents need to monitor all children with craniosynostosis—syndromic and nonsyndromic—for developmental and mental health support longitudinally. Future work should explore risk factors driving utilization, including suture involvement, repair type, and comorbidities.

Free access

Recurrence of cavernous malformations after surgery in childhood

Laura M. Prolo, Michael C. Jin, Tina Loven, Hannes Vogel, Michael S. B. Edwards, Gary K. Steinberg, and Gerald A. Grant

OBJECTIVE

Cavernous malformations (CMs) are commonly treated cerebrovascular anomalies in the pediatric population; however, the data on radiographic recurrence of pediatric CMs after surgery are limited. The authors aimed to study the clinical presentation, outcomes, and recurrence rate following surgery for a large cohort of CMs in children.

METHODS

Pediatric patients (≤ 18 years old) who had a CM resected at a single institution were identified and retrospectively reviewed. Fisher’s exact test of independence was used to assess differences in categorical variables. Survival curves were evaluated using the Mantel-Cox method.

RESULTS

Fifty-three patients aged 3 months to 18 years underwent resection of 74 symptomatic CMs between 1996 and 2018 at a single institution. The median length of follow-up was 5.65 years. Patients most commonly presented with seizures (45.3%, n = 24) and the majority of CMs were cortical (58.0%, n = 43). Acute radiographic hemorrhage was common at presentation (64.2%, n = 34). Forty-two percent (n = 22) of patients presented with multiple CMs, and they were more likely to develop de novo lesions (71%) compared to patients presenting with a single CM (3.4%). Both radiographic hemorrhage and multiple CMs were independently prognostic for a higher risk of the patient requiring subsequent surgery. Fifty percent (n = 6) of the 12 patients with both risk factors required additional surgery within 2.5 years of initial surgery compared to none of the patients with neither risk factor (n = 9).

CONCLUSIONS

Patients with either acute radiographic hemorrhage or multiple CMs are at higher risk for subsequent surgery and require long-term MRI surveillance. In contrast, patients with a single CM are unlikely to require additional surgery and may require less frequent routine imaging.

Free access

Incorporating patient-centered quality-of-life measures for outcome assessment after Chiari malformation type I decompression in a pediatric population: a pilot study

Solomiia Savchuk, Michael C. Jin, Stephanie Choi, Lily H. Kim, Jennifer L. Quon, Anthony Bet, Laura M. Prolo, David S. Hong, Kelly B. Mahaney, and Gerald A. Grant

OBJECTIVE

Optimal management of pediatric Chiari malformation type I (CM-I) is much debated, chiefly due to the lack of validated tools for outcome assessment, with very few tools incorporating patient-centered measures of health-related quality of life (HRQOL). Although posterior fossa decompression (PFD) benefits a subset of patients, prediction of its impact across patients is challenging. The primary aim of this study was to investigate the role of patient-centered HRQOL measures in the assessment and prediction of outcomes after PFD.

METHODS

The authors collected HRQOL data from a cohort of 20 pediatric CM-I patients before and after PFD. The surveys included assessments of selected Patient-Reported Outcomes Measurement Information System (PROMIS) health domains and were used to generate the PROMIS preference (PROPr) score, which is a measure of HRQOL. PROMIS is a reliable standardized measure of HRQOL domains such as pain, fatigue, depression, and physical function, which are all relevant to CM-I. The authors then compared the PROPr scores with Chicago Chiari Outcome Scale (CCOS) scores derived from time-matched clinical documentation. Finally, the authors used the PROPr scores as an outcome measure to predict postsurgical HRQOL improvement at 1 year on the basis of patient demographic characteristics, comorbidities, and radiological and physical findings. The Wilcoxon signed-rank test, Mann-Whitney U-test, and Kendall’s correlation were used for statistical analysis.

RESULTS

Aggregate analysis revealed improvement of pain severity after PFD (p = 0.007) in anatomical patterns characteristic of CM-I. Most PROMIS domain scores trended toward improvement after surgery, with anxiety and pain interference reaching statistical significance (p < 0.002 and p < 0.03, respectively). PROPr scores also significantly improved after PFD (p < 0.008). Of the baseline patient characteristics, preexisting scoliosis was the most accurate negative predictor of HRQOL improvement after PFD (median −0.095 vs 0.106, p < 0.001). A correlation with modest magnitude (Kendall’s tau range 0.19–0.47) was detected between the patient-centered measures and CCOS score.

CONCLUSIONS

The authors observed moderate improvement of HRQOL, when measured using a modified panel of PROMIS question banks, in this pilot cohort of pediatric CM-I patients after PFD. Further investigations are necessary to validate this tool for children with CM-I and to determine whether these scores correlate with clinical and radiographic findings.

Free access

Development of an integrated risk scale for prediction of shunt placement after neonatal intraventricular hemorrhage

Michael C. Jin, Jonathon J. Parker, Adrian J. Rodrigues, Gabriela D. Ruiz Colón, Cesar A. Garcia, Kelly B. Mahaney, Gerald A. Grant, and Laura M. Prolo

OBJECTIVE

Neonatal intraventricular hemorrhage (IVH) is a major cause of mortality and morbidity, particularly following premature birth. Even after the acute phase, posthemorrhagic hydrocephalus is a long-term complication, frequently requiring permanent ventriculoperitoneal shunt (VPS) placement. Currently, there are no risk classification methods integrating the constellation of clinical data to predict short- and long-term prognosis in neonatal IVH. To address this need, the authors developed a two-part machine learning approach for predicting short- and long-term outcomes after diagnosis of neonatal IVH. Integrating both maternal and neonatal characteristics, they developed a binary classifier to predict short-term mortality risk and a clinical scale to predict the long-term risk of VPS placement.

METHODS

Neonates with IVH were identified from the Optum Clinformatics Data Mart administrative claims database. Matched maternal and childbirth characteristics were obtained for all patients. The primary endpoints of interest were short-term (30 day) mortality and long-term VPS placement. Classification of short-term mortality risk was evaluated using 5 different machine learning approaches and the best-performing method was validated using a withheld validation subset. Prediction of long-term shunt risk was performed using a multivariable Cox regression model with stepwise variable selection, which was subsequently converted to an easily applied integer risk scale.

RESULTS

A total of 5926 neonates with IVH were identified. Most patients were born before 32 weeks’ gestation (67.2%) and with low birth weight (81.2%). Empirical 30-day mortality risk was 10.9% across all IVH grades and highest among grade IV IVH (34.3%). Among the neonates who survived > 30 days, actuarial 12-month postdiagnosis risk of shunt placement was 5.4% across all IVH grades and 31.3% for grade IV IVH. The optimal short-term risk classifier was a random forest model achieving an area under the receiver operating characteristic curve of 0.882 with important predictors ranging from gestational age to diverse comorbid medical conditions. Selected features for long-term shunt risk stratification were IVH grade, respiratory distress syndrome, disseminated intravascular coagulation, and maternal preeclampsia or eclampsia. An integer risk scale, termed the Shunt Prediction After IVH in Neonates (SPAIN) scale, was developed from these 4 features, which, evaluated on withheld cases, demonstrated improved risk stratification compared with IVH grade alone (Harrell’s concordance index 0.869 vs 0.852).

CONCLUSIONS

In a large cohort of neonates with IVH, the authors developed a two-pronged, integrated, risk classification approach to anticipate short-term mortality and long-term shunt risk. The application of such approaches may improve the prognostication of outcomes and identification of higher-risk individuals who warrant careful surveillance and early intervention.

Free access

An integrated risk model stratifying seizure risk following brain tumor resection among seizure-naive patients without antiepileptic prophylaxis

Michael C. Jin, Jonathon J. Parker, Laura M. Prolo, Adela Wu, Casey H. Halpern, Gordon Li, John K. Ratliff, Summer S. Han, Stephen L. Skirboll, and Gerald A. Grant

OBJECTIVE

The natural history of seizure risk after brain tumor resection is not well understood. Identifying seizure-naive patients at highest risk for postoperative seizure events remains a clinical need. In this study, the authors sought to develop a predictive modeling strategy for anticipating postcraniotomy seizures after brain tumor resection.

METHODS

The IBM Watson Health MarketScan Claims Database was canvassed for antiepileptic drug (AED)– and seizure-naive patients who underwent brain tumor resection (2007–2016). The primary event of interest was short-term seizure risk (within 90 days postdischarge). The secondary event of interest was long-term seizure risk during the follow-up period. To model early-onset and long-term postdischarge seizure risk, a penalized logistic regression classifier and multivariable Cox regression model, respectively, were built, which integrated patient-, tumor-, and hospitalization-specific features. To compare empirical seizure rates, equally sized cohort tertiles were created and labeled as low risk, medium risk, and high risk.

RESULTS

Of 5470 patients, 983 (18.0%) had a postdischarge-coded seizure event. The integrated binary classification approach for predicting early-onset seizures outperformed models using feature subsets (area under the curve [AUC] = 0.751, hospitalization features only AUC = 0.667, patient features only AUC = 0.603, and tumor features only AUC = 0.694). Held-out validation patient cases that were predicted by the integrated model to have elevated short-term risk more frequently developed seizures within 90 days of discharge (24.1% high risk vs 3.8% low risk, p < 0.001). Compared with those in the low-risk tertile by the long-term seizure risk model, patients in the medium-risk and high-risk tertiles had 2.13 (95% CI 1.45–3.11) and 6.24 (95% CI 4.40–8.84) times higher long-term risk for postdischarge seizures. Only patients predicted as high risk developed status epilepticus within 90 days of discharge (1.7% high risk vs 0% low risk, p = 0.003).

CONCLUSIONS

The authors have presented a risk-stratified model that accurately predicted short- and long-term seizure risk in patients who underwent brain tumor resection, which may be used to stratify future study of postoperative AED prophylaxis in highest-risk patient subpopulations.

Free access

Stereotactic radiosurgery for recurrent pediatric brain tumors: clinical outcomes and toxicity

Elyn Wang, Paulina M. Gutkin, Justin Oh, Erqi Pollom, Scott G. Soltys, Gerald A. Grant, Laura M. Prolo, Steven Chang, Gordon Li, Paul G. Fisher, Sonia Partap, Cynthia J. Campen, Iris C. Gibbs, and Susan M. Hiniker

OBJECTIVE

Recurrence of brain tumors in children after the initial course of treatment remains a problem. This study evaluated the efficacy and safety of reirradiation using stereotactic radiosurgery (SRS) in patients with recurrent pediatric primary brain tumors.

METHODS

This IRB-approved retrospective review included pediatric patients with recurrent primary brain tumors treated at Stanford University from 2000 to 2019 using frameless SRS. Time to local failure (LF) and distant intracranial failure (DIF) were measured from the date of SRS and analyzed using competing risk analysis. Overall survival (OS) and progression-free survival (PFS) were analyzed with the Kaplan-Meier method.

RESULTS

In total, 37 patients aged 2–24 years (median age 11 years at recurrence) were treated for 48 intracranial tumors. Ependymoma (38%) and medulloblastoma (22%) were the most common tumor types. The median (range) single fraction equivalent dose of SRS was 16.4 (12–24) Gy. The median (range) follow-up time was 22.9 (1.5–190) months. The median OS of all patients was 36.8 months. Eight of 40 (20%) lesions with follow-up imaging locally recurred. The 2-year cumulative incidence of LF after reirradiation with SRS was 12.8% (95% CI 4.6%–25.4%). The 2-year cumulative incidence of DIF was 25.3% (95% CI 12.9%–39.8%). The median PFS was 18 months (95% CI 8.9–44). Five (10.4%) patients developed toxicities potentially attributed to SRS, including cognitive effects and necrosis.

CONCLUSIONS

Reirradiation using SRS for recurrent pediatric brain tumors appears safe with good local control. Innovations that improve overall disease control should continue because survival outcomes after relapse remain poor.