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OBJECTIVE: To determine practice variation in surgical management of co-morbidities in pediatric patients with Congenital Diaphragmatic Hernia (CDH). BACKGROUND: A higher percentage of CDH patients are surviving to discharge, accompanied by an increase in morbidity requiring surgical interventions such as tracheostomy and gastrostomy tube insertion. The frequency, trends, and regional variations in operative management of these co-morbidities in this population are unclear. METHODS: Neonates who underwent CDH repair between 2012-2022 in the United States Pediatric Health Information System database were identified. Multivariable regression identified predictive factors for additional surgical morbidity after CDH repair, defined by an additional surgical intervention during index hospitalization or within one year after discharge. To narrow the spectrum of severity of disease, only patients with an intensive care unit admission on index hospitalization were included. Secondary analysis compared frequency of operations and hospital resource utilization by region. RESULTS: 4003 patients underwent CDH repair and were discharged from their index hospitalization. 1939 (48%) underwent at least one additional surgical procedure after the index CDH repair. Most performed surgeries were gastrostomy tube (28%), fundoplication (13%), and tracheostomy (5%). Covariates associated with additional surgical morbidity included: prematurity (OR 1.38; 95% CI: 1.20-1.59), cardiac co-morbidity (OR 1.31; 95% CI: 1.14-1.49), and chromosomal anomalies (OR 1.76, 95% CI: 1.30-2.40). Northeast (OR: 2.43; CI 1.42-3.52), Midwest (OR 2.11; 95% CI: 1.45-3.07), and South (OR 1.45, 95% CI 1.02-2.12) regions were associated with additional surgical morbidity. Patients who required additional surgical procedures had longer initial inpatient length of stays (71 versus 31 d) and higher associated costs ($357,000 versus $161,000). CONCLUSIONS: Surgical morbidity exists in CDH patients after initial CDH repair. Counseling families on these outcomes is important in establishing expectations for management. Establishing guidelines for optimal surgical management will require continued reporting from multi-institutional studies.

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OBJECTIVES: Our study examines if the disease severity profile of our Congenital Diaphragmatic Hernia (CDH) patient cohort adherent to long-term follow-up differs from patients lost to follow-up after discharge and examines factors associated with health care utilization. METHODS: Retrospective review identified CDH survivors born 2005-2019 with index repair at our institution. Primary outcome was long-term follow-up status: "active" or "inactive" according to clinic guidelines. Markers of CDH disease severity including CDH defect classification, oxygen use, tube feeds at discharge, and sociodemographic factors were examined as exposures. RESULTS: Of the 222 included patients, median age [IQR] was 10.2 years [6.7-14.3], 61% male, and 57 (26%) were insured by Medicaid. Sixty-three percent (139/222) of patients were adherent to follow-up. Seventy-six percent of patients discharged on tube feeds had active follow-up compared to 55% of patients who were not, with similar findings for oxygen at discharge (76% vs. 55%). Kaplan-Meier analysis showed patients with smaller defect size had earlier attrition compared to patients with larger defect size. Other race (Hispanic, Asian, Middle Eastern) patients had 2.87 higher odds of attrition compared to white patients (95% CI 1.18-7.0). Medicaid patients had 2.64 higher odds of attrition compared to private insurance (95% CI 1.23-5.66). CONCLUSION: Loss to follow-up was associated with race and insurance type. Disease severity was similar between the active and inactive clinic cohorts. Long-term CDH clinic publications should examine attrition to ensure reported outcomes reflect the discharged population. This study identified important factors to inform targeted interventions for follow-up adherence. LEVEL OF EVIDENCE: Level III.

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Neonates with congenital diaphragmatic hernia encounter a number of surgical and medical morbidities that persist into adulthood. As mortality improves for this population, these survivors warrant specialized follow-up for their unique disease-specific morbidities. Multidisciplinary congenital diaphragmatic hernia clinics are best positioned to address these complex long-term morbidities, provide long-term research outcomes, and help inform standardization of best practices in this cohort of patients. This review outlines long-term morbidities experienced by congenital diaphragmatic hernia survivors that can be addressed in a comprehensive follow-up clinic.

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CASE: Popliteal artery entrapment syndrome (PAES) is rarely on the differential for exertional lower extremity pain in the young athlete. This article illustrates a case of a 20-year-old female National Collegiate Athletic Association (NCAA) Division-I college basketball player who was diagnosed with PAES after conservative treatment of medial tibial stress syndrome and comprehensive evaluation for chronic exertional compartment syndrome. She received bilateral popliteal artery releases through a posterior approach and made an asymptomatic return to Division-I collegiate basketball. CONCLUSION: PAES is a rare potentially limb-threatening disease that must be included on the differential of young athletes who present with exertional lower extremity pain.

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OBJECTIVE: HPV status is an important prognostic factor for patients with oropharyngeal, anal and cervical cancers treated with radiotherapy. This study evaluates the association between HPV and p16 status and outcome in a radiation-treated cohort with vulvar squamous cell carcinoma (SCC). METHODS: Patients with vulvar SCC who received radiotherapy with or without surgical resection between 1985 and 2011 were identified retrospectively. Immunostaining for p16 and multiplex PCR for HPV genotyping were performed using archival tumor tissue from 57 patients. Actuarial estimates of PFS, OS and in-field recurrence were calculated using the Kaplan-Meier method. Cox proportional hazards models were used for multivariable analysis. Median follow-up was 58months among the 57 patients with an available tumor specimen. RESULTS: HPV prevalence was implied in 37% by (diffuse linear) p16 immunostaining and confirmed in 27% by HPV PCR with good agreement (κ=0.7). HPV-16 was identified in 80% of HPV-positive tumors. Women with p16-positive tumors had significantly higher 5-year PFS (65% vs. 16%, p<0.01) and OS (65% vs. 22%, p=0.01) rates, as well as lower in-field relapse rates (19% vs. 75%, p<0.01) compared to those with p16-negative disease. On multivariable analysis adjusted for age and stage, p16 positivity was significantly associated with better PFS (HR 0.4, 95% CI 0.2-0.9) and lower rates of in-field relapse (HR 0.2, 95% CI 0.06-0.6). Results were similar when analyzed by HPV DNA status. CONCLUSION: In this study, the presence of HPV or its surrogate of p16 immunostaining was an independent prognostic factor for in-field relapse and survival in women with vulvar SCC treated with radiotherapy. This finding warrants validation in larger cohorts or the prospective setting.

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OBJECTIVE: Lower extremity lymphedema adversely affects quality of life by causing discomfort, impaired mobility and increased risk of infection. The goal of this study is to investigate factors that influence the likelihood of lymphedema in patients with endometrial cancer who undergo adjuvant radiation with or without chemotherapy. METHODS: A retrospective chart review identified all stage I-III endometrial cancer patients who had a hysterectomy with or without complete staging lymphadenectomy and adjuvant radiation therapy between January 2006 and February 2013. Patients with new-onset lymphedema after treatment were identified. Logistic regression was used to find factors that influenced lymphedema risk. RESULTS: Of 212 patients who met inclusion criteria, 15 patients (7.1%) developed new-onset lymphedema. Lymphedema was associated with lymph-node dissection (odds ratio [OR], 5.6; 95% CI, 1.01 to 105.5; p=0.048) and with the presence of pathologically positive lymph nodes (OR, 4.1; 95% CI, 1.4 to 12.3; p=0.01). Multivariate logistic regression confirmed the association with lymph-node positivity (OR, 3.2; 95% CI, 1.0007 to 10.7; p=0.0499) when controlled for lymph-node dissection. Median time to lymphedema onset was 8 months (range, 1 to 58 months) with resolution or improvement in eight patients (53.3%) after a median of 10 months. CONCLUSION: Lymph-node positivity was associated with an increased risk of lymphedema in endometrial cancer patients who received adjuvant radiation. Future studies are needed to explore whether node-positive patients may benefit from early lymphedema-controlling interventions.

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BACKGROUND AND PURPOSE: The goals of this study were to determine the rate and risk factors of rectal bleeding (RB) after external beam radiotherapy and vaginal brachytherapy (EBRT +VB), and to compare these data to previously unreported RB rates from PORTEC-2 patients receiving EBRT or VB alone. MATERIALS AND METHODS: Retrospective chart review identified 212 endometrial cancer patients receiving adjuvant EBRT+VB between 2006 and 2013. Patient-reported RB data were also obtained from PORTEC-2 patients randomized to EBRT (n=166) or VB (n=182). The two populations were compared using an RB scale of symptom severity. RESULTS: After a median 35months, 17.9% of EBRT+VB patients (n=38) experienced any RB with 1.9% (n=4) having bleeding requiring intervention. Age ⩽70years was the only predictor of RB (OR 2.8; 95% CI 1.1-8.7; p=0.027). Rates of patient-reported RB after EBRT were similar with 15.0% (n=25) having any RB and 0.6% (n=1) having "very much" bleeding. On regression analysis, any EBRT (either EBRT alone or EBRT+VB) increased the risk of RB compared to those who received VB alone (OR 3.0; p=0.0028; 95% CI 1.4-6.7). The rates of more severe RB were low and did not significantly differ between treatments. CONCLUSIONS: Significant RB is rare after radiation. EBRT has higher rates of rectal bleeding than VB. The addition of VB to EBRT does not significantly alter bleeding rates.

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OBJECTIVE: Uterine carcinosarcoma (CS) is an aggressive malignancy and the optimal adjuvant treatment is not well-established. We report outcomes with vaginal brachytherapy (VB) for women with early-stage CS. METHODS AND MATERIALS: A multi-institutional retrospective study of Stage I-II CS treated with hysterectomy, surgical staging, and adjuvant high-dose-rate VB without external-beam pelvic radiotherapy was performed. Rates of vaginal control, pelvic control, locoregional control, disease-free survival, and overall survival were determined using the Kaplan-Meier method. RESULTS: 33 patients were identified. Prescribed VB dose was 21 Gy in three fractions (n = 15 [45%]) or 24 Gy in six fractions (n = 18 [55%]). Eighteen patients (55%) received chemotherapy. Median followup was 2.0 years. Twenty-seven patients (82%) underwent pelvic lymphadenectomy, 5 (15%) had nodal sampling, and 1 (3%) had no lymph node assessment. Relapse occurred in 11 patients (33%), all of whom had lymph node evaluation. Locoregional relapse was a component of failure in 6 patients (18%), of whom 3 (9%) failed in the pelvis alone. Three patients (9%) had simultaneous distant and locoregional relapse (two vaginal, one pelvic). Five additional patients (15%) had distant relapse. Six of the 11 patients (55%) with disease recurrence received chemotherapy. Two-year vaginal control and pelvic control were 94% and 87%. Two-year locoregional control, disease-free survival, and overall survival were 81%, 66%, and 79%. CONCLUSIONS: Despite having early-stage disease and treatment with VB, patients in this series had relatively high rates of local and distant relapse. Patients who undergo lymphadenectomy and VB remain at risk for relapse. Novel treatment strategies are needed.

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