Publications by authors named "Randal Olshefski"

25 Publications

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A quest for meaning: A qualitative exploration among children with advanced cancer and their parents.

Psychooncology 2020 Nov 23. Epub 2020 Nov 23.

The Research Institute at Nationwide Children's Hospital, Columbus, Ohio, USA.

Objective: Meaning-making may assist individuals in adaptation to stressful life events, particularly bereavement. However, few studies have examined meaning-making among pediatric populations with advanced illness to understand how this process unfolds before the child's death. This study explores meaning-making pre-bereavement among children with advanced cancer and their parents.

Methods: As part of a larger study examining shared decision-making near the end of life, 24 children with advanced cancer and/or high-risk cancer, 26 mothers, and 11 fathers participated in individual, semi-structured interviews. Analyses focused on questions regarding meaning-making. Four coders analyzed the data via directed content analysis.

Results: Three major meaning-making themes emerged: (1) sense-making (i.e., unknown, no sense/meaning, religious/spiritual explanations, scientific explanations), (2) benefit-finding, and (3) purpose/legacy. Some stated they were unable to make sense of the diagnosis, because there was no reason, they were not there yet, or they were dealing with the situation and moving forward. Others reported finding meaning through spiritual and scientific explanations. Many identified benefits related to the child's illness, such as personal growth and stronger relationships. Some parents expressed their purpose in life was to live for their children, while others shared their child's legacy as a way to find meaning.

Conclusions: Our findings highlight the struggle children and parents often face when attempting to make sense of the child's advanced or high-risk illness. Clinicians might consider if meaning-centered interventions designed for use in adults at the end of life and bereaved parents may be helpful for children with advanced or high-risk cancer and their parents.
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http://dx.doi.org/10.1002/pon.5601DOI Listing
November 2020

Impact of a Best Practice Prevention Bundle on Central Line-associated Bloodstream Infection (CLABSI) Rates and Outcomes in Pediatric Hematology, Oncology, and Hematopoietic Cell Transplantation Patients in Inpatient and Ambulatory Settings.

J Pediatr Hematol Oncol 2021 Jan;43(1):e64-e72

Department of Pediatrics, Division of Infectious Diseases.

Background: Pediatric hematology, oncology, and hematopoietic cell transplantation (HCT) patients are at increased risk for bloodstream infections. The authors sought to evaluate the influence of a standardized best practice central venous catheter (CVC) maintenance bundle on the burden of and risk factors for mucosal barrier injury (MBI) and non-MBI central line-associated bloodstream infections (CLABSIs) across a common inpatient and ambulatory continuum in this high-risk population.

Methods: A retrospective cohort study of patients with underlying malignancy, hematologic disorders, and HCT recipients with a CVC in place at the time of CLABSI diagnosis in both inpatient and ambulatory settings from January 1, 2012 to December 31, 2016. Descriptive, nonparametric statistics were used to describe patient characteristics and outcomes. Logistic regression analyses were applied to identify potential risk factors for inpatient versus ambulatory and MBI versus non-MBI CLABSI.

Results: During the 5-year period, 118 of 808 (14.6%) patients had 159 laboratory-confirmed CLABSIs for ambulatory and inpatient CLABSI rates of 0.27 CLABSI/1000 and 2.2 CLABSI/1000 CVC days, respectively. CLABSI occurred more frequently in hospitalized patients after HCT and with underlying leukemia, most frequently caused by Gram-negative bacteria. MBI CLABSI accounted for 42% of all CLABSI with a 3-fold higher risk in hospitalized patients. Having multiple CVC or a CVC that was not a port independently associated with higher CLABSI risk.

Conclusions: In our cohort, non-MBI CLABSI continued to account for the majority of CLABSI. CVC type is independently associated with higher overall CLABSI risk. Further studies are needed to reliably define additional prevention strategies when CLABSI maintenance bundles elements are optimized in this high-risk population.
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http://dx.doi.org/10.1097/MPH.0000000000001950DOI Listing
January 2021

Excellent outcome of young children with nodular desmoplastic medulloblastoma treated on "Head Start" III: a multi-institutional, prospective clinical trial.

Neuro Oncol 2020 12;22(12):1862-1872

Division of Pediatric Hematology-Oncology, Nationwide Children's Hospital, Columbus, Ohio, USA.

Background: "Head Start" III, was a prospective clinical trial using intensive induction followed by myeloablative chemotherapy and autologous hematopoietic cell rescue (AuHCR) to either avoid or reduce the dose/volume of irradiation in young children with medulloblastoma.

Methods: Following surgery, patients received 5 cycles of induction followed by myeloablative chemotherapy using carboplatin, thiotepa, and etoposide with AuHCR. Irradiation was reserved for children >6 years old at diagnosis or with residual tumor post-induction.

Results: Between 2003 and 2009, 92 children <10 years old with medulloblastoma were enrolled. Five-year event-free survival (EFS) and overall survival (OS) rates (±SE) were 46 ± 5% and 62 ± 5% for all patients, 61 ± 8% and 77 ± 7% for localized medulloblastoma, and 35 ± 7% and 52 ± 7% for disseminated patients. Nodular/desmoplastic (ND) medulloblastoma patients had 5-year EFS and OS (±SE) rates of 89 ± 6% and 89 ± 6% compared with 26 ± 6% and 53 ± 7% for classic and 38 ± 13% and 46 ± 14% for large-cell/anaplastic (LCA) medulloblastoma, respectively. In multivariate Cox regression analysis, histology was the only significant independent predictor of EFS after adjusting for stage, extent of resection, regimen, age, and sex (P <0.0001). Five-year irradiation-free EFS was 78 ± 8% for ND and 21 ± 5% for classic/LCA medulloblastoma patients. Myelosuppression was the most common toxicity, with 2 toxic deaths. Twenty-four survivors completed neurocognitive evaluation at a mean of 4.9 years post-diagnosis. IQ and memory scores were within average range overall, whereas processing speed and adaptive functioning were low-average.

Conclusion: We report excellent survival and preservation of mean IQ and memory for young children with ND medulloblastoma using high-dose chemotherapy, with most patients surviving without irradiation.
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http://dx.doi.org/10.1093/neuonc/noaa102DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7746930PMC
December 2020

Romantic Relationships and Physical Intimacy Among Survivors of Childhood Cancer.

J Adolesc Young Adult Oncol 2020 06 13;9(3):359-366. Epub 2019 Nov 13.

Center for Biobehavioral Health, The Abigail Wexner Research Institute, Nationwide Children's Hospital, Columbus, Ohio, USA.

Childhood cancer survivors are at risk for impaired psychosocial functioning, but limited research has focused on psychosexual outcomes in young adulthood. This qualitative study examined the perceived impact of childhood cancer on adult survivors' romantic relationships and sexual/physical intimacy. Phone interviews were completed with adult survivors of childhood cancer, exploring the impact of cancer on (1) romantic relationships and (2) sexual/physical intimacy. Verbatim transcripts were coded using thematic content analysis until saturation was confirmed ( = 40). Survivors in this study ( = 40) were 23-42 years old ( = 29.8; 63% female) and 10-37 years postdiagnosis ( = 18.4). Regarding romantic relationships, 60% of participants reported a negative impact, while 55% of participants reported positive effects; ∼25% of participants reported no impact of childhood cancer on adult romantic relationships. Negative themes included fertility-related concerns, physical effects (e.g., self-consciousness), feeling emotionally guarded, and delayed dating. Positive themes were creating new perspectives, increased maturity, and stronger bonds with partners. Forty percent of survivors in this study perceived having fewer partners than peers. Regarding sexual/physical intimacy, 68% of participants reported a negative impact (themes: body image, fertility-related concerns, sexual/physical dysfunction), while 33% of participants reported no effects. This study demonstrates both positive and negative effects of childhood cancer on adult survivors' romantic relationships, whereas effects on physical intimacy were predominantly negative. Further research is needed to inform effective psychosexual interventions, and health care providers should routinely address these topics in survivorship care.
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http://dx.doi.org/10.1089/jayao.2019.0114DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7869868PMC
June 2020

A Quality Improvement Approach to Increase Exercise Assessment in Survivors of Childhood Leukemia.

Pediatr Qual Saf 2019 Jul-Aug;4(4):e198. Epub 2019 Jul 29.

Division of Pediatric Hematology/Oncology/BMT, Nationwide Children's Hospital, Columbus, Ohio.

Introduction: Survivors of childhood cancer are at increased risk of treatment-related cardiovascular disease, the severity of which is impacted by the level of regular exercise. Exercise assessments (EAs) are not a routine component of follow-up care.

Methods: We incorporated a quantitative EA tool into the clinic triage during follow-up visits for survivors of acute lymphoblastic leukemia. The nursing staff was surveyed on the use of the EA tool to gauge understanding and level of comfort with addressing patient questions.

Results: Over 27 months, the percentage of off-therapy acute lymphoblastic leukemia patients with documented EA increased from 0% to 80%. We noted degradation in EA completions in the last 6 months of the project, which we attributed to project nursing staff transition and failure to maintain education. Interventions that improved the percentage of completed EA included the incorporation the assessment tool into the electronic medical record and weekly reminders of scheduled eligible patients. A nurse incentive plan did not impact project success. Survey results revealed that the nursing staff were comfortable with the EA and did not view the new process as hurting patient flow.

Conclusion: Implementation of an EA tool into routine clinic follow-up was successful. We met the project goal of assessing greater than 50% of the follow-up patients. This work will serve as the foundation for the next phase of the project, which will be to provide education on the importance of exercise and earlier intervention when a sedentary lifestyle is identified.
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http://dx.doi.org/10.1097/pq9.0000000000000198DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6708644PMC
July 2019

Germinoma Involving the Retina: An Unusual Presentation of Recurrent Intracranial Mixed Germ Cell Tumor.

World Neurosurg 2019 Jan 7. Epub 2019 Jan 7.

The Department of Pathology & Laboratory Medicine, Nationwide Children's Hospital, Columbus, OH 43205 and The Department of Pathology, The Ohio State University, College of Medicine, Columbus, OH 43210 USA.

Background: We report a patient with primary central nervous system mixed malignant germ cell tumor (GCT) who presented with recurrent malignant germinomatous infiltration of the retina.

Case Description: A ten-year-old girl initially presented with a large suprasellar mixed malignant GCT with a near-complete response after initial induction chemotherapy and irradiation. Three and half years after initial therapy, she presented with progressively worsening vision in her left eye. Magnetic resonance imaging showed infiltrative changes within the left optic nerve but no discrete mass. Serum and cerebrospinal fluid (CSF) tumor markers were not elevated and CSF cytology was negative. Left optic nerve biopsy confirmed the presence of mature teratoma and pure germinoma components. She was treated with gross-total resection of the left eye and optic nerve and chemotherapy. Histopathologic evaluation of the optic nerve showed only mature teratoma elements but with pure germinoma cells infiltrating the inner layers of the retina.

Conclusions: Loco-regional extension of suprasellar GCT to the optic nerve is not uncommon; however, infiltration of the tumor into the retina is not reported in the literature. Early detection of optic pathway involvement and proper delineation of the irradiation field may prevent GCT infiltration of the retina with subsequent vision loss.
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http://dx.doi.org/10.1016/j.wneu.2018.12.143DOI Listing
January 2019

Trajectories of health behaviors across early childhood cancer survivorship.

Psychooncology 2019 01 6;28(1):68-75. Epub 2018 Nov 6.

Center for Biobehavioral Health, The Research Institute at Nationwide Children's Hospital, Columbus, Ohio.

Objective: The majority of childhood cancer survivors develop at least one late effect subsequent to treatment (eg, cardiovascular disease and obesity). Consistent engagement in recommended health behaviors may mitigate some of these conditions. Researchers have identified early survivorship as a teachable moment, yet few studies have examined positive health behaviors during this period.

Methods: Families of children with cancer (ages 5-17) were initially recruited following a diagnosis or relapse of cancer. Three years post diagnosis, survivors (n = 82, M  = 13.3, SD = 3.7) and their mothers (n = 103, M  = 41.1, SD = 7.6) completed a questionnaire assessing exercise, dietary, and sleep patterns among survivors. A follow-up assessment was conducted 2 years later. Mixed models tested change in health behavior over time.

Results: At 3- and 5-year post diagnosis, mother and self-report indicated that few survivors engaged in appropriate levels of low-intensity exercise, fruit/vegetable intake, and dairy consumption. However, most survivors engaged in recommended levels of high intensity exercise, fast food restriction, and sleep. Health behaviors remained stable over time, except for mother report of sleep duration, which decreased (b = -0.6, P < 0.001). Brain tumor diagnosis predicted a larger decrease in self-report of sleep duration compared with other diagnoses (P = 0.04). Income predicted fast food intake such that higher income was associated with decreased intake over time, whereas lower income was associated with increased intake (P = 0.04).

Conclusions: During early survivorship, several health behaviors fell short of expectations for exercise and diet and did not improve upon reaching 5-year post diagnosis. Providers should evaluate survivors' health behaviors, including sleep, early and often, intervening when necessary.
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http://dx.doi.org/10.1002/pon.4911DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6545194PMC
January 2019

Complete Remission of an Extracranially Disseminated Anaplastic Pleomorphic Xanthoastrocytoma With Everolimus: A Case Report and Literature Review.

Pediatr Neurol 2018 11 13;88:65-70. Epub 2018 Sep 13.

Division of Hematology, Oncology and Bone Marrow Transplant, Nationwide Children's Hospital, Columbus, Ohio. Electronic address:

Background: Surgical resection is the treatment of choice for pleomorphic xanthoastrocytoma, while chemotherapy and radiation therapy are typically used in patients with anaplasia, metastasis, or sometimes in subtotally resected cases, especially upon recurrence. Extracranial dissemination has been only rarely reported. We describe a five year old boy with the rare occurrence multiply recurrent and extracranially disseminated anaplastic pleomorphic xanthoastrocytoma. A complete resolution of his tumor was achieved for greater than two years thus far after administering everolimus.

Methods: We performed a comprehensive literature review of all pleomorphic xanthoastrocytoma cases; 359 cases were described, and 132 of these individuals were less than 18 years of age.

Results: Gross total resection was achieved in only 132 (36.7%) cases, while additional therapy was administered in 186 patients. Only four patients in additon to our own have been documented with extracranial dissemination (four of five in the pediatric population); two patients who succumbed to their disease underwent subtotal resection of the primary tumor.

Conclusions: We report the first patient with extracranially disseminated anaplastic pleomorphic xanthoastrocytoma to be successfully maintained on everolimus as a single oral chemotherapy agent with complete resolution of the tumor. Pleomorphic xanthoastrocytoma can rarely disseminate extracranially in the pediatric population, hence pathologists and neuro-oncologists should be aware of this possibility.
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http://dx.doi.org/10.1016/j.pediatrneurol.2018.09.004DOI Listing
November 2018

A multiyear quality improvement project to increase influenza vaccination in a pediatric oncology population undergoing active therapy.

Pediatr Blood Cancer 2018 09 1;65(9):e27268. Epub 2018 Jun 1.

Division of Pediatric Hematology/Oncology, Nationwide Children's Hospital/Department of Pediatrics, The Ohio State University College of Medicine, Columbus, Ohio.

Background: In an effort to reduce morbidity and mortality from vaccine preventable influenza infection, national consensus guidelines recommend vaccination of patients who are immunocompromised as a result of receiving cancer therapy. Quality improvement (QI) processes are a proven method used to improve vaccination rates.

Procedure: We conducted a QI initiative aimed at increasing influenza vaccination in oncology patients undergoing active treatment. Primary drivers for the project focused on patient education, staff and provider education, and communication regarding vaccine-eligible patients. We performed a retrospective analysis of influenza infection among the vaccine-eligible population. This approach has validity at our institution because of the consistent follow-up and hospital admission pattern of cancer patients on active therapy such that nearly all follow-up care is delivered at our institution.

Results: We successfully achieved greater than 87% vaccination of eligible patients each vaccine season (September to March). During the recommended timeframe for delivering influenza vaccine between September and December of each vaccine season, we offered the vaccine to 100% of patients on active therapy and vaccinated >90%. Barriers to success, including vaccine refusals, increased late in the vaccine season. Influenza infection was documented in 0.5-7.3% of the vaccine-eligible group.

Conclusion: A robust influenza vaccination program implemented using a standardized QI approach can sustain a high vaccination rate in a pediatric oncology population receiving active treatment. The influenza infection rate was under 10% in the vaccinated group.
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http://dx.doi.org/10.1002/pbc.27268DOI Listing
September 2018

Psychosexual development and satisfaction with timing of developmental milestones among adult survivors of childhood cancer.

Psychooncology 2018 08 16;27(8):1944-1949. Epub 2018 May 16.

Center for Biobehavioral Health, The Research Institute at Nationwide Children's Hospital, Columbus, OH, USA.

Objective: To extend the limited research on psychosexual development among childhood cancer survivors, by not only focusing on the prevalence and age of milestone attainment, but also survivors' attitudes toward the timing of reaching such milestones.

Methods: Adult survivors of childhood cancer (N = 90; M  = 29.8, SD = 5.2), recruited from a US pediatric institution, completed online surveys indicating whether they had reached 5 milestones of psychosexual development (ie, first kiss, first boy-/girlfriend, first physical intimacy, sexual debut, first time in love), age at attainment, and perceptions about the timing (ie, right time, wished it had happened earlier, wished they had waited).

Results: Almost all survivors had reached each milestone (≥90%), except for sexual debut (83.3%). Survivors reported their first kiss as the earliest milestone at age 14.6 (N = 82, 92%) and falling in love as the latest milestone at age 18.8 (N = 80; 90%). This timing did not differ by sex/cancer-specific factors. Most survivors (~60%) felt they reached each milestone at the right time. Compared with US normative data, both male and female survivors were less likely to have experienced their sexual debut and were approximately 1.5 years older at sexual debut. Nevertheless, 59% of survivors felt that this timing was right and 31% wished they had waited longer.

Conclusions: This is the first study to demonstrate that although childhood cancer survivors may delay some aspects of psychosexual development, most are satisfied with this timing. Research and clinical practice should emphasize survivors' perceptions/satisfaction toward psychosexual development rather than focusing only on normative milestone attainment.
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http://dx.doi.org/10.1002/pon.4746DOI Listing
August 2018

Clinical Indices Can Standardize and Monitor Pediatric Care: A Novel Mechanism to Improve Quality and Safety.

J Pediatr 2018 02 6;193:190-195.e1. Epub 2017 Dec 6.

Department of Pediatrics, The Ohio State University College of Medicine, Columbus, OH; Divison of Pediatric Critical Care Medicine, Department of Pediatrics, Nationwide Children's Hospital, Columbus, OH.

Objective: The Cancer Care Index (CCI), a single metric that sums the number of undesirable patient events in a given time frame (either preventable harm events or missed opportunities to provide optimal care), resulted in a 42% improvement in performance. Our objective was to test the index concept in other service lines to determine whether similar performance improvement occurred.

Study Design: Care indices were developed and introduced in 3 additional service lines: Nephrology (Chronic Kidney Disease Care Index; CKDCI), Pulmonology (Lung Transplantation Care Index; LTCI), and Otolaryngology (Tracheostomy Care Index; TCI). After reaching agreement on specific harms to be avoided and elements of optimal care that should be reliably delivered, these items were compiled into indices that were updated monthly. Reports included each element individually and the total for all elements. Baseline performance was calculated retrospectively for the previous year.

Results: Significant improvement in performance occurred in each program following implementation of the clinical indices. The CKDCI was decreased by 63.2% (P < .001), the LTCI was decreased by 89.5% (P < .001), and the TCI was decreased by 53.0% (P < .001). Surveyed staff indicated satisfaction with use of the metric.

Conclusions: Clinical indices are useful for evaluating and managing the overall reliability of a program's ability to deliver optimal care, and are associated with improved clinical performance and satisfaction by service line staff when incorporated into a program's operation.
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http://dx.doi.org/10.1016/j.jpeds.2017.09.073DOI Listing
February 2018

Characterizing temporal genomic heterogeneity in pediatric high-grade gliomas.

Acta Neuropathol Commun 2017 10 30;5(1):78. Epub 2017 Oct 30.

Department of Human Genetics, McGill University, Montreal, QC, H3A 1B1, Canada.

Pediatric high-grade gliomas (pHGGs) are aggressive neoplasms representing approximately 20% of brain tumors in children. Current therapies offer limited disease control, and patients have a poor prognosis. Empiric use of targeted therapy, especially at progression, is increasingly practiced despite a paucity of data regarding temporal and therapy-driven genomic evolution in pHGGs. To study the genetic landscape of pHGGs at recurrence, we performed whole exome and methylation analyses on matched primary and recurrent pHGGs from 16 patients. Tumor mutational profiles identified three distinct subgroups. Group 1 (n = 7) harbored known hotspot mutations in Histone 3 (H3) (K27M or G34V) or IDH1 (H3/IDH1 mutants) and co-occurring TP53 or ACVR1 mutations in tumor pairs across the disease course. Group 2 (n = 7), H3/IDH1 wildtype tumor pairs, harbored novel mutations in chromatin modifiers (ZMYND11, EP300 n = 2), all associated with TP53 alterations, or had BRAF V600E mutations (n = 2) conserved across tumor pairs. Group 3 included 2 tumors with NF1 germline mutations. Pairs from primary and relapsed pHGG samples clustered within the same DNA methylation subgroup. ATRX mutations were clonal and retained in H3G34V and H3/IDH1 wildtype tumors, while different genetic alterations in this gene were observed at diagnosis and recurrence in IDH1 mutant tumors. Mutations in putative drug targets (EGFR, ERBB2, PDGFRA, PI3K) were not always shared between primary and recurrence samples, indicating evolution during progression. Our findings indicate that specific key driver mutations in pHGGs are conserved at recurrence and are prime targets for therapeutic development and clinical trials (e.g. H3 post-translational modifications, IDH1, BRAF V600E). Other actionable mutations are acquired or lost, indicating that re-biopsy at recurrence will provide better guidance for effective targeted therapy of pHGGs.
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http://dx.doi.org/10.1186/s40478-017-0479-8DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5663045PMC
October 2017

Psychosexual development and satisfaction in long-term survivors of childhood cancer: Neurotoxic treatment intensity as a risk indicator.

Cancer 2017 05 6;123(10):1869-1876. Epub 2017 Feb 6.

Center for Biobehavioral Health, The Research Institute at Nationwide Children's Hospital, Columbus, Ohio.

Background: Risk factors for impairment in psychosexual development and satisfaction among adult survivors of childhood cancer are poorly understood. The authors compared psychosexual outcomes between survivors and healthy controls, and tested whether at-risk survivors can be identified by 1) treatment neurotoxicity or 2) diagnosis.

Methods: A total of 144 young adult survivors of childhood cancer and 144 matched controls completed questionnaires regarding psychosexual development, sexual satisfaction, and satisfaction with relationship status. Survivors were aged 20 to 40 years and were 5 to 34 years after diagnosis. Using medical chart data, survivors were divided into non-neurotoxic (48 survivors), low-dose (36 survivors), and high-dose (58 survivors) neurotoxic treatment groups.

Results: Apart from having fewer lifetime sex partners, survivors did not appear to differ from controls. However, survivors of brain tumors and any survivor who received high-dose neurotoxic treatment reported the lowest rates of achieving milestones of psychosexual development, whereas sexual and relationship status satisfaction were found to be related to relationship status. Neurotoxic treatment intensity further distinguished between survivors of brain tumors with and without psychosexual impairment.

Conclusions: The intensity of neurotoxic treatment may be a valuable indicator of risk for psychosexual impairment relative to diagnosis alone. Health care providers should assess romantic/sexual problems among survivors at risk and make referrals if needed. Cancer 2017;123:1869-1876. © 2017 American Cancer Society.
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http://dx.doi.org/10.1002/cncr.30513DOI Listing
May 2017

Family estimates of risk for neurocognitive late effects following pediatric cancer: From diagnosis through the first three years of survivorship.

Pediatr Blood Cancer 2017 Sep 25;64(9). Epub 2017 Jan 25.

Center for Biobehavioral Health, The Research Institute at Nationwide Children's Hospital, Columbus, Ohio.

Background: Families often express a need for additional information about neurocognitive late effects (NCLE) after a pediatric cancer diagnosis. Therefore, we examined: (i) differences in parent, child, and oncologist estimates of risk for NCLE; (ii) whether the estimates of parents and/or children change over time; and (iii) whether estimates are different for children treated with central nervous system (CNS) directed therapies.

Procedure: Mothers, fathers, and children (initial age: 5-17, self-report: >10) from 258 families reported their perceived likelihood of the child developing "thinking/learning problems" on a visual analog scale (0-100%) at 2 months (T1), 1 year (T2), and 3 years (T3) following cancer diagnosis/relapse. Oncologists estimated the likelihood of NCLE at T1. Children were separated into groups based on CNS-directed treatment (n = 137; neurosurgery, intrathecal chemotherapy, and/or craniospinal radiation) or no CNS treatment.

Results: Mother, father, and child estimates of risk for NCLE were similar to oncologists and to one another around diagnosis (T1). Although there were no significant mean differences, a considerable subset of family members either underestimated their child's risk for NCLE (>40%) or overestimated the risk for NCLE (20%) in comparison to oncologists. At T2 and T3, the estimates of mothers were significantly higher than children. Linear growth curves indicated that mothers' estimates for children with CNS-directed treatment significantly increased throughout the first 3 years of survivorship.

Conclusions: Considering that accurate understanding of NCLE is essential to seeking appropriate assessment and intervention, healthcare providers should focus on implementing family-based education early in treatment and throughout survivorship care.
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http://dx.doi.org/10.1002/pbc.26462DOI Listing
September 2017

The Cancer Care Index: A Novel Metric to Assess Overall Performance of a Pediatric Oncology Program.

J Patient Saf 2020 09;16(3):e120-e125

Hospital Administration.

Objective: Childhood cancer metrics are currently primarily focused on survival rates and late effects of therapy. Our objectives were to design and test a metric that reflected overall quality and safety performance, across all cancer types, of an oncology-bone marrow transplant service line and to use the metric to drive improvement.

Method: The Cancer Care Index (CCI) aggregates adverse safety events and missed opportunities for best practices into a composite score that reflects overall program performance without regard to cancer type or patient outcome. Fifteen domains were selected in 3 areas as follows: (1) treatment-related quality and safety, (2) provision of a harm-free environment, and (3) psychosocial support. The CCI is the aggregate number of adverse events or missed opportunities to provide quality care in a given time frame. A lower CCI reflects better care and improved overall system performance. Multidisciplinary microsystem-based teams addressed specific aims for each domain. The CCI was widely followed by all team members, particularly frontline providers.

Results: The CCI was easy to calculate and deploy and well accepted by the staff. The annual CCI progressively decreased from 278 in 2012 to 160 in 2014, a 42% reduction. Improvements in care were realized across most index domains. Multiple new initiatives were successfully implemented.

Conclusions: The CCI is a useful metric to document performance improvement across a broad range of domains, regardless of cancer type. By the use of quality improvement science, progressive reduction in CCI has occurred over a 3-year period.
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http://dx.doi.org/10.1097/PTS.0000000000000267DOI Listing
September 2020

Comparison of Thyroid Nodule Prevalence by Ultrasound in Childhood Cancer Survivors With and Without Thyroid Radiation Exposure.

J Pediatr Hematol Oncol 2016 Jan;38(1):43-8

*Division of Pediatric Hematology/Oncology ‡Department of Pediatric Endocrinology, Nationwide Children's Hospital/The Ohio State University †Center for Gene Therapy ∥Center for Innovation in Pediatric Practice, The Research Institute at Nationwide Children's Hospital §Department of Surgery, Division of Pediatric Surgery, Nationwide Children's Hospital, The Ohio State University College of Medicine, Columbus, OH.

Purpose: Children receiving radiotherapy of the head, neck, or chest as treatment of primary malignancies are at increased risk for secondary thyroid malignancy. We hypothesized that current standards (yearly thyroid physical examination) lead to a substantial number of missed thyroid nodules. Our objectives were: (1) use thyroid ultrasound to assess thyroid nodules in childhood cancer survivors; and (2) compare prevalence of thyroid nodules in thyroid radiation-exposed patients as compared with cancer survivors without radiation exposure.

Methods: We recruited 60 patients with thyroid radiation and chemotherapy exposure (median age at cancer diagnosis 10.8 y) and 59 patients with chemotherapy exposure alone (median age at diagnosis 4.3 y) from our long-term survivorship clinics. Each patient had a thyroid physical examination and thyroid ultrasound performed.

Results: Thirty-three patients (27.7%) had nodules >0.3 cm of which 2 were palpated (6.1%). We found 22 radiated patients (36.7%) with nodules versus 11 nonradiated patients (18.6%) (P=0.03). Eleven patients were biopsied and 1 diagnosis of secondary papillary thyroid carcinoma was confirmed.

Conclusion: Our study supports further examination of incorporating thyroid ultrasounds into long-term survivorship follow-up guidelines in radiation-exposed patients for the detection of thyroid nodules and secondary malignancies.
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http://dx.doi.org/10.1097/MPH.0000000000000473DOI Listing
January 2016

Body issues, sexual satisfaction, and relationship status satisfaction in long-term childhood cancer survivors and healthy controls.

Psychooncology 2016 Feb 8;25(2):210-6. Epub 2015 May 8.

Health Psychology Research Section, University of Groningen, University Medical Center Groningen (UMCG), Groningen, The Netherlands.

Objective: Research on body image and sexual satisfaction after adult onset cancer has shown significant and lasting impairments regarding survivors' sexuality and romantic relationships. However, knowledge about these topics and their associations in adult survivors of childhood cancer is largely lacking.

Methods: Participants completed web-based questionnaires concerning body image, body dissociation, sexual satisfaction, and relationship status satisfaction (i.e., satisfaction with either being in a relationship or being single). Survivors (n = 87) and controls (n = 87) were matched on age and gender, with a mean age of 27 years (range: 20-40). Survivors were most often diagnosed with leukemia (46%), at an average of 16 years prior to study participation (range: 6-33 years).

Results: Similar numbers of survivors and controls were single (n = 24/31), in a committed relationship (n = 33/23), or married (n = 30/33). Survivors and controls reported comparable levels of body image, body dissociation, sexual experiences, and sexual and status satisfaction (d = 0.15-0.28). Higher status satisfaction was associated with being in a relationship (compared with being single, β = 0.439), more positive body image (β = 0.196), and higher sexual satisfaction (β = 0.200).

Conclusions: Adult survivors of childhood cancer were comparable to healthy peers regarding views of their bodies and psychosexual development, which was unexpected. Independent of whether people experienced cancer or not, their status satisfaction was associated with their relationship status, body image, and sexual satisfaction. Future research should explore why sexual and body problems are identified after adult onset cancer, whereas this seems to be less of a problem in childhood cancer survivors.
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http://dx.doi.org/10.1002/pon.3841DOI Listing
February 2016

A multi-institutional experience in pediatric high-grade glioma.

Front Oncol 2015 18;5:28. Epub 2015 Feb 18.

Department of Radiation Oncology, The Ohio State University Wexner Medical Center , Columbus, OH , USA.

Introduction: Pediatric high-grade gliomas are rare tumors with poor outcomes and incompletely defined management. We conducted a multi-institutional retrospective study to evaluate association of clinical, pathologic, and treatment characteristics with outcomes.

Materials And Methods: Fifty-one patients treated from 1984 to 2008 at the Ohio State University or University of Michigan were included. Histologic subgroups were compared. Log-rank and stepwise Cox proportional hazard modeling were used to analyze progression-free survival (PFS) and overall survival (OS) within the whole group, grade III subgroup, grade IV subgroup, and sub-total resection/biopsy subgroup.

Results: Median OS was 27.6 months. Grade III histology, complete tumor resection, and cerebral tumor location correlated with improved PFS and OS. Temozolomide use and chemotherapy after radiotherapy or chemoradiation (CRT) were associated with better PFS while seizure at presentation was associated with better OS. In multivariate analysis, complete resection and chemotherapy following radiotherapy or CRT were independent predictors for improved PFS and OS. For grade III and IV subgroups, complete resection was associated with improved OS (grade III) and seizure presentation was associated with improved OS (grade IV). In the incompletely resection subgroup, temozolomide use and concurrent CRT independently correlated with improved PFS, while higher radiation dose (≥59.4 Gy) and adjuvant chemotherapy were independently associated with improved OS.

Discussion: Total resection and receiving chemotherapy adjuvant to radiation or CRT are most closely associated with improved PFS and OS. For higher risk incompletely resected patients, temozolomide use and treatment intensification with concurrent CRT, adjuvant chemotherapy, and higher radiation dose were associated with improved outcomes.
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http://dx.doi.org/10.3389/fonc.2015.00028DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4332307PMC
March 2015

Late effects in survivors of childhood CNS tumors treated on Head Start I and II protocols.

Pediatr Blood Cancer 2014 Sep 1;61(9):1644-52; quiz 1653-72. Epub 2014 May 1.

New York University Langone Medical Center, New York, New York.

Background: Due to the devastating late effects associated with cranial irradiation in young children with central nervous system (CNS) tumors, treatment for these patients has evolved to include the use of intensive chemotherapy to either avoid or postpone irradiation. While survival outcomes have improved, late effects data in survivors treated on such regimens are needed.

Objective: This multi-institutional study comprehensively describes late effects in survivors treated on the Head Start I/II protocols.

Methods: Survivors of CNS tumors treated on Head Start I/II protocols were enrolled. Late effects data were collected using a validated parent-report questionnaire. Social, emotional, and behavioral functioning and quality of life were assessed using parent-report on the BASC-2 and CHQ-PF50 questionnaires.

Results: Twenty-one survivors (medulloblastoma = 13, sPNET = 4, ATRT = 1, ependymoma = 3) were enrolled. Ten (48%) were irradiation-free. Late effects (frequency; median time of onset since diagnosis) included ≥ grade III hearing loss (67%; 3.9 years), vision (67%; 4.1 years), hypothyroidism (33%; 4 years), growth hormone (GH) deficiency (48%; 4.7 years), dental (52%; 7.1 years), and no cases of secondary leukemia. Irradiation-free (vs. irradiated) survivors reported low rates of hypothyroidism (0/10 vs. 7/11; P = 0.004) and GH deficiency (2/10 vs. 8/11; P = 0.03). The BASC-2 and CHQPF-50 mean composite scores were within average ranges relative to healthy comparison norms. Neither age at diagnosis nor irradiation was associated with these scores.

Conclusions: Irradiation-free Head Start survivors have lower risk of hypothyroidism and GH deficiency. Secondary leukemias are not reported. With extended follow-up, survivors demonstrate quality of life, social, emotional, and behavioral functioning within average ranges.
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http://dx.doi.org/10.1002/pbc.25064DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4714700PMC
September 2014

Prognostic significance of telomere maintenance mechanisms in pediatric high-grade gliomas.

J Neurooncol 2014 Mar 30;117(1):67-76. Epub 2014 Jan 30.

Division of Oncology, Cancer and Blood Diseases Institute, Cincinnati Children's Hospital Medical Center, MLC 7013, 3333 Burnet Ave, Cincinnati, OH, 45229, USA.

Children with high-grade glioma, including diffuse intrinsic pontine glioma (DIPG), have a poor prognosis despite multimodal therapy. Identifying novel therapeutic targets is critical to improve their outcome. We evaluated prognostic roles of telomere maintenance mechanisms in children with HGG, including DIPG. A multi-institutional retrospective study was conducted involving 50 flash-frozen HGG (35 non-brainstem; 15 DIPG) tumors from 45 children (30 non-brainstem; 15 DIPG). Telomerase activity, expression of hTERT mRNA (encoding telomerase catalytic component) and TERC (telomerase RNA template) and alternative lengthening of telomeres (ALT) mechanism were assayed. Cox Proportional Hazard regression analyses assessed association of clinical and pathological variables, TERC and hTERT levels, telomerase activity, and ALT use with progression-free or overall survival (OS). High TERC and hTERT expression was detected in 13/28 non-brainstem HGG samples as compared to non-neoplastic controls. High TERC and hTERT expression was identified in 13/15 and 11/15 DIPG samples, respectively, compared to controls. Evidence of ALT was noted in 3/11 DIPG and 10/19 non-brainstem HGG specimens. ALT and telomerase use were identified in 4/19 non-brainstem HGG and 2/11 DIPG specimens. In multivariable analyses, increased TERC and hTERT levels were associated with worse OS in patients with non-brainstem HGG, after controlling for tumor grade or resection extent. Children with HGG and DIPG, have increased hTERT and TERC expression. In children with non-brainstem HGG, increased TERC and hTERT expression levels are associated with a worse OS, making telomerase a promising potential therapeutic target in pediatric HGG.
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http://dx.doi.org/10.1007/s11060-014-1374-9DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4261223PMC
March 2014

Outcome of infants and young children with newly diagnosed ependymoma treated on the "Head Start" III prospective clinical trial.

J Neurooncol 2013 Jun 19;113(2):285-91. Epub 2013 Mar 19.

Division of Hematology/Oncology, Children's Hospital Los Angeles, Los Angeles, CA 90027, USA.

This study investigates the outcome of children <10 years old with newly-diagnosed ependymoma treated on the prospective multinational "Head Start" III clinical trial. Between April 2004 and July 2009, 19 children with newly-diagnosed ependymoma were enrolled. All children were to receive five induction chemotherapy cycles followed by one consolidation cycle of myelo-ablative chemotherapy and autologous hematopoietic cell rescue. Children between 6 and 10 years of age or with residual tumor prior to consolidation were to receive irradiation thereafter. Median age of 19 children (8 female) was 20 months at diagnosis. Median follow up was 44 months. The primary site was infratentorial in 11 and supratentorial in 8 patients. Gross total resection was achieved in 10 patients. After induction chemotherapy, all three supratentorial ependymoma patients with residual disease achieved a complete response (CR), while only one of six infratentorial patients with residual disease achieved CR. Three infratentorial patients developed progressive disease during induction chemotherapy. All four infratentorial patients with residual disease who underwent autologous hematopoietic cell transplant, failed to achieve CR. Four patients received focal irradiation following chemotherapy. The 3-year event free survival (EFS) and overall survival (OS) for supratentorial ependymoma were 86 ± 13 % and 100 % respectively. The 3-year EFS and OS for infratentorial ependymoma were 27 ± 13 % and 73 ± 13 % respectively. The role of intensive induction and consolidation chemotherapy in deferring irradiation should be investigated further in children with supratentorial ependymoma with residual disease following surgery. This approach appears ineffective in children with infratentorial ependymoma in the absence of irradiation.
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http://dx.doi.org/10.1007/s11060-013-1111-9DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4119804PMC
June 2013

Cribriform neuroepithelial tumor arising in the lateral ventricle.

Pediatr Dev Pathol 2013 Jul-Aug;16(4):301-7. Epub 2013 Mar 15.

Department of Pathology and Laboratory Medicine, Ohio State University College of Medicine, Columbus, OH, USA.

Cribriform neuroepithelial tumor (CRINET) is a recently recognized central nervous system neoplasm that arises in the ventricles of young children and is characterized by primitive, non-rhabdoid SMARCB1-deficient cells with prominent cribriform architecture. We report a 14-month-old male who presented with signs of increased intracranial pressure. Neuroimaging revealed a large solid and cystic mass in the lateral ventricle. Tumor cells were small, primitive appearing, and arranged in cribriform and trabecular patterns with interspersed solid cellular areas. Rhabdoid cells were absent. Immunohistochemical staining showed no SMARCB1 (INI11, BAF47, hSNF5) expression and strong epithelial membrane antigen (EMA) immunoreactivity along luminal surfaces. Electron microscopy showed epithelial characteristics, including abundant basal lamina. Genetic analysis of the tumor revealed deletion of 1 SMARCB1 allele, while the other contained an intronic point mutation predicted to disrupt splicing. This case further illustrates the distinct histopathologic and molecular genetic features of CRINET and identifies distinctive ultrastructural features.
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http://dx.doi.org/10.2350/12-12-1287-CR.1DOI Listing
October 2013

Intensive chemotherapy followed by consolidative myeloablative chemotherapy with autologous hematopoietic cell rescue (AuHCR) in young children with newly diagnosed supratentorial primitive neuroectodermal tumors (sPNETs): report of the Head Start I and II experience.

Pediatr Blood Cancer 2008 Feb;50(2):312-8

Children's Hospital Los Angeles, Los Angeles, California, USA.

Background: Children with newly diagnosed supratentorial primitive neuroectodermal tumors (sPNET) have poor outcomes compared to medulloblastoma patients, despite similar treatments. In an effort to improve overall survival (OS) and event-free survival (EFS) and to decrease radiation exposure, the Head Start (HS) protocols treated children with newly diagnosed sPNET utilizing intensified induction chemotherapy (ICHT) followed by consolidation with myeloablative chemotherapy and autologous hematopoietic cell rescue (AuHCR).

Procedures: Between 1991 and 2002, 43 children with sPNET were prospectively treated on two serial studies (HS I and II). After maximal safe surgical resection, patients on HS I and patients with localized disease on HS II were treated with five cycles of ICHT (vincristine, cisplatin, cyclophosphamide, and etoposide). Patients on HS II with disseminated disease received high-dose methotrexate during ICHT. If the disease remained stable or in response, patients received a single cycle of high-dose myeloablative chemotherapy followed by AuHCR.

Results: Five-year EFS and OS were 39% (95%CI: 24%, 53%) and 49 (95%CI: 33%, 62%), respectively. Non-pineal sPNET patients faired significantly better than those patients with pineal sPNETs. Metastasis at diagnosis, age, and extent of resection were not significant prognostic factors. Sixty percent of survivors (12 of 20) are alive without exposure to radiation therapy.

Conclusions: ICHT followed by AuHCR in young patients with newly diagnosed sPNET appears to not only provide an improved EFS and OS for patients who typically have a poor prognosis, but also it successfully permitted deferral and elimination of radiation therapy in a significant proportion of patients.
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http://dx.doi.org/10.1002/pbc.21307DOI Listing
February 2008

Coincidence FDG-PET in the evaluation of Langerhans' cell histiocytosis: preliminary findings.

Pediatr Radiol 2003 Sep 23;33(9):598-602. Epub 2003 Jul 23.

Department of Radiology, Columbus Children's Hospital, 700 Children's Way, Columbus, OH 43205, USA.

Background: Bone involvement in Langerhans' cell histiocytosis (LCH) is common. Both bone scintigraphy and plain films are used to identify osseous lesions, but lack specificity for disease activity and response to therapy. FDG-PET is a sensitive technique for identifying bone lesions when histiocytes are present. OBJECTIVE. To describe the potential of coincidence FDG-PET (cFDG-PET) for identification of active bone lesions in LCH and to determine whether it can provide more specific information regarding lesional response to therapy than bone scintigraphy or radiography.

Materials And Methods: The clinical data and imaging findings of three patients with osseous lesions of LCH were retrospectively reviewed.

Results: cFDG-PET identified all active LCH osseous lesions in these patients, differentiated active from healed lesions, and demonstrated normalization of uptake in a treated lesion earlier than bone scintigraphy and radiography.

Conclusion: cFDG-PET appears to have greater specificity than bone scintigraphy and radiography for the identification of active osseous lesions in LCH. It also may predict response to treatment earlier than conventional techniques. Its use in the evaluation of LCH warrants further study.
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http://dx.doi.org/10.1007/s00247-003-0943-4DOI Listing
September 2003