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Ten-Year Follow-up of Internal Neurofibroma Growth Behavior in Adult Patients With Neurofibromatosis Type 1 Using Whole-Body MRI
Ly, K Ina; Merker, Vanessa L; Cai, Wenli; Bredella, Miriam A; Muzikansky, Alona; Thalheimer, Raquel D; Da, Jennifer Liwei; Orr, Christina C; Herr, Hamilton P; Morris, Mary E; Chang, Connie Y; Harris, Gordon J; Plotkin, Scott R; Jordan, Justin T
BACKGROUND AND OBJECTIVES:Internal neurofibromas, including plexiform neurofibromas (PNF), can cause significant morbidity in patients with neurofibromatosis type 1 (NF1). PNF growth is most pronounced in children and young adults, with more rapid growth thought to occur in a subset of PNF termed distinct nodular lesions (DNL). Growth behavior of internal neurofibromas and DNL in older adults is not well documented; yet knowledge thereof is important for patient risk stratification and clinical trial design. The primary objective of this study was to evaluate the long-term growth behavior of internal neurofibromas in adults with NF1. Secondary objectives were to correlate tumor growth behavior with patient-specific, tumor-specific, and patient-reported variables. METHODS:In this prospective cohort study, internal neurofibromas were identified on coronal short TI inversion recovery sequences on baseline and follow-up whole-body MRIs (WBMRIs). Tumor growth and shrinkage were defined as a volume change ≥20%. The association between tumor growth and patient-specific (baseline age, sex, and genotype), tumor-specific (morphology, location, DNL presence on baseline WBMRI, and maximum standardized uptake value on baseline PET imaging), and patient-reported variables (endogenous and exogenous hormone exposure, pain intensity, and quality of life) was assessed using the Spearman correlation coefficient and Kruskal-Wallis test. RESULTS:Of 106 patients with a baseline WBMRI obtained as part of a previous research study, 44 had a follow-up WBMRI. Three additional patients with WBMRIs acquired for clinical care were included, generating 47 adults for this study. The median age during baseline WBMRI was 42 years (range 18-70). The median time between WBMRIs was 10.4 years. Among 324 internal neurofibromas, 62.8% (56% of PNF and 62.1% of DNL) shrank spontaneously without treatment and 17.1% (17.9% of PNF and 13.8% of DNL) grew. Growth patterns were heterogeneous within participants. Patient-specific, tumor-specific, and patient-reported variables (including endogenous and exogenous hormone exposure) were not strong predictors of tumor growth. DISCUSSION:Internal neurofibroma growth behavior in older adults differs fundamentally from that in children and young adults, with most tumors, including DNL, demonstrating spontaneous shrinkage. Better growth models are needed to understand factors that influence tumor growth. These results will inform clinical trial design for internal neurofibromas.
PMCID:9969927
PMID: 36332985
ISSN: 1526-632x
CID: 5602102
Relationship of Imaging-guided Corticosteroid Injections to COVID-19 Incidence in the Pandemic Recovery Period
Vicentini, Joao R T; Habibollahi, Sina; Staffa, Steven J; Simeone, Frank J; Kheterpal, Arvin B; Graeber, Adam R; Bredella, Miriam A; Chang, Connie Y
Background Corticosteroids injected for the treatment of musculoskeletal pain are systemically absorbed and can affect the immune response to viral infections. Purpose To determine the incidence of symptomatic COVID-19 disease in individuals receiving image-guided corticosteroid injections for musculoskeletal pain compared with the general population during the pandemic recovery period. Materials and Methods In this prospective cohort multicenter study, adults with a history of musculoskeletal pain who underwent imaging-guided intra-articular and spine corticosteroid injections from April 2020 to February 2021 were consecutively enrolled. Participants were followed for a minimum of 28 days through their electronic medical record (EMR) or by direct phone communication to screen for COVID-19 test results or symptoms. Clinical data, including body mass index (BMI), were also obtained from the EMR. The incidence of COVID-19 in the state was obtained using the Massachusetts COVID-19 Response Reporting website. The Student t test was used for continuous variable comparisons. Univariable analyses were performed using the Fisher exact test. Results A total of 2714 corticosteroid injections were performed in 2190 adult participants (mean age, 59 years ± 15 [SD]; 1031 women). Follow-up was available for 1960 participants (89%) who received 2484 injections. Follow-up occurred a mean of 97 days ± 33 (range, 28-141 days) after the injection. Of the 1960 participants, 10 had COVID-19 within 28 days from the injection (0.5% [95% CI: 0.24, 0.94]) and 43 had COVID-19 up to 4 months after the injection (2.2% [95% CI: 1.6, 2.9]). These incidence rates were lower than that of the population of Massachusetts during the same period (519 195 of 6 892 503 [7.5%], P < .001 for both 28 days and 4 months). Participants diagnosed with COVID-19 (n = 10) within 28 days from the injection had a higher BMI than the entire cohort (n = 1960) (mean, 32 kg/m2 ± 10 vs 28 kg/m2 ± 6; P = .04). Conclusion Adults who received image-guided corticosteroid injections for pain management during the pandemic recovery period had a lower incidence of symptomatic COVID-19 compared with the general population. © RSNA, 2022 Online supplemental material is available for this article.
PMCID:9341274
PMID: 35787202
ISSN: 1527-1315
CID: 5601962
Training and cultivating the translational science workforce: Responses of Clinical and Translational Science Awards program hubs to the COVID-19 pandemic
Bredella, Miriam A; Volkov, Boris B; Doyle, Jamie M
The coronavirus disease 2019 (COVID-19) pandemic has dramatically changed our lives and the delivery of healthcare. The pandemic also led to widespread disruption in the research activities and training of pre-doctoral, post-doctoral, and early career faculty researchers. This mini-review uses the Local Adaptive Capacity Framework to describe successful practices, challenges, and lessons learned on how Clinical and Translational Science Award (CTSA) hubs have used their expertise, resources, and collaborations to advance clinical and translational science research and workforce development while facing and adapting to a pandemic. Data for this mini-review were taken from the scientific literature (23 articles) and the Research Performance Progress Reports of 50 unique CTSA hubs (40 TL1 and 50 KL2 awards). Institutions responded in innovative ways to the disruption of the COVID-19 pandemic. Electronic and virtual platforms were used to overcome challenges related to physical distancing, laboratory closures, and travel bans. The importance of mentorship and well-being led to the creation of new virtual programs to expand mentoring and networking beyond the home institution and to promote well-being and resilience. These solutions to translational workforce development can be implemented to address future public health emergencies.
PMCID:9841297
PMID: 36448246
ISSN: 1752-8062
CID: 5602122
Well-being grants in an academic medical center: A case example
Olson, Lauren E; Bredella, Miriam A; Levy, Anne S; Mehta, Darshan H
Academic medical centers (AMCs) rely on engaged and motivated faculty for their success. Significant burnout among clinical and research faculty has resulted in career disengagement and turnover. As such, AMCs must be vested in cultivating faculty engagement and well-being through novel initiatives that support faculty. The Well-Being Education Grants program was established by the Office for Well-Being within the Center for Faculty Development at Massachusetts General Hospital to provide the impetus many faculty needed to dedicate time to their well-being, demonstrating that investments in multi-component interventions around faculty well-being require resources and funding.
PMCID:10603360
PMID: 37900355
ISSN: 2059-8661
CID: 5736402
Developing adaptive capacity and preparedness in clinical and translational science [Editorial]
Volkov, Boris B; Bredella, Miriam A; Doyle, Jamie Mihoko; Sorensen, Cecilia
PMCID:10052557
PMID: 37008599
ISSN: 2059-8661
CID: 5602072
Adaptive capacity and preparedness of Clinical and Translational Science Award Program hubs: Overview of an environmental scan
Volkov, Boris B; Ragon, Bart; Doyle, Jamie Mihoko; Bredella, Miriam A
The ability of research networks and individual institutions to effectively and efficiently prepare, respond, and adapt to emergent challenges is essential for the biomedical research enterprise. At the beginning of 2021, a special Working Group was formed by individuals in the Clinical and Translational Science Award (CTSA) consortium and approved by the CTSA Steering Committee to explore "Adaptive Capacity and Preparedness (AC&P) of CTSA Hubs." The AC&P Working Group took a pragmatic Environmental Scan (E-Scan) approach of utilizing the diverse data that had been collected through existing mechanisms. The Local Adaptive Capacity framework was adapted to illustrate the interconnectedness of CTSA programs and services, while exposing how the demands of the pandemic forced them to quickly pivot and adapt. This paper presents a synopsis of the themes and lessons learned that emerged from individual sections of the E-Scan. Lessons learned from this study may improve our understanding of adaptive capacity and preparedness at different levels, as well as help strengthen the core service models, strategies, and foster innovation in clinical and translational science research.
PMCID:9947610
PMID: 36845304
ISSN: 2059-8661
CID: 5602202
CT Texture Analysis in Nonalcoholic Fatty Liver Disease (NAFLD)
Dichtel, Laura E; Tabari, Azadeh; Mercaldo, Nathaniel D; Corey, Kathleen E; Husseini, Jad; Osganian, Stephanie A; Chicote, Mark L; Rao, Elizabeth M; Miller, Karen K; Bredella, Miriam A
BACKGROUND/UNASSIGNED:Nonalcoholic fatty liver disease (NAFLD) is the most common form of liver disease worldwide. There are limited biomarkers that can detect progression from simple steatosis to nonalcoholic steatohepatitis (NASH). The purpose of our study was to utilize CT texture analysis to distinguish steatosis from NASH. METHODS/UNASSIGNED:) underwent liver biopsy and abdominal non-contrast CT. CT texture analysis was performed to quantify gray-level tissue summaries (e.g., entropy, kurtosis, skewness, and attenuation) using commercially available software (TexRad, Cambridge England). Logistic regression analyses were performed to quantify the association between steatosis/NASH status and CT texture. ROC curve analysis was performed to determine sensitivity, specificity, AUC, 95% CIs, and cutoff values of texture parameters to differentiate steatosis from NASH. RESULTS/UNASSIGNED: = 0.009) than those with simple steatosis. Entropy values below 4.73 predict NASH with 100% (95%CI: 67-100%) specificity and 80% (50-100%) sensitivity, AUC: 0.88. MPP values below 54.0 predict NASH with 100% (67-100%) specificity and 100% (50-100%) sensitivity, AUC 0.90. CONCLUSION/UNASSIGNED:Our study provides preliminary evidence that CT texture analysis may serve as a novel imaging biomarker for disease activity in NAFLD and the discrimination of steatosis and NASH.
PMCID:10483004
PMID: 37693260
ISSN: 0973-6883
CID: 5602572
What is well-being? A scoping review of the conceptual and operational definitions of occupational well-being
Bautista, Tara G; Roman, Gretchen; Khan, Munziba; Lee, Michele; Sahbaz, Sumeyra; Duthely, Lunthita M; Knippenberg, Alexa; Macias-Burgos, Miracle A; Davidson, Alec; Scaramutti, Carolina; Gabrilove, Janice; Pusek, Susan; Mehta, Darshan; Bredella, Miriam A
Well-being is a multifaceted construct that is used across disciplines to portray a state of wellness, health, and happiness. While aspects of well-being seem universal, how it is depicted in the literature has substantial variation. The aim of this scoping review was to identify conceptual and operational definitions of well-being within the field of occupational health. Broad search terms were used related to well-being and scale/assessment. Inclusion criteria were (1) peer-reviewed articles, (2) published in English, (3) included a measure of well-being in the methods and results section of the article, and (4) empirical paper. The searches resulted in 4394 articles, 3733 articles were excluded by reading the abstract, 661 articles received a full review, and 273 articles were excluded after a full review, leaving 388 articles that met our inclusion criteria and were used to extract well-being assessment information. Many studies did not define well-being or link their conceptual definition to the operational assessment tool being used. There were 158 assessments of well-being represented across studies. Results highlight the lack of a consistent definitions of well-being and standardized measurements.
PMCID:10643923
PMID: 38028344
ISSN: 2059-8661
CID: 5617302
Breast adipose tissue attenuation as a novel imaging biomarker for cardiometabolic risk
Bunnell, Katherine M; Galdamez, Marilyn E; Torriani, Martin; Bredella, Miriam A
BACKGROUND:Certain adipose tissue depots infer higher cardiometabolic risk than body mass index (BMI). PURPOSE/OBJECTIVE:To assess breast adipose tissue (BrAT) attenuation as a novel imaging biomarker for cardiometabolic risk. MATERIAL AND METHODS/METHODS:We studied 151 women (mean age = 56 ± 1 years) across the weight spectrum. BrAT attenuation, abdominal adipose tissue cross-sectional areas (CSA), and attenuation were quantified using non-contrast computed tomography (CT) scans. Cardiometabolic risk factors were assessed from medical records. RESULTS: < 0.001). BrAT attenuation had a sensitivity of 90% but a specificity of only 35% in detecting the metabolic syndrome (area under the curve = 0.63). CONCLUSION/CONCLUSIONS:BrAT attenuation is associated with cardiometabolic risk markers and could serve as an imaging biomarker for opportunistic risk assessment in patients undergoing CT examination of the chest.
PMID: 36560894
ISSN: 1600-0455
CID: 5602142
Bone marrow adipose tissue is associated with fracture history in anorexia nervosa
Dang, T; Faje, A T; Meenaghan, E; Bredella, M A; Bouxsein, M L; Klibanski, A; Fazeli, P K
UNLABELLED:Although bone mineral density (BMD) is decreased and fracture risk increased in anorexia nervosa, BMD does not predict fracture history in this disorder. We assessed BMD, bone microarchitecture, and bone marrow adipose tissue (BMAT) in women with anorexia nervosa and found that only BMAT was associated with fracture history. INTRODUCTION/BACKGROUND:Anorexia nervosa (AN) is a psychiatric disorder characterized by low body weight, low BMD, and increased risk of fracture. Although BMD is reduced and fracture risk elevated, BMD as assessed by DXA does not distinguish between individuals with versus those without prior history of fracture in AN. Despite having decreased peripheral adipose tissue stores, individuals with AN have enhanced bone marrow adipose tissue (BMAT), which is inversely associated with BMD. Whether increased BMAT is associated with fracture in AN is not known. METHODS:H-MRS, and parameters of bone microarchitecture by HR-pQCT. RESULTS:Sixteen women (47.1%) with AN reported prior history of fracture compared to 11 normal-weight women (39.3%, p = 0.54). In the entire group and also the subset of women with AN, there were no significant differences in BMD or parameters of bone microarchitecture in women with prior fracture versus those without. In contrast, women with AN with prior fracture had greater BMAT at the spine and femur compared to those without (p = 0.01 for both). CONCLUSION/CONCLUSIONS:In contrast to BMD and parameters of bone microarchitecture, BMAT is able to distinguish between women with AN with prior fracture compared to those without. Prospective studies will be necessary to understand BMAT's potential pathophysiologic role in the increased fracture risk in AN.
PMCID:9940017
PMID: 35999286
ISSN: 1433-2965
CID: 5602602