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1.
  • Irestorm, Elin, et al. (author)
  • Auditory hypersensitivity and attention in survivors of paediatric brain tumours
  • 2020
  • Conference paper (peer-reviewed)abstract
    • Objective: Both auditory and visual hypersensitivity are clinical features of mental fatigue after acquired brain damage or in neurological disorders. Both types of hypersensitivity are also associated with attention deficits, especially in neurodevelopmental syndromes. The aim of this study was to examine auditory and visual hypersensitivity, and associations to attention, in a group of children and adolescents treated for paediatric brain tumours (PBTs).Participants and Methods: Included in the study were 34 survivors of PBTs, 8–18 years of age (M: 13.6, SD: 3.0). Eighteen participants were female and 16 were male. Mean time since diagnosis was 4.2 years (SD: 2.2). Auditory and visual hypersensitivity were assessed using two items from the questionnaire Mental Fatigue Scale (MFS), scored on a 7-point Likert scale from 0 – 3. Scores above 1 indicate hypersensitivity. Attention was assessed using Conners Continuous Performance Test 3 (detectability, commissions, variability), and T-scores above 60 were considered impaired. Spearman correlations were conducted between the performance-based and self-report measures.Results: Results from the MFS revealed that 53% of the survivors experienced auditory and 18% visual hypersensitivity as a sequela. Regarding attention, elevated scores were more common for detectability (18%) and variability (21%) than commissions (8%). Visual hypersensitivity was not significantly associated with any of the attention measures, whereas auditory hypersensitivity was significantly associated with detectability (r=.42, p=0.013) and variability (r=.57, p<0.001).Conclusions: These results suggest that auditory hypersensitivity is common after treatment for PBT, and that it is associated with decreased attention. This relationship should be taken into consideration when assessing survivors of PBTs.
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2.
  • Irestorm, Elin, et al. (author)
  • Cognitive Fatigue and Processing Speed in Children Treated for Brain Tumours
  • 2021
  • In: Journal of the International Neuropsychological Society. - 1355-6177. ; 27:9, s. 865-874
  • Journal article (peer-reviewed)abstract
    • Objective:The relationship between fatigue and cognition has not been fully elucidated in children and adolescent survivors of brain tumours. The aim of the present study was to investigate the potential relationship between fatigue and cognitive impairments in these survivors, as this group is at risk for both types of deficits.Methods:Survivors of paediatric brain tumours (n = 45) underwent a neuropsychological testing on average 4 years after diagnosis. Mean age at follow-up was 13.41 years. Cognition was assessed with neuropsychological tests, and fatigue with the Pediatric Quality of Life (PedsQL™) Multidimensional Fatigue Scale. Regression analysis, adjusted for cranial radiotherapy and age at diagnosis, was used to investigate the associations between cognitive variables and fatigue subscales. Cognitive variables associated with fatigue were subsequently exploratively assessed.Results:Significant associations were found for cognitive fatigue and measures of cognitive processing speed; Coding: p = .003, r = .583, 95% CI [9.61; 22.83] and Symbol Search: p = .001, r = .585, 95% CI [10.54; 24.87]. Slower processing speed was associated with poorer results for cognitive fatigue. Survivors with the largest decrease in processing speed from baseline to follow-up also experienced the most cognitive fatigue. Survivors expressed more cognitive fatigue compared to other types of fatigue.Conclusions:The association between cognitive fatigue and cognitive processing speed in children and adolescents treated for brain tumours is in concordance with the results previously reported in adults. Some survivors experience fatigue without impairment in processing speed, indicating the need for comprehensive assessments. Moreover, the study supports that fatigue is a multidimensional concept which should be measured accordingly.
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3.
  • Irestorm, Elin, et al. (author)
  • Cognitive fatigue in relation to depressive symptoms after treatment for childhood cancer
  • 2020
  • In: BMC Psychology. - : Springer Science and Business Media LLC. - 2050-7283. ; 8:1
  • Journal article (peer-reviewed)abstract
    • Background: Cognitive fatigue after childhood cancer is frequently overlooked despite guidelines recommending follow-up, and might be mistaken for depression due to overlapping symptoms. Our objectives were: 1) to examine ratings of fatigue in survivors of paediatric brain tumours (BT) and acute lymphoblastic leukaemia (ALL) compared to healthy controls, 2) to examine the relationship between symptoms of depression and cognitive fatigue, and 3) to evaluate parent-child concordance in ratings of cognitive fatigue. Methods: Survivors of BT (n = 30), survivors of ALL (n = 30), and healthy controls (n = 60) aged 8-18 years completed the Pediatric Quality of Life Multidimensional Fatigue Scale and the Beck Youth Inventories. Associations between cognitive fatigue, diagnosis and depression were assessed with general linear modelling. Group differences were analysed using the Kruskal-Wallis test. Parent-child concordance was investigated with internal consistency reliability. Results: Cognitive fatigue was prevalent in 70% of survivors of BT survivors and in 30% of survivors of ALL. Diagnosis was the main predictor of cognitive fatigue (p <.001, ηp2 = 0.178), followed by depression (p =.010, ηp2 = 0.080). Survivors of BT reported significantly more fatigue than healthy controls on all fatigue subscales. While they also expressed more symptoms of depression, we found no evidence for an interaction effect. Parent-child concordance was moderate to good among survivors, but poor for controls. Conclusions: Survivors of BT and ALL suffer from cognitive fatigue, with survivors of BT expressing more problems. Cognitive fatigue and depression should be assessed in survivors of childhood cancer using both self-rated and proxy-rated measures, and appropriate interventions offered. © 2020 The Author(s).
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4.
  • Irestorm, Elin, et al. (author)
  • Pretreatment Cognition in Patients Diagnosed With Pediatric Brain Tumors
  • 2018
  • In: Pediatric Neurology. - : Elsevier BV. - 0887-8994. ; 79, s. 28-33
  • Journal article (peer-reviewed)abstract
    • Background: There is a large body of literature identifying risk factors for the long-term cognitive alterations found in survivors of pediatric brain tumors. Less is known about baseline cognitive functioning in this population, but studies suggest that cognitive dysfunctions are often present at the time of diagnosis. This study aimed to identify potential risk factors for lower cognitive function at the time of pediatric brain tumor diagnosis. Methods: Participants were children and adolescents (n = 101) diagnosed with a pediatric brain tumor between 2006 and 2015, who underwent a pretreatment neuropsychologic assessment. Multivariate regression models were used to estimate the association between gender, age at diagnosis, tumor size and location, increased intracranial pressure, epilepsy, and six different indicators of cognitive functioning. Results: Overall, cognitive performance was relatively intact, with results close to norm means, but impairments were found in memory and cognitive processing speed. Male gender, older age, epilepsy, increased intracranial pressure, and larger tumors were all associated with lower cognitive function at the time of brain tumor diagnosis; whereas tumor location was not. Conclusions: Pretreatment neuropsychologic assessments, with some adjustments, can be carried out with children and adolescents with brain tumors. Our study adds to a small but growing body of literature documenting cognitive impairments at the time of diagnosis; these impairments may partly explain the longer-term deficits that commonly occur in pediatric brain tumor survivors. Consistent with previous research, pretreatment impairments were more common among boys, older children, and those with increased intracranial pressure, epilepsy, and larger tumors. The relationship between baseline and longer-term cognitive deficits requires further examination.
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5.
  • Meurling, Ann Wirsén, et al. (author)
  • Sex differences in strategy and performance on computerized neuropsychological tests as related to gender identity and age at puberty
  • 2000
  • In: Scandinavian Journal of Psychology. - : Wiley. - 1467-9450 .- 0036-5564. ; 41:2, s. 81-90
  • Journal article (peer-reviewed)abstract
    • Neuropsychological sex differences have since long been under debate. Support for the relation between behavioral differences and biological variables like hormone influence is, however, emerging. Sixteen men and sixteen women, all university students, were tested with computerized neuropsychological tests (APT), the Bem Sexual Role Inventory, and asked about pubertal age. The results were in line with earlier findings of sex differences in neuropsychological tests, men being faster and women more cautious. The assumption that women tend to use left-hemispheric, verbal/serial strategies also in spatial tasks was also partly supported. In women, late onset of puberty was related to better spatial performance, and there were also more intercorrelations between verbal and spatial tests in the female than in the male group, indicating that women use less specific strategies (more g-factor intelligence) in problem solving, or that aptitudes are less compartmentalized in women than in men.
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6.
  • Olsson, Ingrid Tonning, et al. (author)
  • Development after pediatric brain tumor-a longitudinal study
  • 2016
  • In: Neuro-Oncology. - : Oxford University Press (OUP). - 1523-5866 .- 1522-8517. ; 18, s. 135-135
  • Journal article (other academic/artistic)abstract
    • Background: Patients treated for Pediatric Brain Tumors (PBTs) often experience a decline/stagnation in their cognitive functioning.Anumber of potential risk factors for cognitive decline have been identified including gender (female), young age-at-diagnosis, higher baseline IQ, hydrocephalus at diagnosis, treatment with Whole Brain Radiation Therapy (WBRT), and larger radiation field and/or dose. The aim of this longitudinal study was to statistically model the rate of cognitive decline in a large population-based sample of PBT survivors and to find risk factors for a faster decline. Methods: Study participants were patients diagnosed with PBTs between 2001-2013 and/or who turned 18 years of age between 2006 and 2013 (n = 151). Measures of verbal ability, perceptual reasoning, general cognitive ability, auditory working memory, cognitive processing speed, visual workingmemory and sustained attention were collected longitudinally. Multilevel Linear Modelling (MLM) was used to evaluate the rate of cognitive decline in the sample and to estimate baseline values (risk factors) influencing the rate of decline. Results: A significant decline was found for most cognitive abilities across all patient categories with only elementary verbal skills either stable or improving. Variables predicting lower cognitive performance at baseline were gender (male), age at diagnosis, supratentorial lateral tumor, larger tumor size, and treatment withWBRTor chemotherapy.Conclusions: Pediatric BT survivors experience a decline in age related cognitive performance, regardless of the treatment received. Young age at PBT diagnosis, male, gender, supratentorial lateral tumors, larger tumors and treatment with WBRT or chemotherapy were all correlated with a lowered baseline performance.
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7.
  • Papini, Chiara, et al. (author)
  • Evolving therapies, neurocognitive outcomes, and functional independence in adult survivors of childhood glioma
  • 2024
  • In: Journal of the National Cancer Institute. - 0027-8874. ; 116:2, s. 288-298
  • Journal article (peer-reviewed)abstract
    • Background: Treatment of childhood glioma has evolved to reduce radiotherapy exposure with the goal of limiting late toxicity. However, the associations between treatment changes and neurocognition, and the contribution of neurocognition and chronic health conditions to attainment of adult independence, remain unknown. Methods: Adult survivors of childhood glioma diagnosed in 1970-1999 in the Childhood Cancer Survivor Study (n ¼ 1284; median [minimum-maximum] 30 [18-51] years of age at assessment; 22 [15-34] years from diagnosis) self-reported neurocognitive impairment and chronic health conditions. Multivariable models evaluated associations between changes in treatment exposures (surgery only, chemotherapy [with or without surgery], cranial radiation [with or without chemotherapy and/or surgery]), and neurocognitive impairment. Latent class analysis with 5 indicators (employment, independent living, assistance with routine and/or personal care needs, driver’s license, marital or partner status) identified classes of functional independence. Path analysis tested associations among treatment exposures, neurocognitive impairment, chronic health conditions, and functional independence. Statistical tests were 2-sided. Results: Cranial radiation exposure decreased over time (51%, 1970s; 46%, 1980s; 27%, 1990s]. However, compared with siblings, survivors with any treatment exposure were at elevated risk for neurocognitive impairment, including surgery only (eg, memory: relative risk ¼ 2.22; task efficiency: relative risk ¼ 1.88; both P < .001). Three classes of functional independence were identified: independent (58%), moderately independent (20%), and nonindependent (22%). Cranial radiation was associated with nonindependence through impaired task efficiency (b ¼ 0.06), sensorimotor (b ¼ 0.06), and endocrine (b ¼ 0.10) chronic health conditions and through the associations between these conditions and task efficiency (each b ¼ 0.04). Sensorimotor and endocrine chronic health conditions were associated with nonindependence through memory. Conclusion: Most long-term glioma survivors achieve adult independence. However, functional nonindependence is associated with treatment-related neurocognitive impairment and chronic health conditions.
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8.
  • Tonning Olsson, Ingrid, et al. (author)
  • Access to Neuropsychologic Services After Pediatric Brain Tumor.
  • 2013
  • In: Pediatric Neurology. - : Elsevier BV. - 0887-8994. ; 49:6, s. 420-423
  • Journal article (peer-reviewed)abstract
    • Background: Increasing survival rates for pediatric brain tumor patients creates a greater need for neuropsychologic follow-up and intervention. The aim of this study was to evaluate rates of referral by medical doctors to neuropsychologic services and patient and treatment factors that differentiated referred and non-referred patients. Methods: Data were retrieved from medical records of all pediatric brain tumor patients in Southern Sweden diagnosed between 1993 and 2004 who survived more than one year (n=132). Characteristics of the patients, the cancer, and treatment received were then compared for patients who were and were not referred for neuropsychologic examination during that period. Results: Sixty-four (48%) of the pediatric brain tumor patients were referred for neuropsychologic evaluation. These patients had significantly larger tumors, more recurrences of cancer, and increased intracranial pressure at diagnosis when compared to the non-referred group (n=68). However most of the patients in the non-referred group either had risk significant factors for cognitive impairment or were reporting impairments that would suggest a referral was warranted. Conclusions: Given the high rates of cognitive impairment found in patients with pediatric brain tumors, survivors should be referred to neuropsychologic services. In addition to improving long-term adjustment, systematic referral can provide data on cognitive impairments that may inform cost-benefit-analyses when deciding on various treatments at the point of diagnosis. Greater efforts are needed to disseminate and raise awareness about published guidelines on the long-term care of pediatric brain tumor patients.
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9.
  • Tonning Olsson, Ingrid (author)
  • Beyond Survival - Cognition after Pediatric Brain Tumor
  • 2015
  • Doctoral thesis (other academic/artistic)abstract
    • Background: Pediatric Brain Tumor (PBT) survivors suffer from cognitive sequelae, especially within the areas of cognitive tempo, attention, executive function and memory. The cognitive difficulties are often accentuated over the years, but knowledge about the long term trajectory is still scarce. Aim: The aim of this thesis was to examine cognitive sequelae after Pediatric Brain Tumor (PBT); risk factors, common difficulties, development and neuroimaging correlates. Methods: In study I, data from medical logs were used to examine characteristics of the patients who got access to neuropsychological services, compared to those who did not. In study II, data from 70 neuropsychological assessments were used to describe common cognitive impairments and to find risk factors. In study III, patients were invited to a follow-up study 10-13 years after diagnosis. Neuropsychological and neuroimaging data were collected and the two were compared. In study IV, longitudinal cognitive data from 173 patients were analyzed in order to describe development over time and to find risk factors for a negative development. Results: Study I: There were few differences between referred and non-referred patients. Study II: Patients had generally suppressed IQ and difficulties with executive function, memory, cognitive processing speed and attention. Risk factors were Whole-Brain Radiation Therapy (WBRT), large tumors, young age at diagnosis and male sex. Study III: Radiated as well as non-radiated patients had white matter abnormalities. Correlation between neuroimaging and cognition was low when group based statistics were used, but increased when a personalized method was used. Study IV: Most cognitive abilities showed a decline in age related scores over time unconsidered treatment given. Risk factors for impaired cognitive function at diagnosis were: male sex, WBRT, supratentorial lateral tumor, young age at diagnosis, larger tumor size and treatment with chemotherapy. Conclusions: A systematic neuropsychological follow-up is important. Risk factors for cognitive impairment and IQ decline are WBRT, large tumors, young age at diagnosis, male sex, supratentorial lateral tumor, and treatment with chemotherapy. A decline in IQ after PBT is common, unconsidered treatment given. Personalized methods of research would contribute significantly to our understanding of cognitive sequelae after PBT and its relation to neuroimaging.
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10.
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11.
  • Tonning Olsson, Ingrid, et al. (author)
  • Comparison of neuropsychological and brain imaging data in pediatric brain tumor patients surviving more than 10 years
  • 2014
  • In: Neuro-Oncology. - : Oxford University Press (OUP). - 1523-5866 .- 1522-8517. ; 16:Suppl 1, s. 99-104
  • Journal article (peer-reviewed)abstract
    • AIM: To explore the relationship between neuropsychological outcome and magnetic resonance imaging (MRI) findings in paediatric brain tumour patients surviving more than 10 years. BACKGROUND: Cognitive late sequelae after paediatric brain tumour have a multifactorial origin. Low age at diagnosis, cranial radiation therapy, intrathecal methotrexate treatment and increased intracranial pressure are known to predict cognitive sequelae. Cognitive sequelae have been related to MRI findings, but the relation between neuroimaging findings and neuropsychological impairment needs to be explored further. METHOD: Sixteen paediatric brain tumour survivors completed an extensive neuropsychological test battery and MRI 10 to 13 years after diagnosis. Patients were first separately classified as positive or negative regarding neuropsychological impairment and MRI findings and then coded as congruent or incongruent, depending on whether neuropsychological outcome was in agreement with MRI also considering clinical data such as type and location of the brain tumour, post-operative status, treatments or premorbidity. RESULTS: Ten patients were classified as either positive or negative for both MRI and cognitive impairment if blinded for the second parameter and clinical data, and were also congruent for these two parameters when not blinded for clinical data and the second parameter. In five out of six initially incongruent patients the neuropsychological profile was in accordance with MRI findings when evaluating all data with the multidisciplinary holistic approach. CONCLUSIONS: Cognitive outcome in survivors of paediatric brain tumours depends on many different detrimental processes and shows high individual variation. Congruity between neuropsychological impairment and MRI findings might at first seem low. However, in the perspective of a clinical holistic evaluation of MRI findings and neuropsychological outcome considering the knowledge of clinical data such as type and location of the brain tumour, type of MRI findings (atrophy, gliosis, post-operative lesion), undergone treatments and radiation field as well as premorbid cognitive impairment congruity increases.
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12.
  • Tonning Olsson, Ingrid, et al. (author)
  • Long-Term Cognitive Sequelae After Pediatric Brain Tumor Related to Medical Risk Factors, Age, and Sex
  • 2014
  • In: Pediatric Neurology. - : Elsevier BV. - 0887-8994. ; 51:4, s. 515-521
  • Journal article (peer-reviewed)abstract
    • BACKGROUND: Young age at diagnosis and treatment with cranial radiation therapy are well studied risk factors for cognitive impairment in pediatric brain tumor survivors. Other risk factors are hydrocephalus, surgery complications, and treatment with intrathecal chemotherapy. Female gender vulnerability to cognitive sequelae after cancer treatment has been evident in some studies, but no earlier studies have related this to tumor size. The purpose of our study was to find factors correlated with lowered IQ in a nationally representative sample of pediatric brain tumor patients referred for neuropsychologic evaluation. METHODS: Sixty-nine pediatric brain tumor patients, diagnosed 1988-2005 and tested 1995-2006, were included in the study. In a series of stepwise multiple regressions, the relationship of IQ to disease, treatment, and individual variables (sex and syndromes) were evaluated. A subanalysis was made of the covariation between sex and tumor size. RESULTS: The patients had generally suppressed IQ and impairments in executive function, memory, and attention. Lowered IQ was associated with young age at diagnosis, being male, tumor size, and treatment with whole-brain radiation therapy. A sex difference was evident for patients with increased intracranial pressure at diagnosis with males having larger tumors. Tumor size was found to be a better predictor of cognitive sequelae than sex. CONCLUSIONS: Whole-brain radiation therapy, large tumors, young age at diagnosis, and male gender are risk factors for late cognitive sequelae after pediatric brain tumors. When examining sex differences, tumor size at diagnosis needs to be taken into account.
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13.
  • Tonning Olsson, Ingrid, et al. (author)
  • Neurocognitive development after pediatric brain tumor - a longitudinal, retrospective cohort study
  • 2024
  • In: Child Neuropsychology. - : Informa UK Limited. - 0929-7049 .- 1744-4136. ; 30:1, s. 22-44
  • Journal article (peer-reviewed)abstract
    • Survivors of Pediatric Brain Tumors (PBTs) treated with cranial radiation therapy (CRT) often experience a decline in neurocognitive test scores. Less is known about the neurocognitive development of non-irradiated survivors of PBTs. The aim of this study was to statistically model neurocognitive development after PBT in both irradiated and non-irradiated survivors and to find clinical variables associated with the rate of decline in neurocognitive scores. A total of 151 survivors were included in the study. Inclusion criteria: Diagnosis of PBT between 2001 and 2013 or earlier diagnosis of PBT and turning 18 years of age between 2006 and 2013. Exclusion criteria: Death within a year from diagnosis, neurocutaneous syndromes, severe intellectual disability. Clinical neurocognitive data were collected retrospectively from medical records. Multilevel linear modeling was used to evaluate the rate of decline in neurocognitive measures and factors associated with the same. A decline was found in most measures for both irradiated and non-irradiated survivors. Ventriculo-peritoneal (VP) shunting and treatment with whole-brain radiation therapy (WBRT) were associated with a faster decline in neurocognitive scores. Male sex and supratentorial lateral tumor were associated with lower scores. Verbal learning measures were either stable or improving. Survivors of PBTs show a pattern of decline in neurocognitive scores irrespective of treatment received, which suggests the need for routine screening for neurocognitive rehabilitation. However, survivors treated with WBRT and/or a VP shunt declined at a faster rate and appear to be at the highest risk of negative neurocognitive outcomes and to have the greatest need for neurocognitive rehabilitation.
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14.
  • Wibroe, Morten, et al. (author)
  • Cerebellar mutism syndrome in children with brain tumours of the posterior fossa
  • 2017
  • In: BMC Cancer. - : BIOMED CENTRAL LTD. - 1471-2407. ; 17
  • Journal article (peer-reviewed)abstract
    • Background: Central nervous system tumours constitute 25% of all childhood cancers; more than half are located in the posterior fossa and surgery is usually part of therapy. One of the most disabling late effects of posterior fossa tumour surgery is the cerebellar mutism syndrome (CMS) which has been reported in up to 39% of the patients but the exact incidence is uncertain since milder cases may be unrecognized. Recovery is usually incomplete. Reported risk factors are tumour type, midline location and brainstem involvement, but the exact aetiology, surgical and other risk factors, the clinical course and strategies for prevention and treatment are yet to be determined.Methods: This observational, prospective, multicentre study will include 500 children with posterior fossa tumours. It opened late 2014 with participation from 20 Nordic and Baltic centres. From 2016, five British centres and four Dutch centres will join with a total annual accrual of 130 patients. Three other major European centres are invited to join from 2016/17. Follow-up will run for 12 months after inclusion of the last patient. All patients are treated according to local practice. Clinical data are collected through standardized online registration at pre-determined time points pre- and postoperatively. Neurological status and speech functions are examined pre- operatively and postoperatively at 1-4 weeks, 2 and 12 months. Pre- and postoperative speech samples are recorded and analysed. Imaging will be reviewed centrally. Pathology is classified according to the 2007 WHO system. Germline DNA will be collected from all patients for associations between CMS characteristics and host genome variants including pathway profiles.Discussion: Through prospective and detailed collection of information on 1) differences in incidence and clinical course of CMS for different patient and tumour characteristics, 2) standardized surgical data and their association with CMS, 3) diversities and results of other therapeutic interventions, and 4) the role of host genome variants, we aim to achieve a better understanding of risk factors for and the clinical course of CMS - with the ultimate goal of defining strategies for prevention and treatment of this severely disabling condition.
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