adolescents – Page 8 – American ME and CFS Society

Long Term Follow up of Young People With Chronic Fatigue Syndrome Attending a Pediatric Outpatient Service

Abstract:

Aim: To determine the reported duration of illness, the functional and educational long-term outcomes, predictive factors for recovery and seek feedback regarding management in pediatric/adolescent myalgic encepahalomyelitis/chronic fatigue syndrome (ME/CFS).

Methods: A cohort observational study of 784 young people, mean age 14.6 (6-18) years, with ME/CFS diagnosed at a specialist pediatric hospital and receiving regular care, was conducted with follow-up for a mean 8 (range 1-21) years after onset. Baseline symptoms, history, depression and anxiety questionnaires were available from 418. The remaining 366, did not have similar standardized baseline information. Questionnaires requested functional rating, persistent symptoms, duration of illness if “recovered,” social engagement and school/work attendance. Feedback was sought regarding management, support services, useful information, helpful interventions or personnel and use of alternative therapies. Reported recovery and function were compared with baseline information and between the two groups.

Results: Follow-up data were returned from 81.8%. There was no significant difference in functional score (if reported recovery) or illness duration related to provision of baseline data. The mean duration of illness was 5 (range 1-15) years in the 50% who reported recovery. By 5 years 38% and by 10 years 68% reported recovery. At 10 years the mean functional score was 8/10 (range 2-10) with 5% scoring <6. Depression, anxiety or severity of illness at diagnosis was not predictive of non-recovery. Designing and monitoring their own management plan that included educational, social, physical and enjoyable activities, as well as having symptom management and understanding professionals were highly valued. However, remaining engaged in an education system that flexibly accommodated their illness and aspirations was consistently reported as crucial for long term functioning.

Conclusions: ME/CFS in young people has a mean duration of 5 years (1-15) with 68% reporting recovery by 10 years. All improved functionally with 5% remaining very unwell and a further 20% significantly unwell. There were no obvious baseline predictors for recovery. However, depression, anxiety, orthostatic intolerance and to a lesser extent pain at follow up were identified as hampering recovery or function. Supportive professionals, remaining engaged in education and management strategies were identified as helpful.

Source: Rowe KS. Long Term Follow up of Young People With Chronic Fatigue Syndrome Attending a Pediatric Outpatient Service. Front Pediatr. 2019 Feb 21;7:21. doi: 10.3389/fped.2019.00021. eCollection 2019. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6393360/ (Full article)

Impaired Health-Related Quality of Life in Adolescent Myalgic Encephalomyelitis/Chronic Fatigue Syndrome: The Impact of Core Symptoms

Abstract:

Objective: The objectives of this study were to compare the health-related quality of life (HRQOL) of a North American population of adolescents and young adults with myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) to (1) healthy controls (HC), (2) adolescents with ME/CFS in other countries, and (3) other forms of pediatric chronic illness, and (4) to examine the influence of the core illness symptoms in the Institute of Medicine (IOM) case definition on impaired HRQOL.

Study design: Cross-sectional study comparing individuals with ME/CFS referred to a tertiary care Chronic Fatigue clinic and HC. Eligible participants were age 10-30 years and met the Fukuda criteria for CFS. HC were eligible if they were age 10-30 years, with self-reported good, very good, or excellent general health. Pediatric HRQOL was measured using the PedsQL (Pediatric Quality of Life Inventory) and other validated instruments.

Results: We enrolled 55 consecutive ME/CFS patients (46 F) aged 10-23 years. From a pool of 69 potential HC we selected 55 with similar age and gender distribution for comparison. The total and subscale scores on the PedsQL and on all other measures of HRQOL indicated significantly worse function among those with ME/CFS (all P < 0.001). The self-reported frequency of post-exertional malaise (PEM) was significantly associated with the severity of impaired HRQOL (P < 0.001). Cognitive impairment had a weaker association with the PedsQL score (P = 0.02). Orthostatic intolerance was present in 96% of the ME/CFS population. Of the 55 who satisfied the Fukuda criteria, 47 (85%) also satisfied the IOM criteria for the diagnosis. Those meeting the IOM criteria had worse PedsQL total scores than those meeting just the Fukuda criteria (P < 0.001).

Conclusions: HRQOL was substantially lower in an ambulatory population of adolescents and young adults with ME/CFS than for healthy controls in North America, consistent with reports from other continents. HRQOL was also lower in ME/CFS than has been described in children with asthma, diabetes mellitus, epilepsy, eosinophilic gastroenteritis, and cystic fibrosis. The findings of this study lend further support to the inclusion of PEM, cognitive impairment, and orthostatic intolerance as core symptoms of pediatric ME/CFS.

Source: Roma M, Marden CL, Flaherty MAK, Jasion SE, Cranston EM, Rowe PC. Impaired Health-Related Quality of Life in Adolescent Myalgic Encephalomyelitis/Chronic Fatigue Syndrome: The Impact of Core Symptoms. Front Pediatr. 2019 Feb 15;7:26. doi: 10.3389/fped.2019.00026. eCollection 2019. https://www.ncbi.nlm.nih.gov/pubmed/30828572

Adolescent’s descriptions of fatigue, fluctuation and payback in chronic fatigue syndrome/myalgic encephalopathy (CFS/ME): interviews with adolescents and parents

Abstract:

OBJECTIVE: As part of a larger qualitative study to explore outcomes important in paediatric chronic fatigue syndrome/myalgic encephalopathy (CFS/ME) and what improvements in fatigue and disability are key, interviews were undertaken with adolescents and their parents. This paper focuses on their descriptions of fatigue, fluctuation of symptoms and payback.

DESIGN AND SETTING: Semistructured qualitative interviews were undertaken between December 2014 and February 2015. Adolescents and parents were interviewed separately. Participants were recruited from a single specialist paediatric chronic fatigue service. Interviews were audio recorded, transcribed verbatim and analysed using thematic analysis.

PARTICIPANTS: We interviewed 21 adolescents and their parents (20 mothers and 2 fathers). The adolescents were aged between 12 and 17 years of age (mean age 14.4 years), mild to moderately affected by CFS/ME (not housebound) and the majority were female (16/21).

RESULTS: All adolescents with CFS/ME reported fatigue, a natural fluctuation of the condition, with good days and bad days as well as an increase in symptoms after activity (payback). However, adolescent’s descriptions of fatigue, symptoms and the associated impact on their daily lives differed. The variations included: fatigue versus a collection of symptoms, constant versus variable symptoms and variable symptom severity. There were differences between participants in the amount of activity taken to cause payback. The impact of fatigue and symptoms on function ranged from: limiting the duration and amount of leisure activities, struggling with daily activities (eg, self-care) to no activity (sedentary).

CONCLUSIONS: Fatigue, fluctuation of the condition and payback after activity are described by all adolescents with CFS/ME in this study. However, the individual experience in terms of how they describe it and the degree and impact varies.

Source: Parslow RM, Anderson N, Byrne D, Shaw A, Haywood KL, Crawley E. Adolescent’s descriptions of fatigue, fluctuation and payback in chronic fatigue syndrome/myalgic encephalopathy (CFS/ME): interviews with adolescents and parents. BMJ Paediatr Open. 2018 Dec 4;2(1):e000281. doi: 10.1136/bmjpo-2018-000281. eCollection 2018 https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6307594/ (Full article)

School Functioning in Adolescents With Chronic Fatigue Syndrome

Abstract:

Background: It is well known that adolescents with chronic fatigue syndrome (CFS) experience greater school absenteeism compared to healthy adolescents. Less is known about other important aspects of school functioning including school participation, school connectedness, and academic performance in students with CFS. The aim of this study was to compare school functioning as a multifaceted construct in adolescents with CFS to healthy adolescent peers. We also explored whether illness factors were associated with school functioning in adolescents with CFS.

Methods: Thirty-nine participants with CFS and 28 healthy controls (aged 13-17 years) completed a range of subjective and objective measures of school functioning, as well as measures of fatigue and emotional symptoms.

Results: Adolescents with CFS demonstrated significantly higher rates of school absence, as well as poorer school-related quality of life, reduced school participation, poorer connectedness with school, and reduced academic performance. Fatigue severity and emotional symptoms were significantly associated with most aspects of school function.

Conclusions: Adolescents with CFS are at increased risk for poor school functioning across a range of indicators which extend beyond school absenteeism.

Source: Knight SJ, Politis J, Garnham C, Scheinberg A, Tollit MA. School Functioning in Adolescents With Chronic Fatigue Syndrome. Front Pediatr. 2018 Oct 16;6:302. doi: 10.3389/fped.2018.00302. eCollection 2018. https://www.ncbi.nlm.nih.gov/pubmed/30460211

Adolescent and parent factors related to fatigue in paediatric multiple sclerosis and chronic fatigue syndrome: A comparative study

Abstract:

BACKGROUND: Fatigue is a disabling, poorly understood symptom in children and adolescents with multiple sclerosis (caMS), for which effective treatments are lacking. In paediatric Chronic Fatigue Syndrome (CFS), effective psychological interventions have been developed based on psychosocial factors associated with fatigue. This study aimed to identify potentially modifiable factors of fatigue in caMS by comparing caMS, adolescents with CFS, healthy adolescents and their parents on measures of fatigue, psychosocial factors, and neurocognitive functioning.

METHODS: 175 participants including 30 caMS (15 fatigued, 15 non-fatigued), 30 adolescents with CFS, 30 healthy controls, and their parents were compared on measures of self- and parent-reported fatigue, adolescent and parent cognitive behavioural responses to symptoms, sleep, psychological difficulties, parental distress and objectively measured neurocognitive functioning.

RESULTS: Fatigue severity, functional impairment and cognitive behavioural responses to symptoms were equivalent in fatigued caMS and adolescents with CFS, and were significantly higher than in healthy controls and non-fatigued caMS. Neurocognitive functioning was impaired in both caMS groups, but was normal in adolescents with CFS and healthy controls. No between-group differences were identified in adolescent sleep behaviour or psychological difficulties. Parents of all illness groups had more unhelpful cognitions than parents of healthy controls. Psychological distress was elevated in parents of both fatigued groups.

CONCLUSIONS: Fifty percent of caMS reported clinically significant fatigue. Similarities between adolescent and parent cognitive behavioural factors in fatigued caMS and adolescents with CFS suggest important potential targets for intervention. Both fatigued and non-fatigued caMS had cognitive difficulties, suggesting that fatigue may need targeted intervention.

Source: Carroll S, Chalder T, Hemingway C, Heyman I, Bear H, Sweeney L, Moss-Morris R. Adolescent and parent factors related to fatigue in paediatric multiple sclerosis and chronic fatigue syndrome: A comparative study. Eur J Paediatr Neurol. 2018 Nov 2. pii: S1090-3798(18)30016-3. doi: 10.1016/j.ejpn.2018.10.006. [Epub ahead of print] https://www.ncbi.nlm.nih.gov/pubmed/30455131

Associations between clinical symptoms, plasma norepinephrine and deregulated immune gene networks in subgroups of adolescent with Chronic Fatigue Syndrome

Abstract:

BACKGROUND: Chronic fatigue syndrome (CFS) is one of the most important causes of disability among adolescents while limited knowledge exists on genetic determinants underlying disease pathophysiology.

METHODS: We analyzed deregulated immune-gene modules using Pathifier software on whole blood gene expression data (29 CFS patients, 18 controls). Deconvolution of immune cell subtypes based on gene expression profile was performed using CIBERSORT. Supervised consensus clustering on pathway deregulation score (PDS) was used to define CFS subgroups. Associations between PDS and immune, neuroendocrine/autonomic and clinical markers were examined. The impact of plasma norepinephrine level on clinical markers over time was assessed in a larger cohort (91 patients).

RESULTS: A group of 29 immune-gene sets was shown to differ patients from controls and detect subgroups within CFS. Group 1P (high PDS, low norepinephrine, low naïve CD4+ composition) had strong association with levels of serum C-reactive protein and Transforming Growth Factor-beta. Group 2P (low PDS, high norepinephrine, high naïve CD4+ composition) had strong associations with neuroendocrine/autonomic markers. The corresponding plasma norepinephrine level delineated 91 patients into two subgroups with significant differences in fatigue score.

CONCLUSION: We identified 29 immune-gene sets linked to plasma norepinephrine level that could delineate CFS subgroups. Plasma norepinephrine stratification revealed that lower levels of norepinephrine were associated with higher fatigue. Our data suggests potential involvement of neuro-immune dysregulation and genetic stratification in CFS.

Source: Nguyen CB, Kumar S, Zucknick M, Kristensen VN, Gjerstad J, Nilsen H, Wyller VB. Associations between clinical symptoms, plasma norepinephrine and deregulated immune gene networks in subgroups of adolescent with Chronic Fatigue Syndrome. Brain Behav Immun. 2018 Nov 9. pii: S0889-1591(18)30796-7. doi: 10.1016/j.bbi.2018.11.008. [Epub ahead of print] https://www.ncbi.nlm.nih.gov/pubmed/30419269

No evidence found for an increased risk of long-term fatigue following human papillomavirus vaccination of adolescent girls

Abstract:

INTRODUCTION: In 2013, the Netherlands Pharmacovigilance Center Lareb published an overview of reports of long-lasting fatigue following bivalent HPV-vaccination (2vHPV). After an update of this overview in 2015, concerns regarding the safety of 2vHPV was picked up by the media, which led to further reports of long-lasting fatigue. Therefore, the Dutch National Institute for Public Health and the Environment (RIVM) investigated a possible association between HPV-vaccination and long-term fatigue.

METHODS: In this retrospective cohort study conducted in the Integrated Primary Care Information database, we investigated the occurrence of chronic fatigue syndrome (CFS), fatigue ≥6 months and 3-6 months in all girls born in 1991-2000 during the follow-up period January 1st 2007-December 31st 2014 (2007-2008 pre-vaccination and 2009-2014 post-vaccination). Patients with certain fatigue ≥6 m were asked for consent to link their primary care information with vaccination data. Incidence rates per 10,000 person years (PY) for 12-16-year-old girls were compared between pre- and post-HPV-vaccine era. A self-controlled case series (SCCS) analysis was performed using consenting vaccinated cases. A primary high-risk period of 12 months after each dose was defined.

RESULTS: The cohort consisted of 69,429 12-16-year-old girls accounting for 2758 PY pre-vaccination and 57,214 PY post-vaccination. Differences between pre- and post-vaccination incidences (CFS: 3.6 (95% CI 0.5-25.7)/10,000 PY and 0.9 (0.4-2.1); certain fatigue ≥6 m: 7.3 (1.8-29.0) and 19.4 (16.1-23.4); certain fatigue 3-6 m: 0.0 and 16.6 (13.6-20.3), respectively) were not statistically significant. SCCS analyses in 16 consenting vaccinated cases resulted in an age-adjusted RR of 0.62 (95%CI 0.07-5.49).

CONCLUSIONS: Fatigue ≥6 m and 3-6 m was frequently found among adolescent girls, but CFS was rarely diagnosed. No statistically significant increased incidence rates were found post-vaccination compared to similar age groups of girls pre-vaccination. The SCCS analysis included a low number of cases but revealed no elevated risk of certain fatigue ≥6 m in the high-risk period.

Source: Schurink-Van’t Klooster TM, Kemmeren JM, van der Maas NAT, van de Putte EM, Ter Wolbeek M, Nijhof SL, Vanrolleghem A5, van Vliet JA, Sturkenboom M, de Melker HE. No evidence found for an increased risk of long-term fatigue following human papillomavirus vaccination of adolescent girls. Vaccine. 2018 Sep 19. pii: S0264-410X(18)31268-4. doi: 10.1016/j.vaccine.2018.09.019. [Epub ahead of print] https://www.sciencedirect.com/science/article/pii/S0264410X18312684?via%3Dihub (Full article)

Predictors of chronic fatigue in adolescents six months after acute Epstein-Barr virus infection: a prospective cohort study

Abstract:

INTRODUCTION: Acute Epstein-Barr virus (EBV) infection is a trigger of chronic fatigue and Chronic Fatigue Syndrome (CFS). This study investigated baseline predictors of chronic fatigue six months after an acute EBV infection.

MATERIALS AND METHODS: A total of 200 adolescents (12-20 years old) with acute EBV infection were assessed for 149 possible baseline predictors and followed prospectively. We performed linear regression to assess possible associations between baseline predictors and fatigue (Chalder Fatigue Questionnaire total score) six months after the acute EBV infection. A total of 70 healthy controls were included for cross-sectional reference. This study is part of the CEBA-project (Chronic fatigue following acute Epstein-Barr virus infection in adolescents).

RESULTS: In the final multiple linear regression model, fatigue six months after acute EBV infection was significantly and independently predicted by the following baseline variables (regression coefficient B[95% CI]): Sensory sensitivity (0.8[0.09 to 1.6]), pain severity (0.2[0.02 to 0.3]), functional impairment (1000 steps/day) (-0.3[-0.5 to -0.08]), negative emotions (anxiety) (0.4[0.2 to 0.6]), verbal memory (correct word recognition) (1.7[0.1 to 3.3]), plasma C-reactive protein (2.8[1.1 to 4.4] for CRP values >0.86) and plasma Vitamin B12 (-0.005[-0.01 to -0.001]).

CONCLUSIONS: Development of fatigue after acute EBV infection is to a larger extent predicted by baseline variables related to symptoms and functions than to baseline variables reflecting infectious and immune processes.

TRIAL REGISTRATION: ClinicalTrials, ID: NCT02335437, ttps://clinicaltrials.gov/ct2/show/NCT02335437.

Source: Pedersen M, Asprusten TT, Godang K, Leegaard TM, Osnes LT, Skovlund E, Tjade T, Øie MG, Wyller VBB. Predictors of chronic fatigue in adolescents six months after acute Epstein-Barr virus infection: a prospective cohort study. Brain Behav Immun. 2018 Sep 24. pii: S0889-1591(18)30625-1. doi: 10.1016/j.bbi.2018.09.023. [Epub ahead of print] https://www.ncbi.nlm.nih.gov/pubmed/30261303

Using the internet to cope with chronic fatigue syndrome/myalgic encephalomyelitis in adolescence: a qualitative study

Abstract:

BACKGROUND: Adolescents are increasingly using online resources for health purposes. Previous studies suggest that online provision of information about chronic fatigue syndrome or myalgic encephalomyelitis (CFS/ME) is neither balanced nor consistent with evidence-based practice. However, little is known about how adolescents with CFS/ME use the internet for their condition and whether this is helpful or harmful.

METHODS: Nine indepth, semistructured, qualitative interviews were conducted with young people (aged 12-17) recruited from a specialist paediatric CFS/ME service. Interviews explored the types of online resources accessed, motivations for doing so and how resource use related to patterns of coping.

RESULTS: Around the time of diagnosis, participants focused on gathering facts about CFS/ME and therefore used official resources (eg, National Health Service sites) that were considered reliable. This transitioned to exploring patient-led and peer-led spaces: health forums, Facebook and YouTube. Participants accessed these regularly, over the long term, and valued these sites for the personal stories, emotional content and interactive technology. Patient-led and peer-led sites supported coping, encouraging active behavioural management, providing social support and addressing stigmatised aspects of the condition. CFS/ME put a strain on normal adolescent life, such as identity and friendships. Online resources allowed participants to adapt and maintain a sense of normality.

CONCLUSIONS: Adolescents who use the internet find online resources helpful in seeking information and social support for their condition. Healthcare services should improve their online resources to meet the needs of younger users, providing evidence-based content in ways that are relevant to adolescents and that can meet the needs for social support, as well as providing information.

Source: Brigden A, Barnett J, Parslow RM, Beasant L, Crawley E. Using the internet to cope with chronic fatigue syndrome/myalgic encephalomyelitis in adolescence: a qualitative study. BMJ Paediatr Open. 2018 Aug 23;2(1):e000299. doi: 10.1136/bmjpo-2018-000299. eCollection 2018. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6109806/ (Full article)

Two-Year Follow-Up of Impaired Range of Motion in Chronic Fatigue Syndrome

Abstract:

OBJECTIVE: To measure changes in range of motion (ROM) over time in a cohort of 55 adolescents and young adults with chronic fatigue syndrome and to determine whether changes in ROM correlated with changes in health-related quality of life.

STUDY DESIGN: Participants underwent a standardized examination of 11 areas of limb and spine ROM at baseline and at 3- to 6-month intervals for 2 years, resulting in a ROM score that ranged from 0 (normal throughout) to 11 (abnormal ROM in all areas tested). We measured the time until the ROM score was ≤2 (the score in healthy age-matched controls). Change in ROM was measured by subtracting the 24-month from the baseline ROM score and by summing the degrees of change in the 10 tests with continuous outcomes. Health-related quality of life was measured using the Pediatric Quality of Life Inventory 4.0 (PedsQL).

RESULTS: The mean age at enrollment was 16.5 years (range 10-23). Two-year follow-up was available for 53 (96%). The proportion with a ROM score of >2 fell gradually over 2 years, from 78% at entry to 20% at 24 months (P < .001). ROM scores improved from a median of 5 at entry to 2 at 24 months (P < .001). The change in the summed degrees of improvement in ROM correlated positively with improvement in the PedsQL physical function subscale (r = 0.30; P < .03).

CONCLUSIONS: In association with multimodal therapy, young people with chronic fatigue syndrome experienced progressively less impairment in ROM over 2 years, correlating with improvements in the physical function subscale of the PedsQL.

Source: Rowe PC, Marden CL, Flaherty MAK, Jasion SE, Cranston EM, Fontaine KR, Violand RL. Two-Year Follow-Up of Impaired Range of Motion in Chronic Fatigue Syndrome. J Pediatr. 2018 Jun 1. pii: S0022-3476(18)30659-0. doi: 10.1016/j.jpeds.2018.05.012. [Epub ahead of print] https://www.ncbi.nlm.nih.gov/pubmed/29866593