Fibromyalgia and chronic fatigue syndrome in children

Abstract:

BACKGROUND: Fibromyalgia (FM) is characterized by widespread persistent pain and the presence of multiple discrete tender points. Chronic fatigue syndrome (CFS) is a syndrome characterized by debilitating fatigue associated with a variable number of non-specific complaints. Because neither condition had necessarily been recognized in children until recently, those patients have been treated as having school refusal without being diagnosed as having either syndrome. There is a considerable overlap of clinical symptoms between these two syndromes. It is therefore controversial as to whether these syndromes have the same pathogenesis or not. The aim of the present study was to clarify the relationship between these syndromes in children.

METHODS: Fifteen patients with FM and 21 patients with CFS were investigated both clinically and immunologically. Immunological assessments included thorough analysis of autoantibodies using several techniques.

RESULTS: Anti-nuclear antibody titers were higher and the prevalence of anti-Sa antibody was far more frequent in CFS patients than in FM patients.

CONCLUSION: CFS and FM are different from each other at least in childhood, from an immunological aspect, although some patients could have both conditions.

© 2011 The Authors. Pediatrics International © 2011 Japan Pediatric Society.

 

Source: Itoh Y, Shigemori T, Igarashi T, Fukunaga Y. Fibromyalgia and chronic fatigue syndrome in children. Pediatr Int. 2012 Apr;54(2):266-71. doi: 10.1111/j.1442-200X.2011.03514.x. Epub 2012 Jan 12. https://www.ncbi.nlm.nih.gov/pubmed/22115414

 

What stops children with a chronic illness accessing health care: a mixed methods study in children with Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME)

Abstract:

BACKGROUND: Paediatric Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME) is relatively common and disabling with a mean time out of school of more than one academic year. NICE guidelines recommend referral to specialist services immediately if severely affected, within 3 months if moderately affected and within 6 months if mildly affected. However, the median time-to-assessment by a specialist service in the UK is 18 months. This study used a mixed-methods approach to examine factors associated with time taken to access specialist services.

METHODS: Time-to-assessment was analysed as a continuous “survival-time” variable in Cox regression models using data from self-completed assessment forms for children attending a regional specialist CFS/ME service between January 2006 and December 2009. Semi-structured interviews about barriers experienced in accessing healthcare for their child were conducted with nine parents of children aged < 17 years (8 individual and one parent couple). Interviews were digitally recorded and analysed using “thematic analysis”.

RESULTS: 405 children were assessed between 2006 and 2009 and information on school attendance was available on 388. Only 1/125 with severe CFS/ME and 49/263 (19%) with mild to moderate CFS/ME were seen within NICE recommended timeframe. Increased fatigue was associated with shorter time to assessment (HR = 1.15; 95% CI 1.03, 1.29 per unit increase in Chalder fatigue score; P = 0.01). Time-to-assessment was not associated with disability, mood, age or gender. Parents described difficulties accessing specialist services because of their own as well as their GP’s and Paediatrician’s lack of knowledge. They experienced negative attitudes and beliefs towards the child’s condition when they consulted GPs, Paediatricians and Child Psychiatrists. Parents struggled to communicate an invisible illness that their child and not themselves were experiencing.

CONCLUSIONS: GPs, Child Psychiatrists and Paediatricians need more knowledge about CFS/ME and the appropriate referral pathways to ensure timeliness in referral to specialist services.

 

Source: Webb CM, Collin SM, Deave T, Haig-Ferguson A, Spatz A, Crawley E. What stops children with a chronic illness accessing health care: a mixed methods study in children with Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME). BMC Health Serv Res. 2011 Nov 11;11:308. doi: 10.1186/1472-6963-11-308. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3228771/ (Full article)

 

Factors affecting duration of chronic fatigue syndrome in pediatric patients

Abstract:

OBJECTIVE: To determine factors affecting duration of chronic fatigue syndrome (CFS) in pediatric patients.

METHODS: This Retrospective cohort consisted of patients with CFS at the regional referral infectious disease clinic for evaluation of fatigue in children and adolescents. Demographic, clinical, and laboratory data were analyzed to identify the impact on duration and severity of pediatric CFS.

RESULTS: A total number of 53 predominantly white (98.1%) patients with CFS, aged 9-18 years, were included in the study. Other than fatigue, headaches and sleep disturbance were the most common symptoms of pediatric CFS. Seropositive status for Borrelia burgdorferi (B. burgdorferi) and Epstein-Barr virus (EBV) was identified in 66% of the patients with the diagnosis of CFS by CDC criteria. No association was found between the CFS symptoms, gender, or age at diagnosis and duration of fatigue symptoms. Duration of CFS was associated with high Body-Mass Index (BMI) in a regression model after adjustment for patient’s age, gender, and seropositive status for B. burgdorferi and/or EBV (0.34 ± 0.15, P < 0.04).

CONCLUSIONS: BMI is significantly associated with prolonged duration of CFS.

 

Source: Petrov D, Marchalik D, Sosin M, Bal A. Factors affecting duration of chronic fatigue syndrome in pediatric patients. Indian J Pediatr. 2012 Jan;79(1):52-5. doi: 10.1007/s12098-011-0463-4. Epub 2011 May 27. https://www.ncbi.nlm.nih.gov/pubmed/21617905

 

What to do about attention and memory problems in children with CFS/ME: a neuropsychological approach

Abstract:

Our recent research has shown that children with chronic fatigue syndrome/myalgic encephalomyopathy (CFS/ME) describe problems with focused attention, sustained attention, recall and stress. Neuropsychological testing demonstrated lower scores for sustained attention, switching attention, divided attention, auditory learning and immediate recall compared to normative data. This paper describes what is currently known about memory and attention problems in children with CFS/ME and suggests a variety of strategies that could be used to overcome these difficulties.

 

Source: Tucker P, Haig-Ferguson A, Eaton N, Crawley E. What to do about attention and memory problems in children with CFS/ME: a neuropsychological approach. Clin Child Psychol Psychiatry. 2011 Apr;16(2):215-23. doi: 10.1177/1359104511403585.https://www.ncbi.nlm.nih.gov/pubmed/2157176

4

Cognitive dysfunction and mental fatigue in childhood chronic fatigue syndrome–a 6-month follow-up study

Abstract:

OBJECTIVES: Cognitive function was investigated in patients with childhood type chronic fatigue syndrome (CCFS) using the modified advanced trail making test (mATMT).

METHODS: mATMT was performed on 19 patients with CCFS and 25 healthy controls of comparable age and sex. The effectiveness of combined treatment with cognitive behavioral therapy (CBT) and pharmacotherapy and its relationship to cognitive function was investigated by evaluation of Chalder’s fatigue scale and behavior state before and after treatment for 6 consecutive months.

RESULTS: All three tasks (motor skill, selective and alternative attention, and spatial working memory) of the mATMT, especially the difference in reaction time of the alternative attention task, could discriminate CCFS patients from control subjects with 70.5% accuracy (P=0.007). CCFS patients showed significantly lower alternative attention and Chalder’s fatigue score before treatment (P=0.037 and 0.002, respectively). A significant improvement in performance status scores was found during the 6 months follow-up period with combined treatment with CBT and medication (P<0.001). Improvement of their cognitive symptoms was significantly correlated with improvement of alternative attention (r=0.653, P=0.002).

CONCLUSIONS: Higher-order level cognitive dysfunction affects CCFS pathogenesis. Alternative attention performance evaluated by the mATMT may be used to monitor improvement in patients with CCFS. Combined treatment with CBT and medication may be effective to improve poor attention characteristics associated with CCFS.

Copyright © 2011 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.

 

Source: Kawatani J, Mizuno K, Shiraishi S, Takao M, Joudoi T, Fukuda S, Watanabe Y, Tomoda A. Cognitive dysfunction and mental fatigue in childhood chronic fatigue syndrome–a 6-month follow-up study. Brain Dev. 2011 Nov;33(10):832-41. doi: 10.1016/j.braindev.2010.12.009. Epub 2011 May 6. https://www.ncbi.nlm.nih.gov/pubmed/21530119

 

Effects of an educational video film in fatigued children and adolescents: a randomised controlled trial

Abstract:

BACKGROUND: In many cases standard management for chronic fatigue syndrome (CFS) in children and adolescents is ineffective.

OBJECTIVE: To evaluate the efficacy of a video film intervention in preventing the development of persistent fatigue and significant school absence in fatigued children and adolescents.

DESIGN: Randomised controlled trial.

PARTICIPANTS: 91 patients with fatigue; 50 were randomly assigned to receive the intervention (video film plus usual care) and 41 to usual care only.

INTERVENTION: A video film on CFS and coping behaviour.

MAIN OUTCOME MEASURES: Self-reported fatigue severity, physical activity, motivation, concentration and school absence.

RESULTS: 79 patients had complete data at 12 months (42 in the video film and 37 in the usual care group). Mean fatigue severity and school absenteeism scores did not differ significantly, but in the intervention group the score for reduced motivation was higher (difference 2.9 (CI 0.1 to 5.7), p=0.038). 18% more patients in the intervention compared to the usual care group also had persistent fatigue with significant school absence. The odds of developing persistent fatigue and of missing >50% of school classes was 3.3 times higher in the intervention than in the usual care group (OR 3.3 (CI 1.0 to 11.3), p=0.046).

CONCLUSION:This particular video film intervention plus usual care in children and adolescents with unexplained fatigue did not prevent an unfavourable outcome and possibly had an adverse effect in that it reduced motivation and increased the incidence of persistent fatigue with significant school absence. The use of this particular film is not recommended.

 

Source: Bakker RJ, van de Putte EM, Kuis W, Sinnema G. Effects of an educational video film in fatigued children and adolescents: a randomised controlled trial. Arch Dis Child. 2011 May;96(5):457-60. doi: 10.1136/adc.2009.172072. Epub 2010 Sep 22. https://www.ncbi.nlm.nih.gov/pubmed/20861404

 

Biochemical and vascular aspects of pediatric chronic fatigue syndrome

Abstract:

OBJECTIVE: To evaluate the biochemical and vascular aspects of pediatric chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME).

DESIGN: Cross-sectional clinical study.

SETTING: Tayside, Scotland, United Kingdom.

PARTICIPANTS: Twenty-five children with CFS/ME and 23 healthy children recruited from throughout the United Kingdom.

INTERVENTIONS: Participants underwent a full clinical examination to establish a diagnosis of CFS/ME and were asked to describe and score their CFS/ME symptoms. Biochemical markers were measured. Arterial wave reflection was estimated to assess systemic arterial stiffness.

MAIN OUTCOME MEASURES: Markers of oxidative stress and free radicals, C-reactive protein level, white blood cell apoptosis, and arterial wave reflection.

RESULTS: Children with CFS/ME had increased oxidative stress compared with control individuals (isoprostanes: 252.30 vs 215.60 pg/mL, P = .007; vitamin C, mean [SD]: 0.84 [0.26] vs 1.15 [0.28] mg/dL, P < .001; vitamin E, 8.72 [2.39] vs 10.94 [3.46] microg/mL, P = .01) and increased white blood cell apoptosis (neutrophils: 53.7% vs 35.7%, P = .005; lymphocytes: 40.1% vs 24.6%, P = .009). Arterial stiffness variables did not differ significantly between groups (mean augmentation index, -0.57% vs -0.47%, P = .09); however, the derived variables significantly correlated with total (r = 0.543, P = .02) and low-density lipoprotein (r = 0.631, P = .004) cholesterol in patients with CFS/ME but not in controls.

CONCLUSIONS: Biomedical anomalies seen in adults with CFS/ME-increased oxidative stress and increased white blood cell apoptosis-can also be observed in children with clinically diagnosed CFS/ME compared with matched controls. Unlike in their adult counterparts, however, arterial stiffness remained within the reference range in these pediatric patients.

Comment in: Chronic fatigue syndrome in adolescence: where to from here? [Arch Pediatr Adolesc Med. 2010]

 

Source: Kennedy G, Khan F, Hill A, Underwood C, Belch JJ. Biochemical and vascular aspects of pediatric chronic fatigue syndrome. Arch Pediatr Adolesc Med. 2010 Sep;164(9):817-23. doi: 10.1001/archpediatrics.2010.157. https://www.ncbi.nlm.nih.gov/pubmed/20819963

 

Physical and functional impact of chronic fatigue syndrome/myalgic encephalomyelitis in childhood

Abstract:

OBJECTIVE: The aim of this study was to compare self-reported and parent-reported quality of life for a group of pediatric patients with chronic fatigue syndrome (CFS)/myalgic encephalomyelitis (ME) and age- and gender-matched healthy control children, to determine the extent of functional and physical impairment.

METHODS: The Child Health Questionnaire was completed by 25 children with CFS/ME, who were recruited throughout the United Kingdom, and by 23 age-, gender-, and Tanner scale-matched control children. In addition, patients were asked questions about the background to their illness (ie, precipitating factors), the status of their illness, and school attendance.

RESULTS: The median illness duration for patients was 3 years. Sixty-eight percent of the children said that their illness developed quickly, and the illness had an infectious onset for 88%. Only 1 child (4%) attended school full-time, whereas 12 (48%) attended school part-time and 8 (32%) received home tuition only. Children with CFS/ME scored significantly lower for 10 of 14 Child Health Questionnaire concepts; the lowest scores were observed for global health (scores of 21.4 and 84.1 for patients and control subjects, respectively; P < .0001) and role/social limitations attributable to physical health problems (scores of 24.9 and 100, respectively; P < .0001). Quality of life for the children with CFS/ME compared unfavorably with previously published results for pediatric patients with type 1 diabetes mellitus or asthma.

CONCLUSION: The quality of life of children with CFS/ME was profoundly reduced, compared with that of their healthy counterparts.

 

Source: Kennedy G, Underwood C, Belch JJ. Physical and functional impact of chronic fatigue syndrome/myalgic encephalomyelitis in childhood. Pediatrics. 2010 Jun;125(6):e1324-30. doi: 10.1542/peds.2009-2644. Epub 2010 May 17. https://www.ncbi.nlm.nih.gov/pubmed/20478937

 

Phenotypes of chronic fatigue syndrome in children and young people

Abstract:

OBJECTIVE: To investigate the heterogeneity of chronic fatigue syndrome (CFS/ME) in children and young people.

SETTING: Regional specialist CFS/ME service Patients Children and young people aged <19 years old.

METHODS: Exploratory factor analysis was performed on symptoms present at assessment in 333 children and young people with CFS/ME. Linear and logistic regression analysis of data from self-completed assessment forms was used to explore the associations between the retained factors and sex, age, length of illness, depression, anxiety and markers of severity (fatigue, physical function, pain and school attendance).

RESULTS: Three phenotypes were identified using factor analysis: muscoloskeletal (factor 1) had loadings on muscle and joint pain and hypersensitivity to touch, and was associated with worse fatigue (regression coefficient 0.47, 95% CI 0.25 to 0.68, p<0.001), physical function (regression coefficient -0.52, 95% CI -0.83 to -0.22, p=0.001) and pain. Factor 2 (migraine) loaded on noise and light hypersensitivity, headaches, nausea, abdominal pain and dizziness and was most strongly associated with physical function and pain. Sore throat phenotype (factor 3) had loadings on sore throat and tender lymph nodes and was not associated with fatigue or pain. There was no evidence that phenotypes were associated with age, length of illness or symptoms of depression (regression coefficient for association of depression with musculoskeletal pain -0.02, 95% CI -0.27 to 0.23, p=0.87). The migraine phenotype was associated with anxiety (0.40, 95% CI 0.06 to 0.74, p=0.02).

IMPLICATIONS: CFS/ME is heterogeneous in children with three phenotypes at presentation that are differentially associated with severity and are unlikely to be due to age or length of illness.

 

Source: May M, Emond A, Crawley E. Phenotypes of chronic fatigue syndrome in children and young people. Arch Dis Child. 2010 Apr;95(4):245-9. doi: 10.1136/adc.2009.158162. Epub 2009 Oct 19. https://www.ncbi.nlm.nih.gov/pubmed/19843509

 

Autoantibodies to lens epithelium-derived growth factor/transcription co-activator P75 (LEDGF/P75) in children with chronic nonspecific complaints and with positive antinuclear antibodies

Abstract:

Autoimmune fatigue syndrome (AIFS) is characterized by chronic nonspecific complaints, consistently positive antinuclear antibodies (ANA), and lack of alternate medical explanations. A newly recognized antibody, named anti-Sa, was detected in approximately 40% of the patients by Western blot (WB) using HeLa extract.

Some patients with AIFS later develop chronic fatigue syndrome (CFS), and most of them are positive for anti-Sa. On the other hand, Muro et al. reported anti-DFS70 in patients with CFS. Anti-Sa and anti-DFS70 were turned out to be same specificities by exchanging studies of blind sera. The target antigen of anti-DFS70 was identified as lens epithelium derived growth factor/transcription co-activator p75 (LEDGF/p75).

The objectives of this study are to confirm whether the target antigen of anti-Sa is also LEDGF/p75, and to develop ELISA system by using recombinant protein. Recombinant protein of LEDGF/p75 was purchased from Protein One (Bethesda, MD, USA). We developed an ELISA system to detect anti-LEDGF/p75 by coating this recombinant protein. 226 sera of AIFS patients (including 36 CFS patients) were applied to this ELISA assay and Western immunoblot, and it was revealed that anti-Sa-positive sera defined by WB and sera positive for anti-LEDGF/p75 on ELISA were identical.

Moreover, reactivities of anti-Sa on WB were inhibited by pre-incubating with recombinant LEDGF/p75, and eluted antibodies from the nitrocellulose membrane could react on the ELISA. These results confirm that the Sa antigen is LEDGF/p75. The ELISA assay using recombinant LEDGF/p75 could be a promising tool for measuring anti-Sa and consequently for diagnosing CFS.

 

Source: Kuwabara N, Itoh Y, Igarshi T, Fukunaga Y. Autoantibodies to lens epithelium-derived growth factor/transcription co-activator P75 (LEDGF/P75) in children with chronic nonspecific complaints and with positive antinuclear antibodies. Autoimmunity. 2009 Sep;42(6):492-6. Doi: 10.1080/08916930902736663. https://www.ncbi.nlm.nih.gov/pubmed/19657776