Category: Pediatric ME/CFS

Association between school absence and physical function in paediatric chronic fatigue syndrome/myalgic encephalopathy

Abstract:

OBJECTIVE: To investigate factors associated with school attendance and physical function in paediatric chronic fatigue syndrome/myalgic encephalopathy (CFS/ME).

DESIGN: Cross-sectional study.

SETTING: Regional specialist CFS/ME service.

PATIENTS: Children and young people aged under 18 years.

OUTCOME MEASURES: Self-reported school attendance and physical function measured using the physical function subscale of the Short Form 36.

METHODS: Linear and logistic regression analysis of data from self-completed assessment forms on children attending a regional specialist service between 2004 and 2007. Analyses were done in two groups of children: with a completed Spence Children’s Anxiety Scale (SCAS) and with a completed Hospital Anxiety and Depression Scale (HADS).

RESULTS: Of 211 children with CFS/ME, 62% attended 40% of school or less. In children with completed SCAS, those with better physical function were more likely to attend school (adjusted odds ratio (OR) 1.70; 95% CI 1.36 to 2.13). This was also true for those with completed HADS (adjusted OR 2.05; 95% CI 1.4 to 3.01). Increasing fatigue and pain and low mood were associated with worse physical function. There was no evidence that anxiety, gender, age at assessment, family history of CFS/ME or time from onset of symptoms to assessment in clinic were associated with school attendance or physical function.

IMPLICATIONS: Paediatricians should recognise that reduced school attendance is associated with reduced physical function rather than anxiety. Improving school attendance in children with CFS/ME should focus on evidence-based interventions to improve physical function, particularly concentrating on interventions that are likely to reduce pain and fatigue.

 

Source: Crawley E, Sterne JA. Association between school absence and physical function in paediatric chronic fatigue syndrome/myalgic encephalopathy. Arch Dis Child. 2009 Oct;94(10):752-6. doi: 10.1136/adc.2008.143537. Epub 2008 Nov 11. https://www.ncbi.nlm.nih.gov/pubmed/19001477

 

Efficacy of cognitive behavioral therapy for adolescents with chronic fatigue syndrome: long-term follow-up of a randomized, controlled trial

Abstract:

OBJECTIVES: The purpose of this work was to assess the long-term outcome of adolescents with chronic fatigue syndrome who received cognitive behavioral therapy and to determine the predictive value of fatigue severity and physical impairments of the adolescent and the fatigue severity of the mother at baseline for the outcome of the treatment at follow-up.

PATIENTS AND METHODS: Sixty-six adolescent patients with chronic fatigue syndrome who previously participated in a randomized, controlled trial that showed that cognitive behavioral therapy was more effective than a waiting-list condition in reducing fatigue and improving physical functioning were contacted for a follow-up assessment. Fifty participants of the follow-up study had received cognitive behavioral therapy for chronic fatigue syndrome (32 formed the cognitive behavioral therapy group in the original trial, and 18 patients received cognitive behavioral therapy after the waiting period). The remaining 16 patients had refused cognitive behavioral therapy after the waiting period. The main outcome measures were fatigue severity (Checklist Individual Strength), physical functioning (Short-Form General Health Survey), and school attendance.

RESULTS: Data were complete for 61 patients at follow-up (cognitive behavioral therapy group: 47 patients; no-treatment group: 14 patients). The mean follow-up time was 2.1 years. There was no significant change in fatigue severity between posttreatment and follow-up in the cognitive behavioral therapy group. There was a significant further increase in physical functioning and school attendance (10% increase). The adolescents in the cognitive behavioral therapy group were significantly less fatigued and significantly less functionally impaired and had higher school attendance at follow-up than those in the no-treatment group. Fatigue severity of the mother was a significant predictor of treatment outcome.

CONCLUSIONS: The positive effects of cognitive behavioral therapy in adolescents with chronic fatigue syndrome are sustained after cognitive behavioral therapy. Higher fatigue severity of the mother predicts lower treatment outcome in adolescent patients.

 

Source: Knoop H, Stulemeijer M, de Jong LW, Fiselier TJ, Bleijenberg G. Efficacy of cognitive behavioral therapy for adolescents with chronic fatigue syndrome: long-term follow-up of a randomized, controlled trial. Pediatrics. 2008 Mar;121(3):e619-25. doi: 10.1542/peds.2007-1488. https://www.ncbi.nlm.nih.gov/pubmed/18310181

 

Chronic fatigue syndrome in children aged 11 years old and younger

Abstract:

Children in primary school can be very disabled by chronic fatigue syndrome or ME (CFS/ME). The clinical presentation in this age group (under 12 years old) is almost identical to that in older children.

AIM: To describe children who presented to the Bath paediatric CFS/ME service under the age of 12 years.

METHOD: Inventories measuring fatigue, pain, functional disability, anxiety, family history and symptoms were collected prospectively for all children presenting to the Bath CFS/ME service between September 2004 and April 2007. Data from children who presented to the service under the age of 12 are described and compared to those who presented at age 12 or older.

RESULTS: 178 children (under the age of 18) were diagnosed as having CFS/ME using the RCPCH criteria out of 216 children assessed. The mean age at assessment for children with CFS/ME was 14.5 years old (SD 2.9). Thirty-two (16%) children were under 12 years at the time of assessment, four children were under 5 years and the youngest child was 2 years old. Children under 12 were very disabled with mean school attendance of just over 40% (average 2 days a week), Chalder fatigue score of 8.29 (CI 7.14 to 9.43 maximum possible score = 11) and pain visual analogue score of 39.7 (possible range 0-100). Comparison with children aged 12 or older showed that both groups were remarkably similar at assessment. Twenty-four out of the 26 children with complete symptom lists would have been diagnosed as having CFS/ME using the stricter adult Centers of Disease Control and prevention (CDC) criteria.

CONCLUSION: Disability in the under-12 age group was high, with low levels of school attendance, high levels of fatigue, anxiety, functional disability and pain. The clinical pattern seen is almost identical to that seen in older children, and the majority of children would also be diagnosed as having CFS/ME using the stricter adult definition.

 

Source: Davies S, Crawley E. Chronic fatigue syndrome in children aged 11 years old and younger. Arch Dis Child. 2008 May;93(5):419-21. doi: 10.1136/adc.2007.126649. Epub 2008 Jan 11. https://www.ncbi.nlm.nih.gov/pubmed/18192312

 

A matched case control study of orthostatic intolerance in children/adolescents with chronic fatigue syndrome

Abstract:

This study aimed to define cardiovascular and heart rate variability (HRV) changes following head-up tilt (HUT) in children/adolescents with chronic fatigue syndrome (CFS) in comparison to age- and gender-matched controls.

Twenty-six children/adolescents with CFS (11-19 y) and controls underwent 70-degree HUT for a maximum of 30 min, but returned to horizontal earlier at the participant’s request with symptoms of orthostatic intolerance (OI) that included lightheadedness.

Using electrocardiography and beat-beat finger blood pressure, a positive tilt was defined as OI with 1) neurally mediated hypotension (NMH); bradycardia (HR <75% of baseline), and hypotension [systolic pressure (SysP) drops >25 mm Hg)] or 2) postural orthostatic tachycardia syndrome (POTS); HR increase >30 bpm, or HR >120 bpm (with/without hypotension).

Thirteen CFS and five controls exhibited OI generating a sensitivity and specificity for HUT of 50.0% and 80.8%, respectively. POTS without hypotension occurred in seven CFS subjects but no controls. POTS with hypotension and NMH occurred in both. Predominant sympathetic components to HRV on HUT were measured in CFS tilt-positive subjects.

In conclusion, CFS subjects were more susceptible to OI than controls, the cardiovascular response predominantly manifest as POTS without hypotension, a response unique to CFS suggesting further investigation is warranted with respect to the pathophysiologic mechanisms involved.

 

Source: Galland BC, Jackson PM, Sayers RM, Taylor BJ. A matched case control study of orthostatic intolerance in children/adolescents with chronic fatigue syndrome. Pediatr Res. 2008 Feb;63(2):196-202. https://www.ncbi.nlm.nih.gov/pubmed/18091356

 

Sympathetic cardiovascular control during orthostatic stress and isometric exercise in adolescent chronic fatigue syndrome

Abstract:

The chronic fatigue syndrome (CFS) has been shown to be associated with orthostatic intolerance and cardiovascular dysregulation. We investigated the cardiovascular responses to combined orthostatic stress and isometric exercise in adolescents with CFS.

We included a consecutive sample of 15 adolescents 12-18 years old with CFS diagnosed according to a thorough and standardized set of investigations, and a volunteer sample of 56 healthy control subjects of equal sex and age distribution. Heart rate, systolic, mean and diastolic blood pressure, stroke index, and total peripheral resistance index were non-invasively recorded during lower body negative pressure (LBNP) combined with two consecutive periods of handgrip. In addition, we measured baseline plasma catecholamines, and recorded symptoms. At rest, CFS patients had higher heart rate, diastolic blood pressure, plasma norepinephrine (P < 0.01), mean blood pressure and plasma epinephrine (P < 0.05) than controls.

During LBNP, CFS patients had a greater increase in heart rate, diastolic blood pressure, mean blood pressure (P < 0.05) and total peripheral resistance index (n.s.) than controls. During handgrip, CFS patients had a smaller increase in heart rate, diastolic blood pressure (P < 0.05), mean blood pressure and total peripheral resistance index (n.s.) than controls.

Our results indicate that adolescents with CFS have increased sympathetic activity at rest with exaggerated cardiovascular response to orthostatic stress, but attenuated cardiovascular response when performing isometric exercise during orthostatic stress. This suggests that CFS might be causally related to sympathetic dysfunction.

 

Source: Wyller VB, Saul JP, Walløe L, Thaulow E. Sympathetic cardiovascular control during orthostatic stress and isometric exercise in adolescent chronic fatigue syndrome. Eur J Appl Physiol. 2008 Apr;102(6):623-32. Epub 2007 Dec 8. https://www.ncbi.nlm.nih.gov/pubmed/18066580

 

Successful use of a primary care practice-specialty collaboration in the care of an adolescent with chronic fatigue syndrome

Abstract:

We report on the successful collaborative care of an adolescent with chronic fatigue syndrome between a primary care pediatrician and an academic chronic fatigue syndrome specialist located in different cities. Regular telephone and e-mail communication and clearly defined patient-care roles allowed for timely management of symptoms and marked clinical improvement. We discuss ways to improve the collaboration of primary care and subspecialty physicians for patients with chronic fatigue syndrome and children with special health care needs.

 

Source: Kuo DZ, Cheng TL, Rowe PC. Successful use of a primary care practice-specialty collaboration in the care of an adolescent with chronic fatigue syndrome. Pediatrics. 2007 Dec;120(6):e1536-9. https://www.ncbi.nlm.nih.gov/pubmed/18055669

 

Actigraphic assessment of sleep disorders in children with chronic fatigue syndrome

Abstract:

Children with chronic fatigue syndrome (CFS) often suffer from sleep disorders, which cause many physiological and psychological problems. Understanding sleep characteristics in children with CFS is important for establishing a therapeutic strategy. We conducted an actigraphic study to clarify the problems in sleep/wake rhythm and physical activity in children with CFS.

METHODS: Actigraphic recordings were performed for 1-2 weeks in 12 CFS children. The obtained data were compared with those of healthy age-matched children used as the control.

RESULTS: Sleep patterns were divided into two groups based on subjects’ sleep logs: irregular sleep type and delayed sleep phase type. Compared to the control group, total sleep time was longer and physical activity was lower in both groups of CFS. Continuous sleep for more than 10h was not uncommon in CFS. In the irregular sleep type, impaired daily sleep/wake rhythms and disrupted sleep were observed.

CONCLUSION: Using actigraphy, we could identify several characteristics of the sleep patterns in CFS children. Actigraphic analysis proved to be useful in detecting sleep/wake problems in children with CFS.

 

Source: Ohinata J, Suzuki N, Araki A, Takahashi S, Fujieda K, Tanaka H. Actigraphic assessment of sleep disorders in children with chronic fatigue syndrome. Brain Dev. 2008 May;30(5):329-33. Epub 2007 Nov 26. https://www.ncbi.nlm.nih.gov/pubmed/18031961

 

Alexithymia in adolescents with chronic fatigue syndrome

Abstract:

BACKGROUND: Alexithymia is postulated as an important factor in the development of medically unexplained physical symptoms. Chronic fatigue syndrome (CFS) is presently medically unexplained. The aim of this study was to investigate whether the prevalence of alexithymia was higher in adolescents with CFS compared to healthy adolescents. Comorbidity such as anxiety and depression were analyzed as possible confounding factors. Secondly, alexithymia was investigated as a prognostic factor for the recovery of CFS.

METHODS: A cross-sectional study was performed among 40 adolescent outpatients diagnosed with CFS and 36 healthy controls. The 20-item Toronto Alexithymia Scale was used to assess all participants for alexithymia. Additionally, all participants completed a number of questionnaires regarding fatigue (Checklist Individual Strength), somatic complaints (Checklist Somatization Inventory), depression (Children’s Depression Inventory), and trait anxiety (Spielberger State Trait Anxiety Questionnaire). A follow-up study was performed among the CFS adolescents 1 1/2 years after the initial assessment.

RESULTS: CFS adolescents scored higher only on the subscale identifying feelings of the TAS-20 [mean difference after adjustment for depression and anxiety 2.8 (95% CI: 0.6; 4.9]. Twelve CFS adolescents (30%) fulfilled criteria for alexithymia. This subgroup was characterized by higher scores for depression and anxiety and equal scores for fatigue and somatic complaints. At follow-up, no differences in recovery were established between the alexithymic and nonalexithymic CFS adolescents.

CONCLUSIONS: Alexithymia neither appears to be a unique correlate of CFS nor to be a prognostic factor for recovery of the CFS illness.

 

Source: van de Putte EM, Engelbert RH, Kuis W, Kimpen JL, Uiterwaal CS. Alexithymia in adolescents with chronic fatigue syndrome. J Psychosom Res. 2007 Oct;63(4):377-80. https://www.ncbi.nlm.nih.gov/pubmed/17905045

 

Abnormal thermoregulatory responses in adolescents with chronic fatigue syndrome: relation to clinical symptoms

Abstract:

OBJECTIVES: Chronic fatigue syndrome is a common and disabling disease of unknown etiology. Accumulating evidence indicates dysfunction of the autonomic nervous system. To further explore the pathophysiology of chronic fatigue syndrome, we investigated thermoregulatory responses dependent on catecholaminergic effector systems in adolescent patients with chronic fatigue syndrome.

PATIENTS AND METHODS: A consecutive sample of 15 patients with chronic fatigue syndrome aged 12 to 18 years and a volunteer sample of 57 healthy control subjects of equal gender and age distribution were included. Plasma catecholamines and metanephrines were measured before and after strong cooling of 1 hand. Acral skin blood flow, tympanic temperature, heart rate, and mean blood pressure were measured during moderate cooling of 1 hand. In addition, clinical symptoms indicative of thermoregulatory disturbances were recorded.

RESULTS: Patients with chronic fatigue syndrome reported significantly more shivering, sweating, sudden change of skin color, and feeling unusually warm. At baseline, patients with chronic fatigue syndrome had higher levels of norepinephrine, heart rate, epinephrine, and tympanic temperature than control subjects. During cooling of 1 hand, acral skin blood flow was less reduced, vasoconstrictor events occurred at lower temperatures, and tympanic temperature decreased more in patients with chronic fatigue syndrome compared with control subjects. Catecholamines increased and metanephrines decreased similarly in the 2 groups.

CONCLUSIONS: Adolescent patients with chronic fatigue syndrome have abnormal catecholaminergic-dependent thermoregulatory responses both at rest and during local skin cooling, supporting a hypothesis of sympathetic dysfunction and possibly explaining important clinical symptoms.

 

Source: Wyller VB, Godang K, Mørkrid L, Saul JP, Thaulow E, Walløe L. Abnormal thermoregulatory responses in adolescents with chronic fatigue syndrome: relation to clinical symptoms. Pediatrics. 2007 Jul;120(1):e129-37. https://www.ncbi.nlm.nih.gov/pubmed/17606539

 

Sympathetic predominance of cardiovascular regulation during mild orthostatic stress in adolescents with chronic fatigue

Abstract:

Haemodynamic abnormalities have been documented in the chronic fatigue syndrome (CFS), indicating functional disturbances of the autonomic nervous system responsible for cardiovascular control. This study was designed to explore the pathophysiology in adolescent CFS-patients by analysing RR-interval (RRI) variability and diastolic blood pressure (DBP) variability during mild orthostatic stress, using an algorithm which accounts for non-stationary biosignals.

A total of 27 adolescents with CFS and 33 healthy control subjects having equal age- and sex distribution underwent 15 min of 20 degrees head-up tilt (HUT). The spectral power densities of RRI and DBP were computed in the low-frequency (LF) band (0.04-0.15 Hz) and the high-frequency (HF) band (0.15-0.4 Hz) using an adaptive autoregressive algorithm to obtain a time-varying spectrum. RMSSD, a time domain index of RRI variability, was also computed. At rest, all indices of variability were similar in the two groups. During tilt, CFS patients had a larger increase in the LF/HF ratio (P<or=0.001) and normalized LF power of RRI (P<or=0.01), and a larger decrease in normalized HF power (P<or=0.01) of RRI than controls. CFS patients also had trends towards a larger decrease in absolute HF power of RRI and a larger increase in normalized LF power of DBP.

These findings suggest that adolescents with CFS have sympathetic predominance of cardiovascular regulation during very mild orthostatic stress. Possible underlying mechanisms are moderate hypovolemia, abnormalities of reflex control or physical de-conditioning.

 

Source: Wyller VB, Saul JP, Amlie JP, Thaulow E. Sympathetic predominance of cardiovascular regulation during mild orthostatic stress in adolescents with chronic fatigue. Clin Physiol Funct Imaging. 2007 Jul;27(4):231-8. https://www.ncbi.nlm.nih.gov/pubmed/17564672