Thirteen-year follow-up of children and adolescents with chronic fatigue syndrome

Abstract:

OBJECTIVE: To describe the educational, social, and symptomatic outcome of children and adolescents with chronic fatigue syndrome 13 years after illness onset.

METHODS: Between January 1984 and December 1987, 46 children and adolescents developed an illness suggestive of chronic fatigue syndrome. Follow-up questionnaires were obtained from 35 participants an average of 13 years after illness onset. Data were obtained concerning subsequent medical diagnoses, amount of school missed, presence and severity of current symptoms, and subjective assessment of degree of illness resolution.

RESULTS: Of the 35 participants, 24 were female (68.6%) and 11 were male (31.4%). Average age at illness onset was 12.1 years. Eight participants (22.9%) had an acute onset of symptoms, 27 (77.1%) had a gradual onset. No participant received an alternative medical diagnosis that could have explained the symptom complex between illness onset and follow-up. Thirteen participants (37.1%) considered themselves resolved of illness at follow-up; 15 participants (42.9%) considered themselves well but not resolved; 4 (11.4%) considered themselves chronically ill; and 3 (8.6%) considered themselves more ill than during the early years of illness. Correlation with the Medical Outcomes Study Short Form Health Survey was good for current level of symptoms and degree of recovery. Eight participants (22.9%) missed >2 years of school, and 5 of these were still ill at follow-up. Amount of school missed correlated with both illness severity at follow-up and perceived social impact of the illness.

CONCLUSIONS: These data demonstrate the presence of an illness consistent with the current definition of chronic fatigue syndrome. Eighty percent of children and adolescents affected had a satisfactory outcome from their fatiguing illness, although the majority of these participants had mild to moderate persisting symptoms. Twenty percent of participants remain ill with significant symptoms and activity limitation 13 years after illness onset. Chronic fatigue syndrome in children and adolescents may result in persistent somatic symptoms and disability in a minority of those affected.

 

Source: Bell DS, Jordan K, Robinson M. Thirteen-year follow-up of children and adolescents with chronic fatigue syndrome. Pediatrics. 2001 May;107(5):994-8. http://www.ncbi.nlm.nih.gov/pubmed/11331676

 

Psychological adjustment of adolescent girls with chronic fatigue syndrome

Abstract:

OBJECTIVE: To examine psychosocial problems and adaptation of adolescent girls with chronic fatigue syndrome (CFS).

METHODOLOGY: Thirty-six adolescent girls with CFS (mean age: 15.2 years; mean syndrome duration: 19.7 months) who fulfilled the criteria of the Centers for Disease Control and Prevention were examined by interviews regarding premorbid problems and by questionnaires regarding psychosocial functioning and distress, psychological attitudes, and coping resources. Data were compared with normative data.

RESULTS: Of the adolescents, 86.1% reported 1 or more premorbid problems (58.3% physical, 38.9% psychological, and 52.8% familial). Normal adjustment was reported for psychosocial self-esteem, social abilities, and attentional abilities. High adjustment to adult social standards of behavior was found, but low perceived competence in specific adolescent domains, such as athletic ability, romance, and participation in recreational activities. The girls reported predominantly internalizing problems. Normal achievement motivation, no debilitating fear of failure, and high internal locus of control were observed. Palliative reaction patterns and optimism were predominantly used as coping strategies.

CONCLUSIONS: The large number of premorbid problems suggests a possible contributing factor to the onset of the syndrome, although there were no reference data of healthy adolescents. In distinct domains of psychosocial adjustment, the adolescent girls with CFS showed strengths such as adequate self-esteem and scholastic and social abilities, and weaknesses such as low competence in adolescent-specific tasks and internalizing distress, which may partly be explained by syndrome-specific somatic complaints. The use of optimistic and palliative reaction patterns as coping strategies in this patient group indicates that the patients with CFS seem to retain an active and positive outlook on life, which may result in a rather adequate psychological adaptation to the syndrome, but also in maintenance of the syndrome by exceeding the physical limits brought about by the CFS. Our results on adjustment and coping strategies may be helpful to implement (individual) rehabilitation programs.

 

Source: van Middendorp H, Geenen R, Kuis W, Heijnen CJ, Sinnema G. Psychological adjustment of adolescent girls with chronic fatigue syndrome. Pediatrics. 2001 Mar;107(3):E35. http://www.ncbi.nlm.nih.gov/pubmed/11230616

 

Autonomic nervous system dysfunction in adolescents with postural orthostatic tachycardia syndrome and chronic fatigue syndrome is characterized by attenuated vagal baroreflex and potentiated sympathetic vasomotion

Abstract:

The objective was to determine the nature of autonomic and vasomotor changes in adolescent patients with orthostatic tachycardia associated with the chronic fatigue syndrome (CFS) and the postural orthostatic tachycardia syndrome (POTS).

Continuous electrocardiography and arterial tonometry was used to investigate the heart rate and blood pressure responses before and 3-5 min after head-up tilt in 22 adolescents with POTS and 14 adolescents with CFS, compared with control subjects comprising 10 healthy adolescents and 20 patients with simple faint. Heart rate and blood pressure variability, determined baroreceptor function using transfer function analysis, and measured cardiac vagal and adrenergic autonomic responses were calculated using timed breathing and the quantitative Valsalva maneuver.

Two of 10 healthy controls and 14 of 20 simple faint patients experienced vasovagal syncope during head-up tilt. By design, all CFS and POTS patients experienced orthostatic tachycardia, often associated with hypotension. R-R interval and heart rate variability were decreased in CFS and POTS patients compared with control subjects and remained decreased with head-up tilt. Low-frequency (0.05-0.15 Hz) blood pressure variability reflecting vasomotion was increased in CFS and POTS patients compared with control subjects and increased further with head-up tilt. This was associated with depressed baroreflex transfer indicating baroreceptor attenuation through defective vagal efferent response. Only the sympathetic response remained. Heart rate variability declined progressively from normal healthy control subjects through syncope to POTS to CFS patients. Timed breathing and Valsalva maneuver were most often normal in CFS and POTS patients, although abnormalities in select individuals were found.

Heart rate and blood pressure regulation in POTS and CFS patients are similar and indicate attenuated efferent vagal baroreflex associated with increased vasomotor tone. Loss of beat-to-beat heart rate control may contribute to a destabilized blood pressure resulting in orthostatic intolerance. The dysautonomia of orthostatic intolerance in POTS and in chronic fatigue are similar.

 

Source: Stewart JM. Autonomic nervous system dysfunction in adolescents with postural orthostatic tachycardia syndrome and chronic fatigue syndrome is characterized by attenuated vagal baroreflex and potentiated sympathetic vasomotion. Pediatr Res. 2000 Aug;48(2):218-26. http://www.ncbi.nlm.nih.gov/pubmed/10926298

 

Personality in adolescents with chronic fatigue syndrome

Abstract:

Our aim was to study the presence of personality traits and disorder in adolescents with Chronic Fatigue Syndrome (CFS). Personality was then compared to other measures of functioning such as presence of psychiatric disorder and rating on the Child Behavior Checklist 4-18 (CBCL) and in relation to CFS outcome.

Twenty-five adolescents with CFS followed-up after contacts with tertiary paediatric/psychiatric clinics were compared with 15 matched healthy controls. Interviews and questionnaires from parents and youngsters included Personality Assessment Schedule (PAS), Kiddie-SADS Psychiatric Interview, Child Behavior Checklist. CFS subjects were significantly more likely than controls to have personality difficulty or disorder.

Personality features significantly more common amongst them were conscientiousness, vulnerability, worthlessness and emotional lability. There was a nonsignificant association between personality disorder and worse CFS outcome. Personality difficulty or disorder was significantly associated with psychological symptoms and decreased social competence on the CBCL but it was distinguishable from episodic psychiatric disorder. Personality difficulty and disorder are increased in adolescents with a history of CFS. Personality disorder may be linked to poor CFS outcome.

 

Source: Rangel L, Garralda E, Levin M, Roberts H. Personality in adolescents with chronic fatigue syndrome. Eur Child Adolesc Psychiatry. 2000 Mar;9(1):39-45. http://www.ncbi.nlm.nih.gov/pubmed/10795854

 

Disturbed neuroendocrine-immune interactions in chronic fatigue syndrome

Abstract:

The present study was designed to investigate the interaction between neuroendocrine mediators and the immune system in chronic fatigue syndrome (CFS). We examined the sensitivity of the immune system to the glucocorticoid agonist dexamethasone and the beta2-adrenergic agonist terbutaline in 15 adolescent girls with CFS and 14 age- and sex-matched controls.

Dexamethasone inhibits T-cell proliferation in healthy controls and in CFS patients. However, the maximal effect of dexamethasone on T-cell proliferation is significantly reduced in CFS patients as compared with controls. The beta2-adrenergic receptor agonist terbutaline inhibits tumor necrosis factor-alpha production and enhances interleukin-10 production by monocytes. Our data demonstrate that the capacity of a beta2-adrenergic agonist to regulate the production of these two cytokines is also reduced in CFS patients.

We did not observe differences in baseline or CRH-induced cortisol and ACTH between CFS patients and controls. Baseline noradrenaline was similar in CFS and controls, whereas baseline adrenaline levels were significantly higher in CFS patients. We conclude that CFS is accompanied by a relative resistance of the immune system to regulation by the neuroendocrine system. Based on these data, we suggest CFS should be viewed as a disease of deficient neuroendocrine-immune communication.

 

Source: Kavelaars A, Kuis W, Knook L, Sinnema G, Heijnen CJ. Disturbed neuroendocrine-immune interactions in chronic fatigue syndrome. J Clin Endocrinol Metab. 2000 Feb;85(2):692-6. http://www.ncbi.nlm.nih.gov/pubmed/10690878

 

Psychiatric adjustment in adolescents with a history of chronic fatigue syndrome

Abstract:

OBJECTIVE: To ascertain psychiatric adjustment in youngsters with a history of childhood chronic fatigue syndrome (CFS).

METHOD: Subjects were 25 children and adolescents with CFS who were seen in tertiary pediatric/psychiatric clinics (mean age 15.6 years, seen a mean of 45.5 months after illness onset; 17 subjects had recovered and 8 were still ill) and 15 healthy matched controls. Youngsters and their parents (usually mothers) were interviewed and completed questionnaires. Instruments used included the Schedule for Affective Disorders and Schizophrenia for School-Age Children (K-SADS), the Child Behavior Checklist (CBCL), and the Harter Self-Esteem Questionnaire.

RESULTS: At assessment, psychiatric disorders (mainly anxiety and depressive disorders) were present in half the subjects with a history of CFS, a rate significantly higher than in healthy controls. On the CBCL youngsters with a history of CFS had an excess of psychological symptoms and decreased social competence. On the Harter Self-Esteem Questionnaire they reported reduced self-esteem, especially in social competence. Anxiety disorders were significantly more common in recovered subjects than in those with active CFS illness status.

CONCLUSIONS: Psychiatric disorders were found to be increased in adolescents with a history of severe CFS; CFS may enhance the risk for or share common predisposing factors with anxiety disorders.

Comment in: Chronic fatigue syndrome. [J Am Acad Child Adolesc Psychiatry. 2000]

 

Source: Garralda E, Rangel L, Levin M, Roberts H, Ukoumunne O. Psychiatric adjustment in adolescents with a history of chronic fatigue syndrome. J Am Acad Child Adolesc Psychiatry. 1999 Dec;38(12):1515-21. http://www.ncbi.nlm.nih.gov/pubmed/10596251

 

Orthostatic intolerance and chronic fatigue syndrome associated with Ehlers-Danlos syndrome

Abstract:

OBJECTIVE: To report chronic fatigue syndrome (CFS) associated with both Ehlers-Danlos syndrome (EDS) and orthostatic intolerance.

STUDY DESIGN: Case series of adolescents referred to a tertiary clinic for the evaluation of CFS. All subjects had 2-dimensional echocardiography, tests of orthostatic tolerance, and examinations by both a geneticist and an ophthalmologist.

RESULTS: Twelve patients (11 female), median age 15.5 years, met diagnostic criteria for CFS and EDS, and all had either postural tachycardia or neurally mediated hypotension in response to orthostatic stress. Six had classical-type EDS and 6 had hypermobile-type EDS.

CONCLUSIONS: Among patients with CFS and orthostatic intolerance, a subset also has EDS. We propose that the occurrence of these syndromes together can be attributed to the abnormal connective tissue in dependent blood vessels of those with EDS, which permits veins to distend excessively in response to ordinary hydrostatic pressures. This in turn leads to increased venous pooling and its hemodynamic and symptomatic consequences. These observations suggest that a careful search for hypermobility and connective tissue abnormalities should be part of the evaluation of patients with CFS and orthostatic intolerance syndromes.

 

Source: Rowe PC, Barron DF, Calkins H, Maumenee IH, Tong PY, Geraghty MT. Orthostatic intolerance and chronic fatigue syndrome associated with Ehlers-Danlos syndrome. J Pediatr. 1999 Oct;135(4):494-9. http://www.ncbi.nlm.nih.gov/pubmed/10518084

 

Patterns of orthostatic intolerance: the orthostatic tachycardia syndrome and adolescent chronic fatigue

Abstract:

OBJECTIVES: To describe the orthostatic tachycardia syndrome (OTS) in adolescents, similarities to and differences from chronic fatigue syndrome (CFS), and patterns of orthostatic intolerance during head-up tilt (HUT).

STUDY DESIGN: Using electrocardiography and arterial tonometry, we investigated the heart rate and blood pressure responses during HUT in 20 adolescents with OTS compared with 25 adolescents with CFS, 13 healthy control subjects, and 20 patients with simple faint.

RESULTS: Of the control subjects, 4 of 13 experienced typical vasovagal faints with an abrupt fall in blood pressure and heart rate, and 14 of 20 patients with simple faint experienced similar HUT responses. All patients with CFS (25/25) experienced severe orthostatic symptoms with syncope in 2 of 25, early orthostatic tachycardia during HUT in 16 of 23 (13/16 hypotensive), and delayed orthostatic tachycardia in 7 of 23 (6/7 hypotensive). Acrocyanosis and edema occurred in 18 of 25. Early orthostatic tachycardia occurred in 10 of 20 patients with OTS. Of these, 9 of 10 were hypotensive, but hypotension was delayed in 4 of 9. Delayed tachycardia occurred in 10 of 20 (all hypotensive). Acrocyanosis and edema occurred in most patients with CFS, fewer patients with OTS, and in one patient with simple faint. Orthostatic symptoms were similar but more severe in patients with CFS compared with patients with OTS.

CONCLUSIONS: Symptoms and patterns of orthostatic heart rate and blood pressure change in OTS overlap strongly with those of CFS. Orthostatic intolerance in OTS may represent an attenuated form of chronic fatigue pathophysiology.

 

Source: Stewart JM, Gewitz MH, Weldon A, Munoz J. Patterns of orthostatic intolerance: the orthostatic tachycardia syndrome and adolescent chronic fatigue. J Pediatr. 1999 Aug;135(2 Pt 1):218-25. http://www.ncbi.nlm.nih.gov/pubmed/10431117

 

Psychological symptoms in chronic fatigue and juvenile rheumatoid arthritis

Abstract:

OBJECTIVE: To determine if psychological morbidity in youth with chronic fatigue is caused by the stress of coping with a chronic illness.

STUDY DESIGN: Case-control study comparing pediatric patients with debilitating chronic fatigue and matched subjects with juvenile rheumatoid arthritis, a chronic medical illness with similar functional sequelae.

SETTING: Pediatric Infectious Diseases Clinic and Juvenile Rheumatoid Arthritis Clinic of Kosair Children’s Hospital.

PARTICIPANTS: Nineteen children and adolescents with debilitating chronic fatigue and 19 age- and sex-matched peers with juvenile rheumatoid arthritis. Outcome. Structured Interview, Kaufman Brief Intelligence Test, Child Behavior Checklist, and Youth Self-Report.

RESULTS: Intellectual functioning on the Kaufman Brief Intelligence Test Composite was average (103, standard score) for both groups. Pediatric patients with chronic fatigue had higher levels of internalizing psychological distress than patients suffering from juvenile rheumatoid arthritis, despite the fact that both groups had a similar pattern of decline in social and physical activities. Duration of illness did not explain the difference in psychological symptoms.

CONCLUSIONS: Psychological factors may play a more active role in debilitating chronic fatigue in pediatric patients than can be explained by the stress of coping with a similar chronic, non-life-threatening illness.

 

Source: Carter BD, Kronenberger WG, Edwards JF, Marshall GS, Schikler KN, Causey DL. Psychological symptoms in chronic fatigue and juvenile rheumatoid arthritis. Pediatrics. 1999 May;103(5 Pt 1):975-9. http://www.ncbi.nlm.nih.gov/pubmed/10224175

 

Orthostatic intolerance in adolescent chronic fatigue syndrome

Abstract:

OBJECTIVES: To demonstrate the association between orthostatic intolerance and the chronic fatigue syndrome (CFS) in adolescents and to delineate the form that orthostatic intolerance takes in these children.

STUDY DESIGN: We investigated the heart rate and blood pressure (BP) responses to head-up tilt (HUT) in 26 adolescents aged 11 to 19 years with CFS compared with responses in adolescents referred for the evaluation of simple faint and to responses in 13 normal healthy control children of similar age.

RESULTS: A total of 4/13 of the controls and 18/26 simple faint patients experienced typical faints with an abrupt decrease in BP and heart rate associated with loss of consciousness. One CFS patient had a normal HUT. A total of 25/26 CFS patients experienced severe orthostatic symptoms associated with syncope in 7/25, orthostatic tachycardia with hypotension in 15/25, and orthostatic tachycardia without significant hypotension in 3/25. Acrocyanosis, cool extremities, and edema indicated venous pooling in 18/25. None of the control or simple faint patients experienced comparable acral or tachycardic findings.

CONCLUSIONS: We conclude that chronic fatigue syndrome is highly related to orthostatic intolerance in adolescents. The orthostatic intolerance of CFS often has heart rate and BP responses similar to responses in the syndrome of orthostatic tachycardia suggesting that a partial autonomic defect may contribute to symptomatology in these patients.

 

Source: Stewart JM, Gewitz MH, Weldon A, Arlievsky N, Li K, Munoz J. Orthostatic intolerance in adolescent chronic fatigue syndrome. Pediatrics. 1999 Jan;103(1):116-21. http://www.ncbi.nlm.nih.gov/pubmed/9917448