Adolescent Chronic Fatigue Syndrome: Prevalence, Incidence...

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  • Adolescent Chronic Fatigue Syndrome: Prevalence,Incidence, and Morbidity

    WHATS KNOWN ON THIS SUBJECT: Adolescent chronic fatiguesyndrome (CFS) is a disabling illness. The majority of adolescentswith CFS miss significant amounts of school. Little is known aboutthe epidemiology of CFS in adolescents.

    WHAT THIS STUDY ADDS: The prevalence of adolescent chronicfatigue syndrome (CFS) diagnosed by general practitioners wasapproximately 111 per 100 000 (0.11%) per year and theincidence of CFS diagnosed by pediatricians was 12 per 100 000(0.012%) per year. Adolescent CFS is a well-accepted diagnosisamong pediatricians but is probably underrecognized by primaryhealth care providers.

    abstractOBJECTIVE: To determine nationwide general practitioner (GP)-diagnosed prevalence and pediatriciandiagnosed incidence rates of ad-olescent chronic fatigue syndrome (CFS), and to assess CFS morbidity.

    DESIGN AND SETTING: We collected data from a cross-sectional na-tional sample among GPs and prospective registration of new patientswith CFS in all pediatric hospital departments in the Netherlands.

    PATIENTS AND METHODS: Study participants were adolescents aged10 to 18 years. A representative sample of GPs completed question-naires on the prevalence of CFS in their adolescent patients. Pediatrichospital departments prospectively reported new cases of CFS in ado-lescent patients. For every new reported case, a questionnaire wassent to the reporting pediatrician and the reported patient to assessCFS morbidity. Prevalence was estimated through the data from GPquestionnaires and incidence was estimated on the basis of casesnewly reported by pediatricians from January to December 2008.

    RESULTS: Prevalence was calculated as 111 per 100 000 adolescentsand incidence as 12 per 100 000 adolescents per year. Of newly re-ported patients with CFS, 91% scored at or above cutoff points forsevere fatigue and 93% at or above the cutoff points for physical im-pairment. Forty-five percent of patients with CFS reported 50%school absence during the previous 6 months.

    CONCLUSIONS: Clinically diagnosed incidence and prevalence ratesshow that adolescent CFS is uncommon compared with chronic fa-tigue. The primary adverse impact of CFS is extreme disability associ-ated with considerable school absence. Pediatrics 2011;127:e1169e1175

    AUTHORS: Sanne L. Nijhof, MD,a Kimberley Maijer, MD,a

    Gijs Bleijenberg, PhD,b Cuno S. P. M. Uiterwaal, MD, PhD,c

    Jan L. L. Kimpen, MD, PhD,a and Elise M. van de Putte, MD,PhDa

    aDepartment of Pediatrics, Wilhelmina Childrens Hospital, andcJulius Center for Health Sciences and Primary Care, UniversityMedical Center Utrecht, Netherlands, and bExpert CentreChronic Fatigue, Radboud University, Nijmegen Medical Centre,Netherlands

    KEY WORDSchronic fatigue, adolescents, epidemiology, incidence,prevalence

    ABBREVIATIONSCFSchronic fatigue syndromeCDCCenters for Disease Control and PreventionGPgeneral practitionerNIVELDutch Institute for Research of Health ServicesDPSUDutch Pediatric Surveillance UnitCIS-20Checklist Individual Strength

    www.pediatrics.org/cgi/doi/10.1542/peds.2010-1147

    doi:10.1542/peds.2010-1147

    Accepted for publication Jan 27, 2011

    Address correspondence to Sanne L. Nijhof, MD, Department ofPediatrics, Wilhelmina Childrens Hospital, University MedicalCenter Utrecht, Netherlands. E-mail: [email protected]

    PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275).

    Copyright 2011 by the American Academy of Pediatrics

    FINANCIAL DISCLOSURE: The authors have indicated they haveno financial relationships relevant to this article to disclose.

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  • Chronic fatigue syndrome (CFS) in ad-olescents often has an extensive dis-ease course that may lead to consider-able school absence and long-termconsequences for educational and so-cial development.13 To assess the ef-fects of chronic fatigue on Dutch soci-ety and adolescent health care, thedetermination of sound prevalenceand incidence rates is mandatory. Al-most all currently available estimatesof incidence and prevalence were de-termined in adult populations. More-over, international research has re-vealed a wide variation in rates, partlybecause of differences in patient agelimits and methods (eg, settings andapplied diagnostic criteria) used invarious studies. In adolescents, popu-lation surveys revealed annual inci-dence rates of 0.5% and prevalencerates of 0.19% to 1.29%,47 with female-to-male ratios that varied from 2:1 upto 3:1.5,8,9 Most of these estimates wereprimarily determined on the basis ofself-reported data obtained from pa-tients by use of questionnaires andtelephone interviews.

    Several diagnostic criteria for CFS ex-ist, of which the 1994 Centers for Dis-ease Control and Prevention (CDC) cri-teria are commonly used in theNetherlands.1013 According to thesecriteria CFS is characterized by severeand disabling new-onset fatigue thatlasts for at least 6 months and is ac-companied by 4 or more of the follow-ing symptoms: impaired memory orconcentration, sore throat, tender cer-vical or axillary lymph nodes, musclepain, multiple joint pain, new head-aches, unrefreshing sleep, and postex-ertional malaise. Somatic and psychi-atric illnesses should be excluded.10

    Accurate diagnosis of CFS is a complextask that requires exclusion of otherillnesses that could cause similar com-plaints but require different treat-ment. Therefore, clinical estimates ofCFS prevalence and incidence can be

    of value in addition to the aforemen-tioned population surveys.

    The primary aim of this study was todetermine adolescent CFS prevalenceand incidence rates as reported bygeneral practitioners (GPs) and pedia-tricians, respectively, in the Nether-lands. We also investigated the sever-ity of symptoms and disability and theextent of school absence associatedwith CFS in adolescents and assessedthe attitude of GPs and pediatricianstoward the diagnosis and manage-ment of this disease.

    METHODS

    Prevalence Estimations ObtainedFrom GPs

    In March 2008 a questionnaire wasmailed to a sample of 735 GPs, 10% ofall GPs in the Netherlands, who wererandomly selected by the Dutch Insti-tute for Research of Health Services(NIVEL). The GPs were given the oppor-tunity to respond bymail, e-mail, fax, ortelephone. Reminders were sent every3 months during the period from No-vember 2008 to May 2009.

    The GPs were asked to submit theirpractice size and the number of pa-tients with CFS aged 10 to 18 years whowere receiving care. Specifically, theGPs were asked to query their ICPCcoding-systemdatabase (InternationalClassification of Primary Care), whichis used by all Dutch GPs, for fatigueand its derivatives.

    Additional questions to the GPs con-cerned the criteria for the diagnosisandmanagement of these patients. If aGP indicated not to have any patientswith CFS, the GP was asked for a pos-sible reason.

    Population estimatesweremade by ex-trapolation of sample data to the pop-ulation level, because the entire Dutchpopulation is obliged to be registeredwithin the practice of a GP and the av-erage GP practice sizes are compara-

    ble (2000 patients). Furthermore, re-ferral by a GP is mandatory forpatients to access hospital care (ie, pe-diatric care) in the Netherlands, and inturn GPs are informed on the progressof diagnostics and/or treatment of pa-tients referred to the hospital andother health care professionals and fa-cilities (eg, psychologists and rehabili-tation centers).

    Incidence Estimations ObtainedFrom Pediatricians

    During the period from July 2007 to De-cember 2008 all new cases of CFS inadolescents (aged 1018 years) wereassessed monthly by the Dutch Pediat-ric Surveillance Unit (DPSU). The DPSUis a national registry for pediatric dis-orders that includes all 103 pediatrichospital departments in the Nether-lands and consequently reaches allpediatricians working in the Nether-lands. The DPSU is 1 of 12 activepediatric-surveillance registries world-wide. Each pediatric department in theNetherlands receives a monthly (elec-tronic) card to report new cases ofvarious pediatrics disorders. Formoreinformation on survey methods seewww.inopsu.com.14 For validation ofthe estimated incidence number, apostal questionnaire was sent to allpediatric departments that had not re-ported any adolescent patients withCFS in 2008 to verify that there were nocases of CFS in adolescent patientsthat had not been reported. Thesequestionnaires also queried for infor-mation on whether management of pa-tients suspected to have CFS by thesepediatricians differed from that of pro-spective reporting departments. Inci-dence was calculated as the total num-ber of patients reported from Januaryto December 2008.

    For every prospectively reported pa-tient with CFS, the pediatrician and pa-tient were requested by the DPSU tocomplete a questionnaire. Pediatri-

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  • cians were asked whether they diag-nosed CFS according to the 1994 CDCcriteria. Patients received, throughtheir pediatricians, an anonymizedshort survey regarding gender, age,duration of complaints before diagno-sis, assessment of fatigue and concen-tration problems, functional impair-ment, type of complaints, and schoolabsence. Fatigue was assessed withthe self-report questionnaire ChecklistIndividual Strength-20 (CIS-20) sub-scales fatigue severity (8 items) andconcentration problems (5 items).The CIS-20 is a reliable assessmenttool with excellent internal consis-tency (Cronbachs : 0.93) and dis-criminative validity for CFS.15,16 The cut-off point for severe fatigue was set at40 on the subscale fatigue sever-ity.16,17 Disabilities were measured byusing the self-report Child HealthQuestionnaire-Child Form subscalephysical functioning (9 items). Thisassessment tool is reliable and hasbeen validated with good internal con-sistency (Cronbachs: 0.560.90).18,19

    The cutoff point for impaired physicalfunctioning was set at 85 (healthypopulations mean of 96.8 minus 2 SDs[2 5.4]).19 As a reference group, stu-dents at a Dutch secondary school (deBreul, Zeist) were invited to partici-pate. Adolescents who were sufferingfrom a chronic illness and adolescentscurrently under treatment were ex-cluded. The participation rate was85%. From this group of students werandomly selected individuals for acontrol group matched for age andgender (n 144; mean age: 15.3 0.6years; 79% girls).

    Ethics

    The medical ethics committee of theUniversity Medical Center Utrecht ap-proved this study. Case data obtainedby the GP registration surveys wereanonymous; investigators did not haveaccess to information that would allowthem to identify or contact these pa-

    tients and their families. The require-ment for informed consent was thuswaived. Informed consent was obtainedin all cases for which data were trace-able to the individual patient, ie, the inwhich questionnaires were sent to thepatients of reporting pediatricians.

    Analysis

    For estimation of the prevalence andincidence rates, the total number ofadolescents in the Netherlands in 2008was determined to be 1786 933 ac-cording to figures of the Dutch Bureauof Statistics.20

    Statistical analyses were performedby using SPSS version 15.0 (SPSS Inc,Chicago, IL). Outcome variables werepresented as means with SDs andpercentages.

    RESULTS

    Prevalence Estimates

    A total of 354 GPs (48%) responded, ofwhom 304 (41%) returned a completedquestionnaire. There were 81 adoles-cent patients with CFS reported by 42GPs. Absolute CFS number was esti-mated to be 1976 adolescents nation-wide. Thepoint prevalence of adolescentCFS was calculated at 111 per 100 000adolescents (0.11%). For details on prev-alence calculation, see Fig. 1.

    Incidence Estimates

    Of 103 Dutch pediatric departments, 92reported 200 adolescent patients withnewly diagnosed CFS cases in 2008. Ofthese patients, a total of 16 were ex-cluded because of age (n 3), doublereport (n 9), or a revised diagnosisby the reporting pediatrician (n 4),leaving 184 newly reported cases.

    Eleven pediatric departments did notrespond to the questionnaires of theDPSU. Therefore, the overall pediatricdepartment response rate was 89%.

    Analysis of the 11 hospitals that did notreport new cases of adolescent CFSdid not reveal trends in hospital size,location (urban versus rural), or orga-nization level (academic versus regu-lar). The locations of the nonreportinghospitals seemed randomly associ-ated, so different incidence rates of pa-tients with CFS in these hospitals arenot expected.

    On the basis of the assumption that thesame number of adolescents with CFSwere referred to nonresponding depart-ments the incidence rate was correctedfor the 11% nonresponse. The CFS inci-dence rate was 12 per 100 000 adoles-cents per year (0.012%). For details onincidence calculation, see Fig. 1.

    From all DPSU-derived reports, com-pleted questionnaires were obtained

    FIGURE 1Incidence and prevalence rate calculations.

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  • for 81% of patient questionnaires and96% of pediatrician questionnaires. Inall reported cases the CFS diagnosiswas in compliance with CDC criteria.

    Demographics and Morbidity

    Demographic characteristics were ob-tained from 184 CFS cases. The aver-age age of illness onset was 15 years(SD: 1.9 years), with a female-to-male ratio of 5:1. Median illness dura-tion from start of complaints until di-agnosis was 17 months (SD: 18.1months), but illness duration rangedwidely (6110 months). In 22% of pa-tients the illness started after an acuteepisode of infectious disease (of thesepatients 52% had a current or recentEpstein Barr virus infection); 10% ofpatients had an acute noninfectiousonset. The remaining majority of pa-tients (68%) had a gradual onset of thesymptom pattern over weeks tomonths. Themorbidity of patients com-pared to their healthy peers is summa-

    rized in Table 1. Most patients (91%)scored at or above the cutoff point of40 for severe fatigue (mean: 49.8) onthe CIS-20 fatigue-severity subscale.Most patients (93%) also scored at orbeyond the cutoff point of 85 onthe Health Questionnaire-Child Formphysical-functioning subscale. Schoolabsence was high: 90% of patientswith CFS reported considerable (de-fined as 15%50% school absence) tocomplete school absence in the previ-ous 2 weeks and 6 months (Fig. 2). Themean number of concurrently presentCDC symptoms was 5, with unrefresh-ing sleep the most commonly re-ported (84.4%) and tender lymphnodes the least reported (31.3%).

    Attitudes to Diagnosis and DiseaseManagement

    Among contacted GPs, 7% indicatedthat they were too busy to cooperateor never cooperated with postal sur-veys. Forty-three percent of GPs re-

    ported that they themselves diagnosedadolescent CFS in their patients, and 1of 3 of these GPs used CDC criteria. CFSin adolescents was accepted as a dis-tinct diagnosis by 51% of all respond-ing GPs. For detailed information on GPattitudes, see Table 2.

    Ninety-six percent of the consulted pe-diatric departments regarded CFS as adistinct diagnosis, and 92% used CDCcriteria for diagnosing CFS. Of the de-partments who had never diagnosedCFS in an adolescent patient, 1 statedthat this diagnose is applicable only inadults and 3 that this diagnosis is in-adequate. For CFS treatment, patientswere referred to a psychologist, phys-ical therapist, rehabilitation center,and/or a tertiary care center.

    DISCUSSION

    This study was the first, to our knowl-edge, in which nationwide cross-sectional prevalence data as well as

    TABLE 1 Morbidity of Adolescents With CFS Reported by Pediatric Departments in 2008

    CFS Healthy Peers P

    Age, mean, SD 15.2 (1.9) 15.3 (0.6) .556a

    Gender, % girls 85 79 .250b

    Duration of symptoms, mean (SD), mo 17 (18.1) Onset of disease, %Acute 10 Gradual 68 Postinfectious 22 CIS-20, mean (SD)Subscale fatigue severity (range: 856) 49.8 (6.4) 22.6 (10.7) .000a

    Subscale concentration problems (range: 535) 26.5 (6.9) 16.3 (7.1) .000a

    Child Health Questionnaire, mean (SD)Subscale physical functioning (range: 0100) 58.6 (20.4) 95.1 (7.6) .000a

    School absence, % Previous 2 wk Previous 6 mo Previous 2 wk Previous 6 moMean (SD) 58.5 (33.0) 54.2 (29.1) 1.5 (3.9) 4.9 (6.9) .000a

    Minimal (5%) 7.1 4.0 91.2 75.6Mild (5%15%) 4.0 5.0 5.9 18.5Considerable (15%50%) 34.3 46.5 2.9 5.9Severe (50%75%) 18.2 15.8 0.0 0.0Almost complete to complete (75%100%) 36.4 28.7 0.0 0.0Reported CDC symptoms, %Mean (SD) (range: 08) 5.0 (1.6) Unrefreshing sleep, % 84.4 Postexertional malaise lasting24 h, % 79.7 Memory and/or concentration problems, % 78.9 Headaches, % 78.1 Muscle pain, % 59.4 Joint pain, % 48.4 Sore throat, % 43.0 Tender lymph nodes, % 31.3

    a Independent Student t test.b 2-test/Fishers Exact test.

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  • prospective incidence data on adoles-cent patients with CFS were collectedand in which CFS diagnosis was pri-marily confirmed by either a GP or apediatrician. We estimated the GP-diagnosed prevalence of adolescentCFS to be 111 per 100 000 adolescents(0.11%) per year and the pediatrician-

    diagnosed incidence of adolescent CFSto be 12 per 100 000 adolescents(0.012%) per year. Fatigue severity andphysical impairment as well as schoolabsence were found to be remarkablyhigh in adolescents with CFS. Thesedata strongly suggest that adolescentCFS should be regarded as a serious

    illness with corresponding conse-quences such as delay in educationaland social development.

    Strengths and Weaknesses

    Both for prevalence rates and incidencerates we made use of the independentinstitutes NIVEL and DPSU, which use re-liable methods to assess the impact ofnationwide public health issues. To de-termine reliable prevalence numbers, arepresentative sample of GPs was se-lectedbyNIVEL to allow for extrapolation.In regard to the validity of prevalencerates, we assumed that the GPs, as agatekeepers of health care, werewell in-formed by health care professionals in-volved in the diagnostic process andtreatment of CFS in adolescents, whichsuggests that the prevalence estimateswereaccurately reported. Thedatapres-ent in Table 2 do indeed show that al-most 75% of the CFS diagnoses in ado-lescents were made by health careprofessionals other than the GP. Unfor-tunately, although we sent several re-minders and GPS had the option to re-spond bymail, e-mail, or telephone, theGP response rate remained low (48%)and possibly introduced a selectionbias.

    The nationwide response rate on adoles-cent CFS incidence among pediatric de-partments that was prospectively de-rived by the DPSU was high (89%).Furthermore, the rate of adherence toCDC criteria (92%) suggested that Dutchpediatricians adequately diagnosed CFSin adolescents. Although CFS seemed tobe diagnosed adequately by pediatri-cians in our study, underreferral to pedi-atricians by the GPsmight have led to anunderestimation of incidence. However,if a GP does not diagnose CFS in adoles-cents this does not necessarilymean theGP is reluctant to refer a severely fa-tigued and disabled adolescent to a pe-diatrician or other health care profes-sional. Our study doesnot supply data onthe referral pattern of the GP.

    FIGURE 2Distribution of school absence.

    TABLE 2 GPs Attitudes Toward and Management of Adolescent CFS According toQuestionnaire Responses

    Answers GP Responses, n (%)

    Total GPs with adolescent patients withCFS

    42 (13.8)

    Who diagnosed the patient? Myself (GP) 18 (42.9)Pediatrician 22 (52.4)Other 10 (23.8)

    If you diagnosed the patient yourselfa

    Which criteria did you use? CDC criteria 6 (33.3)Other 12 (66.7)

    Did you refer this patient? Yes, to a pediatrician 11 (61.1)Yes, but not to a pediatrician 4 (22.2)No 3 (16.7)

    How do you register this chronicallyfatigued patient?b

    CFS 22 (52.4)Chronic fatigue 6 (14.3)Fatigue/weakness 15 (35.7)Malaise 2 (4.8)Other 2 (4.8)

    Total GPs with no adolescent patientswith CFS

    262 (86.2)

    For what reason do you not have anyadolescent patients with CFS in yourpractice?b

    There are no adolescents in mypractice that can be considered tohave this diagnosis

    161 (61.5)

    I only consider this diagnosis in adults 26 (9.9)I find this diagnosis inadequate 98 (37.4)I dont acknowledge this diagnosis 33 (12.6)Other 17 (6.5)

    a Percentages of GPs who diagnosed CFS in their own patients.b Multiple answers could apply per GP.

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  • Determination of prevalence rates in pe-diatricpracticeswouldhave led toanun-derdetermination of CFS prevalence, be-cause after the diagnostic processpediatricians referred adolescents fortreatment to specialized psychologistsor to a rehabilitation center.

    Comparison With Other Studies

    The estimated rates of the occurrenceof CFS in adolescents that we reportwere lower than those found in previ-ous studies. However, most recentstudies on CFS incidence and preva-lence rates in adolescents are basedon population surveys performed withmethods that were different fromthose used in our study.

    Rimes et al used telephone question-naires in a random adolescent popula-tion sample to longitudinally deter-mine the incidence and prevalence ofdisabling fatigue. A validated question-naire was used to assess psychological/psychiatric diagnoses, but no physicalexaminations or investigations to ex-clude physical causes of fatigue wereperformed. In 824 adolescents, 4 newcases of CFS were reported, ie, a inci-dence rate of 0.5%.7

    Chalder et al determined the prevalenceof adolescent CFS in a cross-sectional in-terview study, in which diagnosis basedon CDC criteria (by interview only) wascompared with self-report by patientsand parental report. Prevalence rateswere, respectively, 0.19%, 0.57%, and0.038%. There was no concordance be-tween parental report that a child hadCFS and operationally defined CFS (CDCcriteria).4 Thesefindings illustrate the ef-fect of the use of different diagnosticmethods.

    Jones et al also conducted a randomdigit-dialing survey in which adoles-cents with CFS-like illness were identi-fied. Of all identified adolescents (31 of8586) only 35.5% volunteered to un-dergo clinical evaluation. None of

    these adolescents met the CFS casedefinition. The authors extracted an es-timated adolescent CFS prevalence ofthe adult population of 50 per 100 000.6

    Farmer et al used 2 twin registries toderive life-time prevalence estimatesof chronic fatigue in adolescents. Se-lected families were sent question-naires and parents were interviewedby telephone. Recorded physician diag-noses were reviewed on paper by anindependent doctor, but patients didnot undergo an additional medical ex-amination. When the diagnosis wasmade in adherence to CDC criteria, theCFS incidence was 1.29%. When fatigueduration was only 3 months, the inci-dence increased to 1.90%. For fatiguewithout any of the 4 accompanyingsymptoms the incidence increased to2.43%.5 These findings illustrate the ef-fect of applied criteria on CFS inci-dence. Adherence to the UK NICE (Na-tional Institute for Health and ClinicalExcellence) criteria leads to higher in-cidence rates, because the diagnosisof CFS according to NICE guidelines re-quires symptoms that persist for only3 months instead of 6 months.21

    Demographical data from the newly re-ported patients with CFS regarding ageof onset and gender ratio are in linewithearlier studies.1,4,8,9,22 Our findings sup-port previous evidence on the disablingcharacter of this illness and high level ofschool absence.1,8,9,2224 Our data re-garding GPs attitudes toward adoles-cent CFS are in line with results of pre-vious studies of CFS attitudes in adultsin the Netherlands. Among GPs, 58%98% accepted CFS as a recognizableclinical entity2527 and 48%66% feltunconfident in diagnosing CFS.26,27

    Implications of the Findings

    Although both prevalence and incidencerates are possibly underestimated, thisdoes not change the fact that adolescentCFS seems uncommon in comparisonwith the high prevalence of severe fa-

    tigue in the Netherlands. A recent studyshowed a prevalence rate of adolescentsevere fatigue of 20.5% in girls and 6.5%in boys, of whom80%and 61.5%, respec-tively, reported fatigue lasting1monthand 46.9% and 35.2% for 3 months.17

    Thesedata support the theory that CFS isa specific clinical entity in the spectrumof chronic and severe fatigue. The bur-den to society mainly consists of the as-sociated extensive school absence andits long-lasting and disabling effects.

    Although CFS in adolescent patientsseemed to be diagnosed adequately bypediatricians in our study, underre-porting remains a point of attention inclinical studies. Results of a recentpopulation-based study in the Nether-lands showed a 1% prevalence rate ofadult self-reported CFS. Strikingly, 70%of these adults consulted their GP fortheir complaints of fatigue, but GPs di-agnosed CFS in only 6.7% of these pa-tients.28 We suggest that all adoles-cents who consult their GP for acomplaint of severe and long-lastingfatigue should be referred to a pedia-trician for proper disease diagnosisand initiation of treatment.

    CONCLUSIONS

    Adolescent CFS is an uncommon ill-ness compared with chronic fatigue.The primary adverse impact of CFS inadolescents is its extremely disablingcharacter and associated high rates ofschool absenteeism. In contrast to thehigh acceptance of adolescent CFSamong pediatricians, CFS is probablyunderrecognized by other primaryhealth care providers.

    ACKNOWLEDGMENTSOur research was partially funded bythe DPSU.

    We thank all pediatricians and GPswho contributed to this study fortheir response and Robert Wisse fordesigning our tables and graphs.

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  • REFERENCES

    1. Crawley E, Sterne J. Association betweenschool absence and physical function inpaediatric chronic fatigue syndrome/myalgic encephalopathy. Arch Dis Child.2009;94(10):752756.

    2. Marshall GS. Report of a workshop on theepidemiology, natural history, and patho-genesis of chronic fatigue syndrome in ad-olescents. J Pediatr. 1999;134(4):395405

    3. Dowsett EG, Colby J. Long term sickness ab-sence due to ME/CFS in UK schools: an epi-demiological study withmedical and educa-tional implications. J Chron Fatigue Synd.1997;3(2):2942

    4. Chalder T, Goodman R, Wessely S, Hotopf M,Meltzer H. Epidemiology of chronic fatiguesyndrome and self reported myalgic en-cephalomyelitis in 515 year olds: crosssectional study. BMJ. 2003;327(7416):654655

    5. Farmer A, Fowler T, Scourfield J, Thapar A.Prevalence of chronic disabling fatigue inchildren and adolescents. Br J Psychiatry.2004;184:477481

    6. Jones JF, NisenbaumR, Solomon L, ReyesM,Reeves WC. Chronic fatigue syndrome andother fatiguing illnesses in adolescents: apopulation-based study. J Adolesc Health.2004;35(1):3440

    7. Rimes KA, Goodman R, Hotopf M, Wessely S,Meltzer H, Chalder T. Incidence, prognosis,and risk factors for fatigue and chronic fa-tigue syndrome in adolescents: a prospec-tive community study. Pediatrics. 2007;119(3). Available at: www.pediatrics.org/cgi/content/full/119/3/e603

    8. Bell DS, Jordan K, Robinson M. Thirteen-year follow-up of children and adolescentswith chronic fatigue syndrome. Pediatrics.2001;107(5):994998

    9. van de Putte EM, Uiterwaal CS, Bots ML, KuisW, Kimpen JL, Engelbert RH. Is chronic fa-tigue syndrome a connective tissue disor-der? A cross-sectional study in adolescents.Pediatrics. 2005;115(4). Available at: www.pediatrics.org/cgi/content/full/115/4/e415

    10. Fukuda K, Straus SE, Hickie I, Sharpe MC,

    Dobbins JG, Komaroff A. The chronic fatiguesyndrome: a comprehensive approach toits definition and study. InternationalChronic Fatigue Syndrome Study Group.Ann Intern Med. 1994;121(12):953959

    11. Holmes GP, Kaplan JE, Gantz NM, et al.Chronic fatigue syndrome: a working casedefinition. Ann Intern Med. 1988;108(3):387389

    12. Lloyd AR, Hickie I, Boughton CR, Spencer O,Wakefield D. Prevalence of chronic fatiguesyndrome in an Australian population. MedJ Aust. 1990;153(9):522528

    13. SharpeMC, Archard LC, Banatvala JE, et al. Areport: chronic fatigue syndrome: guide-lines for research. J R Soc Med. 1991;84(2):118121

    14. Hira Sing RA, Rodrigues PR. [The Dutch Pe-diatric Surveillance System; a quality fo-cused instrument for prevention and re-search]. Ned Tijdschr Geneeskd. 2002;146(50):24092414

    15. Vercoulen JH, Swanink CM, Fennis JF,Galama JM, van der Meer JW, Bleijenberg G.Dimensional assessment of chronic fatiguesyndrome. J Psychosom Res. 1994;38(5):383392

    16. Stulemeijer M, de Jong LW, Fiselier TJ,Hoogveld SW, Bleijenberg G. Cognitive be-haviour therapy for adolescents withchronic fatigue syndrome: randomised con-trolled trial. BMJ. 2005;330(7481):14

    17. ter Wolbeek M, van Doornen LJ, Kavelaars A,Heijnen CJ. Severe fatigue in adolescents: acommon phenomenon? Pediatrics. 2006;117(6). Available at: www.pediatrics.org/cgi/content/full/117/6/e1078

    18. Dittner AJ, Wessely SC, Brown RG. The as-sessment of fatigue: a practical guide forclinicians and researchers. J PsychosomRes. 2004;56(2):157170

    19. Raat H, Landgraf JM, Bonsel GJ, Gemke RJ,Essink-Bot ML. Reliability and validity of thechild health questionnaire-child form (CHQ-CF87) in a Dutch adolescent population.Qual Life Res. 2002;11(6):575581

    20. Statistics Netherlands, Centraal Bureauvoor de Statistiek. National Dutch datacol-lection of 2008. Available at: http://cbs.nl.Accessed March 18, 2011

    21. National Institute for Health and Clinical Excel-lence. Chronic Fatigue Syndrome/Myalgic En-cephalomyelitis (or Encephalopathy): Diagnosisand Management of CFS/ME in Adults and Chil-dren. London, UK: National Institute for Healthand Clinical Excellence; 2007. Available at: www.nice.org.uk/nicemedia/pdf/CG53NICEGuideline.pdf. AccessedMarch 18, 2011

    22. van de Putte EM, Engelbert RH, Kuis W, Sin-nema G, Kimpen JL, Uiterwaal CS. Chronicfatigue syndrome and health control in ad-olescents and parents. Arch Dis Child. 2005;90(10):10201024

    23. Patel MX, Smith DG, Chalder T, Wessely S.Chronic fatigue syndrome in children: across sectional survey. Arch Dis Child. 2003;88(10):894898

    24. Rangel L, Garralda ME, Levin M, Roberts H.The course of severe chronic fatigue syn-drome in childhood. J R Soc Med. 2000;93(3):129134

    25. Bazelmans E, Vercoulen JH, Galama JM, vanWC, van der Meer JW, Bleijenberg G. [Preva-lence of chronic fatigue syndrome and pri-mary fibromyalgia syndrome in The Nether-lands]. Ned Tijdschr Geneeskd. 1997;141(31):15201523

    26. Bowen J, Pheby D, Charlett A, McNulty C.Chronic fatigue syndrome: a survey of GPsattitudes and knowledge. Fam Pract. 2005;22(4):389393

    27. Fitzgibbon EJ, Murphy D, OShea K, KelleherC. Chronic debilitating fatigue in Irish gen-eral practice: a survey of general practitio-ners experience. Br J Gen Pract. 1997;47(423):618622

    28. Vant Leven M, Zielhuis GA, van der Meer JW,Verbeek AL, Bleijenberg G. Fatigue andchronic fatigue syndrome-like complaintsin the general population. Eur J PublicHealth. 2009

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  • DOI: 10.1542/peds.2010-1147 originally published online April 18, 2011; 2011;127;e1169Pediatrics

    L. Kimpen and Elise M. van de PutteSanne L. Nijhof, Kimberley Maijer, Gijs Bleijenberg, Cuno S. P. M. Uiterwaal, Jan L.

    Adolescent Chronic Fatigue Syndrome: Prevalence, Incidence, and Morbidity

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  • DOI: 10.1542/peds.2010-1147 originally published online April 18, 2011; 2011;127;e1169Pediatrics

    L. Kimpen and Elise M. van de PutteSanne L. Nijhof, Kimberley Maijer, Gijs Bleijenberg, Cuno S. P. M. Uiterwaal, Jan L.

    Adolescent Chronic Fatigue Syndrome: Prevalence, Incidence, and Morbidity

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    . ISSN:60007. Copyright 2011 by the American Academy of Pediatrics. All rights reserved. Print

    American Academy of Pediatrics, 141 Northwest Point Boulevard, Elk Grove Village, Illinois,has been published continuously since . Pediatrics is owned, published, and trademarked by the Pediatrics is the official journal of the American Academy of Pediatrics. A monthly publication, it

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    Adolescent Chronic Fatigue Syndrome: Prevalence, Incidence, and MorbidityMETHODSPrevalence Estimations Obtained From GPsIncidence Estimations Obtained From PediatriciansEthicsAnalysis

    RESULTSPrevalence EstimatesIncidence EstimatesDemographics and MorbidityAttitudes to Diagnosis and Disease Management

    DISCUSSIONStrengths and WeaknessesComparison With Other StudiesImplications of the Findings

    CONCLUSIONSACKNOWLEDGMENTSREFERENCES