Follow-Up Survey of Lyndonville Children
Most report At Least Partial Recovery From CFS Symptoms
By Mark Giuliucci

Eighty percent of children stricken with chronic fatigue syndrome (CFS) during a cluster outbreak in the 1980s report making at least a partial subsequent recovery, according to a follow-up study published in the journal Pediatrics.

The findings were based on questionnaires returned by 35 of the 46 children and adolescents who developed CFS-like symptoms in Lyndonville, N.Y. from 1984-87. Of the 35 respondents (24 female, 11 male), 13 (37.1%) report they “recovered completely and feel entirely well.” Fifteen (42.9%) said they “have never recovered completely but feel pretty well.” Four participants (11.4%) said they “recovered somewhat but remain ill,” and three (8.6 %) say they are “more ill” than they were 10 years ago.

All 35 respondents retrospectively met the 1994 international research case definition for CFS at the onset of their illnesses. Each respondent developed unexplained, activity-limiting fatigue for at least six months and at least four of the eight case criteria symptoms (see sidebar). None reported a subsequent diagnosis that would explain their symptoms.

The children were part of a group of 214 Lyndonville residents who developed CFS-like symptoms in the 1980s. No cause has been found for the cluster of CFS cases.

The survey questionnaire included the following items:

• Medical Outcomes Study Short Form Health Survey (SF-36);
• Subsequent medical and emotional diagnoses;
• Visual analog scores (VAS) indicating present severity of 12 CFS-related symptoms;
• Subjective effect of the illness on social and educational life.

Of the 13 respondents who reported complete recovery, none said that their symptoms disappeared within one year of onset. Two “recovered” within one or two years; one “recovered” within two or three years; three “recovered” between three and four years after onset; and three “recovered” after four years. The final respondent reported that symptoms disappeared after 9.5 years.
 
Bell says that the 80 percent recovery figure is somewhat misleading. More than half of those who reported improvement still suffer from lingering CFS symptoms. Many of
them have been forced to adopt coping mechanisms that include restricted activity, flexible daily schedules and a lack of sustained activities.

VAS results generally correlated with self-reported recovery rates. The scores used for analysis were the sums of 12 symptom-specific categories that were self-graded between 0 (no symptom) to 10 (severe symptom). Those who reported complete recovery average a VAS of 15.4, while those who said they had grown more ill reported an average VAS of 74.7.

The SF-36 results showed similar correlation. Subjects who reported better recovery scored higher than others in all eight test domains.

The respondents’ perceived recovery also matched their reports of how the illness affected their social lives. Twelve of the 35 (34.3%) said the illness did not have an overall effect on them socially; 10 of those 12 had reported complete recovery. Sixteen respondents (45.7%) said the disease had a mild impact on their social lives, three (8.6%) claimed a moderate effect and four (11.4%) said that CFS severely impacted them socially.

Notes for clinicians
Guidelines for diagnosing CFS do not differentiate between pediatric and adult cases, but Bell says his experience indicates that two symptoms are often more prominent among children. The first is activity limitation. Children with CFS are more likely to report being forced to curtail daily activities than their adult counterparts. Children also appear more likely to suffer from the effects of orthostatic intolerance (OI). Bell says it’s not clear whether children with CFS develop OI more frequently than adult patients. They may simply be more sensitive to the symptoms of OI than adults.

Bell also believes that physicians should not shy away from diagnosing CFS for fear of encouraging illness behavior. Bell and his co-authors write that this would create "severe difficulties in clinical management." Diagnosing CFS can actually help reduce patient and parental anxiety, can reduce the number of unnecessary laboratory tests and evaluations and can help with attempts at symptom control.

The study also addresses similar concerns about "sympathetic" physicians encouraging children to continue to complain about symptoms. Bell was the primary physician for all 35 respondents during the early years of their illnesses, but continued to care for only 10 of them after the initial treatment period. The other 25 either saw another doctor or "recovered" and did not need additional care. Responses from the questionnaire show no differences between group of 10 and the group of 25. Bell says this indicates the attitude of the treating physician about CFS has little to do with the eventual outcome of the disease.

David S. Bell, MD, FAAP; Karen Jordan, PhD; and Mary Robinson, MS. "Thirteen-Year Follow-Up of Children and Adolescents With Chronic Fatigue Syndrome." Pediatrics. 2001; 107(5): 994-998.

(n=35)

Unexplained fatigue
35 (100%)

Recurrent sore throat
32 (91.4%)

Myalgia
32 (91.4 %)

Headache
30 (85.7%)

Lymph node tenderness
29 (82.9%)

Cognitive difficulties
27 (77.1%)

Multijoint pain
26 (74.3%)

Note: Eight children in the follow-up study were ages 5-9 at onset; 16 were ages 10-14; and 11 were ages 15-18.

Source: David S. Bell, MD, FAAP; Karen Jordan, PhD; and Mary Robinson, MS. "Thirteen-Year Follow-Up of Children and Adolescents With Chronic Fatigue Syndrome." Pediatrics. 2001; 107(5): 994-998.

Predicting which children will recover fully from CFS remains impossible, says David S. Bell, MD, FAAP, co-author of a follow-up study of pediatric CFS patients who developed the disease during a localized outbreak in the mid-1980s.

"We’re certainly not that far down the road," says Bell. "But we have at least started to look at the question of prognosis, and we have some early data."

Bell says that the clearest predictor from his study is the amount of time missed from school. High absentee rates during the early years of illness correlated with poorest outcomes. Bell was the attending physician for all 35 Lyndonville, N.Y. children who responded to his follow-up survey, and he says they were all encouraged to attend school as much as possible.

A predominance of neurological symptoms, notably encephalitis, during the first year of illness also predicted poor outcome. Children whose symptoms failed to improve by the third year of illness were less likely to report full recovery at follow-up.
Bell says it's not clear if these trends will hold for all pediatric CFS cases, or whether they will be limited to cluster outbreaks similar to the one in Lyndonville.