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The case history of an elite ultra-endurance cyclist who developed chronic fatigue syndrome. Med. Sci. Sports Exerc., Vol. 30, No. 9, pp. 1345-1348, 1998. An elite ultra-endurance athlete, who had previously undergone physiological and performance testing, developed chronic fatigue syndrome (CFS). An incremental cycling exercise test conducted while he was suffering from CFS indicated decreases in maximum workload achieved (Wmax;−11.3%), the maximum oxygen uptake (V˙O2max; −12.5%), and the anaerobic threshold (AT; −14.3%) compared to pre-CFS data. A third test conducted after the athlete had shown indications of significant improvement in his clinical condition revealed further decreases in Wmax (−7.9%), V˙O2max(−10.2%) and AT (−8.3%). These data, along with submaximal exercise data and muscle biopsy electron microscopic analyses, suggest that the performance decrements were the result of detraining, rather than an impairment of aerobic metabolism due to CFS per se. These data may be indicative of central, possibly neurological, factors influencing fatigue perception in CFS sufferers.