The natural history of eosinophilia-myalgia syndrome in a tryptophan-exposed cohort in South Carolina.

BACKGROUND: In a previous study, we did follow-up on 418 patients who were exposed to tryptophan in 1989, of whom 47 (11%) had definite and 63 (9%) possible eosinophilia-myalgia syndrome (EMS). METHODS: We assessed mortality and clinical spectrum of illness since 1989 for 242 (58%) of the 418 tryptophan-exposed patients from the original study. To assess outcomes, we used hospital and death records, interviewer-administered questionnaires, physical examinations, and laboratory tests. RESULTS: During the follow-up interval, mortality from all causes was 19% in those with definite EMS, 7% in possible EMS, and 3% in those who were not ill. The age- and sex-adjusted mortality in those with definite EMS was more than 3 times that of the general population or of tryptophan users in the practice who were not ill. Six deaths (66%) among the definite EMS case patients occurred during the 18 months immediately after symptom onset. Compared with the tryptophan users who were not ill, survivors with definite EMS continued to report excess morbidity for 6 major EMS symptoms (myalgia, arthralgia, weakness, rash, alopecia, and sclerodermiform skin changes), but they also reported that the symptom number and severity diminished with time. None of the tryptophan users who were not ill in 1989 developed a symptom complex suggesting new EMS during the follow-up interval. CONCLUSIONS: This study assessing a tryptophan-exposed population found those persons who developed EMS during the 1989 epidemic were at increased risk for death, particularly early after disease onset. Survivors reported improvement or resolution of major symptoms, suggesting that the severity of EMS diminishes with time. We found no evidence of delayed onset of EMS in tryptophan users who were not ill in 1989, regardless of the brand used.

Toxic oil syndrome: survival in the whole cohort between 1981 and 1995.

BACKGROUND AND OBJECTIVE: In 1981, toxic oil syndrome (TOS) appeared in Spain, affecting more than 20,000 persons and causing over 2500 deaths to date. Previous studies have addressed mortality only by gender and age. We analyzed possible prognostic factors in the survival of the cohort. METHODS: The study period was 1 May 1981 to 31 December 1995 (31 December 1995 was the cut-off date for survivors). The study population consisted of the entire cohort. Overall mortality and TOS-related deaths were studied. Kaplan-Meier method and Cox regression were used in the analyses. RESULTS: Among the 20,084 subjects in the cohort, 12,164 (60.6%) were women, and 7917 (39.4%) were men. Of the 1799 deaths, 958 (53.3%) were women, and 841 (46.71%) were men; of the 356 TOS-related deaths, 234 (65.7%) were women, and 122 (34.3%) were men. TOS was the leading cause of death among subjects <40 years of age. Among the TOS-related deaths, the shortest survival times were for women and subjects <40 years of age. The major disease manifestations had the highest relative risks (RR) (liver disease, RR 3.83; pulmonary infection, RR 1.54; motor neuropathy, RR 2.24; pulmonary hypertension, RR 3.19; and eosinophilia, RR 1.14.). CONCLUSIONS: The major clinical manifestations showed worse prognosis for overall and TOS-related mortality. Application of these results to the survivors will help clarify the validity of these conclusions.

Eosinophilia-myalgia syndrome: mortality data from the US national surveillance system.

OBJECTIVE: To describe some of the most severe features of eosinophilia-myalgia syndrome (EMS) and identify potential prognostic indicators. METHODS: Systematic review of data from initial case reports and from followup supplemental death report forms forwarded to the national surveillance system administered by the US Centers for Disease Control (CDC). RESULTS: As of August 10, 1991 36 deaths related to EMS had been reported to CDC. Among all patients fitting the surveillance case definition for EMS, we found that patients who died were older, had higher absolute leukocyte and eosinophil counts, and reported a greater frequency of cough or dyspnea, neuropathy, hepatomegaly, leukocytosis, and elevated erythrocyte sedimentation rate. All patients who died had illnesses affecting multiple organ systems. Of the 36 patients who died, 33 (92%) had neuromuscular sequelae, 29 (81%) had pulmonary complications, and 23 (64%) had cardiac manifestations. The most commonly observed disease process leading to death was progressive polyneuropathy and myopathy (24 of the 36 reported deaths) which produced complications of pneumonia and sepsis or respiratory failure due to weakness; cardiomyopathy was the underlying cause of death for 4 patients, primary pulmonary disease for 3, sudden death attributed to arrhythmia for 2, stroke for 2, and septic complications of therapy for one. CONCLUSION: Although neuromuscular complications were the most prominent sequelae among patients reported to have died, this is clearly a multisystemic disease. Older age and involvement of more than one organ system suggest a particularly poor prognosis, and the neuromuscular, pulmonary and cardiovascular sequelae appear to be the most worrisome.