Some Variants of the Backcalculation Method for Estimation of Disease Incidence: An Application to Multiple Sclerosis Data from the Faroe Islands

Abstract

Backcalculation is a technique that was originally developed for the study of HIV incidence. Here we introduce some variants of the estimation technique that allow for (i) correlation of the unobserved disease incidence counts, and (ii) the use of a smoothing step as part of the maximizing step in the EM algorithm to reduce instability due to small diagnosis counts. Both of these issues can be important in the analysis of small "epidemics." In addition, identification of correlation between diagnosis counts provides indirect evidence of correlation among unobserved incidence counts, hinting at the possibility of an infectious agent. We illustrate the ideas by reconstructing an incidence intensity function for the onset of multiple sclerosis, using data from the Faroe Islands. Previously, this data had been examined statistically, by Joseph, Wolfson & Wolfson (1990), to address the issue of infectiousness of multiple sclerosis. We argue that the incidence function cannot directly shed light on the enigmatic origin of multiple sclerosis in the Faroe Islands during World War II, and, in particular, cannot discriminate between hypotheses of an infectious or environmental agent.