Kashin-Beck disease. An analysis of research and public health activities based on a bibliography 1849-1992.
The main feature of Kashin-Beck disease (KBD) is short stature caused by multiple focal necroses in the growth plate of the tubular bones. This leads to a secondary, sometimes severe osteoarthrosis. The disease was first described in Russia from the Bajkal area by Kashin 1848 and later, 1906, by Eugene Beck. Modern literature is mainly from China. The main theory originally proposed by Russian investigators was that KBD was caused by a toxic effect of mycotoxin. The focus on the disease gradually shifted to China where the causal theory has been based on the effects of selenium deficiency and its interaction with mycotoxins. These main hypotheses may be unified in one theory. KBD is appreciated as a public health problem estimated to affect some 1-3 million people across China. National and provincial registers on KBD exist for selected affected areas. Current incidence is probably declining. Preventive efforts have been undertaken but are usually not controlled for effectiveness nor by putative causal mechanisms. More research into the causal mechanisms is required. A combination of difficulties to obtain literature, including primary material together, and cultural and language barriers have been a main obstacle to more active participation by the international research community. This bibliography is an attempt to help improve this situation. The bibliography covers the period 1849-1992. The analysis of this bibliography shows a broad and intensive, though sometimes insufficiently coordinated research. Many authorities and research bodies are involved. The possibility to develop coordinated projects within a common framework, and thus to use research results to prevent KBD, has been a major source of inspiration in making this review. The review covers 499 publications directly related to KBD and 1415 references to these publications. KBD publications have been retrieved from conventional international journals, usually in English, publications in Russian, as well as unpublished reports in Chinese provided at site visits. The papers are classified by key words. The references in these publications have also been similarly classified and are in part analysed. Based on publications identified from several sources, direction of research and public health efforts from 1848 to 1992 are described. Finally, mainly based on questions raised by and recommendations from a WHO workgroup meeting in 1992, some major components of research activities and public health program are proposed.