A systematic review of the etiopathogenesis of Kienböck's disease and a critical appraisal of its recognition as an occupational disease related to hand-arm vibration

Abstract

Background: We systematically reviewed etiological factors of Kienböck's disease (osteonecrosis of the lunate) discussed in the literature in order to examine the justification for including Kienböck's disease (KD) in the European Listing of Occupational Diseases.

Methods: We searched the Ovid/Medline and the Cochrane Library for articles discussing the etiology of osteonecrosis of the lunate published since the first description of KD in 1910 and up until July 2012 in English, French or German. Literature was classified by the level of evidence presented, the etiopathological hypothesis discussed, and the author's conclusion about the role of the etiopathological hypothesis. The causal relationship between KD and hand-arm vibration was elucidated by the Bradford Hill criteria.

Results: A total of 220 references was found. Of the included 152 articles, 140 (92%) reached the evidence level IV (case series). The four most frequently discussed factors were negative ulnar variance (n=72; 47%), primary arterial ischemia of the lunate (n=63; 41%), trauma (n=63; 41%) and hand-arm vibration (n=53; 35%). The quality of the cohort studies on hand-arm vibration did not permit a meta-analysis to evaluate the strength of an association to KD. Evidence for the lack of consistency, plausibility and coherence of the 4 most frequently discussed etiopathologies was found. No evidence was found to support any of the nine Bradford Hill criteria for a causal relationship between KD and hand-arm vibration.

Conclusions: A systematic review of 220 articles on the etiopathology of KD and the application of the Bradford Hill criteria does not provide sufficient scientific evidence to confirm or refute a causal relationship between KD and hand-arm vibration. This currently suggests that, KD does not comply with the criteria of the International Labour Organization determining occupational diseases. However, research with a higher level of evidence is required to further determine if hand-arm vibration is a risk factor for KD.

Figure 1

Flow diagram demonstrating the individual steps in the study-selection process of literature on the causal factors of KD. The full text of 215 articles was studied and the literature was subdivided according to the discussed etiopathological factors.

Figure 2

Illustration of the frequency of discussed etiopathological factors in literature in 153 reviewed articles. The four most frequently discussed hypotheses are marked in blue (negative ulnar variance), red (primary arterial ischemia), green (trauma) and purple (hand-arm vibration exposure); all other hypotheses are marked in yellow.

Figure 3

Percent distribution of the level of evidence of articles acknowledging and refuting the four most frequently discussed and other hypotheses on the etiopathology of KD.

Figure 4

Percentages of the discussed etiopathological hypotheses in literature in relation to all the hypotheses discussed every 20 years from 1910 until July 2012.