Publication bias in Kienböck's disease: systematic review

Abstract

Purpose: Kienböck's disease is considered rare and currently affects fewer than 200,000 people in the United States. Given the inherent challenges associated with researching rare diseases, the intense effort in hand surgery to treat this uncommon disorder may be influenced by publication bias in which positive outcomes are preferentially published. The specific aim of this project was to conduct a systematic review of the literature with the hypothesis that publication bias is present for the treatment of Kienböck's disease.

Methods: We conducted a systematic review of all available abstracts associated with published manuscripts (English and non-English) and abstracts accepted to the 1992 to 2004 American Society for Surgery of the Hand (ASSH) annual meetings. Data collection included various study characteristics, direction of outcome (positive, neutral/negative), complication rates, mean follow-up time, time to publication, and length of patient enrollment.

Results: Our study included 175 (124 English, 51 non-English) published manuscripts and 14 abstracts from the 1992 to 2004 annual ASSH meetings. Abstracts from published manuscripts were associated with a 53% positive outcome rate, which is lower than the 74% positive outcome rate found among other surgically treated disorders. Over the past 40 years, studies have become more positive (36% to 68%, p=.007) and are more likely to incorporate statistical analysis testing (0% to 55%, p<.001). Of the 14 abstracts accepted to ASSH, 11 were published in peer-reviewed journals. Ten of the 14 accepted abstracts were considered positive, and there was no significant difference in publication rate between studies with positive (n = 10) and negative (n = 4) outcomes (p>.999).

Conclusions: The acceptance rate for negative outcomes studies regarding Kienböck's disease is higher than for other surgical disorders. This may indicate a relative decrease in positive outcome bias among published Kienböck's disease studies compared with other surgical disorders. However, the increasing positive outcome rate for published Kienböck's disease studies over time may suggest a trend of increasing publication bias among journals toward Kienböck's disease studies.

Figure 1. Flowchart of primary database search and data collection

Figure 2. Trends in Surgical Kienbock's Research Over Time