Skip to main content
NIHPA Author Manuscripts logoLink to NIHPA Author Manuscripts
. Author manuscript; available in PMC: 2015 Apr 6.
Published in final edited form as: J Hand Surg Am. 2010 Mar;35(3):359–367.e5. doi: 10.1016/j.jhsa.2009.12.003

Publication Bias in Kienbock's Disease: A Systematic Review

Lee Squitieri 1, Elizabeth Petruska 2, Kevin C Chung 3
PMCID: PMC4386875  NIHMSID: NIHMS676122  PMID: 20193856

Abstract

Purpose

Kienbock's disease is considered a “rare disease” and currently affects less than 200,000 people in the U.S. Given the inherent challenges associated with researching rare diseases, the intense effort in hand surgery to treat this uncommon disorder may be susceptible to publication bias in which positive outcomes are preferentially published. The specific aim of this project is to conduct a systematic review of the literature with the hypothesis that publication bias is present for the treatment of Kienbock'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 1992-2004 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 Kienbock's manuscripts and 14 Kienbock's abstracts from the 1992-2004 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 surgical disorders. Over the past 40 years, studies have become more positive (36% to 68%, p = 0.007) and are more likely to incorporate statistical analysis testing (0% to 55%, p <0.001). Of the 14 abstracts accepted to ASSH, 11 (79%) were published in peer-reviewed journals. Ten of the 14 accepted abstracts (71%) were considered positive, and there was no significant difference in publication rate between studies with positive (n = 10) and non-positive (n = 4) outcomes (p = 1.000).

Conclusions

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

Keywords: Kienbock's disease, systematic review, publication bias


Kienbock's disease, or avascular necrosis of the lunate, is classified as a “rare disease” by the Office of Rare Diseases (ORD) of the National Institutes of Health (NIH), and therefore affects less than 200,000 people in the U.S.1 Since the initial description of this condition by Robert Kienbock in 1910, surgeons have described over 20 different procedures aimed at treating this disease.2-4 The low prevalence and unclear etiology of Kienbock's disease present unique challenges with regards to conducting large, clinically meaningful comparative effectiveness trials. As a result, there is little evidence-based data to support any particular method of operative treatment, or to indicate their superiority over conservative measures.3 Thus, hand surgeons may be inadvertently driven to treat Kienbock's patients with procedures that have the most recent or prevalent data presented in the literature, rather than based on objective comparative outcomes.

Publication bias refers to the tendency of researchers, peer reviewers, and journal editors to submit or accept manuscripts for publication based on the direction or strength of study findings.5-6 This includes: 1) factors that influence the undertaking/performance of research (prepublication bias); 2) acceptance or rejection of an abstract/manuscript (publication bias), and; 3) acceptance or interpretation of previously published research as evidenced by citations in future research and inclusion in systematic reviews/meta-analyses (postpublication bias).6-8 The most serious potential consequence of publication bias at any stage would be overestimation of treatment effects or risk-factor associations in published papers that could lead to inappropriate decisions concerning patient management or health policy.9-11 Previous studies have demonstrated an association between positive outcomes, the presence of statistical significance, more advanced study design, study size, and increased likelihood of ultimate publication.9-17 Due to the limited number of individuals affected with Kienbock's disease and the inherent difficulties associated with conducting research for rare disorders, the intense effort in hand surgery to treat this uncommon condition may be susceptible to publication bias.

Although several studies have assessed publication bias in various surgical specialties, there is a lack of specific data evaluating publication bias within the field of hand surgery.9,13-16 The purpose of this study is to conduct a rigorous evaluation of all available literature regarding the surgical treatment of Kienbock's disease and identify potential areas of publication bias. Using data from published abstracts in both the English and non-English language, we will present a systematic review assessing study characteristics, quality of reporting, and direction of outcomes (positive or neutral/negative) for all published Kienbock's studies. In addition to our evaluation of published abstracts, we will collect the same data from abstracts presented at the 48th-60th annual scientific meeting of the American Society for Surgery of the Hand (ASSH; 1992-2004). By comparing the study characteristics and outcomes of unpublished abstracts to abstracts accepted for publication, we aim to determine factors that were positively or negatively associated with achieving ultimate publication.

Materials and Methods

Literature Search and Abstract Selection

Systematic searches of all available literature from the MEDLINE, EMBASE, and SCPOUS databases (1902-2009) were performed using the key words Kienbock, avascular necrosis, revascularization, and lunate. The resulting titles and abstracts were then screened to rule out non-primary, biomechanical, non-surgical, non-human, non-Kienbocks, anatomical, or diagnostic imaging studies. Studies evaluating pediatric patients or patients with concomitant joint, collagen, or vascular disease were also excluded. Relevant abstracts for analysis consisted of primary studies assessing outcomes for specific procedures used to treat Kienbock's disease. Abstracts containing multiple indications were included as long as the results for Kienbock's patients alone were able to be extracted. Due to the paucity of published research regarding Kienbock's disease, we included both English and non-English studies as long as the non-English studies contained a published English abstract.

In addition to our assessment of abstracts in published manuscripts, we also evaluated the same study characteristics and outcome parameters for abstracts accepted to the 48th to 60th annual scientific meeting of the ASSH (1992-2004). Abstracts prior to 1992 and abstracts from other hand conferences were not available for review. Publications corresponding to the accepted abstracts were identified through a computer search of the MEDLINE database using authors' names and key words from the title. We choose to review abstracts presented until the year 2004 based on previous research which demonstrated that over 90% of abstracts that will eventually lead to publication will publish within 5 years of abstract presentation.12

Data Extraction and Analysis

For each published manuscript and ASSH abstract included in our analysis, data regarding study characteristics (primary language, country of journal origin, study size), outcome direction (positive, negative, or neutral), and complications were collected. In addition, the following time-to-event information was collected for studies with available data: 1) length of patient enrollment, 2) mean follow-up time, and 3) time from completion of patient enrollment to publication. The presence of statistical analysis, designated by the presence of a p value (regardless of the value), was used as a crude indicator of study quality.

All manuscripts and abstracts were analyzed by two reviewers (L.S. and E.P.) with discrepancies resolved between each other by consensus. The results of each study were classified as positive, neutral, or negative. Positive outcomes were defined as present if the authors directly recommended a procedure or stated that the intervention was generally beneficial, irrespective of absolute outcome values or the presence of statistical significance; negative outcomes were defined as present when the authors advised against the intervention or presented only negative results; and neutral outcomes were defined by the absence of author recommendations or the presence of both positive and negative comments/results.13-16 For studies comparing more than one intervention for Kienbock's disease, outcomes were considered positive if the authors specifically recommended at least one of the procedures included in the study.

Statistical Analysis

Various categorical (primary language of study, country of study origin, procedure type, outcome, and presence of statistical analysis) and interval (number of Kienbock's wrists per study, complication rate, follow-up time, time to publication, and length of patient enrollment) data parameters were collected for each abstract. The statistical significance of relationships between categorical variables was evaluated using either chi-square tests (if all expected values were > 5) or Fischer's exact tests (if any expected value was ≤ 5). For comparison of mean interval data parameters, either a two-tailed t test (two groups) or analysis of variance (more than two groups) was performed. P values of less than 0.05 were considered statistically significant and all statistical analyses were performed using SAS statistical software (version 9.1; SAS, Cary, North Carolina).

Results

Published Kienbock's Abstracts

One thousand five hundred eighty seven citations were identified through the MEDLINE, EMBASE, and SCPOUS databases (Figure 1). Of the 253 relevant titles that underwent abstract review, 175 studies (124 English and 51 non-English) met all of our inclusion/exclusion criteria and formed the basis for our systematic review (Appendix A). Fourty-five articles were eliminated due to inextractable data for Kienbock's patients and thirty-three articles were eliminated because they lacked an associated English abstract. Although both English and non-English articles were primarily composed of studies from Europe (42% and 86%, respectively), English articles contained a greater proportion of studies from non-European countries (p < 0.001, Table 1). English articles were also more likely to include some form of statistical analysis (40% vs. 3%, p < 0.001, Table 1).

Figure 1. Flowchart of primary database search and data collection.

Figure 1

Table 1. Study Characteristics of Published Kienbock's Abstracts.

Total Abstracts (n = 175) English Article (n = 124) Non-English Article (n = 51) P Value
Country of Journal Origin <0.001δ
 US 63 63 (51%) 0 (0%)
 Europe 96 52 (42%) 44 (86%)
 Asia 12 8 (6%) 4 (8%)‡
 Other 4 1 (1%) 3 (6%)
Procedure Type* 0.257ζ
 Joint Leveling 51 32 (25%) 19 (33%)
 Implant/Transposition Arthroplasty 36 26 (20%) 10 (17%)
 Partial Wrist Fusion 25 17 (13%) 8 (14%)
 Vascular Bone Graft/Pedicle 18 10 (8%) 8 (14%)
 Salvage 22 18 (14%) 4 (7%)
 Other 36 27 (21%) 9 (16%)
Overall Outcomes 0.053ζ
 Positive 92 71 (57%) 21 (41%)
 Neutral/Negative 83 53 (43%) 30 (59%)
Mean Number of Wrists per Studyα 19.8 18.8 22.3 0.415ε
Complication Rateβ 17.7% 17.0% 22.7% 0.435ε
Mean Follow-Up (months) γ 71 68 83 0.276ε
Presence of Statistical Analysis 52 50 (40%) 2 (4%) <0.001ζ
Time to Publication (years)π 5.1 5.4 3.5 0.103ε
Length of Patient Enrollment (years) Ω 10.0 10.4 7.3 0.117ε

Data presented as number of English abstracts (% of total English abstracts)

Data presented as number of non-English abstract (% of total non-English abstracts)

*

For studies including more than one surgical procedure, separate entries were made for each procedure type

α

Only includes wrists corresponding to surgical Kienbock's patients

β

Data presented as the average complication rate for studies with available data; Reported in 78 of 124 English articles (63%) and 11 of 51 non-English articles (22%)

γ

Data reported in 110 of 124 English articles (89%) and 30 of 51 non-English articles (59%)

π

Data presented as time from end of patient enrollment to publication; Reported in 75 of 124 English articles (60%) and 12 of 51 non-English articles (24%)

Ω

Data reported in 75 of 124 English articles (60%) and 12 of 51non-English articles (24%)

δ

Statistical analysis using Fisher's exact test

ε

Statistical analysis using two-tailed t-test

ζ

Statistical analysis using chi-square test

Overall, our data show that 53% of Kienbock's abstracts were associated with positive outcomes, which is much lower than the 74% positive outcome rate found among top orthopaedic and general surgery journals (Table 2).13 Positive outcomes were associated with lower complication rates (p = 0.006, Table 3) and increased presence of statistical analysis (p = 0.001, Table 3). We found no correlation between positive outcomes and study of journal origin (p = 0.071), primary language of article (p = 0.053), study size (p = 0.987), and mean follow-up time (p = 0.481, Table 3). Of note, we also found no statistically significant association between negative outcomes and increased time to publication (p = 0.747) or length of patient enrollment (p = 0.264), which has been suggested in previous research regarding publication bias.21

Table 2. Factors Associated with Positive Outcomes.

Total Abstracts (n = 175) Positive Outcomes (n = 92) Non-Positive Outcomes (n = 83) P value
Country of Journal Origin 0.071ε
 US 63 40 (43%) 23 (28%)
 Europe 96 45 (49%) 51 (61%)
 Asia 12 4 (4%) 8 (10%)
 Other 4 3 (3%) 1 (1%)
Language of Article 0.053ε
 English Article 124 71 (77%) 53 (64%)
 Non-English Article with English Abstract 51 21 (23%) 30 (36%)
Mean Number of Wrists per Study α 19.8 19.8 19.9 0.987δ
Complication Rate β 17.7% 12.4% 25.8% 0.006δ
Mean Follow up Time γ 71.2 67.7 75.5 0.481δ
Presence of Statistical Analysis 52 37 (40%) 15 (18%) 0.001ε
Time to Publication (years) π 5.1 5.2 5.0 0.747δ
Length of Patient Enrollment (years)Ω 10.0 10.8 9.0 0.264δ

Data presented as number of positive abstracts (% of total positive abstracts)

Data presented as number of negative abstract (% of total negative abstracts)

α

Only includes wrists corresponding to surgical Kienbock's patients

β

Data presented as the average complication rate for studies with available data; Reported in 54 of 92 studies with positive outcomes (59%) and 35 of 83 studies with non-positive outcomes (42%)

γ

Data reported in 78 of 92 studies with positive outcomes (85%) and 62 of 83 studies with non-positive outcomes (75%)

π

Data presented as time from end of patient enrollment to publication; Reported in 47 of 92 studies with positive outcomes (51%) and 40 of 83 studies with non-positive outcomes (48%)

Ω

Data reported in 47 of 92 studies with positive outcomes (51%) and 40 of 83 studies with non-positive outcomes (48%)

δ

Statistical analysis using two tailed t-test

ε

Statistical analysis using chi-squared test

Table 3. Outcomes, Complications, and Quality of Reporting According to Country of Journal Origin.

US (n = 63) Europe (n = 96) Asia (n = 12) Other (n = 4) P value
Overall Outcomes 0.071δ
 Positive 40 (63%) 45 (47%) 4 (33%) 3 (75%)
 Neutral/Negative 23 (37%) 51 (53%) 8 (67%) 1 (25%)
Mean Number of Wrists per Study 20.7 19.4 20.4 14.8 0.939ε
Complication Rate α 12.3% 24.3% 7.6% 0.1% 0.067ε
Mean Follow-Up β 73 72 55 66 0.894ε
Statistical Analysis Present 29 (46%) 20 (21%) 3 (25%) 0 (0%) 0.004δ
Time to Publication (years) γ 5.8 4.5 5.4 NA 0.267ε
Length of Patient Enrollment (years) π 11.6 8.1 12.3 NA 0.093ε

Data reported as number of abstracts (% of total abstracts for given geographic region)

Only includes wrists corresponding to Kienbock's patients

α

Data presented as the average complication rate for studies with available data; Reported in 42 of 63 US studies (67%), 42 of 96 European studies (44%), 3 of 12 Asian studies (25%), and 2 of 4 studies published in another country (50%)

β

Data reported in 59 of 63 US studies (94%), 68 of 96 European studies (71%), 9 of 12 Asian studies (75%), and 4 of 4 studies published in another country (100%)

γ

Data presented as time from end of patient enrollment to publication; Reported in 38 of 63 US studies (60%), 42 of 96 European studies (44%), 7 of 12 Asian studies (58%), and 0 of 4 studies published in another country (0%)

π

Data reported in 38 of 63 US studies (60%), 42 of 96 European studies (44%), 7 of 12 Asian studies (58%), and 0 of 4 studies published in another country (0%)

δ

Statistical analysis using Fisher's exact test

ε

Statistical analysis using ANOVA

Figure 2 demonstrates trends in surgical Kienbock's research over the past 40 years. As awareness of Kienbock's disease has grown, the number of primary clinical trials pertaining to Kienbock's disease has increased substantially from 11 studies during 1970-1979 to 71 studies during 2000-2009. Over time, results from published studies have become more positive (36% to 68%, p = 0.007) and are more likely to incorporate statistical analysis testing (0% to 55%, p <0.001, Figure 2). These findings could be the result of improved management of Kienbock's disease and improved study quality over time. However, the increasing positive outcome rate for published Kienbock's studies over time may suggest a trend of increasing publication bias among journals toward Kienbock's studies.

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

Figure 2

Abstracts Accepted to ASSH

Twenty-four abstracts pertaining to Kienbock's disease were identified from the 1992-2004 annual ASSH meetings (Table 4). Ten abstracts were considered inappropriate for review (2 diagnostic/imaging, 2 non-human, 1 non-operative, 5 inextractable data) and thus eliminated from our analysis. Of the 14 abstracts included in our study, 11 (79%) were ultimately published in peer-reviewed journals with an average of 2.2 years to publication. The presence of positive outcomes or statistical analysis had no significant impact on ultimate publication (p = 1.000, p = 1.000, respectively).

Table 4. Study Characteristics of Kienbock's Abstracts Presented at the 1992-2004 ASSH Annual Meeting.

Variables Number of Abstracts Published (%) P value
Country of Study Origin 1.000
 US 6 5 (83%)
 Other 8 6 (75%)
Total Number of Wrists 0.506
 < 20 9 8 (89%)
 > 20 5 3 (60%)
Result 1.000
 Positive 10 8 (80%)
 Neutral/Negative 4 3 (75%)
Statistical Analysis 1.000
 No 7 5 (71%)
 Yes 7 6 (86%)
Complications 0.096
 Not recorded 7 7 (100%)
 ≤ 10% 3 2 (67%)
 > 10% 4 2 (50%)

14 abstracts were included in our analysis; 10 abstracts were eliminated (2 diagnostic/imaging, 2 non-human, 1 non-operative, 5 inextractable data for Kienbock's patients or for specific procedure)

Statistical analysis using Fisher's exact test

When comparing study characteristics of the 14 ASSH meeting abstracts with the manuscripts published during the same time period from our systematic review (n = 128), we found that abstracts presented at ASSH had a slightly higher positive outcome rate (71% vs 59%), a shorter average time to publication (2.2 years vs. 5.4 years), and were more likely to include some form of statistical analysis testing (50% vs 41%) compared to published manuscripts during the same time period. These results may indicate that Kienbock's studies presented at the ASSH have better outcomes and are of better quality than studies ultimately published in the literature which ultimately results in a faster time to publication.

Discussion

Ever since the advent of scientific and medical periodicals in the late 17th century, the publication of medical research has provided a basis for the common understanding of medical diseases and their associated treatment options. However, it was not until the early 1980's that direct evidence of publication bias within the medical literature became widely recognized.5,9,25-31 Over the past three decades, medical researchers have become increasingly conscientious of the potential consequences of publication bias (overestimation of treatment effects and inappropriate risk factor associations) on patient management and health policy. Recent studies have demonstrated a clear bias toward publishing research with positive outcomes, increased sample size, and statistically significant results.5,8,11,14,31 More specifically, evaluation of the surgical literature has found that top surgical journals are at increased risk for publication bias due to the low prevalence of high-level clinical studies and variable complication rates between different surgical centers.13,23-24 This problem is further exacerbated for rare surgical disorders with poorly defined management strategies, such as Kienbock's disease. Due to the low prevalence, unknown etiology, poorly defined treatment algorithm, and lack of outcomes/complication data for Kienbock's disease, hand surgeons are more likely to base management decisions on randomly dispersed case reports/series in the published literature and presentations at national conferences. Thus, the presence of publication bias either in the published literature or among abstracts presented at national meetings has a greater propensity to directly affect patient care.

In this study, we present an analysis of all world literature (English articles and non-English articles with an English abstract) pertaining to the surgical management of Kienbock's disease. Overall, we did not find any evidence of positive outcome bias among published Kienbock's manuscripts. This conclusion is supported by the low percentage of positive outcome studies for Kienbock's disease (53%) compared to other surgical disorders (74%), and the failure to demonstrate a relationship between positive outcomes in abstracts presented at annual ASSH meetings and achieving ultimate publication.13 However, the lower positive outcome rate for Kienbock's studies could be the result of poorer expected outcomes for the surgical management of Kienbock's disease rather than decreased positive outcome bias among published Kienbock's abstracts.

Our results also demonstrated a higher publication rate of Kienbock's studies (79%) compared to the publication rate of other hand disorders (52%) presented at the ASSH annual meetings which could indicate potential selection bias at the national conference level prior to manuscript submission (pre-publication bias).32 This theory is further supported by the slightly higher positive outcome rate (71% vs 59%), shorter average time to publication (2.2 years vs. 5.4 years), and increased likelihood of including some form of statistical analysis testing (50% vs 41%) among ASSH abstracts compared to published manuscripts during the same time period. However, the limited sample size of our analysis (14 ASSH abstracts) makes it impossible to draw any firm conclusions from these results and their interpretations should be reviewed with caution. Our goal of including this analysis in our study was to increase awareness among hand surgeons of potential selection bias among abstracts presented at national conferences and to encourage discretion when using these results to make patient management decisions.

Study Limitations

As with any systematic review, our study was limited by the quality of available literature. This limitation was especially challenging for Kienbock's disease. Forty-five published studies were unable to be incorporated into our analysis because they involved multiple procedures and/or multiple indications without providing specific outcomes data for Kienbock's patients. Of the remaining 175 published studies included in our analysis, most were small non-comparitive case series.

In an attempt to compensate for the lack of published Kienbock's studies and also to reduce publication bias within our own systematic review, we choose to include both English and non-English literature. However, because of language barriers, we were only able to review the English abstracts associated with non-English articles. This hindrance may have considerably reduced our sample size for various data parameters and consequently impaired our ability to determine statistically significant results.

Another limitation unique to studies analyzing positive outcome bias for specific conditions is the fact that various conditions will have different rates of positive outcomes based on inherent differences in management efficacy. For example, one would expect an inherently lower positive outcome rate for studies researching treatment for pancreatic cancer compared to studies researching management for distal radius fractures simply because distal radius fractures are easier to treat successfully. Thus, an apparent publication bias, as measured by positive outcome rate, does not necessarily imply a flaw in the editorial process.

For our study we were unable to determine whether the cause of our observed low positive outcome rate (53%) was the result of poorer expected outcomes among surgically managed Kienbock's patients or decreased publication bias among published Kienbock's studies. Other studies assessing publication bias for other conditions have attempted to correct for this issue by comparing study characteristics between published and non-published manuscripts or abstracts either accepted or not accepted to national conferences.8,12 However, the authors of this study did not have access to non-published literature or ASSH abstract submissions prior to acceptance.

Due to the paucity of available literature, varied outcome measures, and our inability to review the entirety of non-English articles, we were unable to perform a comprehensive comparative analysis of surgical Kienbock's procedures. Although we recognize the need for a formal evidence-based comparative evaluation of Kienbock's procedures, the purpose of our analysis is to provide a systematic, critical review of the existing literature, identify any evidence of publication bias, and highlight research challenges facing rare surgical diseases.

Future Recommendations

Research regarding rare diseases has historically suffered from decreased attention, inadequate funding, and poor patient recruitment.33-36 As a result, clinical studies of rare diseases often lack randomized controlled trials and formal statistical analyses, thus making them more prone to experience publication bias.33-36 This means that physicians treating such rare diseases must form their clinical judgment solely on the basis of (potentially biased) observational studies, experience, and anecdote.33 In 1993 the NIH founded the Office of Rare Diseases Research (ORDR), which coordinates and supports research of rare diseases.1 Recent studies examining publication bias and challenges specific to rare disease research have promoted increased utility of the national clinical trial registry (www.clinicaltrials.gov), which provides information regarding all prospective clinical trials.35-37 However, this design is poorly suited for many surgical disorders due to the predominance of retrospective outcome analysis. Furthermore, our study demonstrates that a considerable portion of surgical Kienbock's research is conducted outside the United States. We recommend the formation of an international patient database for Kienbock's disease that would provide standardized information regarding outcomes, complications, and follow-up for all surgical treatment methods. This would facilitate the development of clear evidence based outcomes regarding surgical management of this rare disease and enable determination of optimal treatment algorithms.

Acknowledgments

Supported in part by a Midcareer Investigator Award in Patient-Oriented Research (K24 AR053120) from the National Institute of Arthritis and Musculoskeletal and Skin Diseases (to Dr. Kevin C. Chung).

Appendix A

English Abstracts

Author Year Journal Outcomes
1 Ozalp 2009 Archives of Orthopaedic and Trauma Surgery Positive
2 Gay 2009 Plastic and Reconstructive Surgery Positive
3 Mathoulin 2009 Microsurgery Positive
4 Aly 2009 Orthopedics Positive
5 Meena 2009 Indian Journal of Orthopaedics Neutral
6 Croog 2008 Journal of Hand Surgery (American) Positive
7 Waitayawinyu 2008 Journal of Hand Surgery (American) Positive
8 Altay 2008 International Orthopaedics Positive
9 Arora 2008 Journal of Hand Surgery (American) Positive
10 Streich 2008 International Orthopaedics Positive
11 Watanabe 2008 Journal of Bone and Joint Surgery (American) Positive
12 Lumsden 2008 Journal of Hand Surgery (American) Positive
13 Jones 2008 Journal of Hand Surgery (Euroepan Volume) Neutral
14 Kawoosa 2007 International Orthopaedics Neutral
15 Hermans 2007 Scandinavian Journal of Plastic and Reconstructive Surgery and Hand Surgery Neutral
16 Tatebe 2007 Journal of Hand Surgery (American) Positive
17 Raven 2007 Clinical Orthopaedics and Related Research Positive
18 Tambe 2007 Acta Orthopaedica Belgica Negative
19 El-Mowafi 2006 Acta Orthopaedica Belgica Neutral
20 Schweizer 2006 Journal of Hand Surgery (American) Neutral
21 Tatebe 2006 Hand Surgery Positive
22 Gong 2006 Journal of Bone and Joint Surgery (British) Positive
23 Vanden Dungen 2006 Chirurgie de la Main Negative
24 Lu 2006 Annals of Plastic Surgery Positive
25 Moran 2005 Journal of Hand Surgery (American) Positive
26 Yasuda 2005 Scandinavian Journal of Plastic and Reconstructive Surgery and Hand Surgery Positive
27 Daecke 2005 Journal of Hand Surgery (American) Neutral
28 Daecke 2005 Journal of Hand Surgery (American) Positive
29 Zafra 2005 Acta Orthopaedica Belgica Positive
30 Tambe 2005 International Orthopaedics Positive
31 Zenzai 2005 Journal of Hand Surgery (British and European Volume) Positive
32 DeSmet 2005 Journal of Hand Surgery (British and European Volume) Positive
33 Sakai 2004 Hand Surgery Neutral
34 Meier 2004 Journal of Hand Surgery (British and European Volume) Positive
35 Yajima 2004 Scandinavian Journal of Plastic and Reconstructive Surgery and Hand Surgery Positive
36 Thomas 2004 Journal of Hand Surgery (American) Positive
37 Leblebicioglu 2003 Arthroscopy Neutral
38 Watson 2003 Journal of Hand Surgery (American) Neutral
39 Koh 2003 Journal of Hand Surgery (American) Positive
40 Kuhlman 2003 Acta Orthopaedica Belgica Positive
41 Gabl 2003 European Surgery Positive
42 Chillemi 2003 Journal of Orthopaedics and Traumatology Positive
43 Iwasaki 2002 Journal of Bone and Joint Surgery (British) Positive
44 Wada 2002 Journal of Hand Surgery (British and European Volume) Neutral
45 Laing 2002 European Journal of Orthopaedic Surgery and Traumatology Positive
46 Gabl 2002 Journal of Hand Surgery (British and European Volume) Neutral
47 Oishi 2002 Plastic and Reconstructive Surgery Positive
48 Minami 2002 Current Opinion in Orthopaedics Positive
49 Soejima 2002 Journal of Hand Surgery (American) Positive
50 Wintman 2001 Orthopedics Positive
51 Takase 2001 Journal of Bone and Joint Surgery (American) Positive
52 Illarramendi 2001 Journal of Hand Surgery (American) Positive
53 Kakinoki 2001 Hand Surgery Neutral
54 Bengoechea-Beeby 2001 Journal of Hand Surgery (American) Neutral
55 Sauerbier 2000 Annals of Plastic Surgery Positive
56 Lamas 2000 Journal of Hand Surgery (American) Neutral
57 Salmon 2000 Journal of Bone and Joint Surgery (British) Positive
58 Makino 2000 Journal of Reconstructive Microsurgery Neutral
59 Menth-Chiari 1999 Arthroscopy Positive
60 Ueba 1999 Journal of Orthopaedic Science Neutral
61 Nakamura 1998 Journal of Hand Surgery (British and European Volume) Positive
62 Kaarela 1998 Journal of Hand Surgery (British and European Volume) Negative
63 Delaere 1998 Journal of Hand Surgery (British and European Volume) Negative
64 Moneim 1998 Iowa Orthopaedic Journal Positive
65 Yajima 1998 Journal of Hand Surgery (American) Positive
66 Garcia-Elias 1998 Annales de Chirurgie de la Main Neutral
67 Shayfer 1998 Orthopedics Neutral
68 Steenwerckx 1997 Acta Orthopaedica Belgica Neutral
69 Carroll 1997 Clinical Orthopaedics and Related Research Neutral
70 Quenzer 1997 Journal of Hand Surgery (American) Neutral
71 Trail 1996 Journal of Hand Surgery (British and European Volume) Positive
72 Watson 1996 Journal of Hand Surgery (American) Positive
73 Zelouf 1996 Journal of Hand Surgery (American) Positive
74 Guo 1996 Annals of Plastic Surgery Neutral
75 Miura 1996 Journal of Hand Surgery (American) Positve
76 Wheatley 1996 Annals of Plastic Surgery Positive
77 Rhee 1996 Journal of Korean Medical Science Neutral
78 Sennwald 1995 Journal of Hand Surgery (American) Positive
79 DeSmet 1995 Acta Orthopaedica Belgica Negative
80 Tomaino 1994 Journal of Hand Surgery (American) Neutral
81 Bochud 1994 Journal of Hand Surgery (British and European Volume) Neutral
82 Yajima 1994 Journal of the Japanese Orthopaedic Association Neutral
83 Begley 1994 Journal of Hand Surgery (American) Positive
84 Minami 1994 Journal of Hand Surgery (American) Positive
85 Amillo 1993 International Orthopaedics Positive
86 Inoue 1992 Archives of Orthopaedic and Trauma Surgery Neutral
87 Voche 1992 Journal of Hand Surgery (British and European Volume) Positive
88 O'Flanagan 1992 Journal of the Royal College of Surgeons of Edinborough Negative
89 Inoue 1992 Acta Orthopaedica Scandinavica Neutral
90 Nakamura 1991 Journal of Bone and Joint Surgery (American) Positive
91 Weiss 1991 Journal of Bone and Joint Surgery (American) Positive
92 Rock 1991 Journal of Hand Surgery (American) Positive
93 Inoue 1990 Acta Orthopaedica Scandinavica Positive
94 Hasselgren 1990 Journal of Hand Surgery (British and European Volume) Positive
95 Alexander 1990 Journal of Hand Surgery (American) Negative
96 Kawai 1988 Journal of Bone and Joint Surgery (American) Neutral
97 Viljakka 1987 Acta Orthopaedica Scandinavica Neutral
98 Schattenkerk 1987 Acta Orthopaedica Scandinavica Neutral
99 Ekerot 1986 Scandinavian Journal of Plastic and Reconstructive Surgery Neutral
100 Kato 1986 Journal of Hand Surgery (American) Positive
101 Evans 1986 Journal of Hand Surgery (British and European Volume) Neutral
102 Watson 1985 Journal of Hand Surgery (American) Neutral
103 Blanco 1985 Journal of Hand Surgery (American) Neutral
104 Backaert 1985 Acta Orthopaedica Belgica Neutral
105 Sundberg 1984 Clinical Orthopaedics and Related Research Neutral
106 Pardini 1984 International Orthopaedics Neutral
107 Ishiguro 1984 Journal of the Japanese Orthopaedic Association Positive
108 Eiken 1984 Scandinavian Journal of Plastic and Reconstructive Surgery Positive
109 Kinnard 1983 Canadian Journal of Surgery Positive
110 Armistead 1982 Journal of Bone and Joint Surgery (American) Positive
111 Ramakrishna 1982 Journal of Bone and Joint Surgery (American) Neutral
112 Almquist 1982 Journal of Hand Surgery (American) Neutral
113 Lichtman 1982 Journal of Hand Surgery (American) Positive
114 Hedeboe 1982 Scandinavian Journal of Plastic and Reconstructive Surgery Positive
115 Bertini 1982 Italian Journal of Orthopaedics and Traumatology Positive
116 Stark 1981 Journal of Bone and Joint Surgery (American) Negative
117 Grassi 1978 Italian Journal of Orthopaedics and Traumatology Positive
118 Inglis 1977 Journal of Bone and Joint Surgery (American) Positive
119 Lichtman 1977 Journal of Bone and Joint Surgery (American) Neutral
120 Roca 1976 Journal of Bone and Joint Surgery (American) Neutral
121 Barber 1974 Journal of Bone and Joint Surgery (British) Positive
122 Codega 1973 International Surgery Neutral
123 Nahigian 1970 Journal of Bone and Joint Surgery (American) Neutral
124 Graner 1966 Journal of Bone and Joint Surgery (American) Positive

Non-English Abstracts

Author Year Journal Outcomes
1 Von-Maydell 2008 Handchirurgie, Mikrochirurgie, Plastische Chirurgie Neutral
2 Lecomte 2007 Revue de Chirurgie Orthopedique et Reparatrice de l'Appareil Moteur Neutral
3 Baronetti 2006 Minerva Ortopedica e Traumatologica Neutral
4 Khorbi 2005 Tunisie Medicale Neutral
5 Adel 2005 Tunisie Medicale Positive
6 Amillo-Garayoa 2005 Revista de Ortopedia y Traumatologia Positive
7 Lu 2003 Chinese Medical Journal Positive
8 Welby 2003 Chirurgie de la Main Negative
9 Das Gupta 2003 Handchirurgie Mikrochirurgie Plastische Chirurgie Positive
10 Altay 2002 Artroplasti Artroskopik Cerrahi Positive
11 Sauerbier 2001 Handchirurgie Mikrochirurgie Plastische Chirurgie Positive
12 Trankle 2000 Handchirurgie Mikrochirurgie Plastische Chirurgie Positive
13 Siala 2000 Revue de Chirurgie Orthopedique et Reparatrice de l'Appareil Moteur Positive
14 Jiang 1999 Chinese Journal of Reparative and Reconstructive Surgery Positive
15 Dautel 1999 Main Neutral
16 Sauerbier 1998 Langenbecks Archiv für Chirurgie. Supplement. Kongressband. Deutsche Gesellschaft für Chirurgie. Kongress Positive
17 Bartelmann 1998 Handchirurgie Mikrochirurgie Plastische Chirurgie Negative
18 Schulz 1998 Handchirurgie Mikrochirurgie Plastische Chirurgie Neutral
19 Renner 1998 Handchirurgie Mikrochirurgie Plastische Chirurgie Positive
20 Wustner-Hofmann 1997 Handchirurgie Mikrochirurgie Plastische Chirurgie Neutral
21 Staudenmaier 1997 Handchirurgie Mikrochirurgie Plastische Chirurgie Positive
22 Thomas 1997 Annales de Chirurgie de la Main et du Membre Superieur Neutral
23 Ducarmois 1997 Annales de chirurgie de la main et du membre supérieur Neutral
24 Garbuio 1996 Annales de chirurgie de la main et du membre supérieur Neutral
25 Wachtl 1994 Schweizerische Rundschau fur Medizin Praxis Negative
26 Gomis 1994 Revue de Chirurgie Orthopedique et Reparatrice de l'Appareil Moteur Positive
27 Allieu 1991 Annales de Chirurgie de la Main et du Membre Superieur Positive
28 Allieu 1991 Annales de chirurgie de la main et du membre supérieur Positive
29 Voche 1991 Revue de Chirurgie Orthopedique et Reparatrice de l'Appareil Moteur Positive
30 Ham 1990 Nederlands Tijdschrift voor Geneeskunde Neutral
31 Buck-Gramcko 1990 Handchirurgie Mikrochirurgie Plastische Chirurgie Negative
32 Kawai 1990 Annales de Chirurgie de la Main et du Membre Superieur Neutral
33 Shibata 1989 Journal of the Japanese Orthopaedic Association Neutral
34 Lesur 1989 Revue de chirurgie orthopédique et réparatrice de l'appareil moteur Neutral
35 Yang 1989 Chinese Journal of Surgery Neutral
36 Ehall 1989 Handchirurgie Mikrochirurgie Plastische Chirurgie Positive
37 Prommersberger 1988 Handchirurgie Mikrochirurgie Plastische Chirurgie Neutral
38 Kern 1988 Zeitschrift fur Orthopadie und Ihre Grenzgebiete Neutral
39 Bruser 1986 Handchirurgie Mikrochirurgie Plastische Chirurgie Neutral
40 Rajani 1985 Ugeskrift for Laeger Positive
41 Erbs 1984 Handchirurgie Mikrochirurgie Plastische Chirurgie Neutral
42 Razemon 1984 Chirurgie Positive
43 Schmitt 1984 Zeitschrift fur Orthopadie und Ihre Grenzgebiete Positive
44 Saffar 1982 Annales de Chirurgie de la Main Neutral
45 Roullet 1982 Annales de Chirurgie de la Main Negative
46 Comtet 1982 Annales de chirurgie de la main Neutral
47 Naett 1981 Handchirurgie Neutral
48 Kerschbaumer 1981 Orthopade Positive
49 Kerschbaumer 1979 Handchirurgie Negative
50 Codega 1973 Polski Przeglad Chirurgiczny Neutral
51 Sommelet 1970 Revue de chirurgie orthopédique et réparatrice de l'appareil moteur Neutral

Footnotes

We appreciate the assistance of Soo Young Kwak with the statistical analyses portion of this review.

References

  • 1.National Institutes of Health Office of Rare Diseases – Rare Disease Terms. [Accessed June 30 2009]; Available at: http://rarediseases.info.nih.gov/Default.aspx.
  • 2.Schuind F, Eslami S, Ledoux P. Kienbock's disease. J Bone Joint Surg (Br) 2008;90B(2):133–139. doi: 10.1302/0301-620X.90B2.20112. [DOI] [PubMed] [Google Scholar]
  • 3.Luo J, Diao E. Kienbock's disease: An approach to treatment. Hand Clin. 2006;22(4):465–73. doi: 10.1016/j.hcl.2006.07.003. [DOI] [PubMed] [Google Scholar]
  • 4.Wagner JP, Chung KC. A historical report on Robert Kienbock (1871-1953) and Kienbock's Disease. J Hand Surg (Am) 2005;30(6):1117–21. doi: 10.1016/j.jhsa.2005.08.002. [DOI] [PubMed] [Google Scholar]
  • 5.Dickersin K. The existence of publication bias and risk factors for its occurrence. JAMA. 1990;263(10):1385–9. [PubMed] [Google Scholar]
  • 6.Chalmers TC, Frank CS, Reitman D. Minimizing the three stages of publication bias. JAMA. 1990;263(10):1392–95. [PubMed] [Google Scholar]
  • 7.Callaham M, Wears RL, Weber E. Journal prestige, publication bias, and other characteristics associated with citation of published studies in peer-reviewed journals. JAMA. 2002;287(21):2847–50. doi: 10.1001/jama.287.21.2847. [DOI] [PubMed] [Google Scholar]
  • 8.Callaham ML, Wears RL, Weber EJ, Barton C, Young G. Positive-outcome bias and other limitations in the outcome of research abstracts submitted to a scientific meeting. JAMA. 2002;280(3):254–7. doi: 10.1001/jama.280.3.254. [DOI] [PubMed] [Google Scholar]
  • 9.Simes RJ. Publication bias: the case for an international registry of clinical trials. J Clin Oncol. 1986;4(10):1529–41. doi: 10.1200/JCO.1986.4.10.1529. [DOI] [PubMed] [Google Scholar]
  • 10.Yoshimoto Y. Publication bias in neurosurgery: lessons from series of unruptured aneurysms. Acta Neurochir. 2003;145:45–48. doi: 10.1007/s00701-002-1036-0. [DOI] [PubMed] [Google Scholar]
  • 11.Easterbrook PJ, Berlin JA, Gopalan R, Matthews DR. Publication bias in clinical research. The Lancet. 1991;337:867–72. doi: 10.1016/0140-6736(91)90201-y. [DOI] [PubMed] [Google Scholar]
  • 12.Krzyzanowska MK, Pintilie M, Tannock IF. Factors associated with failure to publish large randomized trials presented at an oncology meeting. JAMA. 2003;290(4):495–501. doi: 10.1001/jama.290.4.495. [DOI] [PubMed] [Google Scholar]
  • 13.Hasenboehler EA, Choudhry IK, Newman JT, Smith WR, Ziran BH, Stahel PF. Bias towards publishing positive results in orthopedic and general surgery: a patient safety issue? Patient Saf Surg. 2007;1(1):4–9. doi: 10.1186/1754-9493-1-4. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 14.Harris IA, Mourad M, Kadir A. Publication bias in abstracts presented to the annual meeting of the American Academy of Orthopaedic Surgeons. J Orthop Surg. 2007;15(1):62–6. doi: 10.1177/230949900701500114. [DOI] [PubMed] [Google Scholar]
  • 15.Okike K, Kocher MS, Mehlman CT, Heckman JD, Bhandari M. Nonscientific factors associated with acceptance for publication in the Journal of Bone and Joint Surgery (American volume) J Bone Joint Surg Am. 2008;90(8):2432–7. doi: 10.2106/JBJS.G.01687. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 16.Okike K, Kocher MS, Mehlman CT, Heckman JD, Bhandari M. Publication bias in orthopaedic research: an analysis of scientific factors associated with publication in the Journal of Bone and Joint Surgery (American volume) J Bone Joint Surg Am. 90(3):595–601. doi: 10.2106/JBJS.G.00279. [DOI] [PubMed] [Google Scholar]
  • 17.Ioannidis JP. Effect of the statistical significance of results in the time to completion and publication of randomized efficacy trials. JAMA. 1998;279(4):281–6. doi: 10.1001/jama.279.4.281. [DOI] [PubMed] [Google Scholar]
  • 18.Burdett S, Stewart LA, Tierney JF. Publication bias and meta-analyses: a practical example. Int J Technol Assess Health Care. 2003;19(1):129–34. doi: 10.1017/s0266462303000126. [DOI] [PubMed] [Google Scholar]
  • 19.McAuley L, Pham B, Tugwell P, Moher D. Does the inclusion of grey literature influence estimates of intervention effectiveness reported in meta-analyses? Lancet. 2000;356:1228–31. doi: 10.1016/S0140-6736(00)02786-0. [DOI] [PubMed] [Google Scholar]
  • 20.Vickers A, Goyal N, Harland R, Rees R. Do certain countries produce only positive results? A systematic review of controlled trials. Control Clin Trials. 1998;19:159–66. doi: 10.1016/s0197-2456(97)00150-5. [DOI] [PubMed] [Google Scholar]
  • 21.Hopewell S, Clarke M, Stewart L, Tierney J. Time to publication for results of clinical trials. Cochrane Database Syst Rev. 2007;18(2):MR000011. doi: 10.1002/14651858.MR000011.pub2. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 22.Chalmers I. Underreporting research is a scientific misconduct. JAMA. 1990;263(10):1405–8. [PubMed] [Google Scholar]
  • 23.Syin D, Woreta T, Chang DC, Cameron JL, Pronovost PJ, Makary MA. Publication bias in surgery: implications for informed consent. J Surg Res. 2007;143:88–93. doi: 10.1016/j.jss.2007.03.035. [DOI] [PubMed] [Google Scholar]
  • 24.Chang DC, Matsen SL, Simpkins CE. Why should surgeons care about clinical research methodology. J Am Coll Surg. 2006;203:827–30. doi: 10.1016/j.jamcollsurg.2006.08.013. [DOI] [PubMed] [Google Scholar]
  • 25.Dickersin K, Chan S, Chalmers TC, Sacks HS, Smith H., Jr Publication bias and clinical trials. Controlled Clin Trials. 1987;8:343–53. doi: 10.1016/0197-2456(87)90155-3. [DOI] [PubMed] [Google Scholar]
  • 26.Sommer B. The file drawer effect and publication rates in menstrual cycle research. Psychol Women Q. 1987;11:233–42. [Google Scholar]
  • 27.Dudley HAF. Surgical research: master or servant. Am J Surg. 1978;135:458–460. doi: 10.1016/0002-9610(78)90084-3. [DOI] [PubMed] [Google Scholar]
  • 28.Goldman L, Loscalzo A. Fate of cardiology research originally published in abstract form. N Engl J Med. 1980;303:255–9. doi: 10.1056/NEJM198007313030504. [DOI] [PubMed] [Google Scholar]
  • 29.Meranze J, Ellison N, Greenhow DE. Publications resulting from anesthesia meeting abstracts. Anesth Analg. 1982;61:445–8. [PubMed] [Google Scholar]
  • 30.McCormick MC, Holmes JH. Publication of research presented at the pediatric meetings. AJDC. 1985;139:122–6. doi: 10.1001/archpedi.1985.02140040020017. [DOI] [PubMed] [Google Scholar]
  • 31.Chalmers I, Adams M, Dickersin K, Hetherington J, Tarnow-Mordi W, Meinert C, et al. A cohort study of summary reports of controlled trials. JAMA. 1990;263:1401–4. [PubMed] [Google Scholar]
  • 32.Gavazza JB, Foulkes GD, Meals RA. Publication patterns of papers presented at the American Society for Surgery of the Hand Annual Meeting. J Hand Surg (Am) 1996;21A:742–5. doi: 10.1016/S0363-5023(96)80185-7. [DOI] [PubMed] [Google Scholar]
  • 33.Lilford RJ, Thornton JG, Braunholtz D. Clinical trials and rare diseases: a way out of a conundrum. BMJ. 1995;311(7020):1621–5. doi: 10.1136/bmj.311.7020.1621. [DOI] [PMC free article] [PubMed] [Google Scholar]
  • 34.Behera M, Kumar A, Soares HP, Sokol L, Djulbegovic B. Evidence-based medicine for rare diseases: implications for data interpretation and clinical trial design. Cancer Control. 2007;14(2):160–6. doi: 10.1177/107327480701400209. [DOI] [PubMed] [Google Scholar]
  • 35.Hampton T. Rare disease research gets a boost. JAMA. 2006;295(24):2836–8. doi: 10.1001/jama.295.24.2836. [DOI] [PubMed] [Google Scholar]
  • 36.Dickersin K, Rennie D. Registering clinical trials. JAMA. 2003;290(4):516–523. doi: 10.1001/jama.290.4.516. [DOI] [PubMed] [Google Scholar]
  • 37.National Institutes of Health. [Accessed June 30 2009]; ClinicalTrials.gov. Available at: http://www.clinicaltrials.gov/ct2/home.

RESOURCES