Impact of traditional bonesetters on contemporary fracture care in Low and Middle Income Countries (LMICs): a systematic review

Background : The review aimed at systematically examining the evidence in articles that assess the clinical effects and impact of traditional bonesetters on contemporary fracture care in Low and Middle Income Countries (LMICs). Methods : A systematic review was conducted. Articles were identified by database searching ((PubMed, Embase, ScienceDirect, SCOPUS, and Web of Science). Searching, selecting and reporting were conducted according to the PRISMA (Preferred Reporting Items for Systematic reviews and Meta-Analyses) Statement. The key words that were used in search for literature were: “Bonesetter”, “fracture healer” and “traditional bone setting”. Publications included for review were original articles, set in an LMIC and directly talked about the role and/or impact of traditional bonesetters in providing fracture care. Papers that focused on Low and Middle Income (LMIC) settings were reviewed. Results : A total of 176 papers were screened for eligibility and 15 studies were finally included. Nine were prospective studies, while 6 were retrospective studies. Most of the studies focused on clinical impacts of bone setter intervention. The evidence from the publications show that the main clinical effects of traditional bonesetters had been deleterious, but they had the potential to contribute positively when trained. Conclusion : Few well designed studies are available that assessed the impact of traditional bonesetters. Reported cases and reviews indicate their impact to be deleterious. However, the potential exist that when trained, these deleterious impact can be reduced through training for traditional bonesetters who contribute to fracture care in many LMICs.


Introduction
In most Low and Middle Income countries (LMICs), traditional bonesetters (TBS) still play an integral role in trauma care [1]. The practice of traditional bone setting dates back to history and it has found root in many developing countries in Africa, South America and the Indian subcontinent where TBS still play a role in providing services [2]. In many developing countries, traditional care of injuries and diseases has remained popular despite the existence of modern health care services and advancement in various spheres of life [2,3]. Bone setting skills are usually passed down the family line without any documentation. TBS receive no formal training in modern orthopaedic care and their practice is kept a family secret and as part of an ancestral heritage [4].
Despite the presence and availability of modern orthopaedic services (MOS), TBS enjoy high patronage and wide acceptance across different social and educational strata as well as religions [5].The high patronage of TBS is rooted in cultural belief and has no correlation with educational status, income or occupation [3].Cultural beliefs, expectations of quicker healing, cheaper services and fear of amputation have been noted as reasons for TBS patronage in some studies [6,7,8]. The low coverage of health insurance and high out-of-pocket costs of healthcare in most developing countries also contribute to the poor utilization of MOS [9].
In Nigeria, TBS provide about 70%-90% of primary fracture care in certain areas [10] and this method of fracture care cannot be overlooked, as it has a huge impact on health outcomes. Many patients with fractures would preferentially present directly to the TBS or after initial resuscitation in a hospital.
The bone setting techniques adopted by the TBS lack a sound scientific basis which may lead to limb and life-threatening complications [11]. Various complications and failure rates of TBS treatment have been reported. Oginni [12] and OlaOlorun et al. [13] both in southwest Nigeria, have reported complication rates of 66.7% and 83% respectively, while Faheem et al. [14] in India reported 43%.
Onuminya [9]also observed that 50-60% of limb gangrene in Nigeria were due to complications of TBS practice. Some complications from TBS interventions include: non-union, mal-union, chronic osteomyelitis, limb gangrene, compartment syndrome and joint stiffness [9,11,15,16]. These complications constitute management challenges to orthopaedic surgeons in developing countries as they increase the case load and lead to orthopaedic surgeons treating complications of fractures [4,12].
This systematic review aims to provide a concise overview of current knowledge base of the impact of TBS on fracture care, identify gaps in knowledge and potential areas for future research to assess impact of well-designed interventions on outcome for patients who patronize TBS.

Methods
This systematic review was conducted based on the PRISMA (Preferred Reporting Items for Systematic reviews and Meta-Analyses) framework in order to assure quality of the search process and adequate reporting within this paper [17].

Search Strategy
Articles were identified by database searching (PubMed, Embase, ScienceDirect, SCOPUS, and Web of Science). A secondary search was conducted using three WHO indexes: Hinari Access to Research for Health program, Institutional Repository for Information Sharing (IRIS) and the Global Index Medicus (GIM). The time period used was from 1900 to May 2017. Titles, abstracts, key words and full texts were searched with the following search term: ("bonesetter" OR "fracture healer" OR "Traditional bone setting"). The search strategy used for the PubMed search was 'bonesetter' OR 'traditional bone setting' OR 'fracture healer' AND 'Fractures' OR 'injuries' OR 'musculoskeletal injuries' AND 'fracture care' Or 'treatment'. The search strategy was adapted for each database as necessary. Furthermore, a manual search was conducted to identify relevant articles in the reference lists of the identified articles which meet the inclusion criteria.

Inclusion Criteria
Articles which were included in this systematic review had to meet the following criteria: 1) set in an LMIC; 2) published in English; 3) articles published in peer reviewed journals between 1900 and 2018; 4) directly mentioned the impact of traditional bonesetters' treatment of fractures; 5) articles that evaluated the clinical complications of fractures resulting from traditional bonesetters' treatment.

Exclusion Criteria
Articles were excluded if the paper: i) was a literature review, letter, comment or conference abstract ii) described a theoretical concept but did not directly address the role or impact of traditional bonesetters iii) had no abstract. Case reports, case studies and qualitative studies were excluded.
Study eligibility for inclusion was assessed in duplicate by two authors, and in cases of discrepancy, a third author was consulted to reach a consensus.

Data Extraction and Synthesis
Titles and abstracts were initially screened for their fulfilment of inclusion criteria. Subsequently, the remaining potentially eligible articles were appraised in detail by reading the full-text papers.
All the retained papers were reviewed independently by two sets of two authors who examined the full texts using a data extraction pro-forma developed and pre-tested by the review team. The data extraction pro-forma was used to extract the following: the study setting, the study design, the study objectives, study population, sampling techniques, the treatment received, outcome of the TBS treatment, study limitations and conflicts of interest. Cases of non-concordance were resolved by another author. If there were any discrepancies between the two sets of authors, a fifth author adjudicated.

Quality Assessment
We assessed the quality of the included studies using the National Heart, Lung, and Blood Institute (NHLBI) Quality Assessment Tool for Observational Cohort and Cross-Sectional Studies, which is widely used and recommended by Cochrane for quality assessment of observational and crosssectional studies [18]. The tool assessed the internal validity and risk of bias of the included studies using a checklist of 14 questions with responses, yes, no or others (cannot determine, CD; not applicable, NA; and not reported, NR). Two authors applied the tool and independently evaluated the items of the tool as "yes, " "no, " "not applicable, " "cannot determine, " or "not reported." This was used to guide the overall rating for the quality of each study as "good," "fair," or "poor." Where there was a disagreement, consensus was reached through discussion or by consulting a third author.

Study selection
The database search yielded 192 results (Embase-41; PubMed-28; Science Direct-5; Cochrane-1; SCOPUS-91; Web of Science-26). Fifty four articles were identified through other sources. After the exclusion of duplicates, there were 176 articles eligible for further screening. Title and abstract screening according to the selection criteria led to the exclusion of 143 articles, leaving 33 articles for full-text assessment. After a careful review of these 33 articles, 15 were selected for inclusion in the current systematic review. The process of study selection is depicted in a flow diagram ( Figure 1). Table 1

Quality of included studies
We assessed the methodological quality of the studies according to the NHLBI assessment tool. Most of the studies were of intermediate quality (53.3%), 26.7% were of high quality and 20% of poor quality (table 2). Almost all the included studies had clearly stated research questions and objectives with clearly specified and defined study population. The most frequently unreported criteria were the loss to follow-up after baseline of 20% or less and the participation of eligible persons of at least 50%.
In most of the included studies, the exposure and outcome measures were clearly defined, valid, reliable and implemented consistently across all the study participants. Other commons strengths included the clearly defined inclusion and exclusion criteria for being included in the studies which were pre-specified and applied uniformly to all participants. Blinding of the outcome assessors to the exposure status of the participants was considered not to be applicable in the included studies. Only two of the studies reported that key potential confounding variables were measured and adjusted statistically for their impact on the relationship between exposures and outcomes.

Outcomes of selected articles
The outcome measures assessed were the presence of any musculoskeletal complications resulting from the treatment of a fracture by a traditional bonesetter. A range of indicators were employed for evaluation of outcomes. The most common complications that resulted from TBS treatment of fractures were: malunion, non-union, limb gangrene, compartment syndrome, joint stiffness, limb shortening and chronic osteomyelitis. Limb gangrene was a common complication amongst children [4,22,25]. Two studies examined the methods of treatment by TBS that resulted in complications.
The local splints used by TBS in immobilizing limb fractures was noted to be a major cause of gangrene in children [15,25]. One of the studies analyzed the cost effectiveness of TBS treatment. It was noted that the cost of treatment of the TBS ranged from USD 18-380, whereas the treatment of certain fractures at an orthopaedic hospital in Nigeria ranged from USD 34-98 [19]. The impact of TBS on treatment outcomes of fractures was generally found to be negative, reversing gains made by orthodox fracture care and leading to deleterious complications in many LMICs (Table 1).

Discussion
A total of 15 studies were fully reviewed in order to examine the impact of traditional bonesetters on fracture care in LMICs. Given the widespread influence and popularity of traditional bonesetters in many LMICs, this is surprisingly a low number. This revealed to us that the subject of bonesetters is an understudied one. Most of the articles included were set in Asia and Sub-Saharan Africa. This may be explained by the existence of TBS and their wide-spread patronage in these regions. Some of the papers which appeared with our search gave a historical account and examined factors that led to many people choosing TBS over orthodox medical practitioners, with few examining their impact on fracture care. Most of the articles included in this systematic review were set in English-speaking countries. This finding could be due to the inclusion criterion that required all articles to be in English or a greater familiarity with bone setters in English speaking countries than others.
It is worth observing that all the impacts of TBS interventions identified in the studies reviewed were of a clinical nature, with the most common impacts being Malunion, nonunion, limb gangrene, amputations, and compartment syndrome. These may be attributable to the non-scientific nature of their treatment methods which was highlighted by two studies [15,25]. The studies reported that the local splints applied by TBS in immobilizing limb fractures were applied too tightly resulting in compartment syndrome, Volkmann's ischemic contracture and gangrene. The health and socioeconomic impact of these complications are enormous because young adults in the reproductive age are predominantly involved. These complications also pose a management challenge to orthopaedic surgeons in the developing countries resulting in poor outcomes of fracture treatment [4,11,12].
In spite of the generally poorer outcomes associated with traditional bone setters relative to orthodox medical treatment, large numbers of people in LMICs continue to patronize their services. This is due to the absence of orthodox medical services, perceived cheaper services, the perception that they are more competent at treating fractures, discomfort in cast, the fear of surgery, mistrust of doctors among others [1,9,33,34,35,36]. Dada et al [19] in their study also analyzed the differential cost of fracture treatment between the TBS and orthopaedic surgeons at an orthopaedic hospital in Nigeria. It was noted that the cost of fracture treatment by the TBS ranged from USD 18-380, while that in the orthodox hospital ranged from USD 34-98. This is contrary to a widely held opinion in many LMICs that TBS treatment of fractures is cheaper than orthodox treatment.
Another important impact of TBS intervention, as identified in several of the articles reviewed was that patients who sought initial help from TBS before visiting the hospital were more likely to have poorer outcomes than those who sought initial care from orthodox hospitals [24,33]. This is usually as a result of TBS gangrene that may follow treatment by a TBS which might lead to amputation, sepsis, or death in some cases [22].
Many experts have recommended that because of the widespread presence of TBS and the influence and respect they command among a lot of people in LMICs, efforts should be made to retrain them and possibly integrate them into the formal health system [34,35]. Interviews with bone setters on this issue have elicited different responses; with some welcoming the idea and others rejecting it because they consider orthodox medical practitioners their competitors [36]. Onuminya [37] demonstrated that the training of a TBS resulted in a considerable decrease in the rate of gangrenous limbs, infection, non-union and malunion, when the operations of two bone setters were comparedone who had undergone training by orthodox medical practitioners and the other who had not.

Strengths and weaknesses
To the best of our knowledge, this systematic review is the first on the subject of traditional

Conclusion
Our review identifies that currently, TBS provide huge services to individuals with trauma and bone fractures in many LMICs. Most of the reported outcomes have been deleterious. However, there is potential that when TBS are trained, they can contribute positively to fracture care and outcomes.
Innovative interventions that can lead to improvement in the reported negative impact for individuals with fractures who patronize TBS should be tested in well-designed studies, and if proven to work should be adopted.

Consent to publish: Not applicable
Availability of data and materials: The datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request.
Competing interest: The authors declare no competing interests.
Funding: No financial assistance was received for this study. The study was self-funded by the authors.

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Hatipoglu S, Tatar K. The strengths and weaknesses of Turkish bone-setters. In:    Were key potential confounding variables measured and adjusted statistically for their impact on the relationship between exposure(s) and outcome(s)?