Pediatric Osteopathic Manipulative Medicine: A Scoping Review

Given the concern for the amount and consistency of the available literature, a scoping review is needed to evaluate the current state of information in the field and create a baseline foundation of knowledge.^7,8  Although systematic reviews and meta-analyses remain the gold standard when formulating clinical guidelines for treatment of specific conditions based on evidence collected from RCTs, the aggregation of pediatric OMM research to date appeared insufficient for such analysis at this time.^7,8  Our aims for performing a scoping review were to (1) identify and analyze knowledge gaps in the pediatric osteopathic literature, (2) categorize types of research previously performed and assess their methodologic quality, and (3) assess whether there is a level of evidence necessary to make clinical recommendations.

We followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines to conduct a literature search from each included database’s inception to the date of study initiation (September 24, 2019).⁹  In efforts to maintain a broad and comprehensive search, all studies published in peer-reviewed academic journals, regardless of study design, were eligible for inclusion in this review. A clinical librarian (A.G.) searched 8 medical literature databases, including Embase, Cumulative Index to Nursing and Allied Health Literature (CINAHL) Plus and Alt HealthWatch, Web of Science, Cochrane, OVID Medline, Clinicaltrials.gov, Osteomed.dr, and Proquest Dissertation. Additionally, the American Academy of Pediatrics' Section on Osteopathic Pediatricians research database, which serves as the reference database of citations of peer-reviewed pediatric osteopathic research, was reviewed.¹⁰  Last, the cited references from any article ascertained were manually reviewed for any additional research that may have been missed in the online search. For instances in which >1 study used the same data set, only articles that presented novel findings were included, otherwise only the more comprehensive and recent study was chosen for analysis.

The entry terms used were “osteopathic manipulative treatment,” “osteopathic manipulation,” “orthopedic manipulation,” and “osteopathy.” The search was limited, when available, to only include the pediatric patient cohort (ages 0–18 years).¹¹ 

All studies in which researchers investigated an aspect of pediatric OMM were assessed for inclusion. For purposes of this review, data from subjects who were >18 years old were excluded. Studies had to have been conducted in the United States and written in English to be included. No articles were excluded on the basis of year performed, setting of the study (eg, rural versus urban, inpatient versus outpatient), or study design. The aim of this scoping review was to assess publications of primary data; therefore, any editorials, reviews, or published responses to previously conducted research were excluded.

The initial database search yielded 456 articles (CINAHL and Alt HealthWatch: 156, Cochrane: 15, Embase: 51, Journal of the American Osteopathic Association [JAOA]: 10, Medline: 168, Osteomed.dr: 12, ProQuest Dissertations: 12, Web of Science: 5). After computer-identified duplicate articles were removed, a total of 319 references were included (CINAHL and Alt HealthWatch: 74, Cochrane: 9, Embase: 26, JAOA: 3, Medline: 163, Osteomed.dr: 34, ProQuest Dissertations: 12, Web of Science: 0). With the addition of the American Academy of Pediatrics' Section on Osteopathic Pediatricians database (69 articles), a total of 388 articles were then manually assessed for duplication (73 articles), for a final total of 315 articles. Two blinded reviewers (S.D. and A.H.) independently screened titles and abstracts for fit, and an additional third reviewer (J.A.B.) assisted in the case of discrepancies. During the manual search of the bibliographies of the publications as described above, 3 additional articles absent from the original databases met inclusion criteria and were added. Once the preliminary screen was performed, duplicates and unqualified entries were removed on the basis of exclusion criteria, and complete texts were requested and were read in full for further evaluation and validation. The blinded two-reviewer system was again used for agreement for final inclusion of articles into scoping review. Five articles had discrepancies at this second level of evaluation and were resolved by the third reviewer. At final assessment, there were 30 primary articles that were fully analyzed (PRISMA Summary Flow Diagram,¹²  Fig 1); citations for these articles are included in the reference list.^13–42 

Once the final list of articles to be included was established, the predefined data elements were collected and stored in an Excel spreadsheet. Data elements extracted included article title, year published, journal, authors, location of study, study design, disease or condition focus, OMM technique(s) used, primary objective of study, inclusion and exclusion criteria, number of physicians providing OMM, protocolized versus nonprotocolized treatment use, statistical analysis reported, outcomes, and overall strengths, limitations, and potential biases. Protocolized treatment was defined as a systematic-based treatment approach using a predefined prescriptive therapy regardless of the somatic dysfunction or physical findings of the individual subject being assessed. On the basis of the data above, primary outcome measures included (1) methodology of study design, (2) study conclusions, (3) study strengths, and (4) study limitations, including potential biases. Secondary outcomes included (1) conditions treated, (2) OMM techniques used, and (3) overall safety profile based on adverse events, when data were available.

The Grading of Recommendations, Assessment, Development, and Evaluations system was used to critically appraise the strength of evidence for the clinical utility of OMM in the pediatric population for specific conditions based on the original study data presented (Fig 2).^43,44  Through this system, the quality of evidence was assessed for each outcome, and a multistep grading process allowed complete analysis to conclude a strength of recommendation for each study.

Thirty primary research articles were included in the final review. Most articles (23 of 30, 77%) were published in the JAOA, with representation from additional peer-reviewed journals in the field (Table 1). Most (26 of 30, 87%) were conducted in an outpatient setting and had both male and female representation in the study populations. There was a wide spectrum of study designs including 8 (27%) case reports, 7 (23%) cross-sectional surveys, 2 (7%) retrospective cohort studies, 4 (13%) prospective cohort studies, and 9 (30%) RCTs (Figs 3 and 4). Studies ranged in publication dates from 1960 to 2019, with the majority of studies (17 of 30, 57%) being published since 2000 (Fig 5).

A wide range of clinical conditions was investigated, most commonly musculoskeletal disorders (8 of 30, 27% of studies), neurologic syndromes (6 of 30, 20%), and otorhinolaryngologic diagnoses (5 of 30, 17%). Other conditions such as asthma, feeding difficulties, and dysfunctional urinary voiding were also assessed. The majority of articles were focused on OMM as a treatment modality (20 of 30, 66%) for different medical conditions, with the minority assessing its role in the prevention of illness (5 of 30, 17%). Researchers in the remaining articles analyzed baseline characteristics of OMM patients and incidences of somatic dysfunction (4 of 30, 13% of studies), with researchers in 1 study (1 of 30, 3% of studies) reporting safety and adverse outcome data.

Nine of the studies were RCTs (Tables 2 and 3), 2 of which used a sham treatment group,^25,41  and the remaining 7 compared the addition of OMM to treatment as usual.^{15,19,20,31–33,37}  Wait-list controls were used in 2 of the studies^19,20  and OMM was compared with other complementary therapies in 3.^15,20,41  Additionally, researchers in 2 of these RCTs used a 4-pronged factorial design to compare multiple treatment groups.^15,41  In all but one of the studies, authors provided information to determine if intervention and control participants had similar baseline characteristics limiting residual confounding bias. In 7 (78%) of the RCTs, random allocation was adequately used. In 5 (56%) of the RCTs, the authors stated that the assessors were blinded to treatment allocation, and authors of 8 (89%) of the RCTs employed at least single blinding throughout the experimental comparison. Five (56%) of the RCTs were rated as providing strong clinical recommendations, with the other half rated as weak because of overall poor methodologic quality and small sample size (Table 3).

The techniques used to treat these dysfunctions and disorders included a wide variety of OMM treatment modalities (Table 2). Treatments were delivered in either a protocolized (14 of 30, 47% of studies) or nonprotocolized (16 of 30, 53%) manner by 1 (16 of 30, 53%) or multiple (14 of 30, 47%) osteopathic physicians (Table 2). Outcomes ranged from no effect to significant benefit of OMM on the basis of either qualitative or quantitative outcome measures (Table 3). Of the 30 studies analyzed, researchers in 13 reported the statistical analysis required to demonstrate statistically significant results. Previously validated and recognized scoring systems for collection of either quantitative or qualitative data were used in 30% (9 of 30) of the studies reviewed. In 1 study, researchers reported a participant with mild treatment-related aggravations that were self-resolved, requiring no interventions, and no additional treatment-related complications were reported.²⁶  No significant adverse events or deleterious effects associated with OMM were reported in any of the studies (Table 2), and many authors commented on the safety of OMM in their conclusions.

We analyzed the methodologic quality of the research conducted and systematically graded the studies presented to assess the ability to make clinical recommendations based on those results. Twenty-one of the 30 studies reviewed received a final evaluation of weak evidence, with only 9 studies providing a strong level of evidence on which to make clinical recommendations. Among the RCTs, we identified 4 studies that were of high methodologic quality.^19,25,37,41  The strongest grades of research provided evidence of the benefit of OMM in regard to neurologic developmental disorders and otorhinolaryngologic disease prevention and treatment. Weak grades of research were prevalent for all other conditions included in this review. The majority of studies descriptively identify OMM as having a low risk to benefit ratio, but although none of the studies were specifically dedicated and powered to assess the safety of OMM.

Our aim with this systematic scoping review was to summarize and critically evaluate published osteopathic literature in the pediatric field to date. We conducted this review to (1) better understand the size and state of the most recent published literature on osteopathic practices in pediatrics, (2) assess the quality of RCTs to determine if adequate data has been collected, and (3) synthesize results to guide future research by identifying current literature gaps.

Thirty articles met our criteria and were included in the review. There was an upward trend in the number of articles published within the past 2 decades, with only 8 of the articles being published from 1960 to 2000 and the remaining 22 published between 2000 and September 2019. Although this may reflect the overall increasing number of journals and published medical literature, it may also reflect the growing number of practicing osteopaths. This may further corroborate the increasing demand for work to be done in the field, especially given the overall relatively small number of articles found to be reviewed in this article. Comprehensively, OMM for the treatment of musculoskeletal, neurologic, and otorhinolaryngologic disorders have been the most studied conditions in pediatrics to date.

Although the conducted research spanned all tiers of the hierarchical research pyramid, poor methodology of many of these studies and presence of potential biases significantly limited the quality of the evidence provided. As described in the 2006 study by Licciardione and Russo,⁴⁵  clinical outcomes can be heavily influenced by “the complex interaction of treatment credibility, subject expectations, and the influence of nonspecific treatment effects.” This has inherently plagued osteopathic research because sham treatments have concern for a nonnegligible placebo effect with potential to artificially alter results.⁴⁵  A lack of both blinding and allocation concealment were noted in many studies, which lends to a high potential for expectation and retrospective biases.⁴⁵  Furthermore, a protocolized treatment approach was used in less than half of the studies, which contributed to the rating of low research quality given low likelihood of reproducibility. This highlights a continued paucity of high-quality scientific research available for clinicians to make evidence-based clinical recommendations when treating children. In addition, it perpetuates the lack of support for OMM use in the pediatric clinical setting.

Overall, the majority of studies included in this review provided weak evidence to support the use of OMM. Only one-third of the studies were RCTs, and furthermore, only half of these provided data of high enough caliber to make isolated recommendations based on clinical evidence. As concluded by Podazski et al,⁶  there is not currently an indication for which the effectiveness of OMM has been shown consistently enough to make strong clinical recommendations for any particular condition. Given this, OMM has not been studied well enough to be actively recommended, but it appears to be generally safe, with no adverse events reported in any of the included studies. By using a similar format as proposed by Klein et al, currently, OMM can be medically tolerated (Table 4), but dedicated, large population, multi-institutional RCTs will be required to investigate the safety, efficacy, and feasibility of OMM as a recommended adjunctive treatment modality.⁴⁶  Once the safety of OMM practices is established for certain conditions and in specific populations, the efficacy can be further evaluated, and the feasibility of incorporating treatment into practice can be established.

It is our hope that this scoping review provides a framework for future research studies in the osteopathic pediatric literature, to optimally provide the best care for children. Given the identified issues, we suggest the following 3 recommendations for future investigation regarding pediatric osteopathic research in the United States:

• An increase in the volume of research conducted in the field to support evidence-based practice. In the Sicily statement by Dawes et al,⁴⁷  “Evidence-based practice requires that decisions about health care are based on the best available, current, valid and relevant evidence. These decisions should be made by those receiving care, informed by the tacit and explicit knowledge of those providing care, within the context of available resources.” By assessing the studies conducted to date, a minimal number of strong recommendations are able to be made, but solid baseline knowledge can be gleaned and a framework for conducting future research studies can be recommended.

• Future RCTs conducted must be of strongly graded methodologic quality with adherence to the Consolidated Standards of Reporting Trials statement.⁴⁸  Effective, high-quality research must be ascertained to propel the field of osteopathic medicine forward in an increasingly evidence-based world. The standardized process in which research is conducted needs to be upheld without denigrating the pillars of osteopathic practice. To remedy this, we suggest that osteopathic studies be conducted similar to procedural and surgical trials to conduct mixed methodology, prospective, controlled, sufficiently powered studies. Researchers of these studies should first assess feasibility and safety of OMM within a certain patient population. These data could then be used to assess efficacy and then subsequently compare with an active control in a large-scale study to assess benefit.

• Researchers examining OMM must increase their use of evidence-based treatments as comparison or control groups in their research. These research efforts should focus on pediatric osteopathic practices most often delivered, yet understudied, such as treatments of musculoskeletal disorders, common pediatric otorhinolaryngologic diseases, and the use of osteopathic cranial manipulation. Longitudinal, multimodal studies of factorial design proving the benefit of the addition of OMM into the current standard of treatment will be necessary to garner widespread acceptance for integration.

This scoping review has several limitations that should be considered when interpreting these results. First, although our search was extensive, we cannot be certain that all pediatric osteopathic literature was identified. However, to address this, we searched 10 comprehensive databases with guidance from librarian experts. Second, we were only able to include articles in the English language because of limited resources for translational purposes. Given the difference in practice rights between US-trained osteopathic physicians who are trained medical doctors with additional training in musculoskeletal medicine versus European osteopaths who are solely trained in musculoskeletal medicine, we purposefully narrowed our data set to include only US-based studies. By doing so, the scope of practice and the medical training of the treating physicians were presumed to be standardized and allowed the data presented to be extrapolated to the patient population for which we intend to make clinical recommendations. Although minimal inconsistencies in grading the level of evidence of these studies were encountered, attempts were made to negate discrepancies by using 2 independent, blinded reviewers and an additional reviewer to clarify any incongruities.

Although we identified an increasing number of published pediatric osteopathic research studies, there continues to be a paucity of high-quality, methodologically sound literature supporting OMM effectiveness in this population. Based on this, strong clinical recommendations cannot be made at this time, but OMM can be medically tolerated given its low risk profile. More robust, scientifically rigorous studies must be conducted to guide evidence-based recommendations regarding osteopathic medicine and its use as an adjunctive therapy. Osteopathic physicians should focus future research on RCTs for common pediatric conditions to begin investigating efficacy and move the pediatric osteopathic research field forward.

We acknowledge Susan Jones for her assistance in obtaining full texts of difficult to retrieve articles and her dedication to this project.

CAM

complementary or alternative medicine

CINAHL

Cumulative Index to Nursing and Allied Health Literature

D.O.

Doctor of Osteopathic Medicine

JAOA

Journal of the American Osteopathic Association

OMM

osteopathic manipulative medicine

PRISMA

Preferred Reporting Items for Systematic Reviews and Meta-Analyses

RCT

randomized controlled trial