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Volume 15, Issue 3 (2023)
Iran J War Public Health 2023, 15(3): 257-262 |
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Received: 2023/08/9 | Accepted: 2023/09/18 | Published: 2023/10/2
Received: 2023/08/9 | Accepted: 2023/09/18 | Published: 2023/10/2
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Pashmdarfard M, Kalantari M. Splinting in Congenital Hand Anomalies; A Systematic Review. Iran J War Public Health 2023; 15 (3) :257-262
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1- “Physiotherapy Research Center” and “Department of Occupational Therapy, School of Rehabilitation”, Shahid Beheshti University of Medical Sciences, Tehran, Iran
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Introduction
Hand anomaly can be congenital or acquired as a result of neurological damage to the central nervous system during the childhood process [1]. Most hand deformities in children are known as a congenital disorder, or an abnormal condition that is present at birth regardless of its cause [2]. These abnormalities may lead to structural disorders, in which problems are seen in the appearance of a body part, or functional disorders, in which there are problems in the way a part of the body functions [3]. However, the above-mentioned classification has been updated over several periods, and the last updated classification is related to 2020 [4].
In 2010, for the first time, a new classification for upper limb congenital malformations was published under the title of Oberg-Manske-Tonkin (OMT) [5]. Based on a malformation framework, OMT is accepted by the International Federation of Hand Surgery Society and surgeons treating congenital anomalies of the upper extremities. According to the updated OMT classification, upper extremity abnormalities fall into one of the categories of malformations, deformities, dysplasias, and syndromes [6].
Congenital malformations and anomalies of the upper limb, with a reported overall prevalence of 21.5 out of 10,000 live births, may have serious consequences for any patient [7]. The causes of congenital abnormalities in children are unknown in 60% of cases [8]. The greatest effect of congenital anomalies is observed in the fingers, followed by the forearm area, and finally, in the upper parts, such as the arm [9].
The International Classification of Functioning, Disability, and Health (ICF) provides a framework that describes the interrelationship between body function and structure, activities, and participation, as well as the influence of environmental and personal factors [10]. In the meantime, hand function in children with congenital anomalies of the upper limb can lead to a vicious cycle in body structure, function, activity, or participation in different areas of life. However, early and timely interventions in this vicious cycle can create hope to break this vicious cycle and promote children's participation [11].
Performing activities of daily living (ADL) requires the hands to perform different movements in a coordinated manner at the same time [12]. In the meantime, congenital anomalies in the upper limbs, especially in children, lead to emotional stress, failure in manual functions related to ADLs, and a feeling of low self-confidence in the presence of peers and family, which ultimately may affect the life satisfaction and long-term well-being of children [13].
To date, the treatment of pediatric hand malformations has been controversial, and agreement on the gold standard for the treatment of this malformation in children has yet to be reached [14]. Although some hand deformities in children are currently treated with surgery, some researchers have considered success with conservative treatment, including the use of splints, rehabilitation, drug therapy, or the use of assistive technologies [15].
We assessed the effect of using splints on improving the hand function of children under 18 years old with all kinds of anomalies of the hands.
Information and Methods
We used the systematic evidence-based review process [16] and followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines for systematic review studies [17]. ProQuest, PubMed, Scopus, and EMBASE databases were searched using the following keywords: Orthosis, Splint, Brace, Hand, Upper extremity, Congenital Anomalies, Children, and Pediatric. The Boolean operators "AND" and "OR" were also used to combine and define the relationship between these search terms; for example, "Hand Orthosis AND children".
Inclusion and exclusion criteria
Articles included in this study were published in peer-reviewed journals, assessed congenital hand anomalies and orthotic intervention, consisted of hand anomalies based on the OMT classification system of hand anomalies, and were published in the English language from 1990 to 2023. The included articles showed the I, II, III, IV, and V levels of evidence based on the evidence-based model [16, 18]: Level I: randomized controlled trials (RCTs), systematic reviews, meta-analyses, level II: nonrandomized studies (cohorts and case controls) using two groups, level III: nonrandomized studies with one group (e.g., pre-test–post-test stage), level IV: descriptive studies that included the analysis of outcomes (e.g., single-subject design and case series), level V: case reports and expert opinion that included narrative literature reviews and consensus statements. We excluded theses, presentations, and conferences.
The search was undertaken between July 6, 2023, and August 5, 2023. Two researchers (the first author and the second independent researcher) searched and screened the articles individually based on titles, abstracts, and full-text examination. They found 344 articles and screened the articles together again, and after removing duplicate articles, 131 articles remained. These articles were screened by the first and second authors based on the eligibility criteria. The following articles were removed: assessing orthotic interventions in adults and older adults (n=48), the full text was not available (n=22), the language was other than English (n=16), assessing participants with other deformities rather than hand anomalies (n=25), and studies published before 1990 (n=7). Of the 13 remaining articles, 13 articles were found eligible by both authors (an agreement rate of 92%). To assess the agreement rate between the two researchers, the second author screened the articles and confirmed meeting the eligibility criteria for this study. Any disagreements were resolved by discussion with the second author.
Methodological quality of the included studies
The methodological quality of the included systematic reviews was assessed by the Assessment of Multiple Systematic Reviews (AMSTAR) checklist (Table 1). A score of one was recorded for each criterion present, with a total possible score of 11. The AMSTAR has good face and content validity [19]. The methodological quality of the included trials was evaluated by the Physiotherapy Evidence Database (PEDro) scale. This scale has 11 criteria, each scored as zero or one, resulting in a possible total score of 11. The interrater reliability of the PEDro scale is adequate [20]. Table 1 presents the results of the methodological quality assessment of the one and five included RCT and systematic review studies, respectively. All included trials had moderate-to-high quality with scores ≥5 on the PEDro scale. Also, all included reviews had moderate-to-high quality with scores ≥5 based on the AMSTAR scale.
Table 1. The methodological quality of the included systematic reviews using the AMSTAR checklist and randomized controlled trials using the PEDro Scale
Findings
Selection of articles
Among 13 selected articles, six cases were at evidence level I (one RCT and five reviews), two cases were at evidence level II, two cases were at evidence level III, one was at evidence level IV, and two cases were at evidence level V (Figure 1). The description of the included 13 studies is presented in Table 2.
Figure 1. PRISMA flow diagram.
Table 2. Results of the included articles (n=13)
Discussion
The purpose of this study was to review the effect of using upper limb splints on the hand function of children with various congenital hand anomalies. Based on the updated OMT classification, deformities are also classified as upper limb abnormalities [5]. In the present study, out of the 13 included studies, six studies were at evidence level I [22-27], two studies were at evidence level II [29, 34], two studies were at evidence level III [28, 30], one study was at evidence level IV [32], and two studies were at evidence level V [31, 33]. In most aforementioned studies, surgery was identified as the first line of treatment for congenital anomalies in these children, and the use of splints was considered a complementary treatment after surgery for children.
In the study by Zhou et al., surgery in children with recessive dystrophic epidermolysis bullosa (RDEB) greatly affected the children's hand function when immediately after the mentioned surgeries, the child used a splint [34]. Rogers et al. also stated that the first-line treatment in children with preaxial polydactyly is surgery. Surgery for children with preaxial polydactyly and timely prescription of splints, especially thumb spica splints, was presented as the most effective treatment method for these children [26]. However, Ho et al. regarding the first line of treatment in children with obstetric brachial plexus palsy (OBPP) suggested that serial casting and the use of splints can have the best effect, especially in young children. Contracture between 20° and 40°, as well as contracture of less than 20°, can achieve better results from splinting and serial casting [25]. Ho et al.in a case report assessed a 13-year-old girl with brachymetacarpia, for whom the combination of surgery and post-surgical interventions, including scar tissue massage, therapeutic exercises, and the use of splints were provided as complementary treatments. Night composite extension splint (a thermoplastic night splint) and anti-swan neck splint during the day after surgery showed the best outcomes for children with brachymetacarpia [33].
In the study by Lee et al., splinting was suggested as the first line of treatment for non-fixed trigger thumb in children under one year, and surgery for the first line of treatment for fixed trigger thumb in children [27]. Kennedy suggested that for congenital anomalies, such as radial club hand, the first-line treatment is the use of splints, especially the neoprene splint. Because babies welcome splints more at birth, flexibility in muscles and joints provides a good opportunity and possibility for treatment with splints. Surgery is recommended for children after three years who are fixed with a radial club hand [29].
Zucker et al. suggested that the first-line treatment for syndactyly correction of the hand in children with Apert Syndrome is surgery, which should be conducted when the child is six years old. After surgery, maintaining the correction using the static splint is necessary [28].
The use of splints as first-line or maintenance therapy for better surgical outcomes in children with arthrogryposis multiplex congenital (AMC) has been supported by multiple studies [23, 24, 31]. O'Flaherty considered surgery the first-line treatment in AMC children and mentioned that stretching, serial casting, and splints as post-surgical maintenance interventions to increase the range of motion in the joints [23]. Smith and Drennan [24] and Katić et al. [31] reported that serial casting and splinting increase the range of motion in the joints and are the first-line treatment in AMC infants. Nevertheless, it can be said that in newborns with AMC, it is better to try to increase the range of motion of the joints with interventions, such as serial casting and splinting, and prevent fibrosis of the joints, and in older children up to six months and above, interventions, like surgery is recommended.
Conclusion
Based on the results of the present study, it can be said that the use of splints alone cannot be an effective solution for the function of the upper limbs of children with a variety of congenital hand disorders and abnormalities. The use of splints as the first line of congenital hand anomalies in children can be useful if prescribed during infancy and in some congenital anomalies, such as thumb and radial hand. Splinting is most effective when used in conjunction with other evidence-based interventions, such as surgery, and evidence-based interventions in rehabilitation. The best treatment for children with hand anomalies is surgery and timely prescription of splint use.
Acknowledgments: We are grateful to the officials of the School of Rehabilitation Sciences at Shahid Beheshti University of Medical Sciences and those who helped us with this research.
Ethical Permission: This is a review article with no human or animal sample.
Conflicts of Interests: The authors declared no conflicts of interests.
Authors' Contribution: Pahmdarfard M (First Author), Introduction Writer/Methodologist/Original Researcher (50%); Kalantari M (Second Author), Assistant Researcher/Discussion Writer/Statistical Analyst (50%)
Funding/Support: This research did not receive any grant from funding agencies in the public, commercial, or non-profit sectors.
Hand anomaly can be congenital or acquired as a result of neurological damage to the central nervous system during the childhood process [1]. Most hand deformities in children are known as a congenital disorder, or an abnormal condition that is present at birth regardless of its cause [2]. These abnormalities may lead to structural disorders, in which problems are seen in the appearance of a body part, or functional disorders, in which there are problems in the way a part of the body functions [3]. However, the above-mentioned classification has been updated over several periods, and the last updated classification is related to 2020 [4].
In 2010, for the first time, a new classification for upper limb congenital malformations was published under the title of Oberg-Manske-Tonkin (OMT) [5]. Based on a malformation framework, OMT is accepted by the International Federation of Hand Surgery Society and surgeons treating congenital anomalies of the upper extremities. According to the updated OMT classification, upper extremity abnormalities fall into one of the categories of malformations, deformities, dysplasias, and syndromes [6].
Congenital malformations and anomalies of the upper limb, with a reported overall prevalence of 21.5 out of 10,000 live births, may have serious consequences for any patient [7]. The causes of congenital abnormalities in children are unknown in 60% of cases [8]. The greatest effect of congenital anomalies is observed in the fingers, followed by the forearm area, and finally, in the upper parts, such as the arm [9].
The International Classification of Functioning, Disability, and Health (ICF) provides a framework that describes the interrelationship between body function and structure, activities, and participation, as well as the influence of environmental and personal factors [10]. In the meantime, hand function in children with congenital anomalies of the upper limb can lead to a vicious cycle in body structure, function, activity, or participation in different areas of life. However, early and timely interventions in this vicious cycle can create hope to break this vicious cycle and promote children's participation [11].
Performing activities of daily living (ADL) requires the hands to perform different movements in a coordinated manner at the same time [12]. In the meantime, congenital anomalies in the upper limbs, especially in children, lead to emotional stress, failure in manual functions related to ADLs, and a feeling of low self-confidence in the presence of peers and family, which ultimately may affect the life satisfaction and long-term well-being of children [13].
To date, the treatment of pediatric hand malformations has been controversial, and agreement on the gold standard for the treatment of this malformation in children has yet to be reached [14]. Although some hand deformities in children are currently treated with surgery, some researchers have considered success with conservative treatment, including the use of splints, rehabilitation, drug therapy, or the use of assistive technologies [15].
We assessed the effect of using splints on improving the hand function of children under 18 years old with all kinds of anomalies of the hands.
Information and Methods
We used the systematic evidence-based review process [16] and followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines for systematic review studies [17]. ProQuest, PubMed, Scopus, and EMBASE databases were searched using the following keywords: Orthosis, Splint, Brace, Hand, Upper extremity, Congenital Anomalies, Children, and Pediatric. The Boolean operators "AND" and "OR" were also used to combine and define the relationship between these search terms; for example, "Hand Orthosis AND children".
Inclusion and exclusion criteria
Articles included in this study were published in peer-reviewed journals, assessed congenital hand anomalies and orthotic intervention, consisted of hand anomalies based on the OMT classification system of hand anomalies, and were published in the English language from 1990 to 2023. The included articles showed the I, II, III, IV, and V levels of evidence based on the evidence-based model [16, 18]: Level I: randomized controlled trials (RCTs), systematic reviews, meta-analyses, level II: nonrandomized studies (cohorts and case controls) using two groups, level III: nonrandomized studies with one group (e.g., pre-test–post-test stage), level IV: descriptive studies that included the analysis of outcomes (e.g., single-subject design and case series), level V: case reports and expert opinion that included narrative literature reviews and consensus statements. We excluded theses, presentations, and conferences.
The search was undertaken between July 6, 2023, and August 5, 2023. Two researchers (the first author and the second independent researcher) searched and screened the articles individually based on titles, abstracts, and full-text examination. They found 344 articles and screened the articles together again, and after removing duplicate articles, 131 articles remained. These articles were screened by the first and second authors based on the eligibility criteria. The following articles were removed: assessing orthotic interventions in adults and older adults (n=48), the full text was not available (n=22), the language was other than English (n=16), assessing participants with other deformities rather than hand anomalies (n=25), and studies published before 1990 (n=7). Of the 13 remaining articles, 13 articles were found eligible by both authors (an agreement rate of 92%). To assess the agreement rate between the two researchers, the second author screened the articles and confirmed meeting the eligibility criteria for this study. Any disagreements were resolved by discussion with the second author.
Methodological quality of the included studies
The methodological quality of the included systematic reviews was assessed by the Assessment of Multiple Systematic Reviews (AMSTAR) checklist (Table 1). A score of one was recorded for each criterion present, with a total possible score of 11. The AMSTAR has good face and content validity [19]. The methodological quality of the included trials was evaluated by the Physiotherapy Evidence Database (PEDro) scale. This scale has 11 criteria, each scored as zero or one, resulting in a possible total score of 11. The interrater reliability of the PEDro scale is adequate [20]. Table 1 presents the results of the methodological quality assessment of the one and five included RCT and systematic review studies, respectively. All included trials had moderate-to-high quality with scores ≥5 on the PEDro scale. Also, all included reviews had moderate-to-high quality with scores ≥5 based on the AMSTAR scale.
Table 1. The methodological quality of the included systematic reviews using the AMSTAR checklist and randomized controlled trials using the PEDro Scale
Findings
Selection of articles
Among 13 selected articles, six cases were at evidence level I (one RCT and five reviews), two cases were at evidence level II, two cases were at evidence level III, one was at evidence level IV, and two cases were at evidence level V (Figure 1). The description of the included 13 studies is presented in Table 2.
Figure 1. PRISMA flow diagram.
Table 2. Results of the included articles (n=13)
Discussion
The purpose of this study was to review the effect of using upper limb splints on the hand function of children with various congenital hand anomalies. Based on the updated OMT classification, deformities are also classified as upper limb abnormalities [5]. In the present study, out of the 13 included studies, six studies were at evidence level I [22-27], two studies were at evidence level II [29, 34], two studies were at evidence level III [28, 30], one study was at evidence level IV [32], and two studies were at evidence level V [31, 33]. In most aforementioned studies, surgery was identified as the first line of treatment for congenital anomalies in these children, and the use of splints was considered a complementary treatment after surgery for children.
In the study by Zhou et al., surgery in children with recessive dystrophic epidermolysis bullosa (RDEB) greatly affected the children's hand function when immediately after the mentioned surgeries, the child used a splint [34]. Rogers et al. also stated that the first-line treatment in children with preaxial polydactyly is surgery. Surgery for children with preaxial polydactyly and timely prescription of splints, especially thumb spica splints, was presented as the most effective treatment method for these children [26]. However, Ho et al. regarding the first line of treatment in children with obstetric brachial plexus palsy (OBPP) suggested that serial casting and the use of splints can have the best effect, especially in young children. Contracture between 20° and 40°, as well as contracture of less than 20°, can achieve better results from splinting and serial casting [25]. Ho et al.in a case report assessed a 13-year-old girl with brachymetacarpia, for whom the combination of surgery and post-surgical interventions, including scar tissue massage, therapeutic exercises, and the use of splints were provided as complementary treatments. Night composite extension splint (a thermoplastic night splint) and anti-swan neck splint during the day after surgery showed the best outcomes for children with brachymetacarpia [33].
In the study by Lee et al., splinting was suggested as the first line of treatment for non-fixed trigger thumb in children under one year, and surgery for the first line of treatment for fixed trigger thumb in children [27]. Kennedy suggested that for congenital anomalies, such as radial club hand, the first-line treatment is the use of splints, especially the neoprene splint. Because babies welcome splints more at birth, flexibility in muscles and joints provides a good opportunity and possibility for treatment with splints. Surgery is recommended for children after three years who are fixed with a radial club hand [29].
Zucker et al. suggested that the first-line treatment for syndactyly correction of the hand in children with Apert Syndrome is surgery, which should be conducted when the child is six years old. After surgery, maintaining the correction using the static splint is necessary [28].
The use of splints as first-line or maintenance therapy for better surgical outcomes in children with arthrogryposis multiplex congenital (AMC) has been supported by multiple studies [23, 24, 31]. O'Flaherty considered surgery the first-line treatment in AMC children and mentioned that stretching, serial casting, and splints as post-surgical maintenance interventions to increase the range of motion in the joints [23]. Smith and Drennan [24] and Katić et al. [31] reported that serial casting and splinting increase the range of motion in the joints and are the first-line treatment in AMC infants. Nevertheless, it can be said that in newborns with AMC, it is better to try to increase the range of motion of the joints with interventions, such as serial casting and splinting, and prevent fibrosis of the joints, and in older children up to six months and above, interventions, like surgery is recommended.
Conclusion
Based on the results of the present study, it can be said that the use of splints alone cannot be an effective solution for the function of the upper limbs of children with a variety of congenital hand disorders and abnormalities. The use of splints as the first line of congenital hand anomalies in children can be useful if prescribed during infancy and in some congenital anomalies, such as thumb and radial hand. Splinting is most effective when used in conjunction with other evidence-based interventions, such as surgery, and evidence-based interventions in rehabilitation. The best treatment for children with hand anomalies is surgery and timely prescription of splint use.
Acknowledgments: We are grateful to the officials of the School of Rehabilitation Sciences at Shahid Beheshti University of Medical Sciences and those who helped us with this research.
Ethical Permission: This is a review article with no human or animal sample.
Conflicts of Interests: The authors declared no conflicts of interests.
Authors' Contribution: Pahmdarfard M (First Author), Introduction Writer/Methodologist/Original Researcher (50%); Kalantari M (Second Author), Assistant Researcher/Discussion Writer/Statistical Analyst (50%)
Funding/Support: This research did not receive any grant from funding agencies in the public, commercial, or non-profit sectors.
Keywords:
References
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