Treatment of Supination Deformity for Obstetric Brachial Plexus Injury: A Systematic Review and Meta-Analysis

Treatment of Supination Deformity for Obstetric Brachial Plexus Injury: A Systematic Review and Meta-Analysis

SCIENTIFIC ARTICLE Treatment of Supination Deformity for Obstetric Brachial Plexus Injury: A Systematic Review and Meta-Analysis W. P. Metsaars, MD, ...

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SCIENTIFIC ARTICLE

Treatment of Supination Deformity for Obstetric Brachial Plexus Injury: A Systematic Review and Meta-Analysis W. P. Metsaars, MD, J. Nagels, MD, B. G. Pijls, MD, PhD, J. M. Langenhoff, R. G. H. H. Nelissen, PhD, MD

Purpose To conduct a systematic review and meta-analysis of the literature evaluating treatment outcome for supination deformity in obstetric brachial plexus injury. Methods We included studies on brachial plexus and supination deformity with follow-up of at least one year and quantitative measurements of forearm mean passive pronation and position at rest. Meta-analysis was used to explore modifying factors. Results An elaborate search strategy resulted in 366 studies, of which 13 were included totaling 238 patients (157 osteotomies and 71 biceps rerouting). There was a 75 gain in position at rest and a 65 gain in passive pronation for the osteotomy group, compared to a 79 gain in position at rest for the biceps rerouting group. More severe deformities had greater gains. No influence of age was found. Important adverse effects were hardware failure and a biceps rupture. Recurrence in the osteotomy group was 20% to 40%, versus none in the soft-tissue group. Conclusions This review showed an overall benefit for forearm osteotomies and for biceps rerouting for supination deformity with the gain proportionate to the severity of the deformity. (J Hand Surg Am. 2014;39(10):1948e1958. Copyright Ó 2014 by the American Society for Surgery of the Hand. All rights reserved.) Type of study/level of evidence Therapeutic III. Key words Plexus brachialis, obstetric palsy, supination deformity, osteotomy, biceps rerouting.

T

HE INCIDENCE OF OBSTETRIC brachial plexus injury varies from 0.4 to 5.1 per 1000 live births in various countries.1 The majority of these injuries are mild, and spontaneous functional recovery will occur in about 70% of patients within

From the Department of Orthopaedics and Walaeus Library, Leiden University Medical Center, Leiden, the Netherlands. Received for publication October 14, 2013; accepted in revised form June 1, 2014. No benefits in any form have been received or will be received related directly or indirectly to the subject of this article. Corresponding author: W. P. Metsaars, Department of Orthopaedics, Leiden University Medical Center, Postzone J-11-R, Postbus 9600, 2300 RC Leiden, the Netherlands; e-mail: [email protected] 0363-5023/14/3910-0009$36.00/0 http://dx.doi.org/10.1016/j.jhsa.2014.06.003

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4-6 months. The remaining 30% are left with functional deficits2 that depend on the severity of the injury (axonotmesis, neurotmesis, avulsion) and on which roots were involved (C5-T1). Patients with residual deficits can develop a rotation deformity of the forearm, predominantly a supination deformity.3 This deformity may be secondary to complete or partial paralysis of the forearm pronator muscles innervated by the median nerve (C6-8), combined with unopposed supinator and biceps muscle strength (radial and musculocutaneous nerves, respectively [C5-6]).4,5 Aitkin proposed that bowing of the ulna in association with this muscle imbalance as a result of biceps overpower adversely affects rotation of the forearm.6 However, the deformity also could be the result of shortening of the biceps and supinator

TREATMENT OF SUPINATION DEFORMITY IN OBPI

fields] OR transposition[all fields] OR transpositions [all fields]))

independent of muscle imbalance, similar to elbow contractures shown in mouse models.7 The incidence of supination deformity occurs in 6% to 10% of children with residual brachial plexus birth injuries.8,9 Although the deformity starts as a dynamic deficit, it progresses to a fixed supination contracture as interosseous membrane stiffness, muscle shortening, and joint stiffness increases.10 The posture in forearm supination presents serious functional disabilities. The affected limb cannot be properly positioned for grasping and cannot effectively assist in a range of 2-handed activities. A neutral or pronation position of the forearm may result in marked improvement of the overall upper extremity functionality provided there is sufficient hand function.11 Reported treatments include radioulnar arthrodesis, forearm osteotomies of radius and/or ulna, biceps or brachioradialis rerouting, interosseous membrane release, tendon transfers, and a combination of these.12 Osteotomies of one or both forearm bones alone might lead to recurrence because muscle imbalance is not corrected. Biceps rerouting is performed in mild cases of deformity without contracture and may therefore have better postoperative functional results and less recurrence. Extensive soft-tissue procedures stimulate the formation of fibrosis and may subsequently diminish active range of motion. Better understanding of the factors predicting outcome might improve recurrence rates and overall functionality of the upper extremity. To this end, we reviewed all literature on residual brachial plexus injury patients with supination deformity who were surgically treated by osteotomy and/or soft-tissue procedure and had a follow-up of at least one year. We focused on gain of pronation by surgical intervention and evaluated age and severity of deformity as possible influences on outcome.

Study selection The abstracts available after the search strategy were reviewed for the possibility of inclusion by title and abstract by 2 independent reviewers (W.M. and J.N.) according to the inclusion criteria of the topic of brachial plexus and supination deformity, follow-up of at least one year, and quantitative measurements of rotation of the forearm. If no consensus could be made to include the study by reviewing title and abstract, the entire article was read. If doubt existed whether to include each article after reading the abstract and, if necessary, the entire article, the 2 reviewers agreed after a discussion in consensus. A third reviewer (R.G.H.H.N.) resolved any remaining disputes. Data extraction Data were extracted independently by the 2 reviewers using a data collection sheet, including authorship, year of publication, number of obstetric brachial plexus injury subjects in the study, age, surgery performed, quantitative indications and outcomes of rotation of the forearm, time points for outcome measurements, complications, and recurrence. When included studies reported nonparametric data (eg, median), mean and standard deviation values were calculated according to internationally accepted guidelines or the standard deviation was estimated according to the weighted variance method.13 Summary measures and synthesis of results Since there is no functional gain to be expected in the natural history of supination deformity, we assumed that all postoperative gain in function was due to the surgical intervention. The meta-analysis was based on the assumption that each patient could be considered to be his or her own control. All data were combined for meta-analysis with the random-effects model for risk differences and the pooled standard error for mean differences. Heterogeneity between studies was tested with the I2 statistic, which describes the variation across studies due to heterogeneity. Possible sources of heterogeneity were explored with metaregression using the random-effects regression model that previously has been used to study the effectiveness of the bacillus CalmetteeGuérin vaccine against tuberculosis.14 This model searches for modifying variables that affect the outcome of interest between studies and therefore can help resolve contradictory outcomes of different studies, as was the case with the bacillus CalmetteeGuérin vaccine. In the present

METHODS Literature search A search was performed of all available evidence in PubMed, MEDLINE, and Web of Science from 1966 to 2012. All English, French, German, and Italian literature was included. The search terms contained: PubMed: 1. “brachial plexus”[MeSHTerms] OR (“brachial”[All Fields] OR “brachialis”[All Fields]) AND “plexus”[All Fields]) OR “brachial plexus neuropathies”[Mesh] OR “paralysis, obstetric”[Mesh] OR “obstetrical paralysis”[all fields]. 2. “osteotomy”[MeSHTerms] OR “osteotomy”[All Fields] OR “osteotomies”[All Fields] OR ((biceps[all fields] OR brachioradialis[All Fields]) AND (rerouting[all J Hand Surg Am.

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study we focused on patient-specific variables (age and preoperative deformity) that could influence the gain/increase in position at rest or passive pronation.

rerouting with interosseus membrane release, or osteotomy is shown in Table 4. Mean preoperative position at rest was respectively 69 , 62 , and 63 . Mean passive pronation was 48 for biceps rerouting, 12 for biceps rerouting with interosseous membrane release, and 2 for forearm osteotomy. Complications were noted in 8 studies. Three nonunions were reported: one case after ulnar osteotomy, which was not fixated with plate; one case of ulna nonunion after both bone osteoclasis; and one case of middistal radius osteotomy, which was fixated by a plate. Two delayed unions were reported in plate-fixated middistal radius osteotomies. There were 7 hardware failures, one pronation contracture after osteotomy, and 2 synostoses. After biceps rerouting, one patient had a biceps tendon rupture and 2 had scar formation. No cases of nerve lesions or compartment syndrome were reported. A PRISMA research checklist is available in Appendix C (available on the Journal’s Web site at http://www.jhandsurg.org).

Quality assessment As there is no formal quality measure available in the literature to appraise the methodological quality case series/cohorts of brachial plexus patients, quality assessment was focused on items relevant to identify selection bias based on the methodological index for nonrandomized studies.15 This index constitutes 7 items: study aim, consecutive patients, prospective collection of data, appropriate end point, unbiased evaluation of end points, appropriate follow-up, and follow-up loss not exceeding 5%. RESULTS A total of 366 articles were extracted from the literature (Appendix A, available on the Journal’s Web site at http://www.jhandsurg.org).16 After we read the title and abstract of each article, 32 studies concerning supination deformity were included for further assessment (Table 1). After we read the fulltext article, another 19 studies were excluded from further analysis. Eleven excluded studies did not include pronation measurements at follow-up or had no recorded follow-up for at least one year.8,17e26 Eight excluded studies described modifications or preferred surgical treatment without quantitative patient data.2,5,27e32 In 13 studies with a total of 238 patients, quantitative measurements of postoperative forearm rotation were presented and follow-up was at least one year. Assessment of study quality is shown in Appendix B (available on the Journal’s Web site at http://www.jhandsurg.org). Ten studies evaluated forearm osteotomies,4,9,10,33e39 and 6 studies evaluated biceps rerouting.4,33,39e42 Articles describing both osteotomy and biceps rerouting were analyzed according to surgical procedure. Osteotomies with different techniques were performed in 157 included cases (Table 2). Soft-tissue procedures were performed in 71 included patients (Table 3). Indications for surgery varied between studies. The indications for forearm osteotomy were a passive pronation deficit with a preoperative passive pronation ranging from 11 to 59 . The position at rest ranged from 20 to full supination. Biceps rerouting was indicated to treat absence of active pronation beyond neutral, or 47 to full supination position at rest, with 48 of passive pronation. Interosseus membrane release was added to biceps rerouting in the presence of passive pronation ranging from 7 to 16 . A summary of mean indications for biceps rerouting alone, biceps J Hand Surg Am.

Meta-analysis Osteotomy: After osteotomy a 65 average gain in passive pronation was shown with meta-analysis in 5 studies including 68 patients (95% CI 52 to 77 ; P < .001) (Fig. 1). For every 10 of reported preoperative deformity, the gain in passive pronation increased by 6 (95% CI 0.2 to 1 ; P ¼ .14). Clinical outcome showed that 3 studies had a mean passive pronation of more than 30 and 2 of more than 50 after osteotomy (Fig. 2). A 75 average gain in position at rest after osteotomy was shown with meta-analysis on 6 studies including 139 patients (95% CI 64 to 86 ; P < .001) (Fig. 1). The results of the meta-regression indicated that preoperative deformity greatly influenced the postoperative gain in position at rest. For every 10 greater preoperative deformity, the gain in position at rest increased by 7 (95% CI 2 to 12 ; P ¼ .01). Clinical outcome showed that 2 studies had a position at rest of more than 10 of supination and 4 studies had a neutral or pronation position at rest after osteotomy (Fig. 2). No influence of age was found in these analyses. Biceps rerouting: After biceps rerouting a 79 average gain in position at rest was shown with meta-analysis in 4 studies that included 50 patients (95% CI 67 to 91 ; P < .001) (Fig. 1). The results of the metaregression indicated that preoperative deformity greatly influenced the postoperative gain in position at rest. For every 10 greater preoperative deformity, the gain in position at rest increased by 6 (95% CI 2 to 10 ; P ¼ .003). Clinical outcome showed that all studies had a neutral or pronation position at rest after r

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TABLE 1.

All Retrieved Articles Concerning Surgical Procedures for Supination Deformity Author, Year

N 17

Maillet and Romana (2009) 18

Nath et al (2009)

10

Prospective

Age (y) at Intervention

Follow-Up (mo)

4

N/A

Procedure Radius OT and tendon transfer

Prospective

10

16

Radius OT

Rolfe et al (2009)36

11*

Retrospective

11

26

Distal ulna and midradius OT

Ruhmann and Hierner (2009)41

11*

Retrospective

6

36

Bicepsrerouting and membrana interosseus release

Yam et al (2009)9

42*

Prospective

7

60

Middistal radius OT

9

23

Kooten et al (2008)

5

Study Type

37

Retrospective

0

Descriptive

Hankins et al (2006)10

9*

Retrospective

Kozin (2006)32

0

Descriptive

N/A 10

16

N/A

Allende and Gilbert (2004)33

66*

Retrospective

6

Brunelli (2004)19

21

Middistal radius OT OT and biceps rerouting Proximal ulna and distal radius OT Proximal ulna and distal radius OT/biceps rerouting

67

Middistal radius OT/biceps rerouting/ þ/- membrana interosseus release

r

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Retrospective

N/A

4*

Retrospective

8

Ruhmann et al (2004)

3

Descriptive

N/A

Ulna OT/distal radioulnar artrodesis

Bahm (2004)30

0

Descriptive

N/A

Overall treatment plexus

Ozkan et al (2004)40 21

4

Bahmn and Gilbert (2002)

7

1

Case report

3

9

Retrospective

15

Waters (1997)2

0

Overall review

N/A

Lin et al (1995)34

4*

Prospective

7

96

Retrospective

7

> 24

Proximal ulna and distal radius OT þ/- membrana interosseus release Biceps rerouting, membrana interosseus release, proximal and distal radioulnair capsulotomy

Eberhard (1997)

22

Lipskeir and Weizenbluth (1993)35

10*

48

Brachioradialis and membrana interosseus release

Retrospective

Ruhmann et al (2001)20

40*

Oblique radius OT 27

Middistal radius OT/percutaneous osteoclasis/biceps rerouting Distal ulna OT and humeral OT

30

Biceps tuberositas transposition membrana interosseus release, reinsertion supinator or pronator teres Overall treatment plexus Distal radius and proximal ulna osteoclasis, 10 days rotate

Gilbert (1991)5

0

Descriptive

N/A

Martini (1986)23

12

Retrospective

N/A

N/A

Manfrini and Valdiserri (1985)24

19

Retrospective

10

96 (n ¼ 14)

Supinator transposition-artrolysis-membrana interosseus release-2x radioulnar arthrodesis

9

Retrospective

14

24 (n ¼ 5)

Distal radius OT

Romanini et al (1983)25

TREATMENT OF SUPINATION DEFORMITY IN OBPI

J Hand Surg Am.

8*

D’Emilio (2006)31

1951

(Continued)

1952

8

38

Osteoclasis

DISCUSSION Supination deformity can develop after obstetric brachial plexus injury, and the incidence is higher with more severe neurological lesions. Depending on the severity of the brachial plexus injury, the incidence ranges from 7% in patients with mild neurological deficits to 23% in the more severely involved Narakas group IV.9,43 All surgical interventions led to a significant clinical gain. The greater the preoperative deformity, the greater was the postoperative gain. This reached significance for the position at rest after osteotomy and after biceps rerouting. Hence, the results of our systematic review underscore an overall better pronation after either of the performed surgical procedures.2,4,5,8e10,17e37,39e41 Whether a surgical procedure is also creating a functional benefit is only partly clarified from the reviewed studies. The importance of functional acquired motion of the upper extremity for daily activities has been previously described. A mean rotation arc from 30 of pronation to 20 of supination and a mean elbow flexion arc between 38 and 135 were needed for activities of daily living.11 Only one included study presented the active pronation at baseline, whereas others only mention position at rest or with passive pronation. Most authors did not claim significant improvement of active pronation after any procedure, and this should not be the goal of surgery. The position of the forearm in a functionally pronated position seems more important than active motion for the functionality of the hand, which has been confirmed by an improvement of wrist and finger extension33 and by an improvement of Raimondi score after surgery.9 The rate of recurrence was mentioned in 7 of the reviewed studies. It varied from 20% to 42% in osteotomies, whereas for biceps rerouting no recurrences were reported. Although both groups of patients had similar supinated positions at rest, passive pronation was more affected in the osteotomy group. Confounding in all articles existed, since recurrence was not clearly defined in any. Furthermore, only one study reported on the time of recurrence (median 3 years, with a range of 1e8 years). Some authors discussed the value of adding biceps rerouting to a forearm osteotomy in the presence of severe cases of supination deformity to prevent recurrences.2,33 This

N, number of patients with supination deformity; OT, osteotomy. *Inclusion in the quantitative assessment and meta-analysis.

Retrospective 4*

338

Blount (1962)38

Total

Biceps rerouting and membrana interosseus release

Proximal radius OT þ/- membrana interosseus release 6 to 36

38 10

7 Retrospective

Retrospective 8*

6

Biceps rerouting

Overall treatment plexus

biceps rerouting (Fig. 2). No influence of age was found in these analyses. There was a nonsignificant difference between osteotomy and biceps rerouting, with an average of 6 more gain in position at rest for the biceps rerouting group (95% CI 4 to 15, P ¼ .24).

Zancolli (1967)42

Zaoussis (1963)

8

5

N/A N/A

N/A Owings et al (1971)26

Descriptive 0

15

Masse (1972)29

Descriptive

Overall treatment plexus

Biceps rerouting/osteoclasis

Overall treatment plexus N/A

N/A N/A

N/A Overall review

Descriptive 0

0

Follow-Up (mo)

39 6 Retrospective 11*

Mallet (1972)28

Eng et al (1978)

27

39

Age (y) at Intervention Study Type N Author, Year

Manske et al (1980)

TABLE 1.

All Retrieved Articles Concerning Surgical Procedures for Supination Deformity (Continued)

Procedure

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TABLE 2.

Forearm Osteotomy With Follow-Up > 1 Year and Quantative Follow-Up Measurements Mentioned Preoperative ( )

Author (y)

N

Age (y)

Follow-Up (y)

Procedure

Position at Rest

Passive

Intraoperative ( )

Postoperative ( ) Position at Rest

Passive

Recurrence 17 (40%)

Yam (2009)

42

7 [3;14]

5 [1;15]

dR

90

0 [30;90]

88,5 [120-0]

N/A

70 [100;0]

Rolfe (2009)

11

11 [6;14]

2,2 [0,8;6,9]

dR, dU

76 [35;90]

N/A

N/A

26 [0;60]

N/A

N/A

9 [4;13]

1,9 [0,6;4,3]

dR

20 [30;0]

19 [30;10]

49 [20;90]

8 [0;45]

46 [0;90]

0

9

10 [6;16]

1,3 [0,5;2,8]

dR, pU

70 [45;90]

N/A

81 [45;120]

3 [15;25]

N/A

2 (22%)

Allende (2004)

44

6

5.3

dR

59  20,4

N/A

24  15,4

2  23,3

N/A

9 (20%)

Bahm (2002)

23

7 [2;20]

4 [1;12]

dRþ4pU**

90

N/A

29 [0;70]

17

N/A

7 (30%)

4

6 [3;11]

11 [2,4;20,3]

dR, pU**

N/A

59 [15;90]

34 [0;45]

N/A

36 [25;90]

0

Lipskeir (1993)

10

7 [3;12]

7,3 [3;12]

dR, pU

81 [70;90]

11 [90;0]

N/A

6 [10;20]

46 [20;90]

0$

Manske (1980)

2

6 [3;9]

6.7 [6.3;7]

dR, pU*

90 [90;90]

25 [20;30]

N/A

30 [20;40]

N/A

4

8 [4;12]

3.1 [0,5;4]

dR, pU**

N/A

6 [45;30]

85 [70;90]

N/A

55 [40;70]

Blount (1940) Total

ˇ

Lin (1995)

ˇ

N/A 1 (25%)

157

Mean pronation in degree  std or range between [ ]. If range not noted, not available. Position at rest indicates the rotational position of the forearm when no active or passive movement is applied to the arm. A minus indicates supination. Intraoperative degree of pronation indicates the maximum passive pronation during surgery. Postoperative pronation indicates the pronation at last follow up. d, distal; N, number of patients with supination deformity in obstetric brachial plexus injury in the mentioned study; p, proximal; R, radiusosteotomy; U, ulna osteotomy. *Percutaneous osteoclasis performed in 2 cases. **Percutaneous osteoclasis performed in 4 cases. 2 cases additional biceps rerouting. $One case of synostosis in 45 of pronation and one additional biceps rerouting. ˇ

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8

Hankins (2006)

ˇ

r

Kooten (2008)

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Passive

1953

1954

16 [10;30] 60 [30;90] BR, IMR 3,1 [1;5]

9

8

71

Zancolli (1967)

Total

10 [5;23]

17

Manske (1980)

rerouted biceps then works as a pronator and diminishes and counteracts the supinator forces of the forearm rotators, which was one of the factors causing the initial deformity.2,9 However, the authors did not show patient data on the combined procedure, and conclusions could not be drawn. In cases with partial pronation deficit, one might advocate biceps rerouting with interosseous membrane release. Stiffness and shortening of the interosseous membrane have important roles in the development of the fixed supination contracture.10 Although an interosseous membrane release is a difficult procedure requiring meticulous technique, no recurrences after rerouted biceps combined with interosseus membrane release have been reported.33,41 Based on the meta-analysis and cumulative expert opinion recommendations derived from the included articles, some considerations should be made. Supination position at rest was similar at baseline in biceps rerouting, biceps rerouting with interosseous membrane release, and osteotomy. As such, pronation at rest does not differentiate patients in the different treatment groups. Although the position at rest is a good tool to record improvements of the rotational position at follow-up, it is less suitable to guide the clinician to a treatment strategy. On the contrary, passive pronation at baseline was different for the 3 indications. Although the data on passive pronation are based on few studies, the literature suggests that the different indications for surgery were based on the extent of passive pronation deficit. As surgery significantly improved pronation at rest for both biceps rerouting and osteotomy, we assume that the mentioned indications in the literature can be used in guiding the clinician. However, as there are so little data in the literature, the clinician should always individualize the approach based on clinical findings. Furthermore, experts have suggested treating shoulder deformities before treating supination contracture.2,33 An internally rotated limb that is surgically corrected into a pronation position gains less in overall functionality, as active external rotation is needed for grasping, typing, and writing. Also, a lack of active wrist extension leads to less functionality of the hand after correction of a supination contracture.2,33 The absence of wrist extension in patients with C7 lesions can lead to a dropped hand posture when the forearm is pronated. None of these studies addressed the optimal age for treating supination contractures. Expert opinion suggested that the optimal time was between 6 and 8 years, mainly because of better compliance and functional benefit at this age possibly associated with

Mean pronation in degree  std or range between [ ]. If range not noted, not available. Rest position indicates the rotational position of the forearm when no active or passive movement is applied to the arm. A minus indicates supination. Postoperative values indicate the pronation at last follow up. In none of the studies passive pronation was mentioned at follow-up. BR, biceps rerouting; BRR, brachioradialis rerouting; IMR, interosseus membrane release; N, number of patients with supination deformity in obstetric brachial plexus injury in the mentioned study.

14 [0;50]

6 [0;20]

0 “some”

11 [0;45] 0 [0;0]

22 none

none 48 [0;90]

48 [0;90]

90 BR

BR 4

BR,12x IMR

Bahm (2002)

7

2.5 [0.8;6.8]

49 [30;75] 18  22.4 35 [20;70] N/A, “limited” 2.25 [1;3.3]

6

4 Ozkan (2004)

8 [5;9]

22 Allende (2004)

5.8

BRR, IMR

47  22.8

7 [0;20] 81 [60;90] BR,IMR 3 [0,8;4,6] 6 [4;11] 11 Ruhmann (2009)

5.7 [3;10]

0

0 15 [10;50] 6 [20;30]

Active Position at Rest Active

Preoperative ( )

Passive Position at Rest Procedure Follow-Up (y) Age (y) N Author (y)

TABLE 3.

Soft Tissue Procedures With Follow-Up > 1 Year and Quantative Follow-Up Measurements Mentioned

Postoperative ( )

Recurrence

TREATMENT OF SUPINATION DEFORMITY IN OBPI

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TABLE 4.

Indications for Surgery on Supination Deformity Mean

SD

Patients

Studies

95% CI

Recurrence (Mean,%)

Follow-Up (Mean, Year)

0

4.3

0

4.0

19

4.1

Biceps rerouting Age (y)

6.0

Position in rest ( ) Passive pronation ( )

1.8

36

3

5.2 to 6.8

69

30.4

19

2

76 to 61

48

32.8

9

1

26 to 69

3.2

35

4

5.8 to 8.1

32.6

31

3

74 to 51

Biceps rerouting and interosseus membrane release Age (y)

7.0

Position in rest ( ) Passive pronation ( ) Active pronation ( )

62 12

20

2

9 to 15

20.6

4

1

55 to 15

3.0

157

10

7.0 to 8.0

63

23.7

149

8

66 to 61

2

28.1

70

6

35

6.36

Radius and ulna osteotomy, or both Age (y)

7.5 

Position in rest ( ) Passive pronation ( )

7 to 2

Position at rest and age at baseline was not different in the 3 groups. CI, confidence interval; patients, number of included patients; SD, standard deviation; studies, number of included studies.

a better outcome.30 We could not confirm influence of age in our review. As patient numbers were small, no studies have focused on a particular age, and consequently our results were the cumulative mean of all age groups. In the face of imbalance of supinators and pronators, in younger children with potentially more time for growth and remodeling, an osteotomy alone might have a higher recurrence rate because of persistent supinator overpower (mainly the biceps), which also was a cause of the deformity. Bowing of the ulna with rotational deficit by remodeling through unopposed biceps power is one of the causes of rotational deficits.6 Also, no recurrence was present in a cohort of 109 adults with traumatic brachial plexus injury treated with forearm osteotomy for supination deformity. These results indicate that the absence of growth reduces the risk of recurrence.21 Several authors have proposed adding biceps rerouting in young children, especially those with severe pronation deficits.2,9,33,41 Although we were not able to show differences with age, young age might predispose to recurrences by remodeling after osteotomy and should be taken into account when a young child presents with supination contracture. As randomized trials or comparable large cohort studies for this condition are absent, we have been restricted to a summary of the published data. Due to the relative low prevalence of supination deformity in obstetric brachial plexus injuries, this condition will probably only be amenable for case studies or small J Hand Surg Am.

series studies. The latter stresses even more the importance of evaluating the cumulative experience of current available information in literature (ie, >200 patients). This enables better decision-making for the complex deformities in these children compared to a treatment policy based on low-volume personal experience of surgeons. Since obstetric brachial plexus injury is rare and these secondary sequelae are even far less frequent, a systematic approach is essential to gain functionality for the child. Although our meta-analysis did not definitively identify the treatment of choice concerning location of osteotomy, choice of soft-tissue procedure, and preferred age to perform surgery; the cumulative knowledge guides us to current treatment of choice based on expert opinion. Limitations of this systematic review were the heterogeneity of available studies, not only in patient selection or indication for surgery but also in outcome measurements. Small patient numbers, differences in age, lack of information on severity of neurological deficit, study design, and performed treatment options limited the possibilities for drawing clear conclusions. Risk assessment showed that follow-up was not clearly predefined in 12 of 13 studies. However, studies included overall well-constructed cohorts of patients with low numbers lost to follow-up. Because of the complexity of problems in obstetric brachial plexus patients, which continue throughout growth, we assumed that regular follow-up was present in almost all studies. The cumulative mean follow-up of more than r

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FIGURE 1: Forrest plots showing the gain in passive pronation (PAS) and position at rest (POS) for the osteotomy and biceps rerouting procedures with studies sorted on preoperative deformity. A Osteotomy: there was a significant postoperative increase in passive pronation of 65 in the osteotomy group. B Osteotomy: there was a significant postoperative increase in position at rest of 75 in the osteotomy group. C Biceps rerouting: there was a significant postoperative increase in position at rest of 79 . The I2 was 93% in A and B and 88% in C, indicating consideral heterogenerity. This heterogenerity was largely explained by the amount of preoperative deformity. Greater pre-operative deformity resulted in larger corrections.

4 years confirmed this. Moreover, despite heterogeneity in patient cohort as well as study design, gain in pronation was significant for both osteotomy and biceps rerouting procedures. The latter supports the beneficial effects of surgical treatment for supination deformity in obstetric brachial plexus patients. Based on the results obtained in this review containing the data of 238 patients and expert opinions from several specialists in this field, and taking into J Hand Surg Am.

account that in each case treatment should be individualized based on the clinical findings, we recommend the following strategy for the treatment of supination deformity. First, a correction of a supination deformity should be considered when there is a supination position at rest of more than 60 . Second, treatment should be based on the extent of passive pronation deficit: (1) biceps rerouting in patients with complete to nearly complete preoperative r

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FIGURE 2: Clinical data showing preoperative and postoperative outcome in passive pronation and position at rest for the osteotomy and biceps rerouting procedures as mean with the 95% confidence interval. A Osteotomy: passive pronation. Clinical outcome showed that all studies had a mean passive pronation at follow-up of more than 30 and 2 studies of more than 50 . B Osteotomy: pronation at rest: Clinical outcome showed that 2 studies had a mean position at rest of more than 10 of supination and 4 studies had a neutral or pronation position at rest at follow-up. C Biceps rerouting: pronation at rest: Clinical outcome showed that all studies had a neutral or pronation position at rest after biceps rerouting.

passive pronation (eg, > 70 ); (2) biceps rerouting and interosseous membrane release when there is a partial preoperative passive pronation deficit (eg, > 50 ); (3) either an osteotomy or an interosseous membrane release in cases with a passive pronation between 0 and 50 based on individual expertise and personalized approach; (4) forearm osteotomy in patients J Hand Surg Am.

without passive pronation (eg, < 0 ); or (5) forearm osteotomy and biceps rerouting in children with severe and progressive deformities (eg, passive pronation < 50 ) and/or age less than 6 years, or when a high risk of recurrence is expected. Finally, pronation at rest should not be used to differentiate between treatment strategies. It can be of value to record r

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improvements of rotational position at follow-up. Active pronation should not be a treatment goal. Preoperative active wrist extension and active external rotation should be taken into account when considering supination deformity surgery.

21. Ruhmann O, Schmolke S, Bohnsack M, Carls J, Flamme C, Wirth CJ. Reconstructive operations for the upper limb after brachial plexus palsy. Am J Orthop. 2004;33(7):351e362. 22. Eberhard D. Transposition of the bicipital tuberosity for treatment of fixed supination contracture in obstetric brachial plexus lesions. J Hand Surg Br. 1997;22(2):261e263. 23. Martini AK. Restoration of pronation of the forearm. (Experiences with the operation according to Zancolli) [German]. Handchirurgie Mikrochirurgie Plastische Chirurgie. 1986;18(5):271e274. 24. Manfrini M, Valdiserri L. Proximal radio-ulnar arthrorisis in the treatment of supination deformity resulting from obstetrical paralysis [Italian]. Ital J Orthop Traumatol. 1985;11(3):309e313. 25. Romanini L, Calvisi V, Di CE. Surgical management of the supinatory syndrome in birth palsy [Italian]. Minerva Ortopedica. 1983;34(4):227e232. 26. Owings R, Wickstrom J, Perry J, Nickel VL. Biceps brachii rerouting in treatment of paralytic supinatio contracture of the forearm. J Bone Joint Surg Am. 1971;53(1):137e142. 27. Eng GD, Koch B, Smokvina MD. Brachial plexus palsy in neonates and children. Arch Phys Med Rehabil. 1978;59(10):458e464. 28. Mallet J. Obstetrical paralysis of the brachial plexus. II. Therapeutics. Treatment of sequelae. Results of different therapeutic technics and indications [French]. Rev Chir Orthop Reparatrice Appar Mot. 1972;58(Suppl):6. 29. Masse P. Obstetrical paralysis of the brachial plexus. II. Therapeutics. Treatment of sequelae. Surgical possibilities for the elbow and the hand [French]. Rev Chir Orthop Reparatrice Appar Mot. 1972;58(Suppl):20. 30. Bahm J. Secondary procedures in obstetric brachial plexus lesions [German]. Handchirurgie Mikrochirurgie Plastische Chirurgie. 2004;36(1):37e46. 31. D’Emilio S. Preoperative and postoperative therapeutic management of the supination deformity in the pediatric brachial plexus patient. Tech Hand Up Extrem Surg. 2006;10(2):96e99. 32. Kozin SH. Treatment of the supination deformity in the pediatric brachial plexus patient. Tech Hand Up Extrem Surg. 2006;10(2):87e95. 33. Allende CA, Gilbert A. Forearm supination deformity after obstetric paralysis. Clin Orthop Relat Res. 2004;(426):206e211. 34. Lin HH, Strecker WB, Manske PR, Schoenecker PL, Seyer DM. A surgical technique of radioulnar osteoclasis to correct severe forearm rotation deformities. J Pediatr Orthop. 1995;15(1):53e58. 35. Lipskeir E, Weizenbluth M. Derotation osteotomy of the forearm in management of paralytic supination deformity. J Hand Surg Am. 1993;18(6):1069e1074. 36. Rolfe KW, Green TA, Lawrence JF. Corrective osteotomies and osteosynthesis for supination contracture of the forearm in children. J Pediatr Orthop. 2009;29(4):406e410. 37. van Kooten EO, Ishaque MA, Winters HA, Ritt MJ, van der Sluijs HA. Pronating radius osteotomy for supination deformity in children with obstetric brachial plexus palsy. Tech Hand Up Extrem Surg. 2008;12(1):34e37. 38. Blount WP. Osteoclasis of the upper extremity in children. Acta Orthop Scand. 1962;32:374e382. 39. Manske PR, McCarroll HR Jr, Hale R. Biceps tendon rerouting and percutaneous osteoclasis in the treatment of supination deformity in obstetrical palsy. J Hand Surg Am. 1980;5(2):153e159. 40. Ozkan T, Aydin A, Ozer K, Ozturk K, Durmaz H, Ozkan S. A surgical technique for pediatric forearm pronation: brachioradialis rerouting with interosseous membrane release. J Hand Surg Am. 2004;29(1):22e27. 41. Ruhmann O, Hierner R. Z-plasty and rerouting of the biceps tendon with interosseous membrane release to restore pronation in paralytic supination posture and contracture of the forearm [German]. Oper Orthop Traumatol. 2009;21(2):157e169. 42. Zancolli EA. Paralytic supination contracture of the forearm. J Bone Joint Surg Am. 1967;49(7):1275e1284. 43. Narakas A. Surgical treatment of traction injuries of the brachial plexus. Clin Orthop Relat Res. 1978;133:71e90.

REFERENCES 1. Malessy MJ, Pondaag W. Obstetric brachial plexus injuries. Neurosurg Clin N Am. 2009;20(1):1e14. 2. Waters PM. Obstetric brachial plexus injuries: evaluation and management. J Am Acad Orthop Surg. 1997;5(4):205e214. 3. Sibinski M, Sherlock DA, Hems TE, Sharma H. Forearm rotational profile in obstetric brachial plexus injury. J Shoulder Elbow Surg. 2007;16(6):784e787. 4. Bahm J, Gilbert A. Surgical correction of supination deformity in children with obstetric brachial plexus palsy. J Hand Surg Br. 2002;27(1):20e23. 5. Gilbert A, Brockman R, Carlioz H. Surgical treatment of brachial plexus birth palsy. Clin Orthop Relat Res. 1991;264:39e47. 6. Aitken J. Deformity of the elbow joint as a sequel to Erb’s obstetrical paralysis. J Bone Joint Surg Br. 1952;34(3):352e365. 7. Weekley H, Nikolaou S, Hu L, Eismann E, Wylie C, Cornwall R. The effects of denervation, reinnervation, and muscle imbalance on functional muscle length and elbow flexion contracture following neonatal brachial plexus injury. J Orthop Res. 2012;30(8):1335e1342. 8. Zaoussis AL. Osteotomy of the proximal end of the radius for paralytic supination deformity in children. J Bone Joint Surg Br. 1963;45:523e527. 9. Yam A, Fullilove S, Sinisi M, Fox M. The supination deformity and associated deformities of the upper limb in severe birth lesions of the brachial plexus. J Bone Joint Surg Br. 2009;91(4):511e516. 10. Hankins SM, Bezwada HP, Kozin SH. Corrective osteotomies of the radius and ulna for supination contracture of the pediatric and adolescent forearm secondary to neurologic injury. J Hand Surg Am. 2006;31(1):118e124. 11. Magermans DJ, Chadwick EK, Veeger HE, van der Helm FC. Requirements for upper extremity motions during activities of daily living. Clin Biomech (Bristol, Avon). 2005;20(6):591e599. 12. Tubiana R, Masquelet A. Extensive paralysis of the upper limb. Functional classification and palliative treatment [French]. Chirurgie. 1987;113(3):246e251. 13. Hozo SP, Djulbegovic B, Hozo I. Estimating the mean and variance from the median, range, and the size of a sample. BMC Med Res Methodol. 2005;5:13. 14. Colditz GA, Brewer TF, Berkey CS, et al. Efficacy of BCG vaccine in the prevention of tuberculosis. Meta-analysis of the published literature. JAMA. 1994;271(9):698e702. 15. Slim K, Nini E, Forestier D, Kwiatkowski F, Panis Y, Chipponi J. Methodological index for non-randomized studies (MINORS): development and validation of a new instrument. ANZ J Surg. 2003;73(9):712e716. 16. Moher D, Liberati A, Tetzlaff J, Altman DG. Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. Ann Intern Med. 2009;151(4):264e269. W64. 17. Maillet M, Romana C. Complete obstetric brachial plexus palsy: surgical improvement to recover a functional hand. J Child Orthop. 2009;3(2):101e108. 18. Nath RK, Somasundaram C, Melcher SE, Bala M, Wentz MJ. Arm rotated medially with supination—the ARMS variant: description of its surgical correction. BMC Musculoskelet Disord. 2009;10:32. 19. Brunelli GA. Technique: Oblique radial osteotomy for supination syndrome. J Am Soc Surg Hand. 2004;4:50e54. 20. Ruhmann O, Wirth CJ, Bohnsack M, Flamme C. Corrective bone operations for treatment of deformities caused by obstetrical brachial plexus palsy [German]. Zeitschrift fur Orthopadie und Ihre Grenzgebiete. 2001;139(6):469e472.

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469 hits

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2 hits reference checking

105 dupicates removed

Screening

Identification

TREATMENT OF SUPINATION DEFORMITY IN OBPI

366 unique hits

319 abstracts excluded not supination deformity

Eligibility

37 eligible 5 excluded not supination deformity

Included

32 qualitative assessment n=338

13 quantitative assessment n=228

19 excluded 8 no patient data 11 follow-up < 1 year and/or no quantitative rotation

Osteotomy n=157 Osteotomy

Biceps rerouting n=71

n=157

-Radius -Radiusn=113 n=113 -Radius UlnaUlna n=30n=30 -Radius&and -Osteoclasis n=14

-Biceps rerouting n=26 -Biceps rerouting with membrana interosseus release n=45

-Osteoclasis n=14

APPENDIX A: PRISMA flow chart for retrieved articles found by search strategy on supination deformity in obstetric brachial plexus injury. Number of included obstetric brachial plexus patients added for reviewed articles.

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APPENDIX B. Quality Assessment Using the Methodological Index for Nonrandomized Studies (MINORS) MINORS Scoring MINORS Items

0

1

Clearly stated aim Consecutive patients included Prospective collection data

13 3

10 10

Endpoints appropriate Unbiased assessment endpoints

3 13

13

Follow-up appropriate Loss to follow-up < 5%

2

13 1

12

Number of studies in every quality assessment item and its score is given. Scoring system: 0: not reported, 1: reported but inadequate, 2: reported and adequate.

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