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Available online at www.sciencedirect.com

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Neonatal brachial plexus palsy: Incidence, prevalence, and temporal trends Suneet P. Chauhan, MDa,n, Sean B. Blackwell, MDa, and Cande V. Ananth, PhD, MPHb,c a

Department of Obstetrics and Gynecology, University of Texas, Houston, TX Department of Obstetrics and Gynecology, College of Physicians and Surgeons, Columbia University, New York, NY c Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY b

article info

abstract

Keywords:

Epidemiological knowledge of the incidence, prevalence, and temporal changes of neonatal

Brachial plexus injury

brachial plexuses palsy (NBPP) should assist the clinician, avert unnecessary interventions,

Shoulder dystocia

and help formulate evidence-based health policies. A summary of 63 publications in the

Cesarean delivery

English language with over 17 million births and 24,000 NBPPs is notable for six things. First, the rate of NBPP in the US and other countries is comparable: 1.5 vs. 1.3 per 1000 total births, respectively. Second, the rate of NBPP may be decreasing: 0.9, 1.0 and 0.5 per 1,000 births for publications before 1990, 1990–2000, and after 2000, respectively. Third, the likelihood of not having concomitant shoulder dystocia with NBPP was 76% overall, though it varied by whether the publication was from the US (78%) vs. other countries (47%). Fourth, the likelihood of NBPP being permanent (lasting at least 12 months) was 10–18% in the US-based reports and 19–23% in other countries. Fifth, in studies from the US, the rate of permanent NBPP is 1.1–2.2 per 10,000 births and 2.9–3.7 per 10,000 births in other nations. Sixth, we estimate that approximately 5000 NBPPs occur every year in the US, of which over 580–1050 are permanent, and that since birth, 63,000 adults have been afflicted with persistent paresis of their brachial plexus. The exceedingly infrequent nature of permanent NBPP necessitates a multi-center study to improve our understanding of the antecedent factors and to abate the long-term sequela. & 2014 Elsevier Inc. All rights reserved.

Introduction Described as uncommon, unpredictable, and unpreventable,1 neonatal brachial plexus palsy (NBPP) is flaccid paresis of an upper extremity because of injury to the brachial plexus, with the passive range of motion greater than the active.2 NBPP is classically defined as Erb's palsy and involves cranial nerves C5 and C6. It is recognizable by the characteristic “waiter's

tip” arm position caused by muscle imbalance, which holds the shoulder in an adducted, internally rotated position with the elbow in extension and forearm in pronation. Though most cases of NBPP resolve, approximately 1 in 10 persist for over 1 year3 and may require microsurgical reconstruction. They have financial burden and quality-oflife implications4 and, consequently, may lead to litigation, especially when it occurs in conjunction with shoulder

n Correspondence address: Department of Obstetrics and Gynecology, University of Texas Health Science Center at Houston, 6431 Fannin, Suite 3.264, Houston, TX 77030. E-mail address: [email protected] (S.P. Chauhan).

http://dx.doi.org/10.1053/j.semperi.2014.04.007 0146-0005/& 2014 Elsevier Inc. All rights reserved.

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Table 1 – The rate of neonatal brachial plexus palsy. Number of publications

Total births USA Foreign countries Study period Before 1989 1990–1999 2000 or later Vaginal births USA Foreign countries Study period Before 1989 1990–1999 2000 or later Cesarean delivery USA Other countries

53 28 25

13 11 8 29 18 11

References

NBPP (N)

6–17,19–27,29–31,34–48,40–46,48–56, 58–60,63–65 6,8,9,11,13,14,16,19–21,25,27,29– 31,34,35,42,46,48,49,51,54,58–60,62,65 7,10,12,15,17,22,23,24,26,36–38,40,41, 43–45,50,52,53,55,56,63,64,67 6,79,10–17,22,23 24,26,31,34–38,40,41,43 48,51,56,58,59,60,64,67 1,7,18,19,21,22,24,29,31,32,34,37,43, 46–48,50,51,53,55,57,59,60–63,65,66 1,18,19,21,25,29,31,32,34, 46–48,51,59,60,62,65,66 7,22,24,37,43,50,53,55,57,61,63

3 6 5 7

7,18,22 24,31,32,34,37,43 4,48,51,59,60,66 7,28,29,34,46,63,65

5 2

28,29,34,46,65 7,63

dystocia.5 Epidemiological data on NBPP is valuable for an understanding of the frequency of its occurrence, determining temporal trends, and acknowledging the difficulty in minimizing the injury. The purpose of the review was to ascertain (i) the incidence and prevalence rates of NBPP; (ii) the likelihood of the palsy occurring without shoulder dystocia (SD); and (iii) the rate of permanent (lasting 12 months or more) NBPP. For these three scenarios, we compared the rates for US-based publications versus other countries and determined the temporal trends.

Starting with the publication by Rubin in 1964, we identified 63 publications1,2,6–66 that provided the rate of NBPP. For a specific study period, these articles provided either the total number of deliveries or the total number of vaginal births or both. Since the rate of NBPP is lower with cesarean delivery (CD: 7, 28, 29, 34, 46, 63, and 65) it is important to differentiate between rates of NBPP per 1000 total and vaginal births. Of the 63 publications cited, 53 provided sufficient information to calculate the rate of NBPP per 1000 total births. Among 17 million total births, there were over 24,000 NBPPs. The overall rate of NBPP was 1.4 per 1000 births, with the reported range being 0.1–6.3, a variation of 6000%. The rate of NBPP for articles published from the US was 1.5 per 1000 vs. 1.3 per 1000 total births for publications from other countries, a difference of 13%. To assess temporal trends, we grouped the publications into those whose study periods were 1989 or

Rate (per 1000)

Reported range (per 1000)

24,471

17,034,521

1.4

0.1–6.3

20,843

14,148,214

1.5

0.3–6.3

3628

2,886,307

1.3

0.1–3.6

451 2163 531,834 3038

513,878 2,068,087 261 1,873,330

0.9 1.0 0.5 1.6

0.5–3.6 0.1–3.0 0.2–4.1 0.1–8.1

2539

1,402,740

1.8

0.5–8.1

499

470,589

1.1

0.1–3.5

120 1763 294 84

151,604 980,748 228,471 250,948

0.8 1.8 1.3 0.3

0.7–1.2 0.1–3.3 0.5–5.5 0.3–1.5

75 9

234,399 16,549

0.3 0.5

0.3–1.5 0.4–0.8

United States

Other Countries

7

6

5 NBPP per 1,000 total births

Rates of NBPP

Deliveries (N)

4

3

2

1

0 1960

1970

1980

1990

2000

2010

Published in United States: Y = 4399.9- 4.446(x) + 0.0011(x ); R =0.09% Other Countries Y = -0.0016x + 6.2989x - 6261.8); R =0.07%

Fig. 1 – The rate of neonatal brachial plexus per total births.

212

SE

United States

M I N A R S I N

P

E R I N A T O L O G Y

Other Countries

9

8

NBPP per 1,000 vaginal births

7

6

5

4

3

2

1

0 1970

1980

1990

2000

2010

United States: Y = - 18416 + 18.328(x) - 0.0046(x ); R =0.06% Other Countries Y = - 6266 + 6.2738(x) - 0.0016(x ); R =0.05%

Fig. 2 – The rate of neonatal brachial plexus per vaginal births.

38 (2014) 210–218

before, 1990–1999, and after 2000. For these 3 epochs, the rates of NBPP per 1000 total births were 0.9, 1.0, and 0.5, respectively (Table 1). To compare the temporal trends of NBPP in publications from the US vs. other countries, we plotted the rate according to the year in which the article was published. The rate is increasing in the US and decreasing in other countries, but the R2 value for the second-degree polynomial equations is low (0.09% and 0.07%, respectively), indicating that the correlation is minimal (Fig. 1). Of the 63 publications, 29 provided data to calculate the rate of NBPP per 1,000 vaginal births. In almost 2 million vaginal births, there were over 3000 NBPPs. The overall rate of NBPP was 1.6 per 1000 vaginal deliveries, with the reported range being 0.1–8.1, a variation of 8000%. The rate of NBPP for articles from the US was 1.8 vs. 1.1 for studies from other countries. When the study period of reports was categorized into three groups, the rate of NBPP was 0.8 per 1000 vaginal births when done before 1989, 1.8 for studies done in 1990–1999, and 1.3 for those done in 2000 or later (Table 1). To ascertain the temporal trends, we plotted the rate of NBPP per 1000 VB for publications from the US vs. other countries. The rates in both groups increased and are trending downward. The R2 value for both quadratic regression equations was o1%, indicating minimal correlation between the year of publication and the rate (Fig. 2). We identified 7 publications that provided the rate of NBPP with cesarean delivery. In over 250,000 cesarean deliveries, there were 75 NBPPs. The overall rate was 0.3 NBPP per 1000 cesarean births. When segregated according to the country in which the study was done in, the rates were 0.3 vs. 0.5 per

Table 2 – Neonatal brachial plexus without shoulder dystocia in the US. Total birth

NBPP

Rate BPP/ 1000 TB

NBPP without SD

NBPP without SD (%)

Study

Country

Study period

Published

Levine et al.11

USA

1984

13,870

36

2.6

32

89

Gonik et al.18 Jennett et al.20 Nocon et al.21 Perlow et al.25 Ecker et al.27 Graham et al.29 Gherman et al.32 Ouzounian et al.33 Gilbert et al.34 Chauhan et al.41 Gurewitsch et al.45 Weizsaecker et al.50 Foad et al.53

USA USA USA USA USA USA USA USA

1974–1977; 1979–1981 1979–1987 1977–1990 1986–1990 1985–1990 1985–1993 1987–1991 1995–1996 –

1991 1992 1993 1996 1997 1997 1998 1998

57,597 14,297 19,370 77,616 14,358 NA NA

30 39 33 17 89 15 40 63

NA 0.7 2.3 0.9 1.1 1.0 NA NA

15 22 5 4 37 7 17 4

50 56 15 24 42 47 43 6

USA USA USA

– 1980–2002 1993–2004

1999 2005 2006

1,094,298 89,978 20,478

1611 89 128

1.5 1.0 6.3

757 39 45

47 44 35

USA

2000–2004

2007

11,001

45

4.1

15

33

USA

2008

11,555,823

17,334

1.5

14,254

82

Ouzounian et al.64 Overall

USA

1997, 2000, and 2003 1995–2004

2013

16,071

97

6.0

55

57

12,984,757

19,666

1.5

15,308

78a

TB, total births. a The overall rate of NBPP without SD is appreciably influenced by the study by Foad et al.53 Excluding this publication, the overall rate of NBPP without SD is 45% (1054 NBPP without SD/2332 NBPP).

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Table 3 – Neonatal brachial plexus without shoulder dystocia in the other countries. NBPP without SD

NBPP without SD (%)

Study

Country

Study period

Published

Soni et al.12 Salonen and Uusitalo17 Gonen et al.24 Dawodu et al.26

Libya Finland

1983 1981–1987

1985 1990

7829 14,265

28 16

3.6 1.1

25 8

89 50

Israel United Arab Emirates Israel Israel Ireland UK and Ireland Sweden

1994–1995 1993–1995

1996 1997

4480 9231

11 27

2.8 2.9

3 7

27 26

1995–1999 1996–1999 1994–1998 1998–1999

2000 2001 2002 2003

16,416 24,000 35,796 776,618

17 13 54 323

1.0 0.5 0.4

7 2 7 117

41 15 13 36

1990–1996

2005

16,743

51

3.3

32

63

1987–1997 1989–1995

2005 2005

1,213,987 25,855

2399 27

2 1.0

1166 4

49 15

1991–2000 1994–1998, 2004–2008 2005–2009

2008 2011

30,574 77,624

91 121

3.0 1.6

74 39

81 32

2013

16,697 2,270,115

19 3197

1.1 1.4

6 1497

32 47

Gonen et al.36 Kees et al.38 Donnelly et al.40 Evans-Jones et al.2 Gudmundsson et al.42 Mollberg et al.43 Tandon and Tandon44 Backe et al.51 Walsh et al.62 Jaggat et al.66 Overall

Sweden United Kingdom Norway Ireland Trinidad

1000 cesarean births for the US vs. other countries, respectively (Table 1). There were insufficient publications to determine the temporal trends by the epochs.

Number of births

NBPP

Rate BPP per 1000 TB

NBPP without shoulder dystocia

shoulder dystocia is 45%, which is akin to the rate of 47% from other countries. To determine the temporal trend of NBPP without shoulder dystocia, we plotted the likelihood versus the year in which the study was published (Fig. 3). The trend for publications from the US and other countries is U-shaped: high in early

Occurring in up to 1.5% of vaginal births, shoulder dystocia is the requirement of additional maneuvers rather than gentle downward traction on the fetal head to affect the delivery of shoulders.3 According to both the American Congress of Obstetricians and Gynecologists (ACOG) and the Royal College of Obstetricians and Gynaecologists (RCOG), the most injurious complication of shoulder dystocia is NBPP.3,67,68 Compared to when it occurs with an impacted shoulder, when NBPP occurs without shoulder dystocia it is considered mechanistically different,69 less likely to have 3rd or 4th degree episiotomy,41 as a concomitant fracture of the clavicle or the humerus,70 and to be permanent and be litigated.41,65,71 Hence, it is important to have an overview of how often NBPP occurs without concomitant shoulder dystocia. We identified 29 publications that provided the data on the likelihood of NBPP without shoulder dystocia, with 15 being US-based reports (Table 2) and the remaining 14 from other countries (Table 3). In the US, 78% of over 19,000 NBPPs occurred without concomitant shoulder dystocia; in other countries, with over 3000 NBPPs, 47% of brachial plexus palsy occurred without an impacted shoulder. The disparate rates between the two groups are primarily due to one report, which culled the data from a nationwide Kids' Inpatient Database.53 Since the study by Foad et al.53 is based on the International Classification of Disease (ICD) codes and not on chart reviews, it is feasible that there was ascertainment bias. Without this ICD-based study, the likelihood of NBPP without

Fig. 3 – Neonatal brachial plexus palsy without shoulder dystocia.

214

Table 4 – Total births and the rate of permanent neonatal brachial plexus palsy.

??? 1963–1972 1983–1986 1986–1990 1991–1995 1996–1998 1980–2002

1973 1975 1988 1993 1998 1999 2005

USA USA USA USA USA USA USA

31,700 19,314 8258 14,297 58,565 3008 89,978 225,120

59 11 21 33 48 6 89 267

1.9 0.6 1.9 2.3 0.8 2.0 1.0 1.2

88% 73% 90% 88% 94% 100% 96% 91%

(52) (8) (19) (29) (45) (6) (85) (244)

8% 0% 25% 25% 9% 33% 12% 10% 18%

(4) (0) (4) (1) (4) (2) (10) (25) (48)

1.3 0.0 4.8 0.7 0.7 6.6 1.1 1.1 2.2

1985 1988 2000 2002 2005

7829 25,736 16,416 35,796 25,855

28 48 17 54 27

3.6 1.9 1.0 1.5 1.0

64% 100% 65% 100% 100%

(18) (48) (11) (54) (27)

33% 25% 9% 19% 11%

(6) (12) (1) (10) (3)

7.7 4.7 0.6 2.8 1.2

39,220

30

0.8

100%(30)

27% (8)

2.0

2008

Libya Sweden Israel Ireland United Kingdom United Kingdom Norway

30,574

91

3.0

95% (86)

17% (15)

4.9

2011

Ireland

77,624

121

1.6

100% (12)

16% (19)

2.4

259,050

416

1.6

95% (395)

484,170

683

1.4

95% (639) 100% (683)

19% 23% 15% 21%

2.9 3.7 2.0 2.9

If all newborns lost to FUP had permanent NBPPa Other countries 1983 Soni et al.12 Sjö berg et al.15 1973–1982 Gonen et al.36 1995–1999 Donnelly et al.40 1994–1998 Tandon and 1989–1995 Tandon44 1991–2000 Draycott et al.52 Backe et al.51 Walsh et al.62

1996–1999; 2001–2004 2004–2008; 1994–1998

Subtotal If all newborns lost to FUP had permanent NBPPa Total If all newborns lost to FUP had permanent NBPPa Data presented as N or % (N). FUP, follow-up (of newborns with NBPP). a Lasting 12 months or more.

2008

Permanent NBPPa

(74) (95) (99) (143)

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United States Gordon et al.8 Specht9 Jackson et al.16 Nocon et al.21 Gherman et al.31 Turrentine et al.35 Chauhan et al.41 Subtotal

FUP Z12 months

E R I N A T O L O G Y

TB

P

Country

M I N A R S I N

Published

SE

Study period

NBPP

Rate NBPP per 1000 TB

Permanent NBPPa per 10,000 TB

Study

1

0 1970

1980

1990

2000

2010

United States: Y = – 31735+ 31.875(x) -0.008(x2); R2 = 18.3% Other Countries: Y = 83124 - 83.069(x) + 0.0208(x2); R2 = 71.6%

Fig. 4 – The rate of permanent neonatal brachial plexus palsy.

2.8 5.7 0.9 2.9 3.0 3.9 2.7 3.6 (8) (18) (3) (19) (48) (62) (62) (83) 95% (384) 100% (405) 1.8 405 230,980

29,025 31,828 32,312 66,098 159,263

30 112 21 121 284

1.0 3.5 0.6 1.6 1.8

100% 88% 100% 100% 95%

(30) (98) (21) (121) (270)

27% 16% 14% 16% 18% 22% 16% 20%

0.8 0.8 9.9 1.9 2.9 (1) (4) (9) (14) (21) 12,532 50,114 9071 71,717

33 48 40 121

2.3 1.0 4.4 1.7

88% 94% 100% 94%

(29) (45) (40) (114)

3% 9% 23% 12% 17%

United Kingdom Sweden Saudi Arabia Ireland

Data presented as N or % (N). VB, vaginal births. a Lasting 12 months or more.

2

If all newborns lost to FUP had permanent NBPPa

3

If all newborns lost to FUP had permanent NBPPa Total

4

2008 2008 2009 2011

5

If all newborns lost to FUP had permanent NBPPa Other countries 1996–1999; 2001–2004 Draycott et al.52 Mollberg et al.54 1999–2001 Rahman et al.56 1990–2005 Walsh et al.62 2004–2008; 1994–1998 Subtotal

Permanent NBPP per 10,000 total births

6

Country

7

Table 5 – Vaginal births and the rate of permanent neonatal brachial plexus palsy.

8

Published

Other Countries

Total VB

NBPP

Rate NBPP per 1000 VB

FUP Z 12 months

Permanent NBPPa

NBPP lasting 12 months or more is considered permanent and is linked with muscle imbalance, subluxed or dislocated joints, internal rotation, and adduction contractures about the shoulder, resulting in an inability to externally rotate and abduct the shoulder.72–74 The persistence of a paretic arm also affects the overall health and psychosocial condition of the children and their parents.75 They are, thus, one of the leading causes of obstetric litigation in the United States5 and England.67 We identified 15 publications that reported on the rate of permanent NBPP per total births. Of these 15 articles, 8 were US-based and the remaining 7 were from other countries. In over 225,000 total births in the US, there were 266 NBPPs, and 91% of these newborns were followed up for at least 12 months. Overall, only 10% of the NBPPs lasted for a year or more. If we assume that all the newborns who were lost to follow-up had permanent NBPP, then 18% of injuries to the brachial plexuses are permanent. The rate of permanent NBPP in the US was 1.1 per 10,000 total births and 2.2 per 10,000 if we assume that all newborns who were lost to follow-up had the injury last for a year or more (Table 4). The

USA USA USA

Permanent NBPP

1993 1998 1998

Permanent NBPPa per 10,000 VB

reports, a gradual decline with nadir in late 1990s, and continuing to incline after 2000.

United States

215

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1986–1990 1991–1995 1995–1996

E R I N A T O L O G Y

United States Nocon et al.21 Gherman et al.31 Gherman et al.32 Subtotal

P

Study period

E M I N A R S I N

Study

S

M I N A R S I N

P

E R I N A T O L O G Y

data from 7 publications from other countries is notable for the following: 416 NBPPs in over 259,000 total births; followup rate of 95%; the injury being permanent in 19% of those followed up and 23% if we assume that all those who were lost to follow-up had persistence of palsy; and the rate of permanent NBPP being 2.9 and 3.7 per 10,000 total births for those who were followed up and assuming all newborns who were lost to follow-up had persistent injury. To ascertain the temporal trend, we plotted the year of the 15 publications that provided data on permanent NBPP, separated into US-based vs. other countries. In the United States, the rate increased but subsequently declined. In comparison, the rate in other countries started high and then decreased but has increased in recent reports (Fig. 4). Lastly, we identified 7 publications that provided information on the rate of permanent NBPP per 10,000 vaginal births. While 3 of the reports were from the United States, 4 were from other countries. The rates for NBPP lasting a year or more for US-based and other countries are provided in Table 5.

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80

70,000 Permanent NBPP of 10% Permanent NBPP of 18%

78

Life expectancy

60,000

76 50,000 74

40,000

72

70

30,000

Average Life Expectancy (years)

SE

Cumulave Number Living with Brachial Plexus Injury

216

68

20,000 66

Newborns and adults with NBPP in the United States It is estimated that 175,000–800,000 Americans are afflicted with cerebral palsy.76,77 In our review of the literature, we were unable to find a similar estimate for NBPP in the US. Despite the potential for some imprecision in calculation, such an approximation is needed if we are to estimate the burden of disease and to design trials to decrease its occurrence. With the following data, it is feasible to calculate the period of prevalence of NBPP for 60 years: (i) from 1950 to 2010, the number of births per year; (ii) the life expectancy for a newborn in a given year78,79; (iii) the likelihood of NBPP is 1.5 per 1000 total births (Table 1); and (iv) the likelihood of NBPP being permanent is 10–18% (Table 4). Based on this data, we estimate that there are over 5000 NBPPs every year, of which between 580 and 1050 are permanent. Since 1950, the cumulative number of adults with NBPP is 35,000–63,000 (Fig. 5). We verified our methodology by comparing the actual number of NBPP cases reported by Foad et al.53 vs. estimates based on our four assumptions. For 1997, 2000, and 2003, the investigators noted there were over 11 million births in the US and there were 17,334 NBPPs; based on our calculations, for the specified period there should be 18,044 NBPPs, a difference of 4%.

Conclusions The likelihood of NBPP is just over 1 per 1000 births. The infrequent nature of palsy and the concordance of the rates between the reports from the United States and other countries suggest that it may not be secondary to a specific evanescent maneuver done by clinicians. Since the likelihood of NBPP without shoulder dystocia follows a similar pattern in the US as in other countries (Fig. 3), this trend suggests that the palsy can occur without difficulties in delivering an

10,000 64

0

62 1950

1960

1970

1980

1990

2000

2010

Fig. 5 – The prevalence of permanent neonatal brachial plexus palsy. impacted shoulder. Since permanent NBPP is a morbidity that obstetric interventions should curtail, it is astounding that since 1973, only seven US-based reports, with fewer than 300 cases, have been published. With such limited numbers of permanent NBPP in literature, it is unlikely we know how to optimally decrease the burden of persistent palsy. Despite the paucity of data on permanent NBPP, clinicians and research should acknowledge that an injury that lasts at least a year occurs in approximately 2–3 per 10,000 vaginal births. Due to the infrequent nature, interventions like induction or primary cesarean are unlikely to diminish the overall rate. Akin to the Maternal–Fetal Medicine Units network's cesarean registry,80 what would be pragmatic is a prospective cohort study that elucidates the modifiable factors, if any, associated with permanent NBPP. Without such an understanding, unnecessary diagnostic tests, interventions, trials, and litigations are likely to persist and will derail opportunities to advance our understanding of the causes and consequences of brachial plexus injury and lead to insurmountable burden to the health care economy.

re fe r en ces

1. Hammad IA, Chauhan SP, Gherman RB, Ouzounian JG, Hill JB, Abuhamad AZ. Neonatal brachial plexus palsy with vaginal birth after cesarean delivery: a case–control study. Am J Obstet Gynecol. 2013;208(229):e1–e5.

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Neonatal brachial plexus palsy: incidence, prevalence, and temporal trends.

Epidemiological knowledge of the incidence, prevalence, and temporal changes of neonatal brachial plexuses palsy (NBPP) should assist the clinician, a...
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