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Available online at www.sciencedirect.com
www.elsevier.com/locate/semperi
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.
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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.
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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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