DEVELOPMENTAL MEDICINE & CHILD NEUROLOGY SYSTEMATIC REVIEW

Risk factors for neonatal brachial plexus palsy: a systematic

review and meta-analysis
RUTH VAN DER LOOVEN 1 | LAURA LE ROY 2 | EMMA TANGHE 2 | BIEKE SAMIJN 2 | ELLEN ROETS 3 |
NELE PAUWELS 4 | ELLEN DESCHEPPER 5 | MARTINE DE MUYNCK 6 | GUY VINGERHOETS 7 |
CHRISTINE VAN DEN BROECK 2

1 Department of Physical and Rehabilitation Medicine, Child Rehabilitation, Ghent University Hospital, Ghent; 2 Department of Rehabilitation Sciences and
Physiotherapy, Ghent University, Ghent; 3 Department of Obstetrics and Gynaecology, Prenatal Diagnosis Centre, Ghent University Hospital, Ghent; 4 Knowledge
Centre for Health Ghent, Ghent University Hospital, Ghent; 5 Biostatistics Unit, Department of Public Health, Ghent University, Ghent; 6 Department of Physical and
Rehabilitation, Ghent University Hospital, Ghent; 7 Department of Experimental Psychology, Faculty of Psychological and Educational Sciences, Ghent University, Ghent,
Belgium.
Correspondence to Ruth Van der Looven at Department of Physical Medicine and Rehabilitation, Child Rehabilitation Centre, Ghent University Hospital, C. Heymanslaan 10, route 445, 9000
Ghent, Belgium. E-mail: ruth.vanderlooven@uzgent.be

PUBLICATION DATA AIM To provide a comprehensive update on the most prevalent, significant risk factors for
Accepted for publication 17th September neonatal brachial plexus palsy (NBPP).
2019. METHOD Cochrane CENTRAL, MEDLINE, Web of Science, Embase, and ClinicalTrials.gov
Published online were searched for relevant publications up to March 2019. Studies assessing risk factors of
NBPP in relation to typically developing comparison individuals were included. Meta-analysis
ABBREVIATION was performed for the five most significant risk factors, on the basis of the PRISMA
NBPP Neonatal brachial plexus palsy statement and MOOSE guidelines. Pooled odds ratios (ORs), 95% confidence intervals (CIs),
and across-study heterogeneity (I2) were reported. Reporting bias and quality of evidence
was rated. In addition, we assessed the incidence of NBPP.
RESULTS Twenty-two observational studies with a total sample size of 29 419 037 live births
were selected. Significant risk factors included shoulder dystocia (OR 115.27; 95% CI 81.35–
163.35; I2=92%), macrosomia (OR 9.75; 95% CI 8.29–11.46; I2=70%), (gestational) diabetes (OR
5.33; 95% CI 3.77–7.55; I2=59%), instrumental delivery (OR 3.8; 95% CI 2.77–5.23; I2=77%), and
breech delivery (OR 2.49; 95% CI 1.67–3.7; I2=70%). Caesarean section appeared as a
protective factor (OR 0.13; 95% CI 0.11–0.16; I2=41%). The pooled overall incidence of NBPP
was 1.74 per 1000 live births. It has decreased in recent years.
INTERPRETATION The incidence of NBPP is decreasing. Shoulder dystocia, macrosomia,
maternal diabetes, instrumental delivery, and breech delivery are risk factors for NBPP.
Caesarean section appears as a protective factor.

Neonatal brachial plexus palsy (NBPP) is reported worldwide
in 0.1 to 8.1 per 1000 live births.1–6 Incidence rates vary with
study type and the availability of maternal and fetal care.7–9
NBPP is the result of a closed nerve stretch injury to the bra-
chial plexus, mostly occurring during labour. The mecha-
nisms of injury include maternal, obstetric, and infant factors
that apply traction on the anatomically vulnerable plexus.
Early management includes parental counselling, family
support, splinting, and appropriate and supervised rehabili-
tation. Neurosurgical intervention is usually undertaken at
3 to 6 months of age in children who have shown little or
no significant improvement in the affected muscle
groups.2,10–15 Depending on the long-term clinical course,
secondary surgery may be indicated later in life.
The few studies on NBPP prognosis not hampered by selec-
tion bias suggest that residual deficits are estimated at 20% to
35%.3,5,16–20 This finding is at odds with the previous
optimistic view of full recovery in over 90% of affected chil-
dren.21–26 After neonatal occurrence, the upper limb paresis
affects psychomotor development, bone growth, and joint
development. Deformities occur and may cause painful arthro-
sis in adults. Children with incomplete recovery have consider-
able risk for long-term functional limitations, with financial
burden, restricted daily life activities, limited participation, and
important overall quality of life implications.16,27–43
The potential impact of NBPP on the child’s physical
and psychological development, and their socio-economic
future, highlights the need for the development of preven-
tive strategies. Early identification of modifiable risk fac-
tors is therefore imperative. Appropriate management or
avoidance of risk factors might contribute to a reduction in
NBPP morbidity.
The aim of the present study was to determine and
unravel the significant risk factors for NBPP to enable

© 2019 Mac Keith Press DOI: 10.1111/dmcn.14381 1

individualized risk assessment and proper counselling of
individual women at risk. We conducted a meta-analysis
by combining the results from all available studies to: (1)
assess the five most frequently reported significant risk fac-
tors; (2) calculate their pooled odds ratios; and (3) assess
the heterogeneity among studies. In addition, we assessed
the incidence and its evolution.
METHOD
The Cochrane Database of Systematic Reviews was
searched to ensure a similar review had not been under-
taken. The Preferred Reporting Items for Systematic
Reviews and Meta-Analyses (PRISMA) statement,44
MOOSE guidelines,45 and Grant’s review46 were followed
to report this meta-analysis. The protocol was accepted for
registration in PROSPERO on 14th November 2017
(CRD42017076642).
Information sources and search strategy
The search strategy, using the Population, Intervention,
Comparison, and Outcome (PICO)47 format for clinical ques-
tions, was developed systematically in the MEDLINE data-
base (PubMed interface), using medical subject headings as
well as free text words (Fig. S1, online supporting informa-
tion). We searched entries on ClinicalTrials.gov for ongoing
studies. A final literature search was conducted on 1st March
2019. Manual reference screening of retrieved articles was
performed to identify other pertinent studies. Until the time
of submission of this paper for publication, an automatic
e-mail alert was checked for possible additional relevant arti-
cles. A list of all identified studies is available on request. We
imported all citations into bibliographic software (Endnote
X7; Thomson Reuters, Philadelphia, PA, USA).48
Eligibility
We included all published randomized controlled trials
and observational studies (cohort and case–control) that
assessed the risk of NBPP as the primary outcome in rela-
tion to typically developing comparison individuals. NBPP
was defined as a closed nerve stretch injury to the brachial
plexus (C5–T1). To limit population bias, studies with
only ‘upper’ plexus infants or with unclear definition such
as ‘shoulder injury’ were excluded. Language was restricted
to English, Dutch, or French. Studies with non-extractable
data were excluded.
Three authors (RVDL, LLR, and ET) independently
screened all articles by title, abstract, and keywords using
the Covidence web-based platform49 recommended by the
Cochrane Organization for systematic reviews. When all
inclusion criteria (Table S1, online supporting information)
were met, the full text version of the article was assessed.
Disagreements were resolved by consensus.
Data extraction
Data from each included trial were extracted independently
by the three reviewers (RVDL, LLR, ET) on a data extraction
form designed in accordance with the Cochrane Checklist of
2 Developmental Medicine & Child Neurology 2019
What this paper adds
• The overall incidence of neonatal brachial plexus palsy is 1.74 per 1000 live
births.
• The incidence has declined significantly.
• Shoulder dystocia, macrosomia, maternal diabetes, instrumental delivery,
and breech delivery are the main risk factors.
• Prevention is difficult owing to unpredictability and often labour-related risk.
items.50 Any disagreements were solved after discussion.
Details included source, eligibility, study design, participants,
risk factors, results, as well as any other miscellaneous data.
The primary outcome was risk factors. Secondary, significant
risk factors, considered as p<0.05, were extracted from the
articles and listed. The five most frequently reported signifi-
cant risk factors were subjected to meta-analysis. Raw data for
case and control groups were included for further analysis.
Percentages were converted into n-values with rounding up if
≥0.5. Macrosomia was defined as birthweight above 4000g.51
Birthweight was categorized as 4000 to 4499g, 4500 to
4999g, and 5000g or more.
Risk of bias and quality assessment
Each reviewer independently assessed the risk of bias by
using the Newcastle-Ottawa Scale52 and the Strengthening
the Reporting of Observational studies in Epidemiology
(STROBE) checklist.53
The Grading of Recommendations Assessment, Devel-
opment and Evaluation (GRADE)54 approach was used for
rating the body of evidence into high, moderate, low, and
very low quality. The quality of evidence for each outcome
(risk factor) across all studies was rated according to a
framework, including five factors that may lead to down-
grading the quality of evidence and three factors that may
lead to upgrading it.
Data synthesis
A first set of meta-analyses was conducted for each risk
factor. The pooled odds ratio of NBPP was calculated with
a 95% confidence interval.55 The weighing coefficients
were computed by the Mantel–Haenszel method with a
Dersimonian–Laird random effects model. Data analysis
used Review Manager 5.3 software (RevMan; Copenhagen,
Denmark: The Nordic Cochrane Centre, The Cochrane
Collaboration, 2014). Where zeros caused problems with
computation of the odds ratio or its standard error, 0.5
was added automatically in RevMan to all cells.56,57 We
used the I2 measure of Higgins and Green58 to assess
across-study heterogeneity with I2 values of 0% to 40%
considered to be low, 30% to 60% moderate, 50% to 90%
substantial, and 75% to 100% considerable. This index
does not depend upon the number of studies. If I2 was at
least 50%, sensitivity analysis was performed by excluding
the trials that potentially biased the results. Funnel plots,
which are scatterplots of treatment effect (odds ratio of
NBPP) against a measure of study precision (the standard
error of the log[odds ratio]), were constructed to assess
publication bias.59
 In an additional meta-analysis, the pooled incidence of
NBPP was calculated. The data from included studies were
divided into corresponding timeframes: 1987 to 1995, 1996
to 2005, and 2006 to 2015. A pooled overall proportion of
the event was calculated per timeframe, by a subgroup
meta-analysis, using a Dersimonian–Laird random effects
model with inverse variance, a logit transformation of the
proportions, and Clopper–Pearson or ‘exact’ binomial con-
fidence intervals for individual study results in the forest
plot. The calculations were performed in R, version
3.5.2,60,61 with the metaprop function in the meta library,
version 4.9-4.
RESULTS
Literature search
MEDLINE (974), Web of Science (747), ClinicalTrials.gov
(0), and Embase (1215) provided a total of 2936 citations.
After screening title and abstracts, 92 articles were included
for full text review. Finally, 22 articles met the eligibility cri-
teria. The main reasons for exclusion were study population
(adult), outcome (other than risk factor), comparison group
(no typically developing individuals), non-extractable data,
and no full text available. Two articles62,63 used an overlap-
ping study population. The paper by Foad et al.63 was there-
fore excluded from the meta-analysis. The Cohen’s j to
evaluate concordance of independent reviewers was 0.70,
indicating a substantial interrater agreement. A detailed
PRISMA flow diagram is presented in Figure S2 (online sup-
porting information).
Study characteristics
The characteristics of the 22 included articles are listed in
Table S2 (online supporting information). Twelve were
cohort studies62–73 and 10 were case–control studies.7,15,74–81
Only four studies had a prospective design.7,15,65,70 Nine
studies were conducted in Europe,7,64–67,72,76–78 seven in
North America,62,63,68,69,71,75,81 five in Asia,15,70,74,79,80 and
one in Australia.73 The primary outcome in each study was
risk factors for NBPP. In eight studies,62–64,66,68,69,75,78
data were derived from nationwide databases whereas in 13
articles7,15,65,70–74,76,77,79–81 data originated from hospital
records. Three studies linked clinical and administrative
data sources from the hospital.71,72,80 The recruitment per-
iod of the studies varied from 5 months79 to 20 years,80
with a mean duration of 7 years 4 months and a standard
deviation of 5 years 2 months. The sample size varied from
162 to 24 159 426 live births.
The historic risk factors for NBPP were listed
(Table S3, online supporting information) and divided into
maternal-, labour-, and fetal/neonatal-related factors.
According to three studies,15,72,74 we added (para)medical-
related as a new category.
The STROBE checklist (Table S4, online supporting
information) showed an overall compliance of 67%.
Matching criteria were mentioned in only 36 of the stud-
ies. Twenty-five per cent or less provided bias, sensitivity
analyses, flow diagram, an explanation of how missing data
were addressed, and the number of participants with miss-
ing data.
The methodological quality assessment of each study by
the Newcastle-Ottawa Scale (Table S5, online
supporting information) showed that 12 articles7,15,62–
64,66,69–73,78
achieved a score of at least seven stars, indicat-
ing high quality.82 The other 10 studies65,67,68,74–77,79–81
received at least five stars. Overall, study designs missed
description of comparability.
Meta-analysis
Twenty-one studies represented a total sample size of
29 419 037. The five most frequently reported significant
risk factors were used for meta-analysis: shoulder dystocia,
birthweight, (gestational) diabetes, instrumental delivery,
and breech delivery. Shoulder dystocia showed the highest
association with NBPP (odds ratio [OR] 115.27; 95% con-
fidence interval [CI] 81.35–163.35; 12 studies;
n=25 825 074)7,15,62,65,71,73,75,77–81 but with considerable
across-study heterogeneity (I2=92%) (Fig. S3a, online sup-
porting information). A sensitivity analysis with exclusion
of three studies65,71,78 resulted in low heterogeneity
(I2=19%; n=24 566 515) with similar strength of associa-
tion (OR 113.58; 95% CI 95.35–135.29).
Macrosomia (≥4000g) was the second-highest associated
factor (OR 9.75; 95% CI 8.29–11.46; 13 studies;
n=2 435 212)15,64,67,69–72,74,76–80 (Fig. S3b). The substantial
heterogeneity (I2=70%) was reduced to 34% (n=2 955 694)
by excluding two studies15,70 and resulted in an odds ratio
of 9.14% and 95% confidence interval 8.28 to 10.10.
When subdividing the birthweight into three subcategories
(4000–4499g, 4500–4999g, and ≥5000g), compared with
normal birthweight of not more than 3999g, the odds
ratios were respectively 6.32 (95% CI 5.48–7.29; six stud-
ies;64,67,72,76–78 I2=59%), 20.77 (95% CI 16.86–25.58; five
studies;64,67,72,77,78 I2=75%), and 55.21 (95% CI 49.7–
61.35; five studies;64,67,72,77,78 I2=0%).
(Gestational) diabetes as a risk factor with an odds ratio of
5.33 (95% CI 3.77–7.55; I2=59%; n=1 651 281) was explored
in 10 studies15,65,71,72,74,75,78–81 (Fig. S3c). Excluding two
studies74,75 reduced heterogeneity to 23% (n=1 274 770) and
an odds ratio of 4.21 (95% CI 3.12–5.68).
With an odds ratio of 3.8 (95% CI 2.77–5.23; seven
studies;7,15,67,71,72,77,78 I2=77%; n=1 849 398) instrumental
delivery, containing vacuum and/or forceps delivery,
occurred as a risk factor for NBPP (Fig. S3d). Sensitivity
analysis affirmed three studies15,67,78 accountable, with an
odds ratio of 2.50 (95% CI 1.81–3.45; n=25 240; I2=0%)
after exclusion. For exploratory reasons a separate pooled
odds ratio was calculated for vacuum (OR 6.03; 95% CI
3.61–10.07; seven studies;62,65,68,75,76,79,80 n=25 666 748;
I2=96%) and forceps delivery (OR 4.97; 95% CI 1.95–
12.66; three studies;62,65,68 n=25 284 297; I2=98%).
The last significant risk factor was breech delivery, with
a pooled odds ratio of 2.49 (95% CI 1.67–3.7; 11 stud-
ies;7,62,63,65–67,72,76–79 n=26 780 930; I2=70%) (Fig. S3e).
The substantial heterogeneity reduced by excluding three
Review 3
studies62,78,79 (n=1 401 993; I2=0%), resulting in a higher
odds ratio of 3.35 (95% CI 2.39–4.70).
Caesarean section emerged as a protective factor for
NBPP, with a pooled odds ratio of 0.13 (95% CI 0.11–
0.16; 11 studies;7,62,64,65,67,68,72,74,75,77,78 n=28 530 257;
I2=41). Heterogeneity was moderate (Fig. S3f). The
heterogeneity reduced to 0% by excluding one study62
(n=4 370 832; I2=0%; OR 0.15, 95% CI 0.13–0.17).
The five risk factors demonstrate an asymmetrical funnel
plot, which could assume publication bias. The limited
number of studies per risk factor, however, inhibits an
appropriate causal interpretation (Fig. 1).59 According to
the assessment by the GRADE (Table 1), Caesarean sec-
tion presented high-quality evidence, shoulder dystocia,
and birthweight moderate quality, whereas (gestational)
diabetes and instrumental delivery showed low-quality evi-
dence. Breech delivery had a very low confidence rating.
Strengths of the studies responsible for upgrading were the
large magnitude of effect. An important limitation leading
to downgrading was inconsistency of results for each out-
come and additionally imprecision for results of breech
delivery. All findings are summarized in Table 2.

Shoulder dystocia Macrosomia

0 SE(log[OR])
0 SE(log[OR])

0.5 0.2

0.4
1
0.6
1.5
0.8
OR OR
2 1
0.001 0.1 1 10 1000 0.01 0.1 1 10 100

(Gestational) diabetes Instrumental delivery

0 SE(log[OR])
0 SE(loga[OR])

0.2
0.5

0.4
1
0.6

1.5
0.8

2 OR OR
1
0.002 0.1 1 10 50 0.02 0.1 1 10 50

Breech delivery Caesarean section

SE(log[OR])
0 SE(loga[OR]) 0

0.5 0.5

1 1

1.5 1.5

OR
2 2 OR
0.05 0.2 1 5 20 0.02 0.1 1 10 50

Figure 1: Funnel plots of the five most reported and significant risk factors. SE, standard error; OR, odds ratio

4 Developmental Medicine & Child Neurology 2019

Table 1: Grading of Recommendations Assessment, Development and Evaluation (GRADE) assessment of risk factors for neonatal brachial plexus palsy

Downgrade Upgrade

Effect of
Large Dose– plausible
Limitations Inconsistency Indirectness Publication magnitude of response residual
Start (RoB) of results of evidence Imprecision bias an effect gradient cofounding End

Shoulder LQ Low ↓ 1 OK OK OK ↑ +2 / OK MQ
dystocia
(Gestational) LQ Low ↓ 1 OK OK OK ↑ +1 / OK LQ
diabetes
Birthweight LQ Low ↓ 1 OK OK OK ↑ +2 / OK MQ
Instrumental LQ Low ↓ 1 OK OK OK ↑ +1 / OK LQ
delivery
Breech LQ Low ↓ 1 OK ↓ 1 OK ↑ +1 / OK VLQ
delivery
Caesarean LQ Low OK OK OK OK ↑ +2 / OK HQ
section

RoB, Cochrane risk-of-bias tool for randomized studies; LQ, low quality; ↓, lower it down; OK, quality met criteria; ↑, grade it up; /, not
applicable; MQ, moderate quality; VLQ, very low quality; HQ, high quality.

The pooled overall incidence of NBPP was 1.74 per
1000 live births (95% CI 1.56–1.94) (Fig. S4, online sup-
porting information). The subgroup meta-analysis demon-
strated a significant difference (p=0.013) in the pooled
proportion of the event, between the three timeframes
(1987–1995, 1996–2005, and 2006–2015). The forest plot
showed highly heterogeneous individual study results
within each timeframe. The estimated pooled proportions
were 0.0021 (95% CI 0.0018–0.0025) for the first time-
frame, 0.0019 (95% CI 0.0014–0.0025) for the second, and
0.0012 (95% CI 0.0009–0.0017) for the third. Although a
considerable overlap was found between the first and sec-
ond, and second and third, confidence intervals, a decrease
in proportion could be concluded between the first and
third timeframes.
DISCUSSION
Consistent with previous research,83–86 shoulder dystocia
was found to be the major risk factor for NBPP. Three
studies were responsible for the considerable heterogene-
ity.65,71,78 First, we assigned the shoulder dystocia inci-
dence variety as a possible source: shoulder dystocia is
imprecisely coded, often underreported, and even unrecog-
nized. The prospective design of Backe et al.65 and perhaps
a more rigorous reporting of Backe et al.65 and Ouzounian
et al.71 might explain their relatively high incidence of
shoulder dystocia compared with previous published
research. In contrast, the remarkably low incidence in the
large retrospective case–control study of Mollberg et al.78
is explained by the authors as a possible underreporting of
shoulder dystocia in Sweden. Second, an observable lower
association of NBPP and shoulder dystocia was noted in
two studies,60,67 which could result from their immediate
resort to the recommended shoulder dystocia delivery
technique73,87 in the case of shoulder dystocia.
Macrosomia, confirmed only after delivery of the neo-
nate, is the second risk factor. The 70% heterogeneity was
mostly caused by two studies.15,70 As both were prospec-
tively performed in Asia, their remarkably higher incidence
of macrosomia might be explained by population bias.
Sixty per cent of infants with macrosomia in the study by
Najafian and Cheraghi70 were of Arab ethnicity. Dawodu
et al.15 revealed a higher frequency of maternal diabetes,
shoulder dystocia, and macrosomia in Arab versus Western
populations. Both studies had a strong association of
NBPP and macrosomia. Interestingly, only 2% of infants
with macrosomia in the study by Najafian and Cheragi70
were associated with NBPP compared with 70% in that by
Dawodu et al.15 Possibly, their birthweight distribution
was different, with those in the study by Najafian and

Table 2: Summary of findings for the five most significant risk factors

Number of Number of
Number of Total cases within cases in total OR (95% CI) Strength of
Outcomea studiesb population risk group population for NBPP I2 (%) recommendationc

Shoulder dystocia 12 25 825 074 6563 33 984 115.27 (81.35–163.35) 92 Moderate

Macrosomia 13 2 975 874 3896 5892 9.75 (8.29–11.46) 70 Low
(Gestational) diabetes 10 1 651 281 175 3209 5.33 (3.77–7.55) 59 Moderate
Instrumental delivery 7 1 849 398 824 3112 3.8 (2.77–5.23) 77 Low
Breech delivery 11 26 780 930 183 34 960 2.49 (1.67–3.7) 70 Very low
a
Risk factors implemented in the meta-analysis. bIncluded in the meta-analysis. cBased on the Grading of Recommendations Assessment,
Development and Evaluation (GRADE) quality of evidence assessment. OR, odds ratio; CI, confidence interval; NBPP, neonatal brachial
plexus palsy.

Review 5
Cheragi70 tending to have the lower weight range: 85%
birthweight between 4000 and 4449g, 13% between 4500
and 4999g, and only 2% having at least 5000g. Obstetric
practices might also differ: all cases of NBPP in the study
by Dawod et al.15 were delivered vaginally. Therefore, geo-
graphical differences, study design, and population bias
might have been responsible for the heterogeneity. In
agreement with previous studies,88–90 calculated odds ratios
of NBPP expand powerfully with increasing birthweight:
6.32 for birthweight group 4000 to 4499g, 20.77 for 4500
to 4999g, and particularly 55.21 for those with a birth-
weight of at least 5000g. Birthweight of at least 5000g
seems to be a commonly found risk factor, with I2=0%.
In accordance with previous reports,63,89,91–93 maternal
diabetes is the third significant risk factor. Two stud-
ies,74,75 showing higher odds for NBPP in the diabetic
group, were responsible for the 59% heterogeneity. As the
incidence of NBPP, respectively diabetes, in these studies
was comparable with the included studies, other factors
should be accountable. We believe that birthweight, ratio
of head-to-abdominal circumference of the fetus, maternal
weight, and age difference between diabetic versus non-
diabetic groups are important characteristics possibly
responsible for population bias. Disproportionate growth
of trunk, measured as a smaller ratio of head circumference
to abdominal circumference, is well-known in women with
diabetes.94–96 Unfortunately, the included studies published
none of these data. In the large population-based study by
Freeman et al.,75 61% of diabetic mothers underwent Cae-
sarean section, a finding also reported in other studies.97–99
The reason for Caesarean section was not mentioned but,
knowing the diabetes diagnosis, the obstetrician may have
preferred Caesarean section for fear of shoulder dystocia
or NBPP.
Instrumental delivery (vacuum extraction and/or forceps
delivery) augments the risk for NBPP 3.8-fold, with vac-
uum extraction showing an odds ratio of 6.03. The 77%
heterogeneity was attributable to three studies.15,67,78 Dif-
ferent reporting numbers, depending on the use of mater-
nal versus neonatal records, result in a probable source of
bias. Instrumental delivery is often noted in the mother’s
record as a procedure and only in the newborn infant’s
record as a diagnosis if its use resulted in an injury. Possi-
ble population bias following different prevalence of other
risk factors such as diabetes, macrosomia, shoulder dysto-
cia, or increased length of second stage of labour in the
studied population might cause heterogeneity. The differ-
ent studies, despite reporting adjusted results, did not
examine the same confounders. In addition, no information
could be extracted on the sequential use of instruments,
and certainly temporal trends are important.
Our meta-analysis confirms the previously reported
NBPP risk of breech delivery.100–105 NBPP incidence and
severity are increased with the tendency to develop more
upper palsy and a higher percentage of bilateral palsy.106
Five out of 11 articles7,65,71,76,79 had a wide confidence
interval, mainly because they included relatively few
6 Developmental Medicine & Child Neurology 2019
patients. Three studies were liable for the 70% hetero-
geneity.62,78,79 The contribution of Okby and Sheiner79 of
21% was possibly due to the presence of only one breech-
delivered infant with NBPP. The low risk factor preva-
lence of 0.12% in the study by Abzug et al.62 might be
explained by a higher rate of Caesarean section of 27%. In
contrast, the study by Mollberg et al.78 presented 2.51%
breech deliveries with a small complication rate, which
could be ascribed to a more homogenous and better selec-
tion for trial of vaginal breech delivery.
Caesarean section seems to be a protective factor, with
an odds ratio of 0.13. Low heterogeneity was reduced to
0% by excluding one study.62 The high prevalence of Cae-
sarean section in the study by Abzug et al.62 might be the
contributor.
The pooled overall incidence of NBPP was 1.74 per
1000 live births. The subgroup meta-analysis demonstrates
a significant decrease of incidence over time (1987–1995,
1996–2005, and 2006–2015). The heterogeneity could be
partly explained by the difference in type of obstetric care,
in Caesarean section rate, and in average birthweight of
neonates in different geographical regions.
In practice, clinicians are confronted with a combination
of different risk factors, which are interrelated (Fig. 2). The
risk of shoulder dystocia is highly correlated with fetal
macrosomia,75,107 a history of previous shoulder dysto-
cia7,108 or macrosomia, maternal diabetes and obesity,109
induction of labour,73 abnormalities of labour, the number
and type of manoeuvres used,73 and instrumental deliv-
ery.75,110–112 Infants with macrosomia who are vaginally
delivered are at risk of shoulder dystocia, with the highest
association in the birthweight group greater than 5000g.78
Macrosomia is an important risk factor for instrumental
delivery and increases the risk of Caesarean section. Most
studies confirm the higher incidence of maternal obesity,
macrosomia, disproportional fetal growth, abnormal labour,
and shoulder dystocia among diabetic pregnancies.96,113,114
Other major risk factors for macrosomia are increased
maternal age (>35y) and obesity, a positive history of previ-
ous macrosomia, prolonged pregnancy, and multipar-
ity.64,70,113,115,116 Given the potential for severe neonatal
and long-term morbidity of NBPP, as well as the significant
medico-legal implications, it is essential to recognize
patients who are at high risk (for instance by using a risk
factor scoring system). However, prevention of NBPP is a
challenge owing to the poor predictive value of the risk fac-
tors. Estimation of fetal weight, often used as a measure of
risk stratification, remains, even with modern ultrasound
equipment and in experienced hands, an inaccurate task.116
A sensitivity and specificity of respectively 60% and 90%
for identifying fetuses of at least 4000g makes it insuffi-
ciently reliable.86,90 Therefore, the target in antenatal pre-
vention of NBPP should be on nutrition and physical
activity guidance,117,118 strict glycaemic and weight control
in case of (gestational) diabetes, and appropriate counselling
of women at high risk. Counselling should focus on the
risks and alternatives to vaginal delivery.
 Diabetes
Maternal age
Previous history of macrosomia
Weight gain
Multiparity
Gestational age
Caesarean section
Dysfunctional labour
Prolonged second stage
Figure 2: Interrelation of risk factors. Factors in bold type were implemented in the meta-analysis
Intrapartum risk management should include presence of
consistent intrapartum guidelines, ensuring availability of
regularly and well-trained staff at delivery and minimizing
manoeuvres with excessive traction on the fetal neck. For
macrosomia, recent research suggests that early-term
induction significantly reduces the risk of shoulder dysto-
cia.119–121 The clear association between NBPP and
macrosomia in diabetic pregnancies, with higher occur-
rence of total palsies114 and therefore more permanent dis-
ability,116 is a major concern in the prevention of NBPP.
Elective Caesarean section should be offered where there is
an estimated fetal birthweight greater than 5000g in non-
diabetic pregnancies90,122 and greater than 4500g in those
with diabetes according to guidelines.83,90 In all others, a
trial of labour is recommended. This requires preparedness
for operative delivery, shoulder dystocia, and newborn
asphyxia.114 In the case of shoulder dystocia, the risk for
severity of NBPP is critically linked to a timely recogni-
tion, and increases with the number and types of manoeu-
vres required.73 Several authors123–127 reported evidence
that a systematic approach with simulation training of
specific manoeuvres can reduce cases of NBPP signifi-
cantly.127,128 For mothers with a previous shoulder dysto-
cia history, an estimated fetal weight less than the previous
dystocia delivery, or a lack of history of permanent NBPP,
a trial of vaginal delivery may be reasonable.109
Independent or sequential use of forceps and vacuum
extractor greatly increases the risk of NBPP, especially
among obese and diabetic pregnancies.78,129 The choice
between vacuum and forceps has recently shifted, with a
preference for vacuum as the instrument of first choice.130–
133
Caesarean section performed after failed instrumental
delivery carries increased risk.29 Therefore, obstetricians
should aim to complete all operative vaginal delivery safely
with a single instrument.134
The routine to deliver almost all term breech cases by
elective Caesarean section is a continuing debate. In fact,
there are only three randomized controlled trials that have
investigated neonatal outcomes in term breech by mode of
Abnormal labour
Maternal obesity
Previous history of
Macrosomia Shoulder dystocia
shoulder dystocia
Induction of labour
Number and type
of manoeuvres
Instrumental delivery
delivery.135–137 A Cochrane review138 reported a reduced
perinatal or neonatal mortality among singleton infants
delivered by a planned Caesarean section. However, nei-
ther elective Caesarean section nor vaginal delivery for
breech presentation is risk free.105 Eligibility criteria, bal-
ancing all risks and benefits, for vaginal breech delivery
should be set at the national level to guide best practice.
Although Caesarean section seems to be protective, it
does not totally prevent NBPP68,139 and carries significant
neonatal and maternal morbidities (an increased risk for
repeat Caesarean section, abnormal placentation, uterine
rupture, and ectopic pregnancy).140,141 This opens the
commonly raised issue of the cost:benefit ratio.142–144 For
the USA population, more than 1000 elective Caesarean
sections, costing US$4 million to US$8 million, would be
required to prevent one case of NBPP.75,86 By comparison,
the lifetime costs for a case of NBPP, excluding potential
loss in productivity and earning capacity, is estimated at
more than US$1 million.143 Caesarean section is only indi-
cated in selected cases: women with previous children with
permanent NBPP,23,145 macrosomic pregnancies compli-
cated by diabetes,74,142,143 and a high global risk identified
by the obstetrician.
The decreasing incidence might be the result of aug-
mented awareness of the problem and improved obstetric
techniques and strategies. This encourages further research
to determine predictable and modifiable risk factors. A
future worldwide meta-analysis of incidence could be
important to evaluate geographical differences and their
influence on the risk of NBPP.
Strengths and limitations
The present study is, to our knowledge, the largest meta-
analysis to investigate incidence and risk factors for NBPP
with data obtained from 29 419 037 live births. The method-
ology used was rigorous, following the PRISMA statement.
Baseline characteristics of the patients were largely compara-
ble, which suggested that the population of patients was rep-
resentative. The GRADE approach showed a high-quality
Review 7
body of evidence for Caesarean section, and moderate quality
of evidence for shoulder dystocia and birthweight.
We recognize limitations. First, having investigated only
the five most significant risk factors, we recommend fur-
ther research for all previously mentioned, but also undis-
covered, risk factors. Second, because most of the included
studies were retrospective and observational by design,
they were prone to bias. Specifically, some data might not
have been accurately recorded or were underreported.
Unclear definition of NBPP and its risk factors, but also
increasing medical litigation related to birth trauma, might
have contributed to reporting bias. Source bias (maternal
vs neonatal, administrative vs medical, local hospitals vs
national birth registers) was present. Nationwide databases
are clearly more specific and include home deliveries. The
divergent sample sizes caused different weight influence on
the overall odds ratio. Temporal influences such as study
duration and timing might contribute to imprecision.
Third, as NBPP can be caused by inappropriate delivery
technique, information about quality of delivery is neces-
sary. Unfortunately, we could not assess to what extent
poor obstetric technique contributed to heterogeneity.
Fourth, we might have missed important information by
excluding studies with non-extractable data and those
solely investigating Erb palsy (defined as C5–6–[7]). Fifth,
all included studies were from high-income countries,
which may not be representative of all risk factors. Finally,
lack of information about severity and duration of NBPP
is a major limitation in stratifying the importance of each
risk factor. Heterogeneity, important to the validity of con-
clusions, was therefore carefully analysed.
The additional incidence assessment is prone to potential
selection bias as the search strategy was primarily intended
for risk factors. Studies are restricted to four out of seven con-
tinents with therefore questionable generalizability of these
data worldwide, where demographic variables may differ.
CONCLUSION
Shoulder dystocia, macrosomia, maternal diabetes, instru-
mental delivery, and breech delivery are the main risk
factors for NBPP, with shoulder dystocia presenting the
highest risk. Prevention remains difficult owing to the
unpredictability of these factors and their often labour-
relatedness. Moreover, many risk factors are interrelated.
Caesarean section is a protective factor for NBPP. The
incidence of NBPP has decreased over time, possibly as a
result of increased awareness of the risk and improved
obstetric strategies. In the view of its lifelong impact, the
risk for NBPP, its severity, and morbidity should be fur-
ther lowered. The focus needs to be on risk stratification,
antenatal counselling, enhanced labour surveillance, and
simulation training. This study highlights the need for fur-
ther research to determine predictable and modifiable risk
factors with emphasis on their relationship. As this meta-
analysis integrates results of known risk factors, future
research should also focus on undiscovered ones.
A CK N O W L E D G E M E N T S
We thank Kristine Oostra and Wim Vanhove for their scientific
support and sharing their clinical expertise. The authors have sta-
ted that they had no interest that could be perceived as posing a
conflict or bias.
SUPPORTING INFORMATION
The following additional material may be found online:
Figure S1: Search strategy in the systematic review and meta-
analysis of risk factors for NBPP.
Figure S2: PRISMA flow diagram of study selection process in
the systematic review and meta-analysis of risk factors for NBPP.
Figure S3: (a) Forest plot of shoulder dystocia. (b) Forest plot
of macrosomia. (c) Forest plot of diabetes. (d) Forest plot of
instrumental delivery. (e) Forest plot of breech delivery. (f) Forest
plot of caesarean section.
Figure S4: Forest plot of incidence with subgroups per time-
frame.
Table S1: Eligibility criteria for study selection in the system-
atic review and meta-analysis of risk factors for NBPP.
Table S2: Characteristics of included studies in the systematic
review and meta-analysis of risk factors for NBPP.
Table S3: List of historic risk factors derived from 22 included
articles in the systematic review and meta-analysis of risk factors
for NBPP.
Table S4: Reporting bias across studies evaluated by STROBE
in the systematic review and meta-analysis of risk factors for
NBPP.
Table S5: Individual study risk of bias assessment for the stud-
ies using the Newcastle-Ottawa Scale in the systematic review and
meta-analysis of risk factors for NBPP.

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