Global health

CASE REPORT

Birth brachial plexus palsy: a race against time

Sambeet Patra,1 Jayakrishnan K Narayana Kurup,2 Ashwath M Acharya,2 Anil K Bhat2
1
Kasturba Medical College, SUMMARY Examination findings
Manipal University, Manipal, A 5-year-old child presented to us with weakness of the On examination, the child was cooperative and
Karnataka, India
2
Hand and Microsurgery Unit,
left upper limb since birth. With the given history of obeyed our commands with average intelligence.
Department of Orthopedics, obstetric trauma and limb examination, a diagnosis of His left shoulder was in an attitude of 10° abduc-
Kasturba Medical College, birth brachial plexus palsy was made. Brachial plexus tion, elbow flexed at 30°, wrist extended at 50°,
Manipal University, Manipal, exploration along with microsurgery was performed at and metacarpophalangeal joints flexed at 70° with
Karnataka, India
the same time which included extrinsic neurolysis of the thumb in palm deformity. The modified Mallet
Correspondence to roots and trunks and nerve transfer for better shoulder Scores were grade IV for shoulder abduction, grade
Dr Jayakrishnan K Narayana external rotation and elbow flexion. Both the movements III for external rotation, and grade II for hand to
Kurup, jkdoctorjk@gmail.com were severely restricted previously due to co-contractures spine, hand to neck and internal rotation (figures 1
with the shoulder internal rotators and triceps. The and 2, video 1). The Gilbert and Raimondi Scale
Accepted 20 June 2016
problem of birth brachial plexus palsy is proving to be a for elbow function revealed a score of 2 for
global health burden both in developed countries and in flexion, 1 for extension and −1 for extension
developing countries such as India. The lack of deficit. The Raimondi scale for hand function score
awareness among the general public and primary was 2. Except for finger extension, active move-
healthcare providers and inadequate orthopaedic and ment could be demonstrated in all the joints.
neurosurgeons trained to treat the condition have Except for the shoulder joint, the excursion of
worsened the prognosis. This case lays stress on the other joints was very poor. Perhaps the most disab-
delayed complications in birth brachial palsy and its ling feature was that of a hallmark severe
effective management. co-contracture between the muscles of the shoul-
der, elbow, forearm and hand. For every movement
of the hand, there was simultaneous movement of
CASE PRESENTATION the elbow and shoulder and vice versa. The clas-
A 5-year-old firstborn male child was brought by sical trumpet sign was present. On giving instruc-
his parents with symptoms of multiple deformities tion to take an object towards the mouth the child
of the left upper limb since birth. The mother had could neither complete flexion nor extension of the
an uneventful antenatal history. The child was born elbow in spite of presence of fairly good palpable
at full term by assisted vaginal delivery using contraction in the arm as both the biceps and
forceps in view of shoulder dystocia during labour. triceps were firing simultaneously. In effect, there
The birth weight of the child was 3.9 kg and he did was no useful function in the left upper limb. The
not cry immediately after birth, following which he only use of the limb was in holding small objects
was intubated and referred to a higher centre. He with poor grasp. A diagnosis of birth brachial
was admitted to a neonatal intensive care unit for plexus palsy (BBPP) of the left upper limb was
6 days and resuscitated, following which he was made and the parents were counselled regarding
discharged. Parents noticed the inability of the the need for microsurgery to improve the func-
child to move his left upper limb at around tional status of the child’s left upper limb. The
1 month but did not seek any treatment for 1 year. parents had recently come to know about the
They then took the child to a family physician who health insurance schemes of the state government
advised physiotherapy which was continued for and hence agreed for the surgery.
1 month and then stopped. He was later taken to a
traditional complementary medicine practitioner Operative procedure
who treated the child by immobilisation with splint Brachial plexus exploration and microsurgery was
and weights which did not result in improvement done at the same time. On exploration of the bra-
of his function. At around 2 years of age, they con- chial plexus, extensive fibrosis was observed involv-
sulted a paediatrician who suggested seeking an ing all the roots and trunks. Extrinsic neurolysis of
opinion for surgical correction of the deformity for the roots and trunks were done. It was decided to
which the parents were reluctant due to financial do a nerve transfer to get a better shoulder external
constraints. rotation and elbow flexion which was severely
restricted due to co-contractures of the shoulder
Social and personal history of the child internal rotators and triceps. The spinal accessory
To cite: Patra S, Narayana The child was doing well and was attending an nerve was transferred to the suprascapular nerve;
Kurup JK, Acharya AM,
et al. BMJ Case Rep
Anganwadi school daily in spite of his disability. He four intercostal nerves were harvested from the
Published online: [please had good social interaction with friends and played third to sixth intercostal space and transferred to the
include Day Month Year] games and sport which involved minimal involve- musculocutaneous nerve. Since there was no exten-
doi:10.1136/bcr-2016- ment of the affected hand. His general growth and sion at the digits, it was decided to do tendon trans-
215996 development was appropriate for his age. fer as the nerve transfer option was not available for
Patra S, et al. BMJ Case Rep 2016. doi:10.1136/bcr-2016-215996 1
 Global health

Video 1 Shows the incomplete movements of the affected shoulder,

elbow and wrist due to the weakness and co-contractures.

therapy after a month. At 4 months postsurgery follow-up, the

Figure 1 Note the child’s inability to complete shoulder abduction child was able to open up his hand for grip due to the tendon
and elbow extension.
transfer which was done along with the nerve transfer.
finger extension. The extensor carpi radialis longus tendon was
transferred to the extensor digitorum communis and extensor GLOBAL HEALTH PROBLEM LIST
pollicis longus. The postoperative period was uneventful and the 1. Lack of comprehensive global and regional data of the
child was discharged on the fifth day and reviewed for hand problem.

Figure 2 Note that the child is unable to externally rotate the shoulder and also internal rotate completely.
2 Patra S, et al. BMJ Case Rep 2016. doi:10.1136/bcr-2016-215996
 Global health

2. Poor prognosis and outcomes due to delay in presentation Delay in presentation and seeking care
and seeking care. Nerve reconstruction procedures are best performed in infancy
3. Lack of awareness regarding diagnostic criteria for treatment to allow the greatest potential for recovery of muscle strength.
options among the healthcare providers and the public. Coroneos et al14 assessed the time of referral of a case of BBPP
4. Social impact and stress on the family due to problems of to a multidisciplinary centre and found that the majority (55–
accessibility and affordability in seeking appropriate 60%) of patients with BBPP identified at birth were not referred
healthcare. for multidisciplinary care. They classified the referred patients
on the basis of the month in which referrals were done into first
GLOBAL HEALTH PROBLEM ANALYSIS month (good), third month (satisfactory) and beyond 3 months
Epidemiology: global and Indian (poor) referral and found ‘good’ in 28%, ‘satisfactory’ in 66%,
BBPP, in spite of significant advances in medical healthcare, con- and ‘poor’ in 34% of the patient referrals. Primary care provi-
tinues to be a major burden reported worldwide with a fre- ders lack the expertise and can underestimate the injury and
quency not very different from the records published since six provide guardians with inaccurate information and educa-
decades.1 Recent trends continue to show an incidence of just tion.15–17 This results in guardian distress and delayed specialist
over 1 per 1000 births both in developed countries such as the referral.15
USA and in developing countries such as India.2 3 There is
sparse literature evidence on the incidence and prevalence of
Lack of regional data and usage of services
this injury in India. Bhat et al3 in their study among 32 637
The fact that hardly half of the patients have one or more of
deliveries over a period of 10 years noted an incidence of 1/
modifiable risk factors suggests an unknown mechanism which
1000 live births, which is comparable to other studies done
needs further elaboration and research involving multiple
worldwide. However, this may represent just the tip of the
centres.2 18 Systematic analysis on the natural history of BBPP
iceberg due to the lack of coordinated reporting of cases, lack of
has shown the fallacy and lack of scientific evidence in the often
BBPP National registries and due to a substantial amount of
quoted excellent prognosis of over 90% in such birth injuries.19
non-hospital deliveries in rural areas. The incidence of BBPP is
These studies also mention the presence of permanent residual
estimated to range from 0.4 to 4 per 1000 live births.4–6 The
deficits which may be as high as 30% needing microsurgical
incidence in the USA and Canada is 1.5/1000 and 1.24/1000
intervention for prevention in the first year of life.5 18–20 Even
live births, respectively. There are no global or Indian data avail-
in developed countries, studies have shown underutilisation of
able on the severity, long-term effects of disability and specific
microsurgical services and ‘limited access to care’ for such con-
coordinated healthcare services available for brachial plexus
ditions where only neonates with private insurance had access
palsy, even though its incidence is equivalent to that of autism
to necessary facilities.21 In India, the burden is even more in the
and Down’s syndrome.
presence of disparities due to socioeconomic status, rural/urban
divide, rising hospital costs compounded by poor emergency
Risk factors
services in primary health systems with high out-of-pocket
Shoulder dystocia, prolonged or difficult labour, fetal macroso-
expenditures met by more than three-fourth of the house-
mia, instrumental deliveries, etc, are the most important peri-
holds.22 23 In India, three-fourth of healthcare facility and
natal risk factors for BBPP. Chauhan et al2 suggested that
resources are available in urban areas where hardly 27% of the
concordance of the rates between various countries indicate that
population lives.24
the reason for BBPP may not be due to a specific manoeuvre
done by the clinicians and that the palsy can occur without diffi-
culties in delivering an impacted shoulder. Lack of awareness among the public and healthcare
providers
Types of BBPP and prognosis Lack of awareness of healthcare facilities is another problem
Narakas has classified the severity of BBPP from type I to type among the rural poor who do not seek treatment due to cul-
IV. Narakas’s7 type I involves C5–C6 nerve root involvement tural, socioeconomic and geographic reasons added on by state’s
and is the most common type of BBPP, carrying the most favour- difficulty to integrate primary centres referrals with tertiary
able prognosis. Type II is an injury to the C5, C6 and C7 nerve centres as seen with our patient. Our patient’s parents were in
roots and carries a poorer prognosis than type I. Type III is a the below poverty level group hailing from a rural background,
global palsy involving the C5, C6, C7, C8 and T1 nerve roots. residing 500 km away from our hospital. The parents were
Type IV is the most severe form with global palsy along with unaware of the government subsidy provided for surgeries
Horner syndrome. Neurapraxias recover fully in the first few related to neurological and congenital deficits for children.
months of life. Avulsion injuries ( preganglionic) in which the Unfortunately, these problems are further aggravated by a lack
nerve roots are completely disconnected from the spinal cord of information among the medical fraternity about the recent
and characterised by Horner syndrome, winging of scapula, advances in the management of BBPP, particularly among pae-
phrenic nerve palsy and cannot recover. Approximately 60% of diatricians and orthopaedic surgeons who mainly deal with this
neonatal injuries are mild and spontaneously resolve.4 Active condition in our country. An internal audit among paediatricians
Movement Scores grading system is available, which is reliable and obstetricians in our institution showed that one-third were
in infants and guides the treating doctor about surgical plan- unaware of the current guidelines for management for BBPP
ning.8 9 Many of the caregivers are not aware of such a scoring with respect to microsurgical reconstruction (unpublished data).
system. Early microsurgical intervention is recommended for Those who were aware suggested that the reasons for the delay
those with Narakas type IV BBPP. Return of elbow flexion were: reluctance of the parents and lack of knowledge in recent
strength is the key factor which determines the need for brachial advancement among surgeons who send these children for hand
plexus exploration and nerve reconstruction with most surgeons therapy. We regularly come across older children and adults like
recommending this procedure when antigravity elbow flexion the one mentioned in our case who might have benefited from
has not returned by 3–9 months of age.10–13 early surgery in infancy but were denied the opportunity due to
Patra S, et al. BMJ Case Rep 2016. doi:10.1136/bcr-2016-215996 3
 Global health

various reasons including lack of timely referral and inadequate for the children and their caregivers to meet similar people and
training and skill of the medical personnel. share their experience and provide mutual support.

Impact on family
Having a child with BBPP can affect a family through social, Patient’s perspective
financial and emotional factors. Most often, the impact of the
child’s disability is underestimated. Focus of care often gets con-
centrated on treating the child’s injury, and attention is diverted We belong to a lower middle-class family. This disease has
from the impact on the family. Louden et al studied the impact brought an upheaval on me and my family because of its
of BBPP on the family using the Impact on Family Scale (IFS). prolonged course. I feel sad that my son could not move his
The IFS is a self-administered four point (strongly agree to hand like other children. I did not realise it earlier but noticed
strongly disagree) scale consisting of 27 questions which mea- when he failed to hold objects and play with his hand like his
sures a parent’s perception of their child’s health condition on peers. I do not know clearly the reason for my child’s disability.
family life,25–27 and has four dimensions: financial burden; Doctors mentioned that it had something to do with the
familial/social challenges; personal strain of the primary care- difficult delivery I had. I have taken my son to many hospitals
taker’s and the family’s ability to cope with the stress through before coming here. Initially, nobody advised surgery for my
mutual support.26 27 A total of 102 caregivers were included in child and we wasted a lot of precious time. The doctors here
the study. According to them, the areas which were most have diagnosed and operated on my son. The treating doctor
affected were strain for the family members, social interactions told us that if surgery had been done very early in his life, our
and economic burden. The income and level of education corre- son would have got very good results with the movement of his
lated negatively with the impact on the family. The distance left upper limb. There was no one to guide us properly
travelled for BBPP treatment services was a major concern for regarding the seriousness of his problem. We feel guilty now
the caregivers and caused a huge economic burden, especially that at the age of 2 years, when another doctor advised surgery
for those who travelled nationally compared with those travel- for him, we were reluctant due to financial constraints and did
ling locally. Mothers of children with BBPP were found to have not go ahead with the surgery. We had difficulty covering the
a lower quality of life than healthy controls and psychological expenses, but the insurance policy offered by the state
stressors were more for them.28 29 Akel et al30 reported poorer government provided some help. I understand that he has to
quality of life scores in children with BBPP compared to their undergo regular physiotherapy exercises and follow-up, which
healthy peers. Parental distress and dissatisfaction with commu- will be challenging because of the lack of local physiotherapy
nication with the treating doctor on details of diagnosis and centres and I will have to carry him all the way here for
treatment information has been reported in BBPP. This situation follow-up.
is even worse among illiterate parents and in rural areas of
India.

Healthcare and insurance system in India Learning points

In India, healthcare is mainly the responsibility of state govern-
ments with assistance from the central government. The total
expenditure on public health expenditure as a percentage of
▸ Birth brachial plexus palsy continues to prove a burden for
gross domestic product in India as per the World Bank data is
developed and developing countries due to the involvement
4%, but it is far below the global median of 8.7%%.31 The lack
of multiple risk factors.
of adequate funding for public health forces people to purchase
▸ Detailed and clear guidelines regarding diagnostic criteria for
services from the private sector. The private medical sector is
surgical intervention versus conservative approach should be
the mainstay of healthcare in India providing service to ∼70%
made aware to all healthcare providers.
of the population in urban and rural areas.32 Most of the
▸ Measures should be taken to spread awareness among the
expenses for healthcare are paid out of pocket by the patients.
general public regarding the presentation and outcome of
As per the World Bank data, only about 25% of the Indian
the disease.
population had some form of health insurance in 2010.33
▸ Affordable and easily accessible healthcare should be
However, a study by the Indian government done in 2014
provided to treat these children to avoid late complications
found that the World Bank data was an overestimate and only
and help them achieve full recovery potential.
about 17% of the population had health insurance coverage.34
A universal healthcare system is still not in place in India. This
has led to a detrimental effect on the basic standard of living of Contributors All authors contributed to conception and design of the paper, SP
people, especially in rural areas. and JKN has done the acquisition of data, drafting the article AA and AKB has done
This calls for a multidisciplinary approach to address the corrections in the final manuscript, and was involved in the final approval of the
lacunae in treatment, education and creating awareness, and version to be published.
providing resources and financial support for these families. Competing interests None declared.
Creating proper awareness through clinician-led camps, family Patient consent Obtained.
programmes and other public teaching modalities may prove to Provenance and peer review Not commissioned; externally peer reviewed.
be an effective first step in helping families. Squitieri et al,21 in
their study on adolescents who experienced BBPP, have stressed
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