Abstract
Background: The long-term results of surgical treatment of brachial
plexus birth palsy have not been reported. We present the findings of a
nationwide study, with a minimum five-year follow-up, of the outcomes of
surgery for brachial plexus birth palsy in Finland.
Methods: Of 1,717,057 newborns, 1706 with brachial plexus birth
palsy requiring hospital treatment were registered in Finland between 1971 and
1997. Of these patients, 124 (7.3%) underwent surgery on the brachial plexus
at a mean age of 2.8 months (range, 0.4 to 13.2 months). The most commonly
performed surgical procedure was direct neurorrhaphy after neuroma resection.
One hundred and twelve patients (90%) returned for a clinical and radiographic
follow-up examination after a mean of 13.3 years. Activities of daily living
were recorded on a questionnaire, and the affected limb was assessed with use
of joint-specific functional measures.
Results: Two-thirds (63%) of the patients were satisfied with the
functional outcome, although one-third of all patients needed help in
activities of daily living. One-third of the patients, including all nine with
a clavicular nonunion from the surgical approach, experienced pain in the
affected limb. All except four patients used the hand of the unaffected limb
as the dominant hand. Shoulder function was moderate, with a mean Mallet score
of 3.0. Both elbow and hand function were good, with a mean score on the
Gilbert elbow scale of 3 and a mean Raimondi hand score of 4. Incongruence of
the glenohumeral joint was noted in sixteen (16%) of the ninety-nine patients
in whom it was assessed, and incongruence of the radiohumeral joint was noted
in twenty-one (21%). The extent of the brachial plexus injury was found to be
strongly associated with the final shoulder, elbow, and hand function in a
multivariate analysis.
Conclusions: Following surgical treatment of brachial plexus birth
palsy, substantial numbers of the patients continued to need help performing
activities of daily living and had pain in the affected limb, with the pain
due to a clavicular nonunion in one-fourth of the patients. The strongest
prognostic factor predicting outcome appears to be the extent of the primary
plexus injury.
Level of Evidence: Therapeutic Level III. See
Instructions to Authors for a complete description of levels of evidence.
Brachial plexus birth palsy was first described by
Smellie1 in 1764.
Erb2 presented his
classic description of the injury in 1874, and his name became the term for
paralysis of the upper nerve roots of the brachial plexus. In previous
studies, the reported incidence of brachial plexus birth palsy has ranged from
0.42 to five per 1000
newborns3-7.
Most commonly, the C5 and C6 nerve roots are affected, and nerve roots from C7
to T1 are also injured, resulting in total palsy, in less than half of
patients8,9.
The severity of the neural injury ranges from mild neurapraxia to total
nerve-root avulsion.
Despite past debate about the cause of this brachial plexus
injury10-12,
trauma during delivery is now the generally accepted etiology. According to
Sever's13
experience in the early twentieth century, most children recover
spontaneously. However, the latest study concerning the natural history of
brachial plexus birth palsy, by Hoeksma et
al.14, suggested
that complete neurological recovery occurs in only 66% of the patients.
It has been estimated that close to 15% of patients with brachial plexus
birth palsy have permanent
disability15,16.
Brachial plexus surgery was first performed in patients with brachial plexus
birth palsy in the early twentieth century by
Kennedy17. However,
surgical intervention was abandoned because of high mortality rates and
unconvincing
results18. Brachial
plexus reconstruction gained more popularity with advances in microsurgery and
children's anesthesia in the 1970s. The results of adult brachial plexus
surgery and the initial favorable reports by
Narakas19 as well
as Meyer20 offered
convincing evidence of the benefits of surgery in properly selected patients
with brachial plexus birth palsy. Gilbert and
Tassin21
popularized the use of the absence of biceps contraction at three months of
age as the criterion for operative treatment. However, other authors have
questioned the timing of plexus repair and have recommended a longer
observation
time22,23.
Despite microsurgical repair of the brachial plexus during early infancy,
residual deformities of the shoulder, elbow, and hand are often noted. In
previous studies, results and follow-up time have varied, and the long-term
results of surgery for brachial plexus birth palsy have not been reported, to
our knowledge.
The purpose of our study was to determine the long-term results of surgery
for brachial plexus birth palsy, with special reference to the clinical and
radiographic status of the shoulder, elbow, and hand.
We conducted a population-based retrospective follow-up study of 112
patients who had undergone surgical treatment for brachial plexus birth palsy.
Data were collected from medical records and from the information obtained
during follow-up visits between September 2002 and October 2003.
The National Research and Development Center of Welfare and Health (STAKES)
collects data on all birth injuries in Finland into two registers: the
Hospital Discharge Register, which lists patients with birth injuries
requiring hospital treatment, and the Medical Birth Register, which lists all
births. During the twenty-seven-year study period from 1971 to 1998, 1,717,057
infants were born in Finland and 1706 with brachial plexus birth palsy were
listed in the Hospital Discharge Register; thus, the incidence of brachial
plexus birth palsy according to this register was one per 1000 newborns.
Combining the data (1996 through 2002) from the Medical Birth Register and the
Hospital Discharge Register revealed an incidence of 3.05 per 1000 newborns.
Medical Birth Register data are based on the findings of the clinical
examination performed by a pediatrician for all newborns when they are two
days of age. According to our recent (unpublished) prospective epidemiological
study, the incidence based on the combined Medical Birth Register and Hospital
Discharge Register data appears to be a slight overestimation since these data
include some patients with a birth fracture of the clavicle that was reported
as a brachial plexus birth palsy. A total of 124 patients who had undergone
surgery for brachial plexus birth palsy were identified from the medical
records of nine hospitals that treat brachial plexus birth palsy in Finland.
All of the procedures in the study period were performed by eleven Finnish
surgeons. Since the population of Finland is very stable, all 124 patients
could be contacted. The Ethics Research Board of Helsinki University Hospital
approved the study. Permission to contact the subjects was granted by the
various hospital districts in Finland.
Patients
All patients (fifty-three males and seventy-one females) were asked to
participate in the present study, and 112 (90%; forty-seven males and
sixty-five females) agreed. The mean postoperative follow-up time was 13.3
years (range, 5.0 to 31.5 years). The average birth weight was 4457 g (range,
2890 to 6300 g). The left side only was affected in fifty-two patients (46%),
the right side only was affected in fifty-six (50%), and the injury was
bilateral in four (4%). Brachial plexus surgery was performed by eleven
consultant orthopaedic or hand surgeons in nine hospitals from August 1971 to
December 1997. Two of them performed sixty (54%) of the operations
(Table I). Those surgeons are
referred to as experienced surgeons, and the remainder are considered
inexperienced (performing less than ten operations) in the subsequent
analysis.
General indications for brachial plexus surgery were either total paralysis
at two months of age or no recovery of elbow flexion at three months of age.
However, during the early years of this study, indications were not well
established and the four pioneering surgeons preferred to operate earlier. The
mean age at the time of the operation was 2.8 months (range, twelve days to
13.2 months). The extent of the injury was defined on the bases of collective
data obtained from preoperative electromyography (100 of the 112 patients),
cervical myelography (forty-nine patients), intra-operative findings, and
physical examination at the time of follow-up. Electromyographic findings
showed a C5-C6 lesion in forty-seven patients (47%), a C5-C7 lesion in
thirty-two (32%), and total involvement in twenty-one (21%). According to the
operative findings, sixty-three patients (56%) had a C5-C6 injury, thirty-five
(31%) had a C5-C7 injury, and fourteen (13%) had a total injury. Collective
data including the findings at the follow-up examination, which were used in
the subsequent analysis, showed that fifty-four patients (48%) had a C5-C6
palsy, thirty-one (28%) had a C5-C7 palsy, and twenty-seven (24%) had a total
injury. None of the patients had an isolated lower plexus injury. The
operations are listed in Table
II. Neuroma resection and endto-end neurorrhaphy were used in
sixty-five patients (58%). Osteotomy of the clavicle, which was performed
routinely in the early years and in patients with a total injury, was done in
thirty-seven patients (33%).
During the follow-up period, secondary operations for contractures or
deformities of the limb were performed in sixty-three patients (56%), with
thirty-one (28%) having more than one operation. A total of ninety secondary
operations were performed (Table
III).
Other neurological comorbidities were recorded in the files for eight
patients (7%). Four of them had attention deficit, two were mentally retarded,
and two had hemiparesis on the unaffected side. The prevalence of neurological
comorbidities in this series did not differ from that in the general pediatric
population in
Finland24.
Assessment of Patients
The patients were assessed with a questionnaire that inquired about general
health, hand dominance, fractures or burn injuries, the need for help in
activities of daily living, and satisfaction with the present situation. A
visual analogue score, in which 0 indicated poor and 10 indicated excellent,
was used to evaluate the patient's function and the appearance of the affected
limb.
Physical and Radiographic Examination
Physical examination was performed by an independent observer (M.K.).
Shoulder function was evaluated with use of a modified Mallet scoring
scale25 (see
Appendix), in which each of five steps can be classified as II, III, or IV.
The sum of these steps was divided by five, and the mean value was calculated.
Stability of the glenohumeral joint was classified clinically as stable,
subluxated, or dislocated. The patients were observed for shoulder muscle
atrophy, an abnormal position of the scapula, and possible pseudarthrosis of
the clavicle.
Anteroposterior and axillary radiographs were made for ninety-nine (88%) of
the 112 patients. Radiographs could not be made for thirteen patients because
of pregnancy or the patient's refusal. The shape of the osseous articular
surface of the glenoid was subjectively classified on an arbitrary scale as
normal, concave; blunt posteriorly; or convex, deformed. Congruity of the
glenohumeral joint was estimated subjectively to be congruent, posteriorly
subluxated, posteriorly dislocated, anteriorly subluxated, or anteriorly
dislocated. Radiographs of the clavicle were made if pseudarthrosis was
suspected clinically.
Elbow function was assessed with use of the Gilbert
scale26 (see
Appendix), and hand function was evaluated with use of the Raimondi
scale27 (see
Appendix). Dynamic stability of the radial head in pronation and supination
was assessed clinically. Congruency of the radiohumeral joint was estimated
from the anteroposterior and lateral elbow radiographs to be congruent,
subluxated, or dislocated.
Statistical Methods
Values are given as the mean and range. The Mann-Whitney U test was used to
examine differences between continuous variables. Correlations were analyzed
with use of the Spearman rank correlation test. Odds ratios for the presence
of a good clinical outcome and their 95% confidence intervals for the
different patient groups were derived with logistic regression models (NCSS
6.0; NCSS Statistical Software, Kaysville, Utah). The age of the patient at
the time of the operation, the sex of the patient, the extent of the injury
(C5-C6, C5-7, or C5-C8 [total]), and the experience of the surgeon were
considered as independent covariates in the analyses. The surgical procedure
(neurolysis, neurorrhaphy, grafting, or neurotization) was closely associated
with the type of neural injury (nerve root avulsion or no avulsion) and the
extent of the neural injury; therefore, neither of these covariates could be
included in the same logistic regression model. Two-tailed p values of
=0.05 were considered to be significant.
The mean age of the 112 patients at time of the final clinical and
radiographic follow-up was 13.5 years (range, 5.6 to 31.5 years).
Patient Assessment
The unaffected upper limb was dominant in all but four (4%) of the
patients. Thirty-five patients (31%) had pain in the affected upper limb. Pain
was experienced daily by three patients, two or three times a week by five,
weekly by eight, and at least once a month by nineteen. Pain was not related
to age, the extent of the injury, the type of surgery, radiographic findings,
or secondary operations. However, nine patients with radiographically
confirmed clavic ular nonunion had local pain at the clavicle. The mean visual
analogue score for function and appearance of the affected upper limb was 5.2
(range, 1 to 10) and 6.1 (range 1 to 10), respectively. Twelve patients (11%)
reported burn injuries on the affected side, and seventeen (15%) reported
other injuries. Thirty-nine (35%) of the patients needed help with activities
of daily living, including sixteen of the twenty-nine patients who were
younger than ten years of age and twenty-three of the eighty-three older
patients. Detailed information on activities of daily living is given in
Table IV. The main problems
were with washing hair, cutting food, and buttoning a shirt. The patients'
satisfaction with their functional outcome is presented in
Figure 1.
Results of the Physical and Radiographic Examinations
Shoulder
The mean Mallet scale score for shoulder function was 3.0 (range, 2.0 to
4.0). Twenty of the fifty-four patients with a C5-C6 palsy and four of the
thirty-one with a C5-C7 palsy were observed to have good shoulder function
(Mallet class IV). The Mallet shoulder scores ranged from 2 to 3 for the
patients with a total palsy. The majority of the patients had limited active
external rotation of the adducted shoulder: sixty-two (55%) had <0°,
twenty-three (21%) had 0° to 20°, and twenty-seven (24%) had
>20°. Fourteen patients (13%) had posterior instability of the
glenohumeral joint, and three of them could voluntarily dislocate the joint
posteriorly. With the numbers available, there was no association between the
extent of the injury and instability of the glenohumeral joint or between
posterior instability of the shoulder and secondary surgical procedures. All
except two patients had atrophy of the shoulder muscles and a hypoplastic
cranially positioned scapula.
The glenohumeral joint was congruent in eighty-three patients (84%),
posteriorly subluxated in nine (9%), posteriorly dislocated in three (3%), and
anteriorly subluxated in four (4%). Anterior dislocation was not noted. The
shape of the glenoid cavity was normally concave in sixty-five patients (66%),
blunt posteriorly in eighteen (18%), and convex, deformed in sixteen (16%).
Patients with a congruent glenohumeral joint had a higher mean Mallet score
(3.1) than patients with an incongruent joint (2.7), although this difference
was not significant (p = 0.067). Radiographic findings were not associated
with secondary procedures on the shoulder.
Osteotomy of the clavicle was performed in thirty-seven patients (33%), and
pseudarthrosis of the clavicle was suspected on the basis of the physical
examination in nine (24%) of these patients. The diagnosis was confirmed on
the radiographs. The pseudarthrosis was symptomatic in all of these
patients.
Elbow
The mean score on the Gilbert scale for elbow function was 3 (range, -1 to
5). An extension deficit of the elbow exceeding 29° was noted in
thirty-four patients (30%). Patients without a subluxated or dislocated radial
head had a mean Gilbert elbow rating of 2.5, and the remainder had a mean
rating of 3.0. Radial head instability did not correlate with pain.
Instability of the radial head was suspected clinically in three patients with
total palsy, but none reported pain at the elbow. Radiographically, the
radiohumeral joint was congruent in seventy-eight patients (79%), subluxated
in sixteen (16%), and dislocated in five (5%). Patients with a congruent
radiohumeral joint had a significantly higher mean Gilbert score than patients
with incongruency (3.9 compared with 2.2, p = 0.001). Secondary surgical
procedures at the elbow or forearm were performed in 10% and 33% of the
patients, respectively.
Hand
The mean score on the Raimondi scale for hand function was 4 (range, 1 to
5). Hand function was impaired (a score of 0 to 4 on the Raimondi scale) in
twenty-six patients (31%) with no previously diagnosed C8 nerve-root injury.
However, the patients with poor hand function (a score of 0, 1, or 2 on the
Raimondi scale) did have C8 nerve-root injury.
Function According to Extent of Injury
As assessed with the Mallet, Gilbert, and Raimondi scores, the group with a
C5-C6 injury did not differ significantly from the group with a C5-C7 injury,
whereas the patients with a total injury had significantly lower scores
according to all of the assessment scales
(Fig. 2).
Correlations
In the univariate analysis, there was a significant inverse correlation
between the extent of the injury and the Mallet score (rs = -0.59,
95% confidence interval = -0.70 to -0.45, p < 0.0001), the Gilbert score
(rs = -0.68, 95% confidence interval = -0.77 to -0.56, p <
0.0001), and the Raimondi score (rs = -0.54, 95% confidence
interval = -0.66 to -0.39, p < 0.0001)
(Fig. 2). Similarly, the type
of surgery was associated with the Mallet score (rs = -0.21, 95%
confidence interval = -0.38 to -0.02, p = 0.0302), the Gilbert score
(rs = -0.21, 95% confidence interval = -0.39 to -0.02, p = 0.029),
and the Raimondi score (rs = -0.47, 95% confidence interval = -0.60
to -0.31, p < 0.0001). Correlations between the type of surgery and
function were investigated with a scoring system in which 0 indicated
exploration or neurolysis; 1, neurorrhaphy; 2, grafting; 3, neurotization; and
4, a combination of the aforementioned techniques. The Mallet, Gilbert, and
Raimondi scores after the different surgical procedures are shown in
Table V. Patients with nerve
root avulsion injury had inferior results for shoulder (p = 0.028), elbow (p =
0.0056), and hand function (p = 0.0024). After we adjusted for the age and sex
of the patient and the experience of the surgeon, the extent of injury was
closely associated with a satisfactory outcome
(Table VI). Similarly, after
the same adjustment, an age of more than three months at the time of the
operation was associated with satisfactory hand function (odds ratio = 4.18,
95% confidence interval = 1.09 to 16.01, p = 0.036). The end result for the
group of patients who needed secondary surgery was inferior to that for the
remainder of the patients, despite the reconstructive soft-tissue or bone
procedures (p = 0.02 for shoulder function, p = 0.23 for elbow function, and p
= 0.061 for hand function). With the numbers studied, the experience of the
surgeon was not significantly associated with satisfactory hand function (p =
0.089).
Brachial plexus birth palsy is a complication of delivery that resolves
spontaneously in the majority of patients. It is still unclear if surgery can
alter the natural history of this condition in patients with incomplete
resolution of the neural injury. Surgically treated patients often have
permanent disabilities, and secondary operations are frequently required,
especially for deformities of the shoulder. The early results of brachial
plexus surgery are well documented in the
literature19,20,28-31,
and three previous reports have presented mid-term results, after five to
seven years of
follow-up23,32,33.
We present the results of a retrospective population-based study with
long-term follow-up. The follow-up rate was high, exceeding 90%. Our results
were not obtained from one tertiary center; instead, the surgical procedures
were performed in nine hospitals by eleven different surgeons. Furthermore,
the indications for primary and secondary surgery as well as the surgical
techniques varied during the study period, which reflects the inherent
complexity of the diagnosis and treatment of brachial plexus birth palsy.
The final outcome was evaluated clinically with use of the modified Mallet
scale for shoulder function, the Gilbert scale for elbow function, and the
Raimondi scale for hand function as well as by patient assessment. There is no
one uniformly accepted outcome measurement for the entire upper extremity. In
addition, the Mallet scale has been used in many different
ways25. However,
several centers currently use these scales. Clinical and radiographic
examinations were done by independent observers in the present study. The
effect of each independent variable on the outcome was analyzed with use of a
logistic regression model.
According to our findings, the unaffected hand was nearly always dominant.
The prevalence of left-handedness is estimated to be around 10% in the normal
population34. In
the present study the prevalence was 50%, and the right upper limb had been
affected by the birth palsy in all patients with left-hand dominance. Patients
with brachial plexus birth palsy are susceptible to burn and other injuries,
probably because of the restricted range of motion and impaired sensation of
the hand. One-third (35%) of the patients needed help with activities of daily
living, which is a high prevalence considering their average age of 13.5 years
at the time of final follow-up. This need of help with activities of daily
living by patients with brachial plexus birth palsy has not been reported
previously, to our knowledge.
In this study, 31% of the patients had either occasional or daily pain in
the affected upper limb. However, 54% of the patients who experienced pain
were very satisfied or satisfied with the outcome, and pain is probably an
underestimated symptom among these patients. In contrast, Anand and Birch did
not report evidence of chronic or neuropathic pain in their series of
twenty-four
patients35. In the
early years of our study, osteotomy of the clavicle was routinely performed,
especially in patients with total palsy, in order to improve surgical
exposure. Osteotomy was rarely performed during the later years of the study,
and according to our present practice it is not necessary. Clavicular nonunion
resulting from the surgical approach was the main reason for the pain in nine
of our patients.
It is still unclear if brachial plexus surgery can improve the natural
history of shoulder function in patients with brachial plexus birth palsy.
Only 37% of our patients with a C5-C6 injury had good shoulder function
(Mallet class IV), a finding that is in agreement with that of Birch et
al.32, who reported
a good result after a C5 repair in 33% of their patients. In contrast, Smith
et al.22 reported
that 66% of conservatively treated patients with upper brachial plexus injury
and without biceps function at three months gained Mallet class-IV shoulder
function. Impairment of shoulder function correlated positively with the
extent of the injury in our series as well as in the study by Birch et al. The
majority (57%) of our patients had an internal rotation contracture of the
shoulder joint, which was probably due to muscle
imbalance36,37.
Glenohumeral incongruency, mainly posterior subluxation or dislocation, was
noted in 16% of the patients in our study. Glenohumeral incongruency has been
previously reported in up to 30% of patients with brachial plexus birth
palsy36,38,39.
Hand function also may be impaired in patients with an injury classified as
upper nerve-root palsy according to preoperative and intraoperative findings.
The possible reasons for the late diagnosis of lower nerve-root injury may be
difficulties with clinical judgment preoperatively or with the intraoperative
evaluation of the extent of the injury. However, the patients with poor hand
function (a score of 0, 1, or 2 on the Raimondi scale) were diagnosed with C8
and T1 nerve-root injury. These findings may be explained by the difficulties
in the reconstruction of the lower brachial plexus, which have been reported
by many
authors18,40,41.
However, Birch et
al.32 as well as
Haerle and
Gilbert33 recently
reported reasonable recovery of hand function, even after avulsion injuries of
the lower roots, at a mean of less than seven years.
In our analysis, the most important prognostic factor for the prediction of
the final outcome was the extent of the brachial plexus injury. The type of
surgery was closely associated with the extent and type of injury, and
therefore those covariates could not be included in the same logistic
regression model. However, in our univariate analysis, direct nerve repair was
associated with better results than was grafting or neurotization. Experienced
surgeons tended to have better results, but this difference was noted only in
the univariate analysis of the results measured with the Raimondi scale. Our
findings are in accordance with those of earlier reports that showed overall
poorer results with global
injuries22,32,33.
End-to-end repair was used in 58% of our patients, a practice that is contrary
to current
opinion42. The mean
age of our patients at the time of surgery was young, possibly facilitating
end-to-end repairs because of the small size of the child. Furthermore, the
most experienced surgeon in this study favored end-to-end repair. We are not
aware of any studies comparing different surgical techniques for treatment of
brachial plexus birth palsy, but greater surgical experience has been shown to
shorten the operation time and possibly improve the final
outcome33.
The patient's age at the time of the surgery was not found to be associated
with the final outcome in the univariate analysis, but the multivariate
analysis showed operative treatment of lower plexus injuries after the age of
three months to result in better hand function. This finding may, however, be
biased by the extent of the injury. Patients with total paralysis were
generally operated on earlier than patients with some intact function in the
injured upper limb. However, the age at the operation did not predict
results.
There is still no consensus concerning the timing of
surgery29,42-47.
According to Smith et
al.22,
reconstruction is not justified in patients with brachial plexus birth palsy
who have isolated absence of biceps function at three months of age. This idea
was supported by Strömbeck et
al.23, who did not
find any difference in the final outcome between brachial plexus
reconstructions performed before and those done after the age of six months.
Our results are in agreement with those two reports.
The long-term results of primary surgery in the patients with brachial
plexus birth palsy in our study seem to be inferior to those in earlier
reports with short or mid-term
follow-up18,19,28-30,32,33,36.
However, because there is no uniformly used outcome measurement scale,
comparison of results from different sources is difficult. It is possible that
the results of surgery for brachial plexus birth palsy deteriorate with time.
Because of the population-based nature of our study, the patients were from
all over the country and not just from one tertiary center; this probably
influenced the outcome as well as the type of surgery. Few of the surgeons in
our study had extensive experience with this type of surgery, which could
partly explain the differences in the results compared with those from centers
with greater
experience33.
During this study period, 7.3% of patients with brachial plexus birth palsy
requiring hospital treatment had an operation on the brachial plexus, which
adds up to less than ten operations per year in the whole country of Finland.
Most of the patients had persistent sequelae, especially in shoulder function.
The extent of the initial injury was the most important prognostic factor for
the prediction of the final functional outcome. The fact that this relatively
small number of operations was performed by eleven surgeons in nine hospitals
probably made the learning curve longer and more cumbersome. It may be that
the results of surgery for brachial plexus birth palsy would be better if the
operative treatment were concentrated at specific centers.
A figure depicting the Mallet classification and tables demonstrating the
Gilbert elbow and Raimondi hand scales are available with the electronic
versions of this article, on our web site at
(go to
the article citation and click on "Supplementary Material") and on
our quarterly CDROM (call our subscription department, at 781-449-9780, to
order the CD-ROM). ?
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