Abstract
This article presents a detailed review of the history, etiology, assessment protocols, and current treatment for children who suffer an obstetric birth palsy. In addition, new issues facing surgeons who deal with these patients and their families are discussed. Copyright © 2003 by the American Society for Surgery of the Hand
The credit for the first clinical description of obstetric palsy goes to Smellie
[1]
in 1764. The term obstetric palsy was provided by Duchenne[2]
of Boulogne in 1872 in his book in which he describes 4 cases of upper brachial plexus birth palsy. The classic description by Erb[3]
in 1874 concerned the upper brachial plexus paralysis in adults and he defined ″Erb's point” in the neck (the spot where the fifth and sixth cervical roots unite). Currently, the terms Erb's palsy and Erb's-Duchenne's Palsy indicates upper brachial plexus injury involving the C5, C6, and C7 roots.Klumpke
[4]
was the first female intern in Paris and she explained in 1885 the Horner's sign in brachial plexus lesions associated with avulsion of the T1 root. Klumpke later married Dejerine and therefore isolated lower plexus palsy (involvement of the C8-T1 roots) also is called the Dejerine-Klumpke paralysis.Establishing a multidisciplinary birth palsy clinic
The first step to be performed in managing birth palsy cases is to establish a team led by the reconstructive surgeon performing the primary surgery (a plastic surgeon in our center), a neonatologist (who manages associated birth injuries such as asphyxia), an obstetrician (who helps gather information about the delivery and the incidence of the injury), an orthopedic surgeon, an occupational therapist, a physiotherapist, and a psychiatrist. Over the past 8 years, our clinic has assessed over 450 cases of birth palsy.
[5]
Classification of the types of birth palsy and the pathophysiologic forces involved
There are 4 types of birth palsy
[6]
:- 1.Upper (Erb's) palsy (injury to C5, C6, and C7 roots). The newborn with Erb's palsy shows the writer's tip posture of the paralyzed limb. The arm lies internally rotated and adducted at the shoulder, the elbow is extended, the forearm pronated, and the wrist flexed. The intrapartum force causing this palsy is excessive lateral flexion of the neck. In cephalic presentations, it is associated with shoulder dystocia, and in breech presentations, it is associated with difficult delivery of the after-coming head. Erb's palsy is the most common type of birth palsy and has the best chance of excellent spontaneous recovery.
- 2.Total birth palsy (injury to roots C5, C6, C7, C8, and T1). Newborns with total palsy present with flaccid paralysis of the whole upper limb, including the hand. The intrapartum force causing this palsy is the same as in Erb's palsy (excessive lateral flexion of the neck) but the force is much greater. The upper roots are injured first and then, because of excessive force, the lower roots also are damaged. Total palsy is the second most common type of birth palsy and spontaneous recovery usually is incomplete.
- 3.The remaining 2 types of birth palsy (Klumpke's palsy involving C8-T1 roots) and intermediate palsy (mainly involving C7) are only of historic interest and are no longer seen with modern obstetric practice.7,8,9,10
Incidence and risk factors for birth palsy
The average incidence of birth palsy is about 1:1,000 deliveries. However, the incidence varies greatly in the literature. Three large series of consecutive live births in U.S. hospitals found 113 cases in 58,000 births (about 2:1,000),
11
, 12
, 13
whereas Camus et al[14]
saw only 17 cases from over 20,000 live deliveries (less than 1:1,000) at the Pitie Hospital.There are several well-known risk factors for birth palsy, including fetal macrosomia, increased birth weight, shoulder dystocia, maternal short stature, gestational diabetes, instrumental (forceps and vacuum) delivery, and vaginal breech delivery. The birth lesion in breech delivery frequently is severe and it is often bilateral although the birth weight of these babies is small.
[15]
Contrary to popular belief, birth palsy is more common in multiparous rather than primiparous mothers.It must be emphasized that although delivery by caesarean section has a protective role, it is not a guarantee against the occurrence of birth palsy. We reviewed our data and the literature and found that 1% of all reported birth palsy cases were delivered by caesarean section.
[16]
The definition of maternal short stature as a risk factor for birth palsy or even as an indication for caesarean section should vary according to race. I studied 5,000 pregnant Saudi women and compared them with North American women. On average, Saudi women were shorter and gave birth to smaller babies.[17]
Finally, I have shown that multiparous mothers who previously had delivered large infants with brachial plexus injury are at extremely high risk for giving birth to babies with birth palsy in subsequent vaginal deliveries.18
, 19
Evaluation of the infant with birth palsy in the clinic
A data sheet should be designed by every birth palsy center. The sheet documents maternal history, method of delivery, gestational diabetes, birth weight, and associated injuries such as asphyxia, fractures, phrenic nerve palsy, Horner's syndrome, shoulder subluxation, and torticollis.
The classification of the type of palsy (generally either Erb's or total palsy) is documented. Phrenic nerve palsy (indicating that the C4 root also is involved) may be seen in both types of birth palsy.
Horner's syndrome (ptosis, myosis, enopthalmos, and anhydrosis on the ipsilateral side of the face) indicates proximal injury, usually avulsion of the T1 root (disrupting the communicating branch supplying sympathetics to the stellate ganglion). Therefore, Horner's syndrome may be seen in total palsy (and the very rare Klumpke's birth palsy), but not in Erb's palsy.
I am involved frequently in the medicolegal assessment of birth palsy cases, and I have put a list of the possible causes, other than traumatic delivery, of upper-limb paralysis in the newborn, which mimics the clinical picture of birth palsy:
Fracture pseudoparalysis
Fractures of the clavicle and humerus should be ruled out by clinical examination (and, if in doubt, by plain radiographs). These fractures can produce a pseudoparalysis similar in initial presentation to a true brachial plexus lesion. Pesudoparalysis is caused by compression of the brachial plexus by the fractured bone, by swelling around the plexus, or because of involuntary splinting of the arm in the presence of pain. Pseudoparalysis characteristically resolves more rapidly than a true obstetric lesion of the plexus.
[20]
Congenital aplasia of the roots of the brachial plexus
Gilbert
[21]
described 4 cases of congenital aplasia of various roots of the brachial plexus. Three cases had no other congenital anomalies but in one there was an ipsilateral thumb aplasia. The clinical picture mimicked obstetric palsy and the diagnosis was made by surgical exploration.Congenital varicella of the upper limb
This also may mimic birth palsy. The varicella virus damages the dorsal root ganglion and the anterior horn of the spinal cord, leading to both sensory deficiencies and atrophy of muscles. The diagnosis of congenital varicella is suspected if there is a small area of congenital skin necrosis in the limb. A positive history of maternal varicella during pregnancy and serologic examination can confirm the diagnosis.
[22]
Cerebral palsy
Cerebral palsy presents with spastic quadriplegia, hemiplegia, or monoplegia. The latter presentation is rare, but I have seen 1 medicolegal case that presented to my clinic as a case of birth palsy. The diagnosis was easy because the paralysis was spastic, which should not be confused with traumatic birth palsy, which is a flaccid paralysis.
Intrauterine upper-limb nerve compression by the umbilical cord or amniotic bands
A common manifestation of umbilical cord palsy is isolated high radial nerve palsy.
[23]
At the time of birth, limb posture (slightly flexed elbow with a wrist drop) may mimic Erb's palsy, but careful examination of the shoulder (shoulder function in these infants is normal) and electrophysiologic studies will confirm the diagnosis.Deformation syndrome in bicornuate uterus
Dunn and Engle
[24]
described a newborn with left brachial plexus palsy and ipsilateral small arm and deformed ribs. These lesions represented a deformation syndrome exerted by the abnormal uterine septum of the bicornuate uterus.Intrauterine maladaption palsy
This term is given to isolated brachial plexus injury of intrauterine origin without evidence of cord compression, amniotic bands, or bicornuate uterus.
[25]
The major evidence for the intrauterine onset of palsy is based on electromyography. It takes approximately 7 to 10 days for muscle to develop electromyographic evidence of denervation. Therefore, such findings in the first few days of life support the intrauterine onset of palsy.[26]
The birth palsy data sheet also includes the center's muscle grading system. Assessment of movement in each joint of the affected limb is performed according to this grading system. Brachial plexus examination and documentation of the muscle grades are performed at monthly intervals in the first year of life. Meanwhile, the infant continues to undergo physiotherapy exercises to avoid muscle and joint contractures.
Motor assessment in birth palsy
This is one of the most controversial points in birth palsy. Different centers use different muscle grading systems. In our center, we use the Toronto Muscle Grading System
[20]
(Table 1) to assess the motor power of the limb in infants and very young children because it is impossible to test power against resistance or to ask such children to perform a specific task. The reliability of this system to evaluate the motor power of the upper limb in infants also has been confirmed recently.[27]
In older children we use modifications of other muscle grading systems.[28]
Table 1The Toronto Muscle Grading System
| Observation | Muscle Grade |
|---|---|
| Gravity eliminated | |
| No contraction | 0 |
| Contraction, no motion | 1 |
Motion ≤  range | 2 |
Motion  range | 3 |
| Full range | 4 |
| Against gravity | |
Motion ≤  range | 5 |
Motion > range | 6 |
| Full motion | 7 |
Most centers use the Mallet's Muscle Grading System for shoulder function
[29]
(Table 2). Other methods of shoulder assessment include the Gilbert[21]
and Birch[30]
Systems (Table 2). The latter system is interesting and perhaps the most complete. Birch[30]
uses Gilbert's muscle grades to stage the shoulder and added the suffix ″+” to indicate sufficient medial rotation (to assess the ability of the hand to come against the opposite shoulder). Second, he modified the mallet's system into 5 main functions (global abduction, global external rotation, hand to neck, hand to back, and hand to mouth) and by adding the scores of these functions, a good shoulder merits 15 points and a bad one merits 5 points. Finally, Birch[30]
believed that forearm rotation should be included in shoulder assessment because it is almost always abnormal in children with shoulder deformities and it improves after surgical correction.Table 2Muscle grading systems to assess shoulder function
| Mallet's System | Gilbert's System | Birch's Triple System |
|---|---|---|
| I. Flail shoulder | O. Flail shoulder | a. Muscle grade documentation by a modified Gilbert's system |
| II. Active abduction ≤ 30° | I. Abduction or flexion to 45°, no active lateral rotation | b. 5- to 15-point score according to the Mallet system |
| Zero degrees of external rotation | II. Abduction <90°, lateral rotation to neutral | c. Measurement of active pronation and supination of the forearm |
| Hand to back of neck impossible | III. Abduction = 90°, weak lateral rotation | |
| Hand to back impossible | IV. Abduction <120°, incomplete lateral rotation | |
| Hand to mouth with marked trumpet gesture | V. Abduction >120°, active lateral rotation | |
| III. Active abduction of 30° to 90° | VI. Normal | |
| External rotation up to 20° | ||
| Hand to back of neck difficult | ||
| Hand to back with difficulty | ||
| Hand to mouth possible with partial trumpet gesture | ||
| IV. Active abduction over 90° | ||
| External rotation over 20° | ||
| Hand to back of neck easy | ||
| Hand to back easy | ||
| Hand to mouth easy | ||
| V. Normal shoulder |
Assessment of elbow function in older children with birth palsy also has been performed by using various systems such as the Medical Research Council grading system and the Gilbert-Tassin muscle grading system
[31]
(Table 3).Table 3Other muscle grading systems commonly used to assess elbow function
| Medical Research Council System | Gilbert-Tassin System |
|---|---|
| 0 = No contraction | 0 = No contraction |
| 1 = Flicker | 1 = Contraction without movement |
| 2 = Active motion with gravity eliminated | 2 = Slight or complete movement with weight eliminated |
| 3 = Active motion against gravity | 3 = Complete movement against the weight of the corresponding segment of extremity |
| 4 = Active motion against gravity and resistance | |
| 5 = Normal power |
There have been lengthy discussions at the obstetric plexus meetings in Heerlen about the motor assessment of the elbow and hand in birth palsy patients and the systems in Table 4 were developed by Raimondi and Gilbert and were adopted by the Obstetric Brachial Plexus Workgroup.
[32]
Table 4Raimondi's Motor Power System for assessment of elbow and hand function
| Elbow Functional Assessment | Hand Functional Assessment |
|---|---|
| A. Elbow flexion | 0 = Complete paralysis or slight finger flexion of no use, useless thumb, no pinch, some or no sensation |
| 1 = Nil or some contraction | 1 = Limited active flexion of fingers, no extension of wrist or fingers, possibility of thumb lateral pinch |
| 2 = Incomplete flexion | 2 = Active extension of wrist with passive flexion of fingers (tenodesis), passive lateral pinch of thumb |
| 3 = Complete flexion | 3 = Active complete flexion of wrist and fingers, mobile thumb with partial abduction-opposition, intrinsic balance, no active supination, good possibilities for secondary surgery |
| B. Elbow extension | 4 = Active complete flexion of wrist and fingers, active wrist extension, weak or absent finger extension, good thumb opposition with active ulnar intrinsics, partial pronation/supination |
| 0 = No extension | 5 = Same as 4, with finger extension and almost complete pronation/supination |
| 1 = Weak extension | |
| 2 = Good extension | |
| C.Extension deficit | |
| 0 = 0° to 30° | |
| −1 = 30° to 50° | |
| −2 = more than 50° |
Assessment of sensory function
Assessment of sensation in infants is difficult and is mainly performed by the response to painful stimuli. Narakas
[33]
classified the sensory response in infants with birth palsy into 4 grades as seen in Table 5. Our group in Saudi Arabia studied the problems resulting from sensory abnormalities in birth palsy patients such as accidental contact burns of the upper limb[34]
and self-mutilation of the hand.[35]
As expected, the incidence of self-mutilation was higher among children with total palsy than Erb's palsy. Furthermore, mutilation with total palsy generally was more severe and usually involved biting the tips of the digits. Finally, we observed some similarities between self-mutilation in these children and animal autotomy after denervation.Table 5Narakas Sensory Grading System in birth palsy
| Sensory Grade | Observation |
|---|---|
| 0 | No reaction to painful or other stimuli |
| 1 | Reaction to painful stimuli, none to touch |
| 2 | Reaction to touch, not to light touch |
| 3 | Apparently normal sensation |
Investigative procedures for birth palsy
Electromyography and nerve conduction studies
Although these tests are used widely in the evaluation of adult brachial plexus injuries, their usefulness has been questioned in birth palsy. Vredeveld
[36]
observed that denervation occurs and disappears much earlier in newborns than in adults. After a traumatic delivery, denervation may be found as early as the fifth day and usually disappears by 4 months of age, unless the lesion is severe with multiple root avulsions or ruptures. Therefore, the electromyography (EMG) in obstetric palsy is far too optimistic compared with the clinical picture. Another major difference in the EMG of birth palsy is the occurrence of early massive collateral sprouting of denervated muscles.[36]
Smith
37
, 38
has acquired great experience from analysis of over 500 infants, assessing babies with a combination of mixed nerve action potentials and EMG of selected muscles. He classified the lesions into 4 groups: conduction block, mild, significant, and severe axonal injury. The latter 2 groups were associated with neurotmesis or intradural injury. Kono and Birch[39]
matched these preoperative projections with findings at surgeries in 150 cases and showed a high level of accuracy.Spinal computed tomography with contrast and magnetic resonance imaging
Computed tomography with contrast (CT myelography) and magnetic resonance imaging (MRI) were found to be useful by Sloof and Blaauw,
[40]
showing hematoma within the canal, atrophy and denervation of muscle, and avulsions of spinal nerves. Miller et al[41]
preferred MRI because it is noninvasive, gives excellent depiction of pseudomeningoceles, and aids in evaluation of the spinal cord and the soft tissues surrounding the brachial plexus. However, many other investigators (including myself) have questioned the usefulness of these tests in birth palsy. Myelography is too invasive and MRI requires heavy sedation or general anesthesia in these babies. Furthermore, both tests are not wholly reliable in defining avulsion. Clarke and Cutis[20]
reviewed 18 birth palsy cases that underwent CT myelography and compared the results with surgical findings. A total of 90 nerve roots were studied. The sensitivity of myelography at an individual root level was 69% and the specificity was 85%. Overall the positive predictive value of myelography was 50%, showing that the presence of pseudomeningocele was a poor predictor of root avulsion. The negative predictive value was 93%, which means that we can use myelography to rule out root avulsion injuries.It is likely that increasing refinements in MRI such as in the fast spin-echo MRI
[42]
prove valuable in the future and may even allow us to perform the test in these babies without the use of anesthesia.Natural history, prognostic factors for spontaneous recovery, and indications for primary brachial plexus exploration in birth palsy
It is important to note that the earlier-described 3 terms are related but have different implications in the management of birth palsy.
The term natural history means, for example, following-up a group of newborns with Erb's palsy to find out the percentage of children with good or poor spontaneous recovery. This will depend on the referral pattern and the age of the patient when first assessed by the birth palsy center. This explains why different studies of the natural history in birth palsy show both favorable and unfavorable outcomes.
43
, , 45
, 46
, 47
Currently, the best classification available to predict the natural history is the Narakas[48]
classification (Table 6). This classification should be applied after about 2 weeks and should not be used as an indication for surgery.Table 6Narakas Classification System of birth palsy according to the likely outcome after spontaneous recovery
| Group | Nerve Roots Injured | Presentation | Likely Outcome |
|---|---|---|---|
| 1 | C5, 6 | Shoulder paralysis, no elbow flexion | Excellent spontaneous recovery in over 80% of patients |
| 2 | C5, C6, and C7 | Shoulder paralysis, no elbow flexion, wrist drop | Good recovery of the shoulder and elbow in 60% of patients |
| 3 | C5, C6, C7, C8, and T1 (no Horner's sign) | Complete paralysis of the limb with no Horner's sign | Good recovery of the shoulder and elbow in 30% to 50%, good recovery of the hand in the majority |
| 4 | C5, C6, C7, C8, and T1 (with temporary Horner's sign) | Complete paralysis of the lower limb with temporary Horner's sign | Intermediate between groups 3 and 5 |
| 5 | C5, C6, C7, C8, and T1 (with persistent Horner's sign) | Complete paralysis of the limb with a Horner's sign; the limb is marbled and cold | Severe defects in the shoulder and elbow, very poor hand |
The most important 2 prognostic factors for spontaneous recovery are the nerve root involvement (Table 6) and the spontaneous recovery of biceps by 4 months of age. Another important prognostic factor for total palsy is the presence of persistent Horner's sign. We studied this factor in 22 infants and none of them had a satisfactory spontaneous recovery.
[49]
The presence of phrenic nerve palsy or clavicular fractures as predictors of spontaneous recovery are controversial in the literature, but our studies concluded that these 2 factors have no prognostic value in birth palsy.50
, 51
Although all the earlier listed studies of natural history and prognostic factors provide us with several useful insights, what we really need is to predict, at a few months of age, the probable outcome and the need for primary brachial plexus exploration.
[20]
Different centers have used different criteria and these different approaches are classified as follows:
The use of a single muscle score
Gilbert et al,
[52]
who have the largest series of obstetric paralysis cases in the modern literature, relied on the spontaneous recovery of the biceps as the indication for surgery. If the recovery of the biceps had not begun at 3 months of age, the functional prognosis was considered poor, and surgical repair of the plexus was warranted.In our center
[53]
we use the lack of active elbow flexion against gravity at 4 months of age as the indication for surgery, which is 1 month later then Gilbert et al's[52]
reference age of 3 months. This delay has allowed us to study the natural history in a new group of patients (who recovered active elbow flexion between 3 and 4 months of age). This group included 11 patients (26% of the total study population). At the final follow-up visit, 5 of the 11 had a satisfactory spontaneous recovery. The remaining 6 had a satisfactory recovery of the elbow, but a poor recovery of the shoulder. The former subgroup probably would have had an unnecessary surgery if they had undergone surgical exploration at 3 months of age. Patients in the latter subgroup had severe C5 lesions, and one might argue that their shoulder recovery might have been better with nerve grafting.The use of multiple scores
Clarke and Cutis
[20]
and Michelow et al[54]
relied on a limb motion score calculated from multiple joint motions as the indication for primary surgery. Zancolli and Zancolli[55]
suggested that for each level of involvement of the plexus, there was a different key muscle to consider as an indicator for direct nerve surgery. For the upper plexus, the muscles were the biceps and deltoid, whereas for the middle plexus it was the triceps, and for the lower plexus it was the finger flexors and thumb extensors. Lack of recovery of key muscles by 6 to 8 months of age was the indication for surgery in their series. Laurent et al[56]
used 3 muscles: the biceps, triceps, and deltoid in Erb's palsy patients. Surgery was indicated if there was no improvement of at least one grade on the Medical Research Council scale in 2 of the 3 muscle groups. The improvement had to be by 4 to 6 months of age, and continued improvement had to occur and be maintained at a level greater than M3 for another 4 months for surgery to be avoided.The use of both muscle scores and other prognostic factors
In a study of the prediction of outcome in Erb's palsy, Nehme et al
[57]
concluded that primary brachial plexus reconstruction is justified when there was initial C7 involvement associated with increased birth weight and poor elbow flexion at 6 to 9 months of age.In total palsy cases, Chuang et al
[58]
found that the presence of 3 factors: multiparity, a heavy baby (birth weight over 4.5 kg), and a positive Horner's sign was a bad prognostic sign for recovery of both the upper and lower roots of the brachial plexus.Primary brachial plexus surgery and results of nerve reconstruction
Primary brachial plexus surgery is performed under general anesthesia.
[59]
A standard supraclavicular incision is made in Erb's palsy cases and is extended along the deltopectoral area in total palsy cases. I perform clavicular osteotomy in total palsy cases to improve exposure. The phrenic nerve is identified and stimulated to assess its integrity. The omohyoid muscle is cut and the transverse cervical vessels are cauterized. The nerve roots are identified and stimulated. Surgical findings include the presence of neuroma-in-continuity, rupture, root avulsions, or a combination. In a few cases it is hard to decide if there is partial or total root avulsion and in these cases intraoperative somatosensory-evoked potentials may be helpful.A very controversial issue in birth palsy is what to do with conducting neuromas-in-continuity (ie, neurolysis v resection and nerve grafting). I was involved in the pioneer work of Clarke et al
[60]
who showed that neurolysis does not provide useful functional recovery. Capek et al[61]
later showed that resection of these neuromas does not significantly diminish motor activity by 3 months after surgery. These interesting findings could not be accounted for by axonal regeneration across the interpositional nerve grafts, and they concluded that nerve regeneration or recovery in the nongrafted segment of the plexus must be sufficient to produce the preoperative motion.Several investigators,
21
, 62
including myself, resect all neuromas except if distinct fascicular architecture is identified. Other investigators56
, 63
, 64
depend on the results of intraoperative nerve stimulation to decide whether to perform neurolysis versus resection and nerve grafting.After identification of the lesion and resection of the neuroma, one should identify intraplexus or extraplexus motor donors. Sural nerve grafts are then harvested via a longitudinal, stepladder, or slightly curved incision on the posterolateral aspect of the calf. Endoscopic harvest of the sural nerves also has been described.
[65]
In the absence of root avulsion, reconstruction usually is performed by intraplexus neurotization only. However, in the presence of root avulsion, a combination of intra- and extraplexus neurotization is performed. The spinal accessory usually is used to reconstruct the suprascapular nerve.59
, 66
Other extraplexus motor donors include the intercostals nerves and the phrenic nerve.[67]
Another option to reinnervate the biceps in cases of isolated C5/C6 avulsion is ulnar nerve transfer to the biceps nerve (performed in the upper arm).[68]
Neurorrhaphy is performed under the operating microscope by using sutures or fibrin glue. After surgery, the limb is immobilized in a cocoon cast, a custom-made brace, or a sling. If the latter is used, the limb also should be taped to the trunk. Occasionally, a halo also is included in the brace to avoid neck movements. Physiotherapy is started 3 weeks after surgery.
The results of surgery largely depend on the severity of the lesion and the number of avulsed roots. Gilbert
[69]
has the largest series of cases undergoing surgery. By 1991, he had explored 350 cases.[69]
In 1995, he reported on the results of 178 cases treated by nerve reconstruction that were followed-up for at least 3 years.[70]
Results for repairs in Erb's palsy generally were excellent in the shoulder and elbow. In 54 children with total palsy, reinnervation of the lower trunk provided useful finger flexion in 75% of cases and useful intrinsic function in 50% of cases. I fully agree with Birch[71]
that we (surgeons treating these children) should give acknowledgement to Gilbert who always stressed the importance of reinnervation of the hand in cases of avulsions of the lower roots of the brachial plexus.Sequelae and secondary deformities after birth palsy
The pathogenesis and treatment of sequelae in obstetric palsy has been the subject of much discussion. They are quite different from the sequelae of other paralyses such as poliomyelitis or adult brachial plexus injuries.
[21]
Reasons for that include: birth palsy patients develop common specific secondary deformities at each joint, cross-reinnervation and muscle cocontraction are frequent, and, finally, growth in the presence of contractures lead to significant osseous and joint deformities in addition to the longitudinal growth disturbance of the whole limb.The shoulder
Internal rotation contracture is the most common secondary deformity in birth palsy and is caused by muscular imbalance between the active internal rotators and the paralyzed external rotators. However, other shoulder deformities are not uncommon.
Classification and treatment of secondary shoulder deformities
Zancolli's
[72]
classification is shown in Table 7. Birch[73]
detailed the secondary deformities of the shoulder that occur after untreated internal rotation contractures and this is shown in Table 8. Finally, Waters et al[74]
offered a radiologic classification of the progressive glenohumeral deformity that occurs with increasing age in children with birth palsy.Table 7Zancolli's classification of secondary shoulder deformities in birth palsy
| Type | Subtype | Treatment |
|---|---|---|
| 1. Shoulder contracture | ||
| a. Internal rotation/adduction contracture | i. Normal joint | Subscapularis release and L'Episcopo muscle transfer |
| ii. Joint deformity | Humerus osteotomy | |
| b. External rotation/abduction contracture | i. Normal joint | Release external rotators of the shoulder |
| ii. Joint deformity | Humerus osteotomy | |
| c. Combined a and b | - | Release both subscapularis and external rotators |
| d. Pure abduction contracture | - | Z-plasty of supraspinatus |
| 2. Flaccid paralysis | - | Shoulder arthrodesis |
Table 8Birch's classification of shoulder deformities in birth palsy after untreated internal rotation contracture
| Type | Treatment | |
|---|---|---|
| I | Internal rotation contracture with a congruent joint | Posterior subscapularis slide |
| II/III | Secondary simple posterior subluxation/dislocation | Anterior subscapularis elongation, relocate humeral head |
| IV/V | Secondary complex posterior subluxation/dislocation with elongation of the coracoid, acromion, and the formation of glenoid abnormalities | Anterior subscapularis elongation, shorten coracoid, osteotomy of acromion, and relocate humeral head |
Management of the internal rotation contractures should include careful clinical and radiologic assessment of the shoulder. As mentioned earlier, this is the most frequent and most important secondary deformity of the shoulder in birth palsy. The deformity is best subclassified into Birch's
[73]
subtypes (Table 8). Treatment of Birch I deformity (with a normal joint) is performed by muscle release such as the posterior subscapularis slide[75]
or the modified anterior Sever's release. Once good passive external rotation is ensured, the child is assessed later for muscle transfers to reconstruct active external rotation, which usually is performed by either a modified L'Episcopo procedure (the insertion of latissimus dorsi ± teres major is transferred from the anterior to the posterior aspect of the humerus),72
, 77
, 78
, 79
, 80
, 81
or by reattaching the latissimus dorsi (± teres major) tendon high up on the posterior humeral head.21
, 82
Some investigators perform the shoulder release and muscle transfer simultaneously.83
, 84
, 85
Birch II through V deformities are treated either by the use of Birch's algorithm of management (Table 8) or by performing a rotational osteotomy of the humerus. Various techniques have been used for humerus osteotomy in Erb's birth palsy. Zancolli[72]
used a lower axillary incision, divided the distal part of the pectoralis major tendon, and performed the osteotomy at the level of the distal border of the pectoralis major tendon. Cuesta et al[86]
and Goddard and Fixsen[87]
used a deltopectoral incision and performed the osteotomy just above the deltoid insertion. Al-Zahrani[88]
performed the osteotomy just below the deltoid insertion and removed an anterior wedge from the proximal segment of the humerus, which helped in bringing the hand further in flexion toward the mouth. I have used Al-Zahrani's osteotomy (below the deltoid insertion) but without removal of an anterior wedge from the proximal humerus.[89]
The flail shoulder in birth palsy is another common deformity seen in birth palsy and usually is seen in children born by breech presentation with C5-C6 avulsion. The flail shoulder has neither bony deformity nor capsular contractures. Several types of muscle transfers have been described to reconstruct shoulder abduction in these children,
[82]
but the trapezius transfer remains the most frequently used.[21]
Raimondi et al[90]
recommended adding the levator scapulae (as a glenohumeral stabilizer) to the trapezius transfer. In late cases presenting as adults, shoulder arthrodesis may be considered.Another shoulder deformity seen in birth palsy is scapular winging. Traditional treatment using fascia lata slings to suspend the scapula is likely to fail because of overstretching with growth in these children. Hence, the deformity in birth palsy is best treated by using transfer of muscles such as the transfer of the contralateral trapezius (± rhomboids) to the affected scapula. Stabilization of the scapula will result in significant gain in both abduction and external rotation of the shoulder.
[91]
External rotation/abduction contractures of the shoulder mentioned by Zancolli
[72]
in his classification (Table 7) are iatrogenic and caused by the old practice of splinting the arm of these infants in abduction and external rotation. These deformities no longer are seen with modern physiotherapy practice.The elbow
In birth palsy, paralysis of elbow extension is rare and is corrected easily by a latissimus dorsi transfer. The 2 common deformities seen in these children are: paralysis (or weakness) of elbow flexion and flexion contracture of the elbow (also known as the extension deficit).
Hentz
[92]
outlined the various treatment options for elbow paralysis in birth palsy. The Steindler procedure (relocation of the flexor pronator origin to the midanterior surface of the humerus) is best performed in children who can exhibit the Steindler effect, produced by pronating the forearm and flexing the wrist and at the same time swinging the arm at the elbow to overcome gravity. Those children lacking good wrist extension are poor candidates for this procedure because it accentuates wrist flexor power.Triceps to biceps transfer is best performed in circumstances in which there is dysfunctional biceps-triceps cocontraction. The results of the earlier-described transfers generally are good, but their indications in birth palsy are limited.
Most children requiring reconstruction of elbow flexion usually undergo either a bipolar latissimus dorsi pedicle muscle transfer or a functional muscle transfer.
[21]
The later transfer is a 2-stage procedure. In the first stage, a nerve graft is sutured to the pectoralis nerve on the healthy contralateral side and the nerve graft is passed subcutaneously in front of the rib cage and is brought out at the level of the shoulder of the paralyzed side. After a period of 1 year, the second stage is performed, in which the gracilis muscle is transferred with its neurovascular pedicle to replace the biceps.Extension deficit or elbow flexion contracture is a very common deformity in birth palsy. The contracture often is as much a functional as an aesthetic disability, especially in view of the limb length discrepancy seen in these children. Elbow flexion contractures can be explained by a stronger biceps than triceps, but Aitken
[93]
noted that severe contractures also may occur in patients with weak biceps and that these contractures are associated with an increase in the proximal ulnar curvature and radial neck clubbing. Another explanation for the elbow flexion contracture is cross-innervation and muscle imbalance, which frequently is found after spontaneous recovery of Erb's palsy. I noted that elbow flexion contracture frequently is associated with the internal rotation contracture of the shoulder. Furthermore, the elbow contracture improves after surgical correction of the shoulder deformity.[89]
I explained these findings by the fact that children with shoulder internal rotation contracture always stand with the shoulder slightly abducted (and hence the elbow falls into slight flexion) to place the limb away from the body in a better functional position. This constant flexed posture of the elbow contributes to contracture. Treatment of elbow flexion contractures depends on the severity of the extension deficit. Mild cases are treated by splinting, moderate cases benefit from surgical release, and severe cases require an extension osteotomy of the distal humerus.[21]
The osteotomy in severe cases is performed at 11 to 13 years of age and preoperative total elbow flexion should be complete because the arc of motion will be moved.The forearm
The most common forearm deformity seen in birth palsy is the posture of slight pronation with limitation of active supination. This does not require any treatment because the posture puts the hand in a functional position.
The second most common deformity is the supination posture, which is seen in total palsy patients. Zancolli and Zancolli
[55]
divided these patients into 2 groups and labeled them as the ″posture in supination” and the ″supination contracture” groups. The former group had good passive forearm pronation and was treated with biceps rerouting (rerouting the biceps tendon around the radius so that it acted as a pronator instead of a supinator). The latter group had contracted interosseous membrane, and was treated with release of the interosseous membrane and either biceps rerouting or fusion of the distal radioulnar joint, depending on the status of the distal radioulnar joint.In our center,
[94]
we treat the supination deformity either with biceps rerouting or rotational osteotomy of the radius. The former procedure is performed in children with good passive forearm pronation (indicating the lack of contracture of the interosseous membrane), strong biceps, and no radial head dislocation. The osteotomy procedure is performed if there is contracture of the interosseous membrane (simultaneous release of the interosseous membrane is performed), weak biceps, or dislocation of the radial head. Other investigators also have reported their experience with the biceps rerouting and the radius osteotomy procedures.95
, 96
Other procedures that also have been used for the supination deformity of birth palsy include: proximal radioulnar arthrodesis,[97]
transposition of the bicipital tuberosity,[98]
and 1-bone forearm fusion.[99]
Finally, severe pronation contractures in birth palsy are rare and may be treated by combinations of muscle lengthening and transfers.
[100]
The wrist and hand
The most frequent wrist deformity in Erb's birth palsy is lack of wrist extension. Although various muscle transfers have been used for reconstruction of wrist extension such as pronator teres and the flexor digitorum superficialis, the preferred muscle transfer uses the flexor carpi ulnaris or radialis muscle.
[101]
In patients with total paralysis, wrist instability usually is associated with severe hand paralysis and is difficult to treat. Wrist stabilization in these patients is performed best by arthrodesis after skeletal maturity and often is combined with an intermetacarpal arthrodesis of the first and second metacarpals that allows a small pinch.
[21]
Patients with C5, C6, and C7 Erb's palsy may have paralysis of finger extensors and these can be treated with the usual transfers.
The choice of reconstruction for other hand paralyses such as lack of finger flexion, claw deformity, and lack of thumb opposition has been outlined by our center
[94]
and others.[102]
Other issues
I would like to end this review by discussing 2 important issues that have only gained attention recently. The first is the psychologic problems that these children face. We have reported
[35]
on young children who burn their insensate hands with charcoal or a lighter in front of their friends to gain attention. We also have reported[94]
on the psychologic status in a group of children with total palsy presenting with 2 socially disabling hand postures: the beggar's hand and/or the unshakable hand. All children had a supinated forearm and were teased by their friends, who called them beggars. When the impairment was severe and involved the right hand and wrist, children also complained that they were not able to shake hands. The main aim of our management was the correction of the abnormal posture. Before surgery, some children refused to go to school because of teasing, and most did not want to interact socially. After surgery, all children attended school regularly, and parents reported much better social interaction. However, the functional gain (from surgery) was never enough to dramatically improve the daily use of the limb. Another cause of psychologic disturbance is the limb length discrepancy seen in these children. Once again, growth disturbances are more severe in children with total rather than Erb's palsy.[103]
The second issue is the treatment of cocontraction of muscles in birth palsy. Cocontractions are the result of an aberrant nerve reinnervation at the site of the lesion or after nerve reconstruction. Diagnosis of cocontraction mainly is performed clinically by palpation of the muscle and its antagonist. The examiner also will note a contracting muscle without adequate motor function (although there is a good passive range of motion). Surgical treatment of cocontractions by muscle transfers have been outlined by Chuang et al.
[104]
More recently, nonsurgical treatment using the botulinum A toxin has been reported.[105]
The toxin acts presynaptically by blocking the release of acetylcholine at the neuromuscular junction. Hierner and Berger[105]
showed the repeated intramuscular injections of the toxin into the cocontracting antagonist resulted in a lasting reduction of imbalance between antagonist and agonist activity, with the possibility of adequate function.References
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© 2003 American Society for Surgery of the Hand. Published by Elsevier Inc. All rights reserved.
