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Neonatal brachial plexus injury with diaphragmatic eventration

Aihole, Jayalaxmi S; Deepak, Javaregowda; Jadhav, Vinay; Ramesh, S

doi: 10.4103/ijssr.ijssr_5_17
Case Report
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Our case report describes obstetric right brachial plexus injury leading to Erb's palsy and Klumpke's palsy with ipsilateral paralysis of diaphragm in a 9-month-old baby without Horner's syndrome. The baby required initial right brachial plexus root exploration and nerve grafts, later requiring surgical plication of diaphragm. Now, the baby is free of bronchopneumonia and is in recovering phase of upper limb motor and sensory function.

Department of Paediatric Surgery, IGICH, Bengaluru, Karnataka, India

Department of Paediatric Surgery, IGICH, Bengaluru, Karnataka, India

Department of Paediatric Surgery, IGICH, Bengaluru, Karnataka, India

Department of Paediatric Surgery, IGICH, Bengaluru, Karnataka, India

Address for correspondence:Jayalaxmi Aihole, Department of Paediatric Surgery, IGICH, Bengaluru, Karnataka, India jayalaxmisaihole@yahoo.com

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Introduction

Phrenic nerve injury as part of obstetric brachial plexus injury leading to paralysis of the ipsilateral diaphragm presenting with respiratory distress may require continuous positive airway pressure (CPAP) or mechanical ventilation; and if unresponsive, surgical plication of diaphragm may be required. Herein, we report a case of phrenic nerve palsy following obstetric brachial plexus injury in an infant presenting with recurrent bronchopneumonia.

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Case Report

A 9-month-old male baby presented to us with recurrent bronchopneumonia since birth. The baby was born by full-term vaginal delivery to gravid 2 mother having normal antenatal scans. Because of prolonged labor, forceps delivery was attempted. Baby weighted 4.5 kg at birth and had respiratory difficulty after birth; hence, he was kept on mechanical ventilator for 10 days. The baby was noticed to have right flail arm Figure 1A; in view of diagnosis of obstetric brachial plexus injury, physiotherapy was started. Baby did not show much improvement after 4 months, and magnetic resonance imaging revealed root sleeve avulsion/pseudomeningocele at brachial plexus roots level Figure 2A and Figure 2B; surgical intervention was contemplated. At 4 months, baby underwent right brachial plexus exploration, neuromas of around 2 cm × 1.5 cm involving upper, middle, and lower trunks excision with nerve grafts; spinal accessory to the suprascapular nerve, C5 root to the upper trunk through medial cutaneous nerve of forearm. C6 and C7 roots to the lower trunks through sural nerve in an orthopediatic center.The baby showed mild improvement on limb movements on follow-up.

Figure 1

Figure 1

Figure 2

Figure 2

Baby presented to us with recurrent bronchopneumonia, and on examination, the baby had right Erb's palsy (C5–C6 injury), the arm was characteristically held adducted and internally rotated with the forearm pronated, hand and wrist flexed (“waiter's tip” position). The muscle power of 2/5 in the right arm and in the forearm;whereas 1/5 in the wrist and fingers. Baby also had hypoesthesia below the right wrist joint along with klumpke's palsy, i.e. paralysis of the lower brachial plexus having loss of motion in the wrist and hand with no features of Horner's syndrome.

Chest X-ray revealed elevation of the right dome of diaphragm Figure 2C. Diaphragmatic eventration was demonstrated on fluoroscopy and ultrasonography chest. The baby underwent thoracoscopic diaphragmatic plication Figure 1B. The baby is doing well for the last 2 years with no fresh episodes of bronchopneumonia and overall good health Figure 2D.

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Discussion

The incidence of obstetrical brachial plexus palsy occurs in 0.4–2/1000 live births. 1 The brachial plexus in neonates can be damaged by being stretched, compressed, infiltrated, or deprived of oxygen. Predisposing factors for obstetrical brachial plexus palsy can parcel as, the maternal, fetal and labor related. 1 , 2 , 3 The most important maternal factors are diabetes gravid and excessive weight gain during pregnancy. 2 The most common labor-related risk factors are shoulder dystocia and instrumented delivery, and prolonged second stage of labor is probably a function of shoulder dystocia and of itself not a risk factor. Shoulder dystocia and instrumented delivery increase the stretch force brought on the brachial plexus as a consequence of propulsive forces, traction forces, or both. 2 Cesarean section has a significant protective effect but by no means prevents obstetrical brachial plexus palsy. 2 , 3 The most common fetal risk factor is macrosomia. 1 , 2 , 3

Brachial plexus spreads from C5 to T1. Upper brachial plexus paralysis, resulting from excessive lateral neck flexion away from the shoulder, is called Erb's palsy or Erb–Duchenne palsy; while the less frequent lower brachial plexus paralysis (C8–T1) Dejerine-Klumpke following a sudden upward pulling on an abducted arm is called Klumpke's paralysis. There can also be total brachial plexus palsy as noted in our case. The phrenic nerve originates mainly from the 4 thcervical nerve but also receives contributions from the 3 rdand 5 thcervical roots (C3–C5). 1 , 2 , 3

The severity of brachial plexus injury ranges from least (neuropraxia: permitting complete prompt recovery) to intermediate (axonotmesis: allowing gradual recovery) to severe, with avulsion of the roots from the spinal cord causing permanent injury (neurotmesis). 1 , 4

Algimantas Otonas Narakas (1927–1993) in 1986 designed the Narakas Classification of Nerves to identify the level of nerve injury based on symptoms identified during a clinical examination 3 , 5 /1. Alain Gilbert and Tassin concluded that absence of recovery in the biceps (even slight contraction) at 3 months was inevitably associated with poor final function. 5 Gilbert has popularized the most commonly used indication for operative management in obstetrical brachial plexus palsy: neuroma excision and interpositional nerve grafting, in patients where there has been no recovery of biceps muscular function even after 3 months of age.{Table 1}

We intervened at 4 months in our patient with brachial exploration, neuroma excision, and nerve graftings. 5 Nerve transfers (neurotization) can be performed in cases of preganglionic or combined preganglionic and postganglionic injuries. Root avulsions are considered to be irreparable. 6

Bowerson et al. report an incidence of 2.4%–4.2% of neonate with brachial plexus palsy presenting significant phrenic nerve palsy. 7 Most cases of diaphragmatic paralysis are unilateral and mostly on the right side. 8 , 3 , 6 , 7 The diagnosis of diaphragmatic eventration is suggested on chest radiograph if the right hemidiaphragm is two intercostal spaces higher than left or if the left hemidiaphragm is one intercostal space higher than right. Paradoxical motion or limitation of motion of involved diaphragm may be seen on fluoroscopy or ultrasound examination. Supportive treatment consists of oxygen administration, nasogastric feeding, and mechanical ventilation.

According to Escande et al., noninvasive nasal CPAP should be proposed for the treatment of phrenic nerve obstetrical palsy before introducing more invasive ventilation techniques. 3 Stramrood et al. in their study showed that a minority of infants suffering from diaphragmatic paralysis due to perinatal phrenic nerve injury recover spontaneously. 2

According to de Vries et al., if after 1 month, no spontaneous recovery of the diaphragmatic paralysis caused by a phrenic nerve injury occurs, plication of the diaphragm is indicated. If the condition of the patient clinically deteriorates during the 1 stmonth of life, the patient should be operated upon immediately. 3

Conventionally, diaphragmatic plication (DP) is performed through a thoracotomy that includes incision of the lower intercostal muscles, which are involved in respiratory movement. This may adversely affect ventilation by causing deterioration of respiratory function and making ventilation less efficient. These problems do not occur with thoracoscopic DP since the lower intercostal muscles are left intact. 3 Since our patient had preganglionic injury (root avulsion) Narakas Group III, which has poor prognosis as such, only mild recovery of function and sensation of upper limb including wrist was noted.

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Conclusion

Severe obstetrical brachial plexus injuries are potentially avoidable with adequate precautions. Preganglionic (root avulsions) brachial plexus injuries have poor outcome in spite of early and adequate surgical intervention. DP is an integral part of management in these infants who fail to wean from ventilatory support and in those who have recurrent bronchopneumonia. 3

Ethical standard statement – parents gave informed consent to the publication of the case report.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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    Keywords:

    Birth trauma; diaphragmatic paralysis; phrenic nerve palsy; respiratory distress

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