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19 December 2010

Dear Editor,

We report an unusual case of birth injury-related diaphragmatic palsy which manifested as recurrent projectile vomiting in addition to protracted dependency on continuous positive airway pressure (CPAP). The case was also unusual because there was no associated brachial plexus palsy.

A 40-week gestation male neonate weighing 3920 g was born by vaginal delivery in a regional hospital of Western Australia. The delivery was very difficult because of breech presentation which was discovered only during the second stage of labour. He was born in poor condition with Apgar scores of 2, 4, 5 and 8 at 1, 5, 10 and 40 min, respectively. He was air lifted to the tertiary neonatal intensive care unit after initial stabilisation in the regional hospital. He underwent systemic hypothermia for neuro-protection in view of stage 2 hypoxic ischemic encephalopathy and made good recovery neurologically.

Chest X-ray performed prior to intubation and within few hours after birth revealed marked elevation of the right hemi-diaphragm (Fig. 1). Ultrasound and fluoroscopy showed paradoxical movement of the right hemi-diaphragm, leading to a diagnosis of phrenic nerve palsy. Neurological assessment did not reveal the presence of brachial plexus palsy. We decided to manage the diaphragmatic palsy conservatively while waiting for spontaneous recovery of the diaphragmatic movements.

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Figure 1. X-ray showing marked elevation of the right hemi-diaphragm.

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He was easily extubated from the ventilator within 3 days, but continued to be completely dependent on CPAP.

In addition, he developed recurrent projectile vomiting for the next 8 weeks which was investigated, and pyloric stenosis, malrotation and gastro-oesophageal reflux ruled out. Trial of hypoallergenic formula, continuous feeds using naso-jejunal tube, anti-reflux medication and proton pump inhibitors did not improve the vomiting.

In view of persistent dependency on CPAP, he underwent surgical plication of the diaphragm on day 65. Within 48 h following the surgery, he was able to be weaned completely off CPAP. Interestingly, projectile vomiting also resolved completely and he was able to be discharged home within 1 week after surgery.

Apart from birth trauma, phrenic nerve injury leading to diaphragmatic paralysis can occur as a complication of cardiac surgery, insertion of intercostal drain and internal jugular vein cannulation. Diaphragmatic palsy can also occur in spinal muscular atrophy, congenital myopathies and congenital myasthenia.1 All these differential diagnoses were ruled out in our index case by careful history taking and physical examination of the baby. Unilateral involvement of the diaphragm and quick resolution of the encephalopathy ruled out infectious/metabolic diseases.

Diaphragmatic paralysis secondary to phrenic nerve injury can lead to respiratory compromise, recurrent pneumonia, failure to thrive and even mortality,2,3 but to our knowledge, protracted vomiting has not been reported to be an associated complication. Literature suggests that 66–80% of cases with phrenic nerve injury have associated brachial plexus injury; conversely only about 2.5–10% of neonates with brachial plexus injuries have associated phrenic nerve injury.2,3 Recent case reports have suggested that prolonged wait for spontaneous recovery may increase morbidities and mortality and, hence, early plication may be preferable,4,5 and our experience supports this argument.

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