Early Thoracoscopic Plication of Diaphragm in a Newborn with Brachial Plexus Palsy and Concurrent Phrenic Nerve Palsy.

Phrenic nerve palsy is a rare cause of respiratory distress in a newborn. When conservative measures fail to achieve adequate ventilation, then early surgical plication has been found to be associated with good outcome. We report a case of neonate with phrenic nerve palsy in whom an early thoracoscopic diaphragmatic plication was done.

INTRODUCTION

Obstetric phrenic nerve injury is a relatively rare disorder, which can cause life-threatening respiratory distress.[1] Phrenic nerve damage may result from a stretch injury due to lateral hyperextension of the neck at birth.[234] This rare cause of respiratory distress of newborn may be missed easily among multiple common etiologies.[256] Expectant management and surgical plication form two modalities of treatment.[7] It is very important to understand when to change the plan of management from expectant to surgical plication in this rare disease. Authors herewith stress that an early diaphragmatic plication can avoid comorbidities associated with prolonged ventilation and hospitalization.

CASE REPORT

A male neonate, weighing 1.8 kg, a full term normal vaginal delivery with breech presentation was managed for respiratory distress in another hospital and was referred intubated to our center on day 10 of life. He had multiple episodes of extubation failures and reintubations. On examination, he had diminished movements of the right hand and asymmetric Moro reflex. His right arm remained in adduction and internal rotation. He had normal grasp reflex. There was Erb's palsy on the right side. Chest X-ray done revealed slight haziness on the right side and the right hemidiaphragm was at a higher level as compared to left [Figure 1]. He was ventilated for a day at our center, and weaning was tried. The patient was maintained on nasal continuous positive airway pressure (CPAP) but had increased oxygen requirement. X-ray chest showed normal lung fields, but the right hemidiaphragm was raised as compared to the left. Suspecting diaphragmatic paralysis as a cause for respiratory distress an ultrasonography (USG) was done. USG revealed diminished right hemidiaphragmatic movements as compared to the left.

Figure 1

Pre- and post-operative chest X-ray showing flattening of diaphragm

After few hours he developed respiratory distress and hypoxemia requiring reintubation and ventilation. He was weaned off ventilator but again had extubation failure. Meanwhile, all other blood investigations were normal. Inflammatory markers for sepsis-like C-reactive protein and erythrocyte sedimentation rate were negative, and blood culture was normal. No other cause of extubation failure was evident, other than inefficient respiration. Thus, the patient was planned for thoracoscopic plication of diaphragm on day 17 of life.

Conventional three-port technique was used. Intraoperatively the right hemidiaphragm was found to be thin, and plication was done using prolene 3-0 sutures. Three sutures were placed from periphery to center [Figure 2]. We left an intercostal drainage tube via the posterior most port.

Figure 2

Intra operative figures showing lax and thinned out diaphragm

Seventy-two hours after the operation the patient was extubated successfully. Chest X-ray showed normal position of the diaphragm [Figure 1]. The intercostal tube was also removed, and the patient discharged home after 7 days after establishing full feeds and oxygen independence. Physiotherapy was advised for Erb's palsy.

After 1 month follow-up, the patient remains stable and is off oxygen. Parents are continuing physiotherapy for Erb's palsy.

DISCUSSION

The incidence of perinatal diaphragmatic paralysis is approximately 1 in 15,000–1 in 30,000 live births and mortality is estimated at 10%–15%.[1234] The incidence of concurrent ipsilateral phrenic nerve palsy with neonatal brachial plexus palsy was 2.4%,[1] 4.2%[3] and 6%.[4] Conversely, 66.7%–71.4% of patients manifesting phrenic nerve palsies were reported to demonstrate some degree of brachial plexus palsy.[15]

The timing for diaphragmatic plication remains controversial.[7] The controversy is between surgery and expectant management. Expectant management ranges from simple oxygen supplementation to noninvasive nasal CPAP to prolonged mechanical ventilation.[1278] Stramrood et al. have reported success with both modalities.[5] Yoshida and Kawabata showed that 20 out of 21 neonates had spontaneous recovery.[4] Previous authors have advised 2–4 weeks as time after which expectant management should be stopped as spontaneous recovery is no longer expected.[1789] However, we feel that waiting for this long can be problematic and the decision should be based on clinical condition. Early plication is recommended.[9]

Many previous authors have advised nonimprovement on ventilation as an indication for surgery.[910] However, its extubation failures (with recurrent intubations), desaturation episodes, prolonged hospital stay due to oxygen dependence and feeding problems which are also of concern.[9] Prolonged mechanical ventilation has its own complications and costs. Complications of prolonged ventilation and recurrent extubation failures increase stress to these respiratory compromised babies. These have implications on the surgery to be performed too.[9] Thus, many a times expectant management for too long and delay in surgery may be detrimental to the patient. With improved and safer anesthetic techniques and minimal invasive thoracoscopic techniques, surgical plication has become safer and less traumatic to these neonates and infants.[78910] All of the previous reports including ours have shown that infants who undergo early plication have a quick recovery and can be extubated successfully within a few days.

Thoracoscopy is nowadays the modality of choice for surgical plication.[310] Studies have shown that the effects of thoracoscopic plication are evident immediately as compared to those done by thoracotomy. Early recovery is evident in the form of decreased postoperative ventilation and hospital stay.

Thoracoscopic repair of the stretched portion of the diaphragm has been previously described by means of different techniques: invagination of the diaphragm and its plication using continuous sutures, mechanical suturing at the bottom of the dome using the endostapler with excision of redundant diaphragm tissue, and the application of multiple U-shaped sutures and using spinal needle.[610] This has been done using five ports, conventional three ports and two ports using spinal needle from both laparoscopic and thoracoscopic routes.[69] Both single stitch and continuous stitch techniques have been used, with single stitch technique being easier.[6910] Previous authors have recommended starting suturing from periphery toward mediastinum, however starting sutures from center to stabilize diaphragm have also been recommended.[6910] In all techniques, radiological success as in flattening the diaphragm and clinical success as in weaning from ventilation and extubation was achieved.[678910] No major complications were reported in previous studies apart from occasional pneumothorax.[10]

CONCLUSION

Early diaphragmatic plication enhances the weaning process and may prevent or minimize the morbidity associated with long-term mechanical ventilation in a neonate with diaphragmatic paralysis.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.