Diaphragmatic Paralysis Following Chest Tube Insertion in an Infant: Case report and literature review

Summary

Diaphragmatic paralysis (DP) can occur due to central nervous system pathology or peripheral nerve injury. Direct injury to the phrenic nerve after intercostal chest drain (ICD) insertion for treatment of pneumothorax is an infrequent complication. We present a 4-month-old infant, ex-preterm 27 weeks, who was admitted to a tertiary care hospital paediatric intensive care unit in Muscat, Oman, in 2023 with severe respiratory syncytial virus bronchiolitis and required intubation and mechanical ventilation (MV). His illness was complicated by right-side pneumothorax that required ICD insertion. Post-extubation, he had persistent tachypnoea with the inability to be weaned from non-invasive ventilation. Chest X-ray (CXR) and fluoroscopy showed a high right diaphragm dome with paradoxical movements. He improved dramatically after the plication of the right diaphragm and was discharged home on the 9th day after the plication.

1. Introduction

The phrenic nerve originates mainly from the 4th cervical nerve. It carries motor, sensory and sympathetic nerve fibres for diaphragmatic function. Activation of the motor innervation causes the diaphragm to contract with inspiration, resulting in a flattened diaphragm. During exhalation, the diaphragm relaxes and returns to the dual dome shape.¹ Phrenic nerve injury can be seen due to central nervous system pathology or peripheral nerve injury. Congenital heart disease surgeries, birth trauma and pneumonia have all been known as reasons for peripheral phrenic nerve paralysis.²

Direct trauma to the phrenic nerve after chest tube insertion has been reported; most reported cases in the literature were in neonates.^2,3,4,5,6 The diagnosis is mainly clinical and can be confirmed by chest X-ray (CXR), ultrasound and fluoroscopy.⁷ It can be further confirmed by trans-diaphragmatic pressure (Pdi) measurements and electromyographic study. A recent study showed that Pdi morphology can identify unilateral paralysis.⁸ However, to obtain these parameters, it is necessary to introduce oesophageal and gastric balloons and to use special equipment and software, thus limiting their use in everyday clinical practice.

The presented case is very unusual case and will enlighten physicians on the importance of proper ICD positioning to avoid the inconvenient and troublesome side effects of diaphragm paralysis.

2. Case report

A 4-month-old ex-preterm at 27 weeks with a corrected age of 6 weeks and a weight of 4.6 kg was admitted to a tertiary care hospital paediatric intensive care unit in Muscat, Oman, in 2023 with respiratory syncytial virus bronchiolitis. His initial CXR showed bilateral infiltration with normal position of the diaphragms [Fig. 1]. He was started on non-invasive ventilation (NIV) with no improvement and required intubation and mechanical ventilation. He developed tension pneumothorax on the right side. A 12 Fr chest tube was inserted in the right 5th intercostal space anterior axillary line using the Seldinger technique [Fig. 2]. The patient's condition gradually improved, and tracheal extubation was done after 24 days. The ICD was removed on the 7th day. At that point in time, the right dome of the diaphragm was found to be elevated [Fig. 3]. The patient continued to need NIV via nasal biphasic airway pressure for a further 19 days. Ultrasound examination off positive pressure ventilation (PPV) showed limited movement of the right diaphragm. In addition, fluoroscopy of PPV showed paradoxical movement of the diaphragm. He was shifted to high flow nasal cannula (HFNC) but could not tolerate it and was put back on NIV.

Fig. 1.

Chest X-ray of a 4-month-old infant at the initial presentation showing a normal diaphragm position.

Fig. 2.

Chest X-ray of a 4-month-old infant after intercostal chest tube insertion showing tube position at the apex (superior) of the right lung.

Fig. 3.

Chest X-ray of a 4-month-old infant showing a high dome of the right diaphragm after extubation.

A diaphragmatic plication was performed, and the child was extubated on the 5^th postoperative day and required NIV for only 1 day. Later, the patient was moved to a low-flow nasal cannula and then to room air. He was discharged home on the 9th day after the diaphragmatic plication.

3. Discussion

Phrenic nerve injury can be seen due to central nervous system pathology or peripheral nerve injury. Congenital heart disease surgeries, birth trauma, and pneumonia have all been known as reasons for peripheral phrenic nerve paralysis. Congenital weakness (eventration) is related to muscular aplasia of the diaphragm and is commonly found on the right side. Acquired forms are usually traumatic, such as birth trauma or lateral thoracotomy, and are often unilateral, though the distribution of cases is equal between both sides.⁹

The phrenic nerve is the longest branch of the cervical plexus and enters the thorax through the superior thoracic aperture, between the subclavian artery and vein.¹⁰ It descends, crossing the apex of the right or left pleural cavity, coursing between the mediastinal pleura and the pericardium.¹¹ The phrenic nerve courses along the pericardium bilaterally; therefore, any chest tube reaching the mediastinal border may cause nerve compression. Nerves can be damaged by compression or stretching; nerve compression studies on rat sciatic and optic nerves showed that ischemia from compression can lead to significant nerve damage.^12,13 Compression injury of the radial nerve due to blood pressure cuff has also been reported.^14,15

We report an uncommon cause of diaphragmatic paralysis (DP), which was associated with blunt trauma caused by chest drain for right side pneumothorax. In the current case, the ICD was inserted in the right 5th intercostal space anterior axillary line and was directed to the apicomedial zone. Ghani et al. conducted a retrospective cohort analysis of over 531 patients and showed a strong association between the right apicomedial chest drains looping at the 2nd right intercostal space level and DP (P < 0.05). They called this zone the “danger zone”.¹⁶

The exact pathophysiology of reversibility of nerve injury after nerve compression or blunt injury is unknown. It is thought to be a local reaction to periods of ischaemia that result in ion-induced conduction blockade. There is initially demyelination of the axon, which eventually recovers. Loss of function occurs until remyelination occurs, sometimes rapidly over days or up to 12 weeks.^17,18 ICD insertion or manipulation may induce phrenic nerve injury, which can be either reversible or irreversible. By the age of 1 year, 50–80% of cases show recovery without intervention.⁹ PPV may obscure the effect of the diaphragmatic injury and make it only discoverable after the removal of support. DP can induce respiratory insufficiency and increase postoperative morbidity and respiratory support dependency, especially in neonates and small infants.¹⁹

In the current case, the suspicion of right-sided diaphragmatic palsy was not raised until the positive end-expiratory pressure was weaned down, targeting extubation when CXR showed a high position of the right diaphragm.

Different modalities to diagnose DP, including radiography, fluoroscopy, ultrasound and electrophysiology, are used. Fluoroscopy is a modality that can visualize the diaphragm continuously during the normal respiratory cycle. It is easy as a procedure, and for interpretation, it has been considered the gold standard for unilateral DP diagnosis, but it needs to be done off PPV.²⁰

Diaphragmatic ultrasound is a portable, non-invasive, easy-to-perform, and well-tolerated test with a linear relationship between diaphragmatic movement and inspired volume, which permits quantitative and qualitative evaluation of diaphragmatic movement.²¹ Therefore, ultrasound has been recommended for evaluating diaphragmatic movement on suspicion of weakness or paralysis.

Ultrasound has been proven beneficial for detecting diaphragmatic dysfunction, with high sensitivity (93%) and specificity (100%) for diaphragmatic neuromuscular disease.^22,23 Phrenic nerve stimulation is not a standard infant modality; in the current case, it was not used.

DP management secondary to phrenic nerve injury should be individualised to the patient as there is no clear and solid rule. Conservative versus surgical treatment of diaphragmatic eventration is still controversial. Being asymptomatic without ventilatory support is an indicator of conservative management. However, to the best of the Authors' knowledge and following a literature review, the duration of dependency on ventilatory support is not specified.

To date, there are a total of 8 reported for non-congenital heart surgery cases of DP secondary to ICD insertion for pneumothorax in infants [Table 1].^2,3,4,5,6 A total of 5 infants required diaphragmatic plication and the other recovered without surgical intervention. Spontaneous recovery is unlikely if there are persistent signs of phrenic nerve palsy after 1 month of insult or extubation failure.²⁴ The current case remained on NIV/HFNC support after extubation for approximately 18 days. Multidisciplinary discussion concluded proceeding with diaphragmatic plication due to its safety profile and lower chances of allowing for lung de-recruitment and strengthening the intercostal and abdominal muscles. Nevertheless, it doesn't prevent functional recovery.²⁵ It was suggested to place the ICD at least 2 cm away from the vertebra or mediastinal border to avoid phrenic nerve injury.^4,26 The current patient underwent diaphragmatic plication, and a few days after surgery, he was discharged home without any noticeable respiratory distress.

Table 1.

Summary of the reported cases of diaphragmatic paralysis.^2-6

Author and year	No. of	Age in
of publication	cases	days	Diagnostic tool	Salient features	ICD position	Outcome
Ayaln et al.2 (1979)	1	18	CXR and fluoroscopy	Right ICD inserted to drain pneumothorax	Low mediastinum	Remained symptomatic till age 18 of life. Diaphragmatic plication done. Follow-up after 2 months showed complete recovery.
Philipps et al.3 (1981)	1	1	CXR and fluoroscopy	Left side ICD inserted to drain pneumothorax	Middle mediastinum	Required NIV weaned gradually and discharged at age of 6 weeks.
Nahum et al.4 (2001)	1	1260	CXR, fluoroscopy and MRI thorax	Right side ICD inserted to drain pneumothorax	Not mentioned	Required MV, weaned after 11 days and discharged home after 24 days with no follow-up.
Williams et al.5 (2003)	1	33	CXR, fluoroscopy, chest ultrasound and percutaneous electric stimulation	Bilateral effusions; Required 2 ICD on each side elevated diaphragm on right side	Middle mediastinum	Failed extubation 4 times. Plication done on day 75.
Odita et al.6 (1992)	1	4	CXR, fluoroscopy and chest ultrasound	Left side ICD inserted for pneumothorax	Overly to the spine	Complete recovery after 5 weeks.
	1	2	CXR, fluoroscopy and chest US	Left side ICD inserted to drain pneumothorax	Overly to the spine	Required plication by day 36 of life.
	1	1	CXR	Left side spontaneous pneumothorax	Overly to the spine	Required plication at age of 1 month.
	1	2	CXR	Recurrent pneumothorax on right side	Overly to the spine	Required plication at age of 1 year.

ICD = intercostal chest drain; CXR = chest X-ray; NIV = non-invasive ventilation; MRI = magnetic resonance imaging; MV = mechanical ventilation; US = ultrasound.

4. Conclusion

ICD insertion is an infrequent cause of phrenic nerve injury and diaphragm paralysis, which can be confirmed by ultrasound and fluoroscopy. Diaphragm plication might be necessary if signs of phrenic nerve palsy persist and the patient has extubation failures or remains dependent on respiratory support. This was demonstrated in the current patient, who remained dependent on NIV support till he had diaphragm plication. ICD reaching the vertebra or mediastinal border may cause phrenic nerve injury.

Ethics Statement

Verbal consent for publication purposes was obtained from the father.

Authors' Contribution

MAG contributed towards conception of the work, acquisition of data, interpretation of data, drafting of manuscript and intellectual content. SAH contributed towards literature review. AEE contributed towards conception of the work, data collection and summarizing the case report. AMF contributed towards conception of the work, data collection and intellectual input. All authors approved the final version of the manuscript.

Data Availability

The data that support the findings of this study are available from the corresponding author upon reasonable request.