- Email: [email protected]
Spontaneous Recovery Following Traumatic Phrenic Nerve Palsy
HLC 2152 No. of Pages 3
Heart, Lung and Circulation (2016) xx, 1–3 1443-9506/04/$36.00 http://dx.doi.org/10.1016/j.hlc.2016.05.124
BRIEF COMMUNICATION
Spontaneous Recovery Following Traumatic Phrenic Nerve Palsy R. Nelson, MBChB a, M.D. Haydock, CertSc, MBChB a,b*, D.A. Haydock, MBChB, FRACS a a
Green Lane Cardiothoracic Surgical Unit, Auckland City Hospital, Auckland, NZ The University of Auckland, Department of Surgery, Auckland, NZ
b
Received 20 May 2016; accepted 29 May 2016; online published-ahead-of-print xxx
This is a case report of unilateral traumatic phrenic nerve palsy with spontaneous recovery over a period of 31 months. This adds to the literature, demonstrating that extended conservative management can lead to successful resolution even in the setting of traumatic aetiology. Keywords
Phrenic nerve Trauma
Case A 58-year-old New Zealand man presented, reporting exertional dyspnoea, orthopnoea and a significant reduction in exercise capacity over a six-month period. Preceding the onset of symptoms he had experienced trauma to his neck after being trampled by an alpaca. Medical history was unremarkable, other than a modest smoking history. On examination, his right lung base was dull on percussion during inspiration and expiration suggestive of right hemidiaphragm paralysis which was confirmed with chest radiographs. Otherwise, cardiorespiratory and neck examination were non-contributory. Computed tomography of his chest confirmed a raised right hemidiaphragm, but identified no specific cause of this abnormality. Computed tomography of his neck was unremarkable. Spirometry showed moderate restriction with forced expiratory volume in one second of 1.80 litres, and a forced vital capacity of 2.10 litres, (50% and 45% of predicted values respectively). Further spirometry demonstrated significant reduction in lung function from lying to standing. Diaphragm screening under fluoroscopy confirmed complete right diaphragm paralysis. After non-resolution (but a degree of improvement) of symptoms over the space of one year, subsequent followup diaphragmatic screening under fluoroscopy showed movement of the right hemidiaphragm with respiration of
a height of about one vertebral body. This was worse than the left hemidiaphragm, but was an improvement compared with the previous study. Furthermore, repeat chest X-ray nine months following this showed a return of the right hemidiaphragm to a normal position; he had a correlated full resolution of symptoms and spirometry (Table 1). Figures 1a-f show serial chest X-rays throughout the course of the patient’s recovery.
Discussion This case report of unilateral phrenic nerve palsy supports a conservative approach to management even in the setting of blunt trauma. Phrenic nerve palsy is relatively rare, and has a range of aetiologies, including trauma, iatrogenesis (with chiropractic manipulation, cardiac surgery and central venous catheter insertion all implicated) [1,2,3,4] and malignancy, with many cases also considered idiopathic. Unilateral phrenic nerve palsy often presents with subtle symptoms, such as exertional dyspnoea, or is identified incidentally on chest X-ray. However, in cases where symptoms are significant enough, or have failed to resolve after extensive periods of conservative management, surgical intervention exists in the form of diaphragmatic plication. This attenuates the paradoxical movement of the hemidiaphragm
*Corresponding author at: Auckland City Hospital 2-20 Park Road, Grafton, Auckland, ph +64 21 111 6286, Email: [email protected] © 2016 Australian and New Zealand Society of Cardiac and Thoracic Surgeons (ANZSCTS) and the Cardiac Society of Australia and New Zealand (CSANZ). Published by Elsevier B.V. All rights reserved.
Please cite this article in press as: Nelson R, et al. Spontaneous Recovery Following Traumatic Phrenic Nerve Palsy. Heart, Lung and Circulation (2016), http://dx.doi.org/10.1016/j.hlc.2016.05.124
HLC 2152 No. of Pages 3
2
R. Nelson et al.
Table 1 Resolution of symptoms and spirometry
FEV1 (% predicted) FVC (% predicted)
1 month
3 month (standing)
3 month (lying)
7 month
35 month
1.8 (50) 2.1 (45)
1.79 (51) 2.84 (62)
1.14 (33) 2.03 (44)
1.84 (48) 2.6 (54)
2.58 (76) 3.65 (81)
Figure 1 A - 0 months B - 3 months C - 7 months D - 18 months E - 20 months F - 31 months.
with respiration. Versteegh and Tjien [5] describe this surgical technique, and provide an overview of outcomes from several case series which illustrate improvement in vital capacity postoperatively, particularly in the supine position. In less symptomatic patients, there is a weight of evidence to suggest that prolonged conservative management is appropriate. There are numerous case reports and series which describe a very long period before return to previous level of function. Douglas [6] describes 40 patients with idiopathic unilateral diaphragmatic paralysis, who had a range of 2 to 19 years until complete recovery. In another series of 10 patients with traumatic phrenic nerve palsy [7], among whom there was a 6–12-month period of recovery. Dernaika et al. [8] compiled cases from 13 reports and series, in which, of 129 cases of bilateral or unilateral nerve palsy, 40 exhibited full recovery with conservative management, which
ranged from 1 month to 19 years. The authors also report on a case of a 57-year-old man who, after presenting with idiopathic right hemidiaphragmatic paralysis, which despite no clinical or radiological improvement after 9 months, demonstrated complete recovery after 21 months. This evidence, as well as that which is presented in this case, illustrates that a prolonged course of conservative management can be appropriate, and that non-resolution after long periods doesn’t necessitate a surgical approach. A delayed course of recovery has to be considered, and we suggest that management should be considerate of how symptomatic a patient is, as well as progression of clinical/radiological status over time. This is supported by animal studies [9] which illustrate that the timeframe of recovery is incredibly variable and is different depending on the mechanism of the palsy and even from different nerves.
Please cite this article in press as: Nelson R, et al. Spontaneous Recovery Following Traumatic Phrenic Nerve Palsy. Heart, Lung and Circulation (2016), http://dx.doi.org/10.1016/j.hlc.2016.05.124
HLC 2152 No. of Pages 3
3
Spontaneous Recovery Following Traumatic Phrenic Nerve Palsy
This case also provides an instance of blunt trauma as being a cause of diaphragmatic nerve palsy, and shows that full resolution of symptoms with conservative management is possible in cases of blunt trauma to the neck.
Declaration There are no conflicts of interest to declare
References [1] Schram DJ, Vosik W, Cantral D. Diaphragmatic paralysis following cervical chiropractic manipulation: case report and review. CHEST Journal 2001 Feb 1;119(2):638–40. [2] Kimura J. Electrodiagnosis in diseases of nerve and muscle: principles and practice. Oxford University Press; 2013 Aug 12.
[3] Rigg A, Hughes P, Lopez A, Filshie J, Cunningham D, Green M. Right phrenic nerve palsy as a complication of indwelling central venous catheters. Thorax 1997 Sep 1;52(9):831–3. [4] Takasaki Y, Arai T. Transient right phrenic nerve palsy associated with central venous catheterization. British Journal of Anaesthesia 2001 Sep 1;87(3):510–1. [5] Versteegh MI, Braun J, Voigt PG, Bosman DB, Stolk J, Rabe KF, Dion RA. Diaphragm plication in adult patients with diaphragm paralysis leads to long-term improvement of pulmonary function and level of dyspnea. European Journal of Cardio-Thoracic Surgery 2007 Sep 1;32(3):449–56. [6] Douglass BE, Clagett T. The prognosis in idiopathic diaphragmatic paralysis. Diseases of the Chest 1960 Mar 31;37(3):294–7. [7] Iverson LI, Mittal A, Dugan DJ, Samson PC. Injuries to the phrenic nerve resulting in diaphragmatic paralysis with special reference to stretch trauma. The American Journal of Surgery 1976 Aug 31;132(2):263–9. [8] Dernaika TA, Younis WG, Carlile PV. Spontaneous recovery in idiopathic unilateral diaphragmatic paralysis. Respiratory Care 2008 Mar 1;53(3):351–4. [9] Ho¨ke A. Mechanisms of Disease: what factors limit the success of peripheral nerve regeneration in humans? Nature Clinical Practice Neurology 2006 Aug 1;2(8):448–54.
Please cite this article in press as: Nelson R, et al. Spontaneous Recovery Following Traumatic Phrenic Nerve Palsy. Heart, Lung and Circulation (2016), http://dx.doi.org/10.1016/j.hlc.2016.05.124
Heart, Lung and Circulation (2016) xx, 1–3 1443-9506/04/$36.00 http://dx.doi.org/10.1016/j.hlc.2016.05.124
BRIEF COMMUNICATION
Spontaneous Recovery Following Traumatic Phrenic Nerve Palsy R. Nelson, MBChB a, M.D. Haydock, CertSc, MBChB a,b*, D.A. Haydock, MBChB, FRACS a a
Green Lane Cardiothoracic Surgical Unit, Auckland City Hospital, Auckland, NZ The University of Auckland, Department of Surgery, Auckland, NZ
b
Received 20 May 2016; accepted 29 May 2016; online published-ahead-of-print xxx
This is a case report of unilateral traumatic phrenic nerve palsy with spontaneous recovery over a period of 31 months. This adds to the literature, demonstrating that extended conservative management can lead to successful resolution even in the setting of traumatic aetiology. Keywords
Phrenic nerve Trauma
Case A 58-year-old New Zealand man presented, reporting exertional dyspnoea, orthopnoea and a significant reduction in exercise capacity over a six-month period. Preceding the onset of symptoms he had experienced trauma to his neck after being trampled by an alpaca. Medical history was unremarkable, other than a modest smoking history. On examination, his right lung base was dull on percussion during inspiration and expiration suggestive of right hemidiaphragm paralysis which was confirmed with chest radiographs. Otherwise, cardiorespiratory and neck examination were non-contributory. Computed tomography of his chest confirmed a raised right hemidiaphragm, but identified no specific cause of this abnormality. Computed tomography of his neck was unremarkable. Spirometry showed moderate restriction with forced expiratory volume in one second of 1.80 litres, and a forced vital capacity of 2.10 litres, (50% and 45% of predicted values respectively). Further spirometry demonstrated significant reduction in lung function from lying to standing. Diaphragm screening under fluoroscopy confirmed complete right diaphragm paralysis. After non-resolution (but a degree of improvement) of symptoms over the space of one year, subsequent followup diaphragmatic screening under fluoroscopy showed movement of the right hemidiaphragm with respiration of
a height of about one vertebral body. This was worse than the left hemidiaphragm, but was an improvement compared with the previous study. Furthermore, repeat chest X-ray nine months following this showed a return of the right hemidiaphragm to a normal position; he had a correlated full resolution of symptoms and spirometry (Table 1). Figures 1a-f show serial chest X-rays throughout the course of the patient’s recovery.
Discussion This case report of unilateral phrenic nerve palsy supports a conservative approach to management even in the setting of blunt trauma. Phrenic nerve palsy is relatively rare, and has a range of aetiologies, including trauma, iatrogenesis (with chiropractic manipulation, cardiac surgery and central venous catheter insertion all implicated) [1,2,3,4] and malignancy, with many cases also considered idiopathic. Unilateral phrenic nerve palsy often presents with subtle symptoms, such as exertional dyspnoea, or is identified incidentally on chest X-ray. However, in cases where symptoms are significant enough, or have failed to resolve after extensive periods of conservative management, surgical intervention exists in the form of diaphragmatic plication. This attenuates the paradoxical movement of the hemidiaphragm
*Corresponding author at: Auckland City Hospital 2-20 Park Road, Grafton, Auckland, ph +64 21 111 6286, Email: [email protected] © 2016 Australian and New Zealand Society of Cardiac and Thoracic Surgeons (ANZSCTS) and the Cardiac Society of Australia and New Zealand (CSANZ). Published by Elsevier B.V. All rights reserved.
Please cite this article in press as: Nelson R, et al. Spontaneous Recovery Following Traumatic Phrenic Nerve Palsy. Heart, Lung and Circulation (2016), http://dx.doi.org/10.1016/j.hlc.2016.05.124
HLC 2152 No. of Pages 3
2
R. Nelson et al.
Table 1 Resolution of symptoms and spirometry
FEV1 (% predicted) FVC (% predicted)
1 month
3 month (standing)
3 month (lying)
7 month
35 month
1.8 (50) 2.1 (45)
1.79 (51) 2.84 (62)
1.14 (33) 2.03 (44)
1.84 (48) 2.6 (54)
2.58 (76) 3.65 (81)
Figure 1 A - 0 months B - 3 months C - 7 months D - 18 months E - 20 months F - 31 months.
with respiration. Versteegh and Tjien [5] describe this surgical technique, and provide an overview of outcomes from several case series which illustrate improvement in vital capacity postoperatively, particularly in the supine position. In less symptomatic patients, there is a weight of evidence to suggest that prolonged conservative management is appropriate. There are numerous case reports and series which describe a very long period before return to previous level of function. Douglas [6] describes 40 patients with idiopathic unilateral diaphragmatic paralysis, who had a range of 2 to 19 years until complete recovery. In another series of 10 patients with traumatic phrenic nerve palsy [7], among whom there was a 6–12-month period of recovery. Dernaika et al. [8] compiled cases from 13 reports and series, in which, of 129 cases of bilateral or unilateral nerve palsy, 40 exhibited full recovery with conservative management, which
ranged from 1 month to 19 years. The authors also report on a case of a 57-year-old man who, after presenting with idiopathic right hemidiaphragmatic paralysis, which despite no clinical or radiological improvement after 9 months, demonstrated complete recovery after 21 months. This evidence, as well as that which is presented in this case, illustrates that a prolonged course of conservative management can be appropriate, and that non-resolution after long periods doesn’t necessitate a surgical approach. A delayed course of recovery has to be considered, and we suggest that management should be considerate of how symptomatic a patient is, as well as progression of clinical/radiological status over time. This is supported by animal studies [9] which illustrate that the timeframe of recovery is incredibly variable and is different depending on the mechanism of the palsy and even from different nerves.
Please cite this article in press as: Nelson R, et al. Spontaneous Recovery Following Traumatic Phrenic Nerve Palsy. Heart, Lung and Circulation (2016), http://dx.doi.org/10.1016/j.hlc.2016.05.124
HLC 2152 No. of Pages 3
3
Spontaneous Recovery Following Traumatic Phrenic Nerve Palsy
This case also provides an instance of blunt trauma as being a cause of diaphragmatic nerve palsy, and shows that full resolution of symptoms with conservative management is possible in cases of blunt trauma to the neck.
Declaration There are no conflicts of interest to declare
References [1] Schram DJ, Vosik W, Cantral D. Diaphragmatic paralysis following cervical chiropractic manipulation: case report and review. CHEST Journal 2001 Feb 1;119(2):638–40. [2] Kimura J. Electrodiagnosis in diseases of nerve and muscle: principles and practice. Oxford University Press; 2013 Aug 12.
[3] Rigg A, Hughes P, Lopez A, Filshie J, Cunningham D, Green M. Right phrenic nerve palsy as a complication of indwelling central venous catheters. Thorax 1997 Sep 1;52(9):831–3. [4] Takasaki Y, Arai T. Transient right phrenic nerve palsy associated with central venous catheterization. British Journal of Anaesthesia 2001 Sep 1;87(3):510–1. [5] Versteegh MI, Braun J, Voigt PG, Bosman DB, Stolk J, Rabe KF, Dion RA. Diaphragm plication in adult patients with diaphragm paralysis leads to long-term improvement of pulmonary function and level of dyspnea. European Journal of Cardio-Thoracic Surgery 2007 Sep 1;32(3):449–56. [6] Douglass BE, Clagett T. The prognosis in idiopathic diaphragmatic paralysis. Diseases of the Chest 1960 Mar 31;37(3):294–7. [7] Iverson LI, Mittal A, Dugan DJ, Samson PC. Injuries to the phrenic nerve resulting in diaphragmatic paralysis with special reference to stretch trauma. The American Journal of Surgery 1976 Aug 31;132(2):263–9. [8] Dernaika TA, Younis WG, Carlile PV. Spontaneous recovery in idiopathic unilateral diaphragmatic paralysis. Respiratory Care 2008 Mar 1;53(3):351–4. [9] Ho¨ke A. Mechanisms of Disease: what factors limit the success of peripheral nerve regeneration in humans? Nature Clinical Practice Neurology 2006 Aug 1;2(8):448–54.
Please cite this article in press as: Nelson R, et al. Spontaneous Recovery Following Traumatic Phrenic Nerve Palsy. Heart, Lung and Circulation (2016), http://dx.doi.org/10.1016/j.hlc.2016.05.124