J Korean Med Sci.  2022 Oct;37(38):e283. 10.3346/jkms.2022.37.e283.

Diaphragmatic Dysfunction due to Neuralgic Amyotrophy After SARSCoV-2 Vaccination: A Case Report

• Kang J
• Cho JY

• KMID: 2533563
• DOI: http://doi.org/10.3346/jkms.2022.37.e283

Abstract

Neuralgic amyotrophy is an idiopathic neuropathy characterized by acute-onset pain, typically in the upper extremity or shoulder, followed by weakness of the associated muscles. Phrenic nerve involvement is rare. We report a 63-year-old man who presented with dyspnea and right shoulder pain after severe acute respiratory syndrome coronavirus 2 (SARSCoV-2) vaccination. His chest radiograph showed an elevated right hemidiaphragm that was absent before vaccination. A pulmonary function test showed a restrictive pattern with a significant reduction (40%) in forced vital capacity in the supine position. Diaphragm ultrasonography revealed a reduction in both diaphragmatic excursion and a thickening fraction of the right hemidiaphragm. Electrophysiological studies suggested a right upper brachial plexopathy. Considering the temporal relationship between the vaccination and absence of other causes, SARS-CoV-2 vaccination was thought to be the reason for neuralgic amyotrophy with diaphragmatic dysfunction. As there was no evidence of hypoventilation or sleep disturbance that may require noninvasive ventilation, the patient was followed with conservative treatment with analgesics. During 8 months of follow-up, his shoulder pain was relieved significantly but dyspnea improved only slightly. Neuralgic amyotrophy is an underdiagnosed etiology of diaphragmatic dysfunction and should be considered in patients with dyspnea and shoulder pain.

Keyword

Diaphragmatic Dysfunction; Neuralgic Amyotrophy; SARS-CoV-2; Vaccination

Figure

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  Fig. 1 Chest radiograph images of the case patient (A) one month before vaccination and (B) one month after vaccination. Elevation of the right hemidiaphragm is noted after vaccination.

Reference

1. Li Q, Guan X, Wu P, Wang X, Zhou L, Tong Y, et al. Early transmission dynamics in Wuhan, China, of novel coronavirus-infected pneumonia. N Engl J Med. 2020; 382(13):1199–1207. PMID: 31995857.

2. Falsey AR, Sobieszczyk ME, Hirsch I, Sproule S, Robb ML, Corey L, et al. Phase 3 safety and efficacy of AZD1222 (ChAdOx1 nCoV-19) COVID-19 vaccine. N Engl J Med. 2021; 385(25):2348–2360. PMID: 34587382.

3. Polack FP, Thomas SJ, Kitchin N, Absalon J, Gurtman A, Lockhart S, et al. Safety and efficacy of the BNT162b2 mRNA COVID-19 vaccine. N Engl J Med. 2020; 383(27):2603–2615. PMID: 33301246.

4. Baden LR, El Sahly HM, Essink B, Kotloff K, Frey S, Novak R, et al. Efficacy and safety of the mRNA-1273 SARS-CoV-2 vaccine. N Engl J Med. 2021; 384(5):403–416. PMID: 33378609.

5. Sadoff J, Gray G, Vandebosch A, Cárdenas V, Shukarev G, Grinsztejn B, et al. Safety and efficacy of single-dose Ad26.COV2.S vaccine against COVID-19. N Engl J Med. 2021; 384(23):2187–2201. PMID: 33882225.

6. Son SA, Kim YJ, Lim SY, Kim HB. Bilateral vocal fold paralysis after COVID-19 mRNA vaccination: a case report. J Korean Med Sci. 2022; 37(25):e201. PMID: 35762144.

7. Kim JG, Kim SY, Oh HS, Jo DH. Parsonage-Turner syndrome following typhoid vaccination. Yonsei Med J. 2021; 62(9):868–871. PMID: 34427074.

8. Kim KK. Acute brachial neuropathy--electrophysiological study and clinical profile. J Korean Med Sci. 1996; 11(2):158–164. PMID: 8835764.

9. Van Eijk JJ, Groothuis JT, Van Alfen N. Neuralgic amyotrophy: an update on diagnosis, pathophysiology, and treatment. Muscle Nerve. 2016; 53(3):337–350. PMID: 26662794.

10. Crespo Burillo JA, Loriente Martínez C, García Arguedas C, Mora Pueyo FJ. Amyotrophic neuralgia secondary to Vaxzevri (AstraZeneca) COVID-19 vaccine. Neurologia (Engl Ed). 2021; 36(7):571–572. PMID: 34330677.

11. Lahrmann H, Grisold W, Authier FJ, Zifko UA. Neuralgic amyotrophy with phrenic nerve involvement. Muscle Nerve. 1999; 22(4):437–442. PMID: 10204776.

12. Blanco-Aparicio M, Montero-Martínez C, Couto-Fernández D, Pernas B, Fernández-Marrube M, Verea-Hernando H. Unilateral painful diaphragm paralysis as the only sign of amyotrophic neuralgia. Arch Bronconeumol. 2010; 46(7):390–392. PMID: 19879033.

13. McEnery T, Walsh R, Burke C, McGowan A, Faul J, Cormican L. Phrenic nerve palsy secondary to Parsonage-Turner syndrome: a diagnosis commonly overlooked. Lung. 2017; 195(2):173–177. PMID: 28138789.

14. van Alfen N, Doorduin J, van Rosmalen MH, van Eijk JJ, Heijdra Y, Boon AJ, et al. Phrenic neuropathy and diaphragm dysfunction in neuralgic amyotrophy. Neurology. 2018; 91(9):e843–e849. PMID: 30054437.

15. Mahajan S, Zhang F, Mahajan A, Zimnowodzki S. Parsonage Turner syndrome after COVID-19 vaccination. Muscle Nerve. 2021; 64(1):E3–E4. PMID: 33890680.

16. Coffman JR, Randolph AC, Somerson JS. Parsonage-Turner syndrome after SARS-CoV-2 BNT162b2 vaccine: a case report. JBJS Case Connect. 2021; 11(3):e21.00370.

17. Tsairis P, Dyck PJ, Mulder DW. Natural history of brachial plexus neuropathy. Report on 99 patients. Arch Neurol. 1972; 27(2):109–117. PMID: 4339239.

18. Hughes PD, Polkey MI, Moxham J, Green M. Long-term recovery of diaphragm strength in neuralgic amyotrophy. Eur Respir J. 1999; 13(2):379–384. PMID: 10065685.

19. Podnar S. Idiopathic phrenic neuropathies: a case series and review of the literature. Muscle Nerve. 2015; 52(6):986–992. PMID: 25846409.