Bilateral vocal cord paralysis can cause obstruction of glottic airflow, resulting in respiratory distress and the need for a surgical airway. We report a case of acute onset bilateral vocal cord paralysis in a healthy adolescent patient 9 days after a mild severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection. This 15-year-old patient presented with dyspnea, tachypnea, and stridor. A fiberoptic laryngoscopy revealed bilateral vocal fold paralysis with limited vocal cord abduction causing respiratory distress. A detailed set of diagnostic test results, including blood work, imaging, and a lumbar puncture, were all negative other than her new onset diagnosis of SARS-CoV-2 infection. The patient underwent tracheostomy placement with the subsequent resolution of her symptoms. Her vocal cord function remains impaired, and the patient continues to be tracheostomy-dependent 13 months after the initial presentation. SARS-CoV-2 infection has multiple well-established neurologic complications in children. The current case suggests that vocal cord paralysis may be an additional neuropathic sequela of the virus. Vocal cord pathology should be an important consideration when constructing a differential for children presenting with voice, swallowing, or breathing complaints after SARS-CoV-2 infection.
Unilateral vocal cord paralysis can cause issues with breathing, voice, and swallowing. Bilateral vocal cord paralysis poses a unique challenge because the bilateral nature of the paralysis can obstruct glottic airflow, resulting in stridor, respiratory distress, and in the most severe cases, necessitate a surgical airway. In pediatric patients, vocal cord paralysis can be caused by central nervous system disease, such as Arnold-Chiari malformation type II, or by peripheral injury of contributing fibers of the superior laryngeal nerve or recurrent laryngeal nerve at any point in their course from the base of the skull to the mediastinum. Post-viral neuropathy is one known cause of vocal cord paralysis. Since the onset of the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) pandemic, the authors of several case reports and case series have described adults with unilateral or bilateral vocal cord paralysis as a complication of SARS-CoV-2 infection. We report a case of an adolescent with bilateral vocal cord paralysis associated with SARS-CoV-2 infection that resulted in long-term tracheostomy dependence.
Patient Presentation
A 15-year-old girl with a history of asthma and anxiety presented to the emergency department 13 days after diagnosis of SARS-CoV-2 infection with acute onset shortness of breath. The patient had developed symptoms of congestion, fever, and fatigue ∼2 weeks earlier, prompting home SARS-CoV-2 antigen testing, which resulted positive. Her acute symptoms resolved after 5 days. Nine days after her positive test, she acutely developed dyspnea that worsened with activity. Because of persistent symptoms, she presented to the emergency department 14 days after her positive test, at which time she was noted to have intermittent inspiratory stridor without desaturation. A respiratory pathogen polymerase chain reaction panel result, including SARS-CoV-2, was negative. She was treated with steroids and bronchodilators for presumed acute asthma exacerbation without improvement.
During initial admission, otolaryngology was consulted for an evaluation of the glottic airway. A laryngeal scope examination revealed paradoxical vocal fold motion (PVFM), and she was referred to outpatient speech therapy. During the outpatient speech therapy session, her signs were not classic for PVFM because laryngeal control postures did not produce significant relief of stridor. At this point, the patient was referred to our multidisciplinary aerodigestive center. Given continued stridor, as well as the new onset of dysphagia, left-sided weakness, paresthesias, and gait unsteadiness, she was admitted for a comprehensive multidisciplinary workup.
During her hospitalization, otolaryngology, neurology, psychiatry, speech language pathology, and neurosurgery were consulted. Repeat laryngeal scope examinations on admission revealed bilateral vocal cord hypomobility with severely limited abduction and adduction. Cerebrospinal fluid analysis results for oligoclonal bands, varicella zoster virus, Lyme disease, mycoplasma, enterovirus, lower extremity electromyography (EMG), and myasthenia gravis panel were negative. An MRI of the brain and the base of the skull did not reveal any abnormality. An MRI of the spine revealed a syrinx from T4–5 to T10, which after review by neurology and neurosurgery, was judged to be unrelated to her presenting signs and symptoms. A video fluoroscopic swallow study revealed no aspiration or penetration. Her left-sided weakness and paresthesias were thought most likely to be functional in nature, although a SARS-CoV-2-related etiology could not be excluded. Her vocal cord paresis was thought likely to be related to the preceding SARS-CoV-2 infection.
In an attempt to relieve her airway symptoms, otolaryngology injected botulinum toxin type A into the bilateral thyroarytenoid and cricothyroid muscles. This approach has been shown to be an alternative to tracheostomy in pediatric patients with bilateral laryngeal paralysis and has been used in patients with refractory PVFM.1 However, stridor and dyspnea persisted. She ultimately underwent a tracheostomy, which relieved her stridor and dyspnea. Subsequent trials of capping her tracheostomy have been unsuccessful, and she remains tracheostomy-dependent 13 months after her initial presentation. Notably, weakness and paresthesias had resolved by the time of her first outpatient neurology follow-up appointment, consistent with a transient SARS-CoV-2-related effect versus a functional disorder precipitated by the significant stress caused by her breathing difficulties at the time of her initial presentation.
Discussion
By February 2023, there had been >15 million reported cases of pediatric SARS-CoV-2 infection.2 Various neurologic sequelae of SARS-CoV-2 infection have been well-described in children, including headache, seizure, peripheral neuropathy, stroke, demyelinating disorders, and encephalopathy.3 In adults, neurologic complications have been reported in 14% to 57% of patients hospitalized with SARS-CoV-2 infection.4 Postulated mechanisms of neurologic injury secondary to SARS-CoV-2 include direct viral injury to neural cells, vascular endothelial injury, inflammatory injury, or a combination of these etiologies, but are overall not well-understood.3
Peripheral nervous system manifestations of SARS-CoV-2 infection are increasingly being recognized and studied. Reported presentations include nerve pain, skeletal muscle injury, Guillain-Barre syndrome, cranial polyneuritis, neuromuscular junction disorders, neuro-ophthalmologic disorders, neurosensory hearing loss, and dysautonomia.5 Cranial nerves most frequently involved include cranial nerves VII, VI, and III, presenting as hypogeusia or ageusia, facial palsy, and ophthalmoparesis, respectively.6 Incidences of such peripheral neuropathies are not yet readily available.
Rare cases of unilateral or bilateral vocal cord paresis or paralysis have been reported in adult patients with SARS-CoV-2 infections, many of whom have required prolonged orotracheal intubation.7–9 Even rarer cases of vocal cord paralysis or paresis have been reported in patients with SARS-CoV-2 infection who have not required orotracheal intubation, suggesting that vocal cord paresis and paralysis may represent additional neuropathic sequelae of SARS-CoV-2 infection unrelated to trauma or compression from intubation.10–12
The largest case series to date describes a series of 16 adult patients who tested positive for SARS-CoV-2, had no history of intubation as a result of their infection, and presented with vocal cord paresis or paralysis.13 The average age was 49.7 years of age (range 28–72 years). Only 2 of these patients presented with bilateral vocal cord dysfunction, both of whom had paresis as opposed to paralysis. None of these patients presented with respiratory distress or required a surgical airway.
One case of vocal cord pathology has been reported in a pediatric patient with SARS-CoV-2 infection. This patient had unilateral vocal cord paralysis and other cranial nerve neuropathies. This patient recovered function and had a normal laryngoscopic examination 10 days after hospital discharge.14
Our adolescent patient developed bilateral vocal cord paralysis, likely as a late sequela of SARS-CoV-2 infection. A detailed battery of diagnostic tests, including blood work, imaging, and extensive analysis of cerebrospinal fluid could not define another etiology. This patient required a tracheostomy for airway protection. Post-viral neuropathy has persisted for 13 months after initial presentation, suggesting that neuropathy in children after SARS-CoV-2 infection may not always be transient.
The differential diagnosis for our patient included PVFM, a condition that can present similarly to bilateral vocal cord paralysis and is characterized by inappropriate adduction of the vocal cords during inspiration. The mainstay of treatment of PVFM in children is voice therapy and psychiatric treatment. The injection of botulinum toxin into the thyroarytenoid muscle can be used in refractory cases of PVFM to provide temporary relief so a child can participate in speech or psychiatric treatment.15,16 In a case series at our institution of 59 children with PVFM, 76% achieved resolution of symptoms with speech therapy and psychiatric treatment alone. None required surgical intervention with botulinum injection.17
Our patient was diagnosed with PVFM before referral to our pediatric aerodigestive center. However, as she worked with speech therapy it became clear that her presentation was not consistent with PVFM given the absence of any improvement in her signs with laryngeal control postures. Her respiratory signs also failed to improve after botulinum toxin injection in the thyroarytenoid muscle. Notably, she has continued to work with speech therapy and continues to have bilateral vocal fold paralysis after 13 months of therapy, further negating PVFM as a plausible diagnosis.
As of May 2023, there have been 20 718 SARS-CoV-2 cases per 100 000 children in the United States.2 Given the prevalence of this virus, the possibility of vocal cord pathology should be an important consideration for children with voice or swallowing issues (in the case of unilateral vocal cord paralysis/paresis) or in patients with stridor or increased work of breathing (in the case of bilateral vocal cord paresis/paralysis) after even mild SARS-CoV-2 infection. Respiratory distress in the setting of a recent SARS-CoV-2 infection may easily be confused for more common pathologies, such as asthma exacerbation. If the patient does not respond to treatment as expected, prompt evaluation by an otolaryngologist for fiberoptic laryngoscopy should be considered.
Conclusions
We present a case of bilateral vocal cord paralysis in an adolescent patient 2 weeks after presentation with acute SARS-CoV-2 infection. Because of respiratory distress, this patient required tracheostomy placement, which relieved her stridor and dyspnea. However, she remains tracheostomy-dependent 13 months after her initial presentation. Children represent 18% of all reported SARS-CoV-2 infections in the United States. The virus has known neurologic complications, including headache, seizure, and peripheral neuropathy. The current case reveals that vocal cord paralysis may be an additional neuropathic sequela of the virus. The possibility of vocal cord pathology should be included in the differential diagnosis of children who present with voice, swallowing, or breathing complaints after SARS-CoV-2 infection.
Drs Larrow and Hartnick conceptualized and designed the project, drafted the initial manuscript, and reviewed and revised the manuscript; and both authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.
FUNDING: No external funding.
CONFLICT OF INTEREST DISCLOSURES: The authors have indicated they have no potential conflicts of interest relevant to this article to disclose.
Comments