Autor :Mastroianni, María Lucila1, Faggionato, Martina1, Cigarra, Cecilia Mariela1, Pintos, Cecilia Inés2, Di María, Marta1
1Physical therapist 2Otolaryngologist Institutions where the authors work: Hospital Interzonal General de Agudos “Petrona Villegas de Cordero”, San Fernando, province of Buenos Aires, Argentina.
https://doi.org/10.56538/ramr.HDUW1663
Correspondencia : mastroiannilucila@gmail.com
ABSTRACT
This article shows a case report
of bilateral vocal cord paralysis (BVCP) in a 64-year-old patient with
Parkinson’s disease (PD), a neurodegenerative disorder that affects motor
coordination and causes alterations in speech, swallowing and breathing. The
patient was admitted to the hospital with respiratory distress, initially
diagnosed as bronchoaspiration pneumonia. After some tests, BVCP in the
paramedian position was diagnosed, thus an emergency tracheostomy was
performed. The patient showed respiratory improvement and subsequent tolerance
to oral feeding.
During her hospitalization,
different strategies for tracheostomy management were implemented and
swallowing evaluations were performed, facilitating the return to oral feeding
of semisolid and liquid consistencies. The patient also received assistance
with motor rehabilitation and had tracheostomy management education both for
her and her family.
Despite the lack of access to
neurology specialists, comprehensive treatment and family training enabled an
adequate recovery and a successful transition to home care, improving the
patient’s quality of life.
The study highlights the rarity
of BVCP in PD patients, the fact that it is frequently misdiagnosed, and the
importance of an interdisciplinary approach to its management.
Key words: Parkinson’s disease, Vocal cords, Differential diagnosis,
Interdisciplinary care team
RESUMEN
Este artículo presenta un caso clínico de
parálisis bilateral de cuerdas vocales (PBCV) en una paciente de 64
años con Enfermedad de Parkinson (EP), un trastorno neurodegenerativo
que afecta la coordinación motora y provoca alteraciones en el habla, la
deglución y la respiración. La paciente ingresó al
hospital con dificultad respiratoria, inicialmente diagnosticada como
neumonía broncoaspirativa. Tras evaluaciones, se diagnosticó PBCV
en posición paramediana, por lo que se optó por realizar una
traqueostomía de urgencia. La paciente mostró mejoría
respiratoria y posterior tolerancia a la alimentación por vía
oral.
Durante su internación, se implementaron distintas
estrategias de manejo de la traqueostomía y se realizaron evaluaciones
deglutorias, facilitando el regreso a la alimentación oral de
consistencias semisólidas y líquidas. La paciente también
fue asistida en su rehabilitación motora y recibió
educación para el manejo de la traqueostomía por parte de su
familia.
A pesar de la falta de acceso a especialistas en
neurología, el tratamiento integral y la capacitación de la
familia permitieron una adecuada recuperación y una transición
exitosa al cuidado domiciliario, mejorando la calidad de vida de la paciente.
El estudio destaca la rareza de la PBCV en pacientes con
EP, su diagnóstico a menudo erróneo y la importancia de un
enfoque interdisciplinario para su manejo.
Palabras clave: Enfermedad de Parkinson, Cuerdas vocales, Diagnóstico diferencial,
Grupo de salud interdisciplinario
Received:10/25/2024
Accepted:11/02/2024
INTRODUCTION
The larynx is a functionally
complex organ that is part of the aerodigestive crossroads; it enables voice
production, and plays a fundamental role in the ventilation and protection of
the airways. Any structural alteration and/or laryngeal dysfunction can have
significant consequences on an individual’s health and quality of life.1 Specifically, neurological conditions represent a risk
factor, as they can interfere with the neuromuscular coordination required for
these vital functions. This is evident in the physiopathology of multiple
neurological disorders that impact speech, swallowing, and/or breathing in
affected individuals.1,2
Parkinson’s Disease (PD) is the second most prevalent neuromuscular disease worldwide,
affecting over 7 million people globally. It is a neurodegenerative disorder
characterized by a progressive loss of motor coordination control, primarily
attributed to a decrease in dopamine secretion by neurons in the basal ganglia.
Its symptoms are classified as motor, neuropsychiatric, and autonomic, with
tremors, hypokinesia, and rigidity being the most prevalent, in addition to
speech and swallowing disorders.3 Laryngeal
alterations manifest as dysphagia, respiratory obstruction, vocal cord problems,
including vocal cord fatigue, voice tremor, dyspnea, rigidity in the voice, and
inability to project the voice, primarily due to vocal cord movement
disorders.3,4 Additionally,
the central control of respiratory muscles can also be affected, which, along
with all other symptoms, can significantly impact the quality of life of
patients with PD.5
Vocal cord paralysis is a rare
diagnosis and is associated with increased morbidity and mortality.4 To our
knowledge, there have been reports of 19 cases worldwide in patients with PD.3,6
The aim of this study is to
report a case of bilateral vocal cord paralysis (BVCP) in a patient with
Parkinson’s disease, which represented a challenge for its diagnosis and
therapeutic definition in the context of hospitalization at a public hospital
in the province of Buenos Aires.
CASE REPORT
The case involves a 64-year-old
female patient with a history of type 2 diabetes treated with oral
hypoglycemics and PD diagnosed in 2021, treated with Levodopa (follow-up
treatment with a private neurologist). Figure 1 shows the timeline with the
relevant events from hospitalization to discharge.
On July 9, 2023, the patient was
admitted to the emergency room of the Hospital Interzonal General de Agudos
“Petrona Villegas de Cordero” located in the northern area of Greater Buenos
Aires. She was admitted due to respiratory difficulty that had begun 48 hours
prior and was progressively worsening. She was alert, responded to simple and
complex commands, had regular ventilatory mechanics, oxygen saturation of 90%
by pulse oximetry (SpO2)
while breathing ambient air, a respiratory rate of 25 breaths per minute, and a
heart rate of 110 beats per minute. The first diagnostic suspicion in the
emergency room was bronchoaspiration pneumonia due to swallowing disorder
associated with PD, thus a swallowing evaluation by the kinesiology team was
initially requested.
During the bedside swallowing
evaluation, inspiratory laryngeal stridor was detected without a stethoscope.
Also, weak voice, ineffective cough, use of accessory muscles for ventilation,
and asymmetry of the soft palate (right-side drooping), with no swallowing or
gag reflex present upon stimulation. Voluntary and spontaneous swallows were
observed.
Given the patient’s clinical
condition, a consultation with the Otorhinolaryngology Department was
requested, which performed a rhinofibrolaryngoscopy (RFL), revealing vocal cord
paralysis in the midline, causing closure of the glottic space (Figure 2),
decreased sensitivity, and tremor in the pharyngeal and laryngeal structures.
After discussing with the treatment team and the patient, it was agreed to perform
an urgent tracheostomy (TQT) to prevent the progression of respiratory failure
(RF). The procedure was carried out on July 11 by the General Surgery
Department using a percutaneous technique, placing a 7.5 cannula with an endotracheal
cuff and a subglottic suction port. Following the procedure, the patient was
transferred to the Intensive Care Unit for monitoring. She was spontaneously
ventilating and hemodynamically stable. Additionally, an enteral feeding tube
(type K108) was placed to ensure proper nutritional intake in a safe manner. In
the following days, the patient responded favorably with improved ventilatory
mechanics and 100% SpO2 on ambient
air.
Various surgical options were
presented to the patient, such as cordopexy, cordotomy, or unilateral partial
arytenoidectomy (procedures that would have required her transfer to another
institution with the capability to handle such complexity) as alternatives to
permanent use of the TQT. But the patient declined any intervention with
potential risks that could prolong her hospital stay. Given this resolution,
the necessary procedures were initiated to manage the required resources for
home hospitalization for patients with TQT.
Starting on July 13, her
hospitalization continued in the Intermediate Care Unit, where the relevant
evaluations were conducted according to the protocol for managing
tracheostomized patients by the kinesiology team, as well as swallowing
assessments in collaboration with otorhinolaryngology.
48 hours after the tracheostomy
was performed, tracheal pressures were measured, showing an inspiratory
pressure of -20 cmH2O
and an expiratory pressure of 5 cmH2O.
With these values, the use of a phonation valve (PV) was tested, showing a good
clinical response, and daytime use was initiated. A few days later, the cannula
was changed to a Biesalski type with an endocannula, and a modified Blue Dye
Test was performed with semisolid foods, which resulted negative, allowing the
start of supervised meals. Due to episodes of mucous blockage in the
endocannula, the use of a passive thermohumidifier filter during the night was
decided, a strategy that proved effective in improving TQT maintenance and the
quality of inspired air.
On the 11th day of
hospitalization, a videofluoroscopy with barium sulfate staining of semisolid
foods, liquids, and solids was performed. During the swallowing of a semisolid
consistency (firm yogurt), the patient demonstrated adequate lip sealing and
proper intraoral handling of the bolus, triggered the swallowing reflex at the
base of the tongue and valleculae, and showed preserved laryngeal mobility and
epiglottic closure, with no residue accumulating in anatomical recesses. No
penetration or aspiration of food was observed, and esophageal transit showed
no particularities. In the evaluation with liquids administered in 5 mL
boluses via syringe, the same results were observed. During the swallowing of
solid food (bread), regular intraoral handling of the bolus was observed, with
slowing down of the preparatory and oral phases, requiring a double swallow,
but no other particularities were noted in the remaining stages of swallowing.
No coughing reflex was triggered during the study. Based on these results, it
was agreed to continue with oral feeding of semisolid, processed, and liquid
consistencies, and gradually progress to solid foods depending on the patient’s
clinical evolution and tolerance. After 8 days of this approach, the patient
progressed to the point where total caloric intake was achieved orally, in
collaboration with the Nutrition Service, allowing for the removal of the K108
tube.
During hospitalization, motor
kinesiology assistance was also provided according to the patient’s abilities
and previous levels of functional independence, achieving slow walking with the
help of family members and encouraging as much self-management as possible in
daily living activities. Additionally, the medical team was advised to begin
the process of requesting supplies for home care through the patient’s health
coverage provider. Family education was provided on the management of the
tracheostomy and its care. This included teaching alarm signs for obstruction
or accidental decannulation, as well as suction techniques for secretions.
The total hospital stay was 33
days until discharge to home care.
Currently, both the patient and
her family continue to attend the hospital monthly for regular follow-up of the
tracheostomy, ensuring the proper maintenance of the artificial airway and
prevention of complications. Moreover, potential changes in the symptoms
associated with BVCP are being evaluated, which might allow for modifications
in the tracheostomy management. Additionally, nutritional follow-up is ongoing
(with scheduled controls through the Nutrition Service). On the other hand,
there are still barriers to accessing follow-up care with a specialized
neurologist, not only to evaluate potential advances in the functional
neuromuscular deterioration due to PD, but also to explore various
pharmacological therapeutic approaches that could improve BVCP. These
limitations stem from the lack of available professionals in the area
accessible to the patient and her family, their economic resources, and the
personal decision to prioritize the clinical stability achieved so far.
DISCUSSION
Bilateral vocal cord paralysis is
rarely observed in patients with PD, being more common with conditions such as
multiple system atrophy. Although cases of this alteration have been reported
in the literature, its incidence in this population is low, suggesting a
pathogenesis that remains poorly understood.6
In the specific case of our patient, the
clinical symptoms presented in the emergency room along with her medical
history initially led to a misdiagnosis. This highlights the importance of an
interdisciplinary approach for a comprehensive evaluation and appropriate
management during hospitalization.
The patient’s initial respiratory
management required an emergency tracheostomy, a strategy similar to what has
been reported in other cases in which maintaining upper airway (UAW) permeability
is prioritized.3,6
A significant limitation in our
case was the inability to use a collaborative medical approach to optimize
neurological medication that could alleviate symptoms associated with BVCP, due
to the lack of specialized professionals at our institution. However, cases in
which BVCP-related stridor has resolved solely with pharmacological treatment—without
the need for surgical intervention—are rare.6-8 In this regard, laryngeal stridor in patients
with PD can have various etiologies beyond BVCP, such as laryngospasm or
dystonia,9 requiring a
thorough evaluation by neurologists, dosage adjustments of different
medications, and individualized follow-up. But this type of specialized care
wasn’t easily accessible for our patient.
In contrast to other reported
cases, our patient did not present with severe dysphagia, which allowed for
oral feeding despite upper airway involvement and typical clinical symptoms
such as dysphonia, vocal tremor, and increased respiratory effort. These
symptoms have also been described in other case reports,3,10 but most of
them involve patients with a longer disease duration since diagnosis.
In the 2018 review by Hamdan et
al, a total of 18 patients with PD were analyzed, reporting a male-to-female
ratio of 2.6:1 for (BVCP), with the most common onset age being 63. 94 years and an average duration of 9.58 years from diagnosis to
symptom onset. In our case, the patient had a similar age to those in
the review but had only a two-year disease progression since diagnosis. This
could suggest a possible late diagnosis in relation to the onset of symptoms.3
The hospital stay was long
despite the resolution of respiratory failure due to limited access to
adequate home care resources for a patient with a tracheostomy. However, during
this time, efforts were made to facilitate the transition home by providing
extensive education and training to the family, who served as the patient’s
primary caregivers.
There are reports of other
laryngeal surgical options performed in patients with PD and BVCP. However, in
our case, these options were offered as a long-term therapeutic strategy after
the tracheostomy (which was the best option in the emergency situation at our
institution). The patient declined these procedures after receiving appropriate
advice in that regard.
Despite the challenges presented
within the context of emergency care of a public hospital, we were able to
assess the case, consider differential diagnoses, and promptly resolve the
emergency situation of a patient with PD presenting with an atypical case of
acute RF due to BVCP. Our institution lacks specialized neurology
professionals to optimally adjust pharmacological treatments and does not have
the resources to offer all surgical alternatives from the onset of symptoms.
However, the interdisciplinary management of the acute episode and subsequent
tracheostomy care allowed the patient to make an informed and timely decision
regarding her therapeutic course. This approach helped prevent severe
complications and ensured a quality of life aligned with her condition and
possibilities.
Conflict of interest
The authors have no conflicts of
interest to declare.
REFERENCES
1. Núñez-Batalla F, Díaz-Molina JP,
Costales-Marcos M, Moreno Galindo C, Suárez-Nieto C.
Neurolaringología. Acta Otorrinolaringol Esp. 2012;63:132-40.
https://doi.org/10.1016/j.otorri.2010.12.003
2. Cámpora H, Falduti A, Hawkes M.
Respiración-deglución: encrucijada aerodigestiva. En: Maddalena
F. Deglución de la A a la Z. Fisiopatología. Evaluación.
Tratamiento. Segunda edición. Ciudad Autónoma de Buenos Aires:
Ediciones Journal; 2019. p. 35-43.
3. Hamdan AL, Khalifee E, Tabet
G. Unilateral Vocal Fold Paralysis in Parkinson Disease: Case Report and Review
of the Literature. J Voice. 2018;32:763-6.
https://doi.org/10.1016/j.jvoice.2017.09.007
4. Brake MK, Anderson J.
Bilateral vocal fold immobility: a 13 year review of etiologies, management and
the utility of the Empey index. J Otolaryngol Head Neck Surg. 2015;44:27. https://doi.org/10.1186/s40463-015-0080-8
5. Monteiro L, Souza-Machado A,
Valderramas S, Melo A. The effect of levodopa on pulmonary function in Parkinson’s
disease: a systematic review and meta-analysis. Clin Ther. 2012;34:1049-55. https://doi.org/10.1016/j.clinthera.2012.03.001
6. Bin Asnawi SA, Bin Mohamed MH,
Bin Doi M. A rare presentation of bilateral vocal cord
paralysis in late stage Parkinson’s disease. Int J Med
Robot [Internet]. 2018;5(r).
https://doi.org/10.15342/ijms.v5ir.244
7. Corbin DO, Williams AC.
Stridor during dystonic phases of Parkinson’s disease. J
Neurol Neurosurg Psychiatry. 1987;50:821-2.
https://doi.org/10.1136/jnnp.50.6.821
8. Tsai CC, Wu MN, Liou LM, Chang
YP. Levodopa reverse stridor and prevent subsequent
endotracheal intubation in Parkinson disease patients with bilateral vocal cord
palsy: A case report. Medicine (Baltimore). 2016;95:e5559. https://doi.org/10.1097/MD.0000000000005559
9. Gan EC, Lau DP,
Cheah KL. Stridor in Parkinson’s disease:
a case of ‘dry drowning’? J Laryngol Otol. 2010;124:668-73.
https://doi.org/10.1017/S0022215109992222
10. Plasse HM, Lieberman AN.
Bilateral vocal cord paralysis in Parkinson’s disease. Arch
Otolaryngol. 1981;107:252-3.
https://doi.org/10.1001/archotol.1981.00790400054013