Sarcoidosis and True Vocal Fold Paresis: 2 Cases and a Review of

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Acta Otorrinolaringol Esp. 2014;65(6):378---380
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CASE STUDY
Sarcoidosis and True Vocal Fold Paresis: 2 Cases and a Review
of the Literature夽
Sarcoidosis y parálisis de cuerdas vocales: a propósito de 2 casos y revisión
de la literatura
Joan Lop Gros,∗ Jacinto García Lorenzo, Miquel Quer Agustí, Carolina Bothe González
Servicio de Otorrinolaringología, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain
Received 15 January 2013; accepted 13 April 2013
Clinical Cases
Case 1
A 61-year-old male reported isolated dysphonia of 3 years’
evolution. Examination revealed bilateral paresis of the
vocal folds in the paramedian position, with no dyspnoea
or other accompanying symptoms.
Cervical CAT scan showed no alterations. A chest X-ray
was performed which revealed bilateral hilar adenopathies.
Since sarcoidosis was suspected, a thoracic CAT scan was
requested which confirmed the presence of pretracheal,
subcarinal and bilateral hilar enlarged lymph nodes.
Differential diagnosis of the adenopathies was started, a
Mantoux test PPD was performed, which resulted negative,
and calcaemia and angiotensin converting enzyme (ACE)
levels were measured, which were normal. The patient
underwent videomediastinoscopy and the adenopathies
夽 Please cite this article as: Lop Gros J, García Lorenzo J, Quer
Agustí M, Bothe González C. Sarcoidosis y parálisis de cuerdas
vocales: a propósito de 2 casos y revisión de la literatura. Acta
Otorrinolaringol Esp. 2014;65:378---380.
∗ Corresponding author.
E-mail addresses: [email protected], [email protected]
(J. Lop Gros).
were biopsied. The pathology report showed non-caseating
granulomas, therefore a diagnosis of sarcoidosis was made.
Treatment was started with prednisone 1 mg/kg/day for
3 months, with complete recovery of left vocal fold (LVF)
mobility, whereas the paralysis of the right vocal fold (RVF)
persisted.
Case 2
A 35-year-old woman reported isolated dysphonia of 2 years’
evolution. Examination revealed paralysis in the paramedian position of the LVF. Bilateral hilar adenopathies were
revealed on cervicothoracic CAT scan, with no alterations
at cervical level. Fibrobronchoscopy was performed with a
biopsy, the histological study confirmed the presence of noncaseating granulomas, a diagnosis of sarcoidosis was made
and treatment was started with prednisone 1 mg/kg/day for
6 months.
After 6 months’ treatment, and despite the radiological
disappearance of the adenopathies, the LVF paralysis persisted.
Discussion
An aetiologic study must be started for patients with a history of dysphonia due to paralysis or paresis of the vocal
folds (VF). Studies show that the most common causes of
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Sarcoidosis and True Vocal Fold Paresis
Table 1
379
Published Cases of Vocal Fold Paralysis Associated With Sarcoidosis.
Reference
Findings
Age,
gender
Aetiology
Treatment
Coffey
et al.
Bilateral VF paralysis
bilateral hilar,
peritracheal
and subcarinal
lymphadenopathies
Bilateral VF paralysis;
mediastinal,
peritracheal and apical
lymphadenopathies;
polyneuritis
LVF paralysis; hilar
lymphadenopathies
35, male
Bilateral
compressive lymphadenopathies
Methylprednisolone 1
Complete
resolution,
2
41,
female
Cranial
polyneuropathy
and compressive
lymphadenitis
Methylprednisolone 36
Complete
resolution,
1
Hughes
and
McGavin
Jaffe
LVF paralysis;
paratracheal,
et al.
pretracheal and hilar
lymphadenopathies
Tobias
LVF paralysis; right-sided
et al.
bilateral hilar
and paratracheal
lymphadenopathies
Kassimi
LVF paralysis;
et al.
paratracheal
lymphadenopathies
Povedano LVF paralysis; bilateral
et al.
hilar adenopathies
64,
female
Compressive
adenopathy
N/A
N/A
N/A
64, male
Compressive lymphadenopathies
Prednisone
N/A
Paralysis
persists,
32
49, male
Compressive lymphadenopathies
Prednisone
N/A
Complete
resolution
45,
female
Compressive lymphadenopathies
Prednisolone
3
53, male
Compressive lymphadenopathies
Prednisone
4
Lop
et al.
61, male
Compressive lymphadenopathies
Prednisone
150
Complete
resolution,
1.5
Complete
resolution,
6
Partial
resolution,
12
35,
female
Compressive lymphadenopathies
Prednisone
100
Witt
Lop
et al.
Bilateral VF paralysis;
bilateral pretracheal,
subcarinal and hilar
lymphadenopathies
LVF paralysis; left
paratracheal and
bilateral hilar
lymphadenopathies
Diagnostic
delay (weeks)
Response,
weeks
Persists,
24
Source: Modified from Coffey CS, Vallejo SL, Farrar EK, Judson MA, Halstead LA. Sarcoidosis presenting as bilateral vocal fold paralysis
from bilateral compression of the recurrent laryngeal nerves from thoracic adenopathy. J Voice. 2009;23:631---4.
vocal fold paralysis in adults are endothoracic neoplasias and
trauma (iatrogenic or otherwise) to the larynx, these 2 cases
constitute more than half the cases of paralysis. However,
between 10% and 42% of vocal fold paralyses are considered idiopathic; this is a percentage greater than 3%---17% of
which represents the group of ‘‘rare’’ causes (e.g. granulomatous diseases). Paralysis of the left vocal fold is the most
common.1
Therefore, aetiologic study of a case of vocal fold paralysis starts by ruling out the presence of endothoracic
oncological processes and laryngeal trauma. Once these
have been discounted, the rare causes of vocal fold paralysis should be considered, such as neurological, rheumatic,
autoimmune or granulomatous disease.
Granulomatous diseases are divided into infectious,
principally tuberculosis, and non-infectious, beriliosis and
sarcoidosis, for example. We perform laboratory tests to
make a differential diagnosis; PPD and ACE and calcaemia
levels are useful. The diagnosis of sarcoidosis is based on
the following criteria: (a) suggestive clinico-radiological
features, (b) non-caseating granulomas shown in histology or a positive Kveim/Siltzbach test, (c) the exclusion
of other granulomatous diseases and (d) compatible clinical evolution.2 However, the definitive diagnosis will be
histopathological.
Sarcoidosis is a granulomatous disease of unknown aetiology which can affect any organ. The intra-thoracic region
is most commonly affected (80%---90% of cases)3 with hilar,
mediastinal lymph node and/or pulmonary infiltration.
Granulomas affecting the cervicofacial region in
sarcoidosis occur in 9% of cases.4 Although laryngeal
involvement can occur primarily due to granulomatous
infiltration,4 several cases have been described of paralysis
due to compression of the recurrent nerves caused by
mediastinal adenopathies.5 Povedano et al.,6 based on the
clinical and pathogenic characteristics of described cases of
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380
the disease, suggest 3 mechanisms of paralysis of the vocal
folds in the context of sarcoidosis: (1) primary fixation due
to granulomas, (2) neuropathy due to primary involvement
of the vagus nerve, and (3) neuropathy due to compression of the recurrent nerve secondary to adenopathies.
The distinction between these 3 possibilities will be made
depending on the findings accompanying the vocal fold
paralysis. We opt for the neuropathy group when other
cranial nerves are involved, and for the group of primary
fixation due to granulomas in the case of sarcoidosis of the
larynx itself. Since the laryngeal CAT scans in the 2 cases
which we present were normal, and there were no other
neurological alterations, we assume that we can include
them in the group of paralysis secondary to compressive
adenopathy of the recurrent nerve.
A great many of the cases published on vocal fold paralysis in the context of sarcoidosis fall into the compression
neuropathy group. Coffey et al.5 revised most of the published cases (Table 1) of recurring paralysis in the context
of sarcoidosis and found that these were largely left-sided
recurrent paralyses; this data is to be expected given the
long intra-thoracic route of the left recurrent nerve.7 However, the same authors describe a case of bilateral recurrent
paralysis which is justified by the existence of adenopathies
at paratracheal level and of the right subclavian artery,
where the lower right laryngeal nerve recurs.5 Witt8 and
Alon9 also presented cases of bilateral or right-sided recurrent paralysis in sarcoidosis, but these were associated with
other cranial neuropathies, and constitute examples of neurosarcoidosis and therefore could be included in group 2 of
those proposed by Povedano et al.6
Despite the disparity in laterality, both the unilateral and the bilateral cases showed an excellent response
to treatment with corticosteroids, given the reversion of
inflammation and lymph node oedema. However, in the
cases where introduction of treatment was delayed,
J. Lop Gros et al.
the response to corticosteroids was reduced.8 This observation is reinforced in the two cases which we present, both
share a long evolution time prior to diagnosis (2 years).
In conclusion, it is important to include granulomatous
diseases in the differential diagnosis of recurrent paralysis.
Conflict of Interests
The authors have no conflict of interest to declare.
References
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editors. Neurologic disorders of the larynx. 2nd ed. New York:
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2. Baughman RP, Culver DA, Judson MA. A concise review of
pulmonary sarcoidosis. Am J Respir Crit Care Med. 2011;183:
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5. Coffey CS, Vallejo SL, Farrar EK, Judson MA, Halstead LA. Sarcoidosis presenting as bilateral vocal cord paralysis from bilateral
compression of the recurrent laryngeal nerves from thoracic
adenopathy. J Voice. 2009;23:631---4.
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Sarcoidosis como causa de parálisis del nervio laríngeo recurrente. Presentación de un caso. An Otorrinolaringol Ibero Am.
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Ann Otol Rhinol Laryngol. 2010;119:641---5.