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Sarcoidosis Presenting as Bilateral Vocal Cord Paralysis From Bilateral Compression of the Recurrent Laryngeal Nerves From Thoracic Adenopathy
Sarcoidosis Presenting as Bilateral Vocal Cord Paralysis From Bilateral Compression of the Recurrent Laryngeal Nerves From Thoracic Adenopathy *Charles S. Coffey, †Stacey L. Vallejo, ‡Emily K. Farrar, †Marc A. Judson, and *Lucinda A. Halstead, Charleston, South Carolina
Summary. Bilateral true vocal fold paralysis is rarely attributable to inflammatory diseases. We describe what appears to be the first case in the medical literature of sarcoidosis presenting as isolated, bilateral true vocal cord paralysis resulting from compressive bilateral mediastinal adenopathy. The presenting symptoms, clinical outcome, radiographs and laryngeal findings are discussed in detail. Sarcoidosis should therefore be added to the differential diagnosis of bilateral vocal fold paralysis. Key Words: Sarcoidosis–Vocal cord–Paralysis–Thoracic lymphadenopathy–Corticosteroids.
INTRODUCTION Vocal cord paralysis is most frequently the result of recurrent laryngeal nerve pathology, and may be caused by injury to the nerve or its proximal pathways. Traumatic injuries and surgical trauma are the most common causes of bilateral vocal cord paralysis, followed by malignancy resulting in compression of the thoracic portion of each of the paired nerves.1 Rarely, bilateral paralysis is attributable to infectious, inflammatory, or neurologic diseases, including both central and peripheral processes. We describe what appears to be the first case in the medical literature of sarcoidosis presenting as isolated, bilateral true vocal cord paralysis. Sarcoidosis should therefore be added to the differential diagnosis of this disorder. CASE REPORT A 35-year-old African American man presented to the emergency department with a 7-day history of dysphagia, dysphonia progressing to aphonia, and low-grade fever. He also complained of nasal congestion, fatigue, and a significant weight loss over the previous week. He went to a physician with these complaints and was treated unsuccessfully with amoxicillin and azithromycin for presumed pharyngitis. He had a history of hypertension. He had no recent illnesses, trauma, travel, or exposure to environmental hazards or sick contacts. Physical examination revealed a well-nourished man in mild airway distress. Mild biphasic stridor was noted with maximal respiratory effort. Frequent throat clearing and choking were noted as he attempted to clear secretions from his airway. The Accepted for publication January 8, 2008. Submitted for presentation: The Voice Foundation’s 37th Annual Symposium, May 28– June 1, 2008, Philadelphia, Pennsylvania. From the *Department of Otolaryngology-Head & Neck Surgery, Medical University of South Carolina, Charleston, South Carolina; yDivision of Pulmonary and Critical Care Medicine, Department of Medicine, Medical University of South Carolina, Charleston, South Carolina; and the zDivision of Rheumatology, Department of Medicine, Medical University of South Carolina, Charleston, South Carolina. Address correspondence and reprint requests to Lucinda A. Halstead, MD, Department of Otolaryngology-Head & Neck Surgery, Medical University of South Carolina, Charleston, 135 Rutledge Avenue, Suite 1130, Charleston, SC 29425. E-mail: [email protected] Journal of Voice, Vol. 23, No. 5, pp. 631-634 0892-1997/$36.00 Ó 2009 The Voice Foundation doi:10.1016/j.jvoice.2008.01.009
patient spoke in a breathy whisper limited to a few words per utterance because of shortness of breath. The nose, oral cavity, and oropharynx were free of visible lesions. The neck was supple without lymphadenopathy or crepitus. Cranial nerve examination revealed no focal deficits. Rigid rod lens laryngoscopy and flexible fiberoptic laryngoscopy demonstrated copious pooling of secretions in the postcricoid region and pyriform sinuses. Both vocal folds were immobile and in the paramedian position, with mild buckling of the folds on effortful phonation (Figure 1A). Computed tomography (CT) of the neck and chest revealed bilateral hilar, peritracheal, and subcarinal lymphadenopathy (Figures 2A, B). A modified barium swallow study showed decreased laryngeal elevation as well as aspiration of thin liquids and laryngeal penetration of thicker liquids. A serum angiotensin-converting enzyme level was elevated at 145 U/L (normal range, 9–67). Titers for antinuclear antibody, Lyme antibody, heterophile antibody, and human immunodeficiency virus (HIV)1 and HIV2 antibodies were negative. Blood tests for erythrocyte sedimentation rate, rheumatoid factor, and antinuclear cytoplasmic antibodies were normal. A purified protein derivative tuberculosis skin test as well as sputum stains and cultures for mycobacteria were negative. Spirometry demonstrated a normal forced vital capacity (FVC), forced expiratory volume in one second (FEV1), and FEV1/FVC ratio. However, the flow-volume loop demonstrated a variable extrathoracic airway obstruction. Endobronchial needle aspiration biopsy of a peritracheal mediastinal lymph node under ultrasound guidance was performed and revealed noncaseating granulomatous inflammation that was stain and culture negative for acid fast organisms and fungi, consistent with a diagnosis of sarcoidosis. The patient was admitted for airway surveillance and nutritional support via nasogastric tube (NGT), but he could not tolerate the NGT because of his narrowed airway and inability to swallow his secretions. The patient was begun on a 3-day course of intravenous methylprednisolone, 1 g intravenous daily, which improved the patient’s airway and swallowing sufficiently to avoid the percutaneous gastrostomy and tracheotomy scheduled for day 3. He was then placed on oral prednisone 40 mg daily. Significant improvement in vocal quality was noted after 1 week, at which time repeat videostroboscopy demonstrated return of left vocal fold function and decreased
632
Journal of Voice, Vol. 23, No. 5, 2009
FIGURE 1. Rigid rod lens laryngoscopy, demonstrating bilateral vocal fold paralysis in the paramedian position with copious postcricoid pooling of secretions (A); return of left vocal fold function and decreased secretions after 1 week (B); and return of bilateral vocal fold function with complete clearance of secretions after 2 weeks (C).
pooling of secretions (Figure 1B). A repeat swallow study showed no aspiration. The patient’s symptoms continued to improve clinically, and laryngeal exam 3 weeks after initial presentation demonstrated recovery of bilateral vocal fold motion and no pooling of secretions (Figure 1C). Three weeks later the patient discontinued prednisone for 4 days and his left true vocal fold motion decreased again. It promptly recovered when the prednisone was resumed.
FIGURE 2. CT imaging of the neck and chest demonstrating likely sites of extralaryngeal compression of the left and right recurrent laryngeal nerves. Axial CT of the chest demonstrates left paratracheal and mediastinal nodes on each side of the aortic arch and a single large right pretracheal node (arrows) (A). Coronal CT of the neck demonstrates two large nodes (arrows) adjacent to the junction of the right subclavian and common carotid arteries (B).
DISCUSSION Sarcoidosis is a multisystem idiopathic granulomatous disease that can affect any organ in the body. Sarcoidosis most commonly affects the respiratory tract, with a predilection for the lower tract. Sarcoidosis of the upper respiratory tract (SURT) is considered uncommon, although it is more frequent than realized.2 SURT may involve the nose, sinuses, larynx, tonsils, and tongue.2 Upper airway abnormalities attributable to sarcoidosis usually are the result of SURT lesions with granulomatous inflammation of the involved structures. Rarely, the upper airway may be affected by sarcoidosis involvement or compression of nerves that affect upper airway musculature. Sarcoidosis has infrequently been described as a cause of unilateral vocal cord paralysis. A search of the literature revealed eight case reports including our own in the English literature of vocal cord paralysis discovered in patients who were subsequently diagnosed with sarcoidosis; of these, only one other case involved paralysis of bilateral vocal cords (Table 1).3–9 The presenting symptom in each of these cases was hoarseness, and several patients were noted to have cough or dysphagia. In each case, a chest radiograph or chest CT scan demonstrated significant enlargement of hilar, peritracheal, or carinal lymph nodes, which were believed to be the cause of recurrent nerve compression and vocal cord paralysis. Oral corticosteroids were used to treat four of the patients with unilateral cord paralysis, three of whom experienced return of vocal cord function.4,5,6,8 The fourth patient, reported by Jaffe et al, suffered persistent unilateral cord paralysis despite 8 months of corticosteroid therapy.4 In addition to compressive lymphadenopathy, sarcoidosis may result in polyneuritis affecting various cranial nerves. A large survey reviewing 115 cases of sarcoidosis affecting the nervous system showed facial nerve paralysis in 50% of cases, as well as frequent involvement of the optic nerve.10 The vagus, auditory, hypoglossal, trigeminal, spinal accessory, and trochlear nerves were less often affected. Colover reported nine cases of ‘‘paralysis of one or both vocal cords.more frequently unilateral than bilateral,’’ although there is no further mention of bilateral paralysis. There is likewise limited information regarding the respective roles of lymphadenopathy and polyneuritis in these cases.
Charles S. Coffey, et al
633
Sarcodosis Presenting as Bilateral TVF Paralysis
TABLE 1. Reported Cases of Vocal Cord Paralysis Associated With Sarcoidosis Reference Coffey et al
Witt
Hughes & McGavin
Jaffe et al
Tobias et al
el-Kassimi et al
Swinburn et al
Chijimatsu et al
Findings Bilateral TVC paralysis; bilateral hilar, peritracheal, and subcarinal lymphadenopathy Bilateral TVC paralysis; mediastinal, paratracheal, and apical lymphadenopathy; polyneuritis Left TVC paralysis; hilar lymphadenopathy and calcification Left TVC paralysis; pretracheal, paraaortic, and hilar lymphadenopathy Left TVC paralysis; bilateral hilar & right peritracheal lymphadenopathy Left TVC paralysis; paratracheal, carinal, and hilar lymphadenopathy Left TVC paralysis; bilateral hilar and paratracheal lymphadenopathy Left TVC atrophy and ‘‘elevation’’ of arytenoid; hilar and paratracheal lymphadenopathy
Age, Sex
Etiology (Presumed)
Therapy
Response
35, Male
Bilateral compressive lymphadenopathy
Methylprednisolone (1 g IV QD 3 3 d), then prednisone (40 mg PO QD)
Resolution of paralysis after 2 wk
41, Female
Cranial polyneuritis and compressive lymphadenopathy
Methylprednisolone (40 mg IV q 6 h)
Resolution of paralysis after 7 d
74, Female
Compressive lymphadenopathy
Not reported
Not reported
64, Male
Compressive lymphadenopathy
Prednisone (60 mg PO QD initial) tapered 3 8 mo
Persistence of paralysis after 8 mo
49, Male
Compressive lymphadenopathy
Prednisone (40 mg PO QD)
Resolution of paralysis
45, Female
Compressive lymphadenopathy
Prednisolone (40 mg PO QD 3 10 d)
Resolution of paralysis after 10 d
25, Male
Compressive lymphadenopathy
No medical management
Spontaneous resolution after 10 mo
17, Male
Compressive lymphadenopathy
Prednisolone (30 mg PO QD 3 2 wk)
Resolution of paralysis after 2 wk
Abbreviations: TVC, true vocal cord; QD, once daily; PO, oral; IV, intravenous.
The only other report in the medical literature that clearly describes bilateral vocal cord paralysis in a sarcoidosis patient was in a patient previously diagnosed with sarcoidosis and polyneuritis who developed paralysis of both vocal cords.9 This was accompanied by facial hypoesthesia, iritis, and mediastinal adenopathy. Vocal cord paralysis was thus attributed to a combination of polyneuritis and recurrent laryngeal nerve compression. The patient’s symptoms resolved after treatment with intravenous corticosteroids. The present report appears to be the first in the literature to describe sarcoidosis presenting as isolated, bilateral true vocal cord paralysis. In contrast to the case described by Witt,9 our patient demonstrated no evidence of polyneuritis or other manifestations of sarcoidosis. Rather, the primary pathology was limited to peritracheal and mediastinal lymphadenopathy,
which we believe led to compression of both of the recurrent laryngeal nerves. Intrathoracic lymphadenopathy is common in sarcoidosis, with radiographic evidence of pathologically enlarged nodes noted in 60–90% of cases.5,8 In each of the six cases of unilateral vocal cord paralysis discussed previously, only the left cord was affected. A similar predilection for left-sided nerve involvement has been described for the more common causes of recurrent laryngeal nerve injury, and is likely because of the longer intrathoracic course of the left nerve as passes around the arch of the aorta.11 In contrast, the right recurrent laryngeal nerve is less vulnerable as it takes a shorter course around the right subclavian artery. However, chest CT images in the current case demonstrated pathologically enlarged peritracheal lymph nodes both at the aortic arch, where the left recurrent
634 laryngeal nerve is vulnerable, and at the junction of the innominate and right subclavian arteries, where the right nerve is at risk. We thus conclude that bilateral nerve compression was responsible for this patient’s bilateral vocal cord paralysis and resulting symptoms. The dramatic recovery of laryngeal function and deglutition with 3 days of steroids further supports the diagnosis of compressive adenopathy. Although operative palpation of the arytenoid joints to determine mobility did not occur due to the patient’s dramatic clinical improvement, the CT scan did not reveal any abnormality of the arytenoid complexes. In summary, we report a case of bilateral vocal cord paralysis from bilateral recurrent laryngeal nerve compression because of sarcoid thoracic adenopathy. Sarcoidosis should be added to the list of causes of this disorder. The condition appears to respond quickly to corticosteroids. REFERENCES 1. Hillel AD. Evaluation and management of bilateral vocal cord immobility. Otolaryngol Head Neck Surg. 1999;121:760-765.
Journal of Voice, Vol. 23, No. 5, 2009 2. Sharma OP. Sarcoidosis of the upper respiratory tract: selected cases emphasizing diagnostic and therapeutic difficulties. Sarcoidosis Vasc Diffuse Lung Dis. 2002;19:227-233. 3. Hughes P, McGavin C. Recurrent laryngeal palsy and mediastinal lymphadenopathy. Respir Med. 1995;89:584-585. 4. Jaffe R, Bogomolski-Yahalom V, Kramer MR. Vocal cord paralysis as the presenting symptom of sarcoidosis. Respir Med. 1994;88:633-636. 5. Tobias JK, Santiago SM, Williams AJ. Sarcoidosis as a cause of left recurrent laryngeal nerve palsy. Arch Otolaryngol Head Neck Surg. 1990;116: 971-972. 6. el-Kassimi FA, Ashour M, Vijayaraghavan R. Sarcoidosis presenting as recurrent left laryngeal nerve palsy. Thorax. 1990;45:565-566. 7. Swinburn CR, Pozniak AL, Davies DG, Treasure T, Johnson NM. Left recurrent laryngeal nerve palsy as the presenting feature of sarcoidosis. Sarcoidosis. 1986;3:67-68. 8. Chijimatsu Y, Tajima J, Washizaki M, Homma H. Hoarseness as an initial manifestation of sarcoidosis. Chest. 1980;78:779-781. 9. Witt RL. Sarcoidosis presenting as bilateral vocal fold paralysis. J Voice. 2003;17:265-268. 10. Colover J. Sarcoidosis with involvement of the nervous system. Brain. 1948;71:451-475. 11. Maisel R, Ogura J. Evaluation and treatment of vocal cord paralysis. Laryngoscope. 1974;84:302-316.
Summary. Bilateral true vocal fold paralysis is rarely attributable to inflammatory diseases. We describe what appears to be the first case in the medical literature of sarcoidosis presenting as isolated, bilateral true vocal cord paralysis resulting from compressive bilateral mediastinal adenopathy. The presenting symptoms, clinical outcome, radiographs and laryngeal findings are discussed in detail. Sarcoidosis should therefore be added to the differential diagnosis of bilateral vocal fold paralysis. Key Words: Sarcoidosis–Vocal cord–Paralysis–Thoracic lymphadenopathy–Corticosteroids.
INTRODUCTION Vocal cord paralysis is most frequently the result of recurrent laryngeal nerve pathology, and may be caused by injury to the nerve or its proximal pathways. Traumatic injuries and surgical trauma are the most common causes of bilateral vocal cord paralysis, followed by malignancy resulting in compression of the thoracic portion of each of the paired nerves.1 Rarely, bilateral paralysis is attributable to infectious, inflammatory, or neurologic diseases, including both central and peripheral processes. We describe what appears to be the first case in the medical literature of sarcoidosis presenting as isolated, bilateral true vocal cord paralysis. Sarcoidosis should therefore be added to the differential diagnosis of this disorder. CASE REPORT A 35-year-old African American man presented to the emergency department with a 7-day history of dysphagia, dysphonia progressing to aphonia, and low-grade fever. He also complained of nasal congestion, fatigue, and a significant weight loss over the previous week. He went to a physician with these complaints and was treated unsuccessfully with amoxicillin and azithromycin for presumed pharyngitis. He had a history of hypertension. He had no recent illnesses, trauma, travel, or exposure to environmental hazards or sick contacts. Physical examination revealed a well-nourished man in mild airway distress. Mild biphasic stridor was noted with maximal respiratory effort. Frequent throat clearing and choking were noted as he attempted to clear secretions from his airway. The Accepted for publication January 8, 2008. Submitted for presentation: The Voice Foundation’s 37th Annual Symposium, May 28– June 1, 2008, Philadelphia, Pennsylvania. From the *Department of Otolaryngology-Head & Neck Surgery, Medical University of South Carolina, Charleston, South Carolina; yDivision of Pulmonary and Critical Care Medicine, Department of Medicine, Medical University of South Carolina, Charleston, South Carolina; and the zDivision of Rheumatology, Department of Medicine, Medical University of South Carolina, Charleston, South Carolina. Address correspondence and reprint requests to Lucinda A. Halstead, MD, Department of Otolaryngology-Head & Neck Surgery, Medical University of South Carolina, Charleston, 135 Rutledge Avenue, Suite 1130, Charleston, SC 29425. E-mail: [email protected] Journal of Voice, Vol. 23, No. 5, pp. 631-634 0892-1997/$36.00 Ó 2009 The Voice Foundation doi:10.1016/j.jvoice.2008.01.009
patient spoke in a breathy whisper limited to a few words per utterance because of shortness of breath. The nose, oral cavity, and oropharynx were free of visible lesions. The neck was supple without lymphadenopathy or crepitus. Cranial nerve examination revealed no focal deficits. Rigid rod lens laryngoscopy and flexible fiberoptic laryngoscopy demonstrated copious pooling of secretions in the postcricoid region and pyriform sinuses. Both vocal folds were immobile and in the paramedian position, with mild buckling of the folds on effortful phonation (Figure 1A). Computed tomography (CT) of the neck and chest revealed bilateral hilar, peritracheal, and subcarinal lymphadenopathy (Figures 2A, B). A modified barium swallow study showed decreased laryngeal elevation as well as aspiration of thin liquids and laryngeal penetration of thicker liquids. A serum angiotensin-converting enzyme level was elevated at 145 U/L (normal range, 9–67). Titers for antinuclear antibody, Lyme antibody, heterophile antibody, and human immunodeficiency virus (HIV)1 and HIV2 antibodies were negative. Blood tests for erythrocyte sedimentation rate, rheumatoid factor, and antinuclear cytoplasmic antibodies were normal. A purified protein derivative tuberculosis skin test as well as sputum stains and cultures for mycobacteria were negative. Spirometry demonstrated a normal forced vital capacity (FVC), forced expiratory volume in one second (FEV1), and FEV1/FVC ratio. However, the flow-volume loop demonstrated a variable extrathoracic airway obstruction. Endobronchial needle aspiration biopsy of a peritracheal mediastinal lymph node under ultrasound guidance was performed and revealed noncaseating granulomatous inflammation that was stain and culture negative for acid fast organisms and fungi, consistent with a diagnosis of sarcoidosis. The patient was admitted for airway surveillance and nutritional support via nasogastric tube (NGT), but he could not tolerate the NGT because of his narrowed airway and inability to swallow his secretions. The patient was begun on a 3-day course of intravenous methylprednisolone, 1 g intravenous daily, which improved the patient’s airway and swallowing sufficiently to avoid the percutaneous gastrostomy and tracheotomy scheduled for day 3. He was then placed on oral prednisone 40 mg daily. Significant improvement in vocal quality was noted after 1 week, at which time repeat videostroboscopy demonstrated return of left vocal fold function and decreased
632
Journal of Voice, Vol. 23, No. 5, 2009
FIGURE 1. Rigid rod lens laryngoscopy, demonstrating bilateral vocal fold paralysis in the paramedian position with copious postcricoid pooling of secretions (A); return of left vocal fold function and decreased secretions after 1 week (B); and return of bilateral vocal fold function with complete clearance of secretions after 2 weeks (C).
pooling of secretions (Figure 1B). A repeat swallow study showed no aspiration. The patient’s symptoms continued to improve clinically, and laryngeal exam 3 weeks after initial presentation demonstrated recovery of bilateral vocal fold motion and no pooling of secretions (Figure 1C). Three weeks later the patient discontinued prednisone for 4 days and his left true vocal fold motion decreased again. It promptly recovered when the prednisone was resumed.
FIGURE 2. CT imaging of the neck and chest demonstrating likely sites of extralaryngeal compression of the left and right recurrent laryngeal nerves. Axial CT of the chest demonstrates left paratracheal and mediastinal nodes on each side of the aortic arch and a single large right pretracheal node (arrows) (A). Coronal CT of the neck demonstrates two large nodes (arrows) adjacent to the junction of the right subclavian and common carotid arteries (B).
DISCUSSION Sarcoidosis is a multisystem idiopathic granulomatous disease that can affect any organ in the body. Sarcoidosis most commonly affects the respiratory tract, with a predilection for the lower tract. Sarcoidosis of the upper respiratory tract (SURT) is considered uncommon, although it is more frequent than realized.2 SURT may involve the nose, sinuses, larynx, tonsils, and tongue.2 Upper airway abnormalities attributable to sarcoidosis usually are the result of SURT lesions with granulomatous inflammation of the involved structures. Rarely, the upper airway may be affected by sarcoidosis involvement or compression of nerves that affect upper airway musculature. Sarcoidosis has infrequently been described as a cause of unilateral vocal cord paralysis. A search of the literature revealed eight case reports including our own in the English literature of vocal cord paralysis discovered in patients who were subsequently diagnosed with sarcoidosis; of these, only one other case involved paralysis of bilateral vocal cords (Table 1).3–9 The presenting symptom in each of these cases was hoarseness, and several patients were noted to have cough or dysphagia. In each case, a chest radiograph or chest CT scan demonstrated significant enlargement of hilar, peritracheal, or carinal lymph nodes, which were believed to be the cause of recurrent nerve compression and vocal cord paralysis. Oral corticosteroids were used to treat four of the patients with unilateral cord paralysis, three of whom experienced return of vocal cord function.4,5,6,8 The fourth patient, reported by Jaffe et al, suffered persistent unilateral cord paralysis despite 8 months of corticosteroid therapy.4 In addition to compressive lymphadenopathy, sarcoidosis may result in polyneuritis affecting various cranial nerves. A large survey reviewing 115 cases of sarcoidosis affecting the nervous system showed facial nerve paralysis in 50% of cases, as well as frequent involvement of the optic nerve.10 The vagus, auditory, hypoglossal, trigeminal, spinal accessory, and trochlear nerves were less often affected. Colover reported nine cases of ‘‘paralysis of one or both vocal cords.more frequently unilateral than bilateral,’’ although there is no further mention of bilateral paralysis. There is likewise limited information regarding the respective roles of lymphadenopathy and polyneuritis in these cases.
Charles S. Coffey, et al
633
Sarcodosis Presenting as Bilateral TVF Paralysis
TABLE 1. Reported Cases of Vocal Cord Paralysis Associated With Sarcoidosis Reference Coffey et al
Witt
Hughes & McGavin
Jaffe et al
Tobias et al
el-Kassimi et al
Swinburn et al
Chijimatsu et al
Findings Bilateral TVC paralysis; bilateral hilar, peritracheal, and subcarinal lymphadenopathy Bilateral TVC paralysis; mediastinal, paratracheal, and apical lymphadenopathy; polyneuritis Left TVC paralysis; hilar lymphadenopathy and calcification Left TVC paralysis; pretracheal, paraaortic, and hilar lymphadenopathy Left TVC paralysis; bilateral hilar & right peritracheal lymphadenopathy Left TVC paralysis; paratracheal, carinal, and hilar lymphadenopathy Left TVC paralysis; bilateral hilar and paratracheal lymphadenopathy Left TVC atrophy and ‘‘elevation’’ of arytenoid; hilar and paratracheal lymphadenopathy
Age, Sex
Etiology (Presumed)
Therapy
Response
35, Male
Bilateral compressive lymphadenopathy
Methylprednisolone (1 g IV QD 3 3 d), then prednisone (40 mg PO QD)
Resolution of paralysis after 2 wk
41, Female
Cranial polyneuritis and compressive lymphadenopathy
Methylprednisolone (40 mg IV q 6 h)
Resolution of paralysis after 7 d
74, Female
Compressive lymphadenopathy
Not reported
Not reported
64, Male
Compressive lymphadenopathy
Prednisone (60 mg PO QD initial) tapered 3 8 mo
Persistence of paralysis after 8 mo
49, Male
Compressive lymphadenopathy
Prednisone (40 mg PO QD)
Resolution of paralysis
45, Female
Compressive lymphadenopathy
Prednisolone (40 mg PO QD 3 10 d)
Resolution of paralysis after 10 d
25, Male
Compressive lymphadenopathy
No medical management
Spontaneous resolution after 10 mo
17, Male
Compressive lymphadenopathy
Prednisolone (30 mg PO QD 3 2 wk)
Resolution of paralysis after 2 wk
Abbreviations: TVC, true vocal cord; QD, once daily; PO, oral; IV, intravenous.
The only other report in the medical literature that clearly describes bilateral vocal cord paralysis in a sarcoidosis patient was in a patient previously diagnosed with sarcoidosis and polyneuritis who developed paralysis of both vocal cords.9 This was accompanied by facial hypoesthesia, iritis, and mediastinal adenopathy. Vocal cord paralysis was thus attributed to a combination of polyneuritis and recurrent laryngeal nerve compression. The patient’s symptoms resolved after treatment with intravenous corticosteroids. The present report appears to be the first in the literature to describe sarcoidosis presenting as isolated, bilateral true vocal cord paralysis. In contrast to the case described by Witt,9 our patient demonstrated no evidence of polyneuritis or other manifestations of sarcoidosis. Rather, the primary pathology was limited to peritracheal and mediastinal lymphadenopathy,
which we believe led to compression of both of the recurrent laryngeal nerves. Intrathoracic lymphadenopathy is common in sarcoidosis, with radiographic evidence of pathologically enlarged nodes noted in 60–90% of cases.5,8 In each of the six cases of unilateral vocal cord paralysis discussed previously, only the left cord was affected. A similar predilection for left-sided nerve involvement has been described for the more common causes of recurrent laryngeal nerve injury, and is likely because of the longer intrathoracic course of the left nerve as passes around the arch of the aorta.11 In contrast, the right recurrent laryngeal nerve is less vulnerable as it takes a shorter course around the right subclavian artery. However, chest CT images in the current case demonstrated pathologically enlarged peritracheal lymph nodes both at the aortic arch, where the left recurrent
634 laryngeal nerve is vulnerable, and at the junction of the innominate and right subclavian arteries, where the right nerve is at risk. We thus conclude that bilateral nerve compression was responsible for this patient’s bilateral vocal cord paralysis and resulting symptoms. The dramatic recovery of laryngeal function and deglutition with 3 days of steroids further supports the diagnosis of compressive adenopathy. Although operative palpation of the arytenoid joints to determine mobility did not occur due to the patient’s dramatic clinical improvement, the CT scan did not reveal any abnormality of the arytenoid complexes. In summary, we report a case of bilateral vocal cord paralysis from bilateral recurrent laryngeal nerve compression because of sarcoid thoracic adenopathy. Sarcoidosis should be added to the list of causes of this disorder. The condition appears to respond quickly to corticosteroids. REFERENCES 1. Hillel AD. Evaluation and management of bilateral vocal cord immobility. Otolaryngol Head Neck Surg. 1999;121:760-765.
Journal of Voice, Vol. 23, No. 5, 2009 2. Sharma OP. Sarcoidosis of the upper respiratory tract: selected cases emphasizing diagnostic and therapeutic difficulties. Sarcoidosis Vasc Diffuse Lung Dis. 2002;19:227-233. 3. Hughes P, McGavin C. Recurrent laryngeal palsy and mediastinal lymphadenopathy. Respir Med. 1995;89:584-585. 4. Jaffe R, Bogomolski-Yahalom V, Kramer MR. Vocal cord paralysis as the presenting symptom of sarcoidosis. Respir Med. 1994;88:633-636. 5. Tobias JK, Santiago SM, Williams AJ. Sarcoidosis as a cause of left recurrent laryngeal nerve palsy. Arch Otolaryngol Head Neck Surg. 1990;116: 971-972. 6. el-Kassimi FA, Ashour M, Vijayaraghavan R. Sarcoidosis presenting as recurrent left laryngeal nerve palsy. Thorax. 1990;45:565-566. 7. Swinburn CR, Pozniak AL, Davies DG, Treasure T, Johnson NM. Left recurrent laryngeal nerve palsy as the presenting feature of sarcoidosis. Sarcoidosis. 1986;3:67-68. 8. Chijimatsu Y, Tajima J, Washizaki M, Homma H. Hoarseness as an initial manifestation of sarcoidosis. Chest. 1980;78:779-781. 9. Witt RL. Sarcoidosis presenting as bilateral vocal fold paralysis. J Voice. 2003;17:265-268. 10. Colover J. Sarcoidosis with involvement of the nervous system. Brain. 1948;71:451-475. 11. Maisel R, Ogura J. Evaluation and treatment of vocal cord paralysis. Laryngoscope. 1974;84:302-316.