Anthracosis: an unusual cause of vocal fold paralysis.
Anthracotic pigmentation in the bronchial mucosa is a bronchoscopic finding of pneumoconiosis, or evidence of heavy atmospheric soot. This pigmentation in the tracheobronchial mucosa is surrounded by calcified or noncalcified lymph nodes. Anthracosis is not a previously known cause of left vocal fold paralysis. We present what we believe to be the first reported case of anthracosis-caused vocal fold paralysis.
Anthracosis, a form of pneumoconiosis, is most commonly seen in coal miners, and it is also caused by environmental factors such as air pollution, biomass smoke, and cigarette smoke. (1,2) Intrapulmonary lymphadenopathy (LAP) often occurs in anthracosis, while mediastinal or axillary LAP is rare. (3) Dark anthracotic pigmentation in the bronchial mucosa is a bronchoscopic finding of pneumoconiosis or evidence of heavy atmospheric soot particles. (4)
In this article, we present a case of vocal fold paralysis (VFP) caused by anthracosis, a rare pathology that, to the best of our knowledge, has not been previously indicated in the etiology of VFP.
The patient was an 82-year-old woman who had been living in a rural area that is highly exposed to biomass smoke because biomass is widely used there as an energy source for household cooking and heating. The patient was admitted to our clinic in June 2005 with complaints of dry cough and hoarseness for the preceding 2 months. She was a nonsmoker and had no history of tuberculosis (TB) exposure. She also had no history of weight loss, hemoptysis, night sweats, or anorexia. Tier body temperature and breathing sounds were normal, and there was no organomegaly or LAP. She had normal skin turgor with no peripheral edema.
Left VFP was observed during both phonation and inspiration in the endoscopic larynx examination (figure 1, A and B). Biochemical laboratory test results were normal. Erythrocyte sedimentation rate was 14 mm, and a purified protein derivative (PPD) skin test was 8 mm. The patient's chest radiograph revealed increasing reticular density bilaterally that was more prominent in the right middle and lower zones. Her pulmonary function tests were normal (FVC: 1.86, [FEV.sub.1]: 1.40, [FEV.sub.1]/ FVC: 75.3, [FEF.sub.25-75%]: 1.20, PEF; 2.84).
Computed tomography (CT) of the thorax (figure 2) revealed increased interstitial reticulonodular density on the posterior segment of the upper lobe of the right lung and conglomerated LAPs in the pretracheal, prevascular, retrocaval, aortic pulmonary window, carinal, subcarinal, and hilar areas. The largest nodule was 35 mm in diameter, and there were low-density calcifications inside the LAPs.
Gastric and bronchial lavages were negative for acidfast microorganisms. Ventilation-perfusion scans were as follows: pH: 7.40 mmHg, [P.sub.O2]: 66.4 mmHg, P[CO.sub.2]:42.3 mmHg,; HC[O.sub.3]: 26.1 mmol/L, saturation [O.sub.2]: 92.8%.
On bronchoscopic evaluation, diffuse anthracosis was found to be present in the right and left bronchial systems. The upper right lobe was edematous and twisted, and the segment openings were open. A dirty-white lesion with a regular surface was observed on a necrotic area located on the medial side of the lower right lobe. A transbronchial biopsy of the lesion indicated chronic inflammation, fibrotic proliferation of the interstitium, anthracosis, necrosis, and mucus collection.
During 4 years of follow-up, the patients respiratory function, chest radiograph findings, and endoscopic laryngeal findings remained stable.
Hoarseness caused by left VFP can be due to diseases of the heart (cardiomegaly or mitral stenosis with gross enlargement of the left atrium), aorta (aortic arch aneurysm), esophagus (carcinoma), thyroid (carcinoma), or mediastinal lymph nodes (bronchial carcinoma, sarcoidosis, TB, lymphoma, or silicosis). (5-9) Any of these pathologies can destroy, compress, or stretch the vagal nerves, the thoracic sympathetic outflow, and the laryngeal recurrent nerves. The left laryngeal recurrent nerve, in particular, is more commonly affected than the right nerve by a pathologic mediastinal process because of its longer intrathoracic course around the aortic arch. (10,11) However, the etiology of VFP is unknown in approximately 15% of cases. (10,12)
Anthracosis is mostly seen in coal miners due to accumulation of coal dust in the lungs, but it can also result from exposure to air pollution, biomass pollution, or cigarette smoke. (1,2) Intrapulmonary, but not mediastinal, LAP is common in anthracosis. (3) In addition, in the elderly, intrathoracic lymph nodes are often anthracotic.
To our knowledge, only one case has been reported with the presence of both anthracosis and TB in the same mediastinal LAP nodules. (13) However, there have been no published reports about mediastinal lymphadenopathy caused exclusively by anthracosis that resulted in left VFP. We are presenting what is to our knowledge the first case of VFP resulting from anthrocotic mediastinal LAP; VFP caused by TB lymphadenitis and mediastinal LAP has been previously reported. (7,14,15)
In developed countries, mediastinal LAP with vocal fold dysfunction is generally related to a malignant process. (7) Anthracosis resulting in VFP due to prevascular LAP, especially in the aortic-pulmonary window, is very rare, (3) but it can occur more frequently in developing or underdeveloped countries because of the high risk of exposure to the causative agents. (14) The traction of the lymph nodes caused by fibrosis and compression may lead to left VFP.
In our case, the patient, a senior woman living in a rural area, was exposed to biomass smoke for about 50 years. Although this case of VFP appears to be strongly related to anthracosis, it is possible that other causes, such as a viral infection, should be considered. However, we did eliminate TB and malignancy as the cause of her VFP because (1) she had no history of TB exposure, (2) her PPD was 8 mm, (3) her chest radiograph had no indication of TB, (4) acid-fast bacillus was not observed in the bronchial and gastric lavage and in her tracheobronchial biopsy, and (5) no malignancy and/or TB was found. The stability of the patients condition during 4 years of follow-up further confirmed our diagnosis.
A study by Chung et al suggests that TB can be the causative agent if bronchial stenosis and fibrosis occur with anthracosis. (16) In his series of 908 patients, there were 28 cases with anthracofibrosis, only 6 cases with anthracosis, and 43 cases with endobronchial TB without anthracotic pigmentation. In addition, 61% of the anthracofibrosis patients had active TB, whereas none of the anthracosis patients did. These findings indicate that anthracosis can be present with TB, although detection of the TB is difficult. In our patient's case, TB also was eliminated because there was no anthracofibrosis or any obstructive mass in the airway lumen.
Another study that was conducted in England, evaluating a database of 700 bronchoscopy reports, found only 7 anthracosis cases in the database. (9) Six of the patients had an occupational history of coal or other dust exposure, and only 1 patient had a history of TB. However, VFP was not present in any of the cases.
In conclusion, we are recommending that if a patient with a history of long-time exposure to biomass smoke or similar air pollutants is diagnosed with dysphonia and left VFP during a laryngeal endoscopic examination, the patient should then be referred to a specialist for mediastinal evaluation and to rule out a diagnosis of anthracosis, TB, or malignancy. Also, as demonstrated in this article, anthracosis should be considered/evaluated as a possible cause of 15% of VFP cases with an unknown etiology. (10,12)
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Sedat Aydin, MD; Ozlem Celebi, MD; Merve Kiroglu, MD; Mehmet Gokhan Demir, MD
From the Department of Otolaryngology, Kartal Teaching and Research Hospital, Istanbul, Turkey (Dr. Aydin, Dr. Celebi, and Dr. Kiroglu); and the ENT Department, Prof. Dr. Celal Ertug Etimesgut State Hospital, Ankara, Turkey (Dr. Demir).
Corresponding author: Sedat Aydin, MD, Department of Otolaryngology, Kartal Teaching and Research Hospital, Istasyon caddesi Merdivenli sok Ozkan, Apt. No. 5 d 6, Kartal, Istanbul, Turkey. Email: firstname.lastname@example.org
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|Title Annotation:||ORIGINAL ARTICLE|
|Author:||Aydin, Sedat; Celebi, Ozlem; Kiroglu, Merve; Demir, Mehmet Gokhan|
|Publication:||Ear, Nose and Throat Journal|
|Article Type:||Case study|
|Date:||Jul 1, 2015|
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