with anthracosis as a cause of vocal cord paralysis
H. Ahmet BİRCAN1, Sema BİRCAN2, Önder ÖZTÜRK1, Songül ÖZYURT1, Ünal ŞAHİN1, Ahmet AKKAYA1
1Süleyman Demirel Üniversitesi Tıp Fakültesi, Göğüs Hastalıkları Anabilim Dalı,
2Süleyman Demirel Üniversitesi Tıp Fakültesi, Patoloji Anabilim Dalı, Isparta.
ÖZET
Vokal kord paralizisi sebebi olarak mediastinal tüberküloz lenfadenit ve antrakozis birlikteliği
Bronşiyal mukozada antrakotik pigment görülmesi pnömokonyoz veya yoğun atmosferik kirliliğin bir bulgusu olarak dü- şünülmektedir. Kalsifik veya kalsifik olmayan lenf nodlarının çevrelediği bronşta antrakotik pigmentasyon ile birlikte bron- şiyal daralma veya tıkanıklığın görülmesi antrakofibrozis olarak adlandırılmaktadır. Tüberküloz ve antrakofibrozis arasın- da da kuvvetli birliktelik saptanmıştır. Süperior mediastende tüberküloz lenfadenitli bir olgunun ses kısıklığı ile başvurma- sı nadir bir durumdur. Bu makalede antrakozis ve mediastinal tüberküloz lenfadenit birlikteliğinin ses kısıklığına neden ol- duğu bir olgu sunulmuştur. Ses kısıklığı, öksürük, halsizlik, iştahsızlık ve gece terlemesi yakınmaları bulunan 66 yaşında- ki kadın hasta mediastinal lenfadenopati etyolojisini araştırmak için kliniğimize yatırıldı. Uygulanan fiberoptik bronkosko- pide sol vokal kord paralizisinin yanı sıra bronşiyal mukozada inflamasyon bulguları ve yaygın antrakotik pigmentasyon görüldü. Bronş lavajı ve mukoza biyopsisinde tüberküloz veya malignite bulgularına rastlanmadı. Ancak daha sonra uy- gulanan torakotomi sonucunda hastada parankimal antrakozis ve çıkarılan lenf nodunda antrakozis ile birlikte kazeifikas- yon gösteren granülomatöz inflamasyon tespit edildi. Parankimal antrakozis ve mediastinal tüberküloz lenfadenit tanısı ile hasta standart antitüberküloz tedavi altına alındı. Ancak tedavisinin ikinci ayında gastrik adenokarsinom operasyonunun abdominal komplikasyonu sonucunda hasta kaybedildi.
Anahtar Kelimeler: Ses kısıklığı, vokal kord paralizisi, mediastinal tüberküloz lenfadenit, pulmoner antrakozis.
Yazışma Adresi (Address for Correspondence):
Dr. H. Ahmet BİRCAN, Süleyman Demirel Üniversitesi Tıp Fakültesi, Göğüs Hastalıkları Anabilim Dalı, Çünür 32200 ISPARTA - TURKEY
e-mail: [email protected]
Anthracosis, a form of pneumoconiosis, is most commonly seen in coal workers (1). Air polluti- on, biomass smoke and cigarette smoke are also known as the other environmental factors for anthracosis (2-4). Anthracosis often causes int- rapulmonary lymphadenopathy but it rarely ca- uses mediastinal or axillary lymphadenopathy (5,6). Dark anthracotic pigmentation in the bronchial mucosa has been regarded as a bronc- hoscopic finding of pneumoconiosis or evidence of heavy atmospheric soot particles (7). Chung et al. stated that anthracotic pigmentation in con- junction with smooth bronchial narrowing or ob- literation, surrounded by calcified or noncalcified lymph nodes, was a typical finding of anthraco- fibrosis (8). There is a potential relationship bet- ween bronchial anthracofibrosis and tuberculosis (8). Tuberculous lymphadenopathy of superior mediastinum is a common condition in underde- veloped countries but presentation with hoarse- ness due to paralysis of recurrent laryngeal ner- ve (RLN) is rare (9,10). Only a few case reports about this subject had been reported in English
literature. In this report, a case of tuberculous mediastinal lymphadenitis with anthracosis is presented as an unusual cause of hoarseness.
CASE REPORT
A 66-year-old woman was admitted to our clinic with the symptoms of dry cough and hoarseness that had started two months ago. Her dry cough was deteriorated especially when she was tal- king. She had also constitutional symptoms such as malaise, anorexia and night sweats and a his- tory of pulmonary infection which was etiologi- cally undiagnosed when she was 7-year-old. She denied an active or passive exposure to cigaret- te or biomass smoke. Also there was no history of any contact with an active tuberculosis pati- ent. On clinical examination her blood pressure was 130/80 mmHg; temperature was 36.2°C and pulse 96 beats/min. Breathing sounds were diminished at the right lung base and there were bilateral scarce end-expiratory crackles. Labora- tory findings were as fallows; Hb: 13.8 mg/dL, Hct: 39%, WBC count: 8630/mm3, MCV: 77.8 fl., SUMMARY
Mediastinal tuberculous lymphadenitis with anthracosis as a cause of vocal cord paralysis
H. Ahmet BİRCAN1, Sema BİRCAN2, Önder ÖZTÜRK1, Songül ÖZYURT1, Ünal ŞAHİN1, Ahmet AKKAYA1
1Department of Chest Diseases, Faculty of Medicine, Süleyman Demirel University, Isparta, Turkey,
2Department of Pathology, Faculty of Medicine, Süleyman Demirel University, Isparta, Turkey.
Anthracotic pigmentation in the bronchial mucosa has been regarded as a bronchoscopic finding of pneumoconiosis or evi- dence of heavy atmospheric soot. Anthracotic pigmentation with bronchial narrowing or obliteration, surrounded by cal- cified or noncalcified lymph nodes is typical finding of anthracofibrosis. There is a potential relationship between bronchi- al anthracofibrosis and tuberculosis. Tuberculous lymphadenopathy of superior mediastinum presentation with hoarseness is very rare. The paper reports a case of tuberculous mediastinal lymphadenitis with anthracosis causing vocal cord pa- ralysis. A 66-year-old woman was admitted to our clinic with the symptoms of dry cough, hoarseness, malaise, anorexia, night sweats and with the multiple mediastinal lymphadenopathy. Fiberoptic bronchoscopy revealed left vocal cord pa- ralysis, bronchial mucosal inflammation and multiple anthracotic plaques. Bronchial lavage and mucosal biopsy were ne- gative for malignancy and tuberculosis. The thoracotomy was performed and a mediastinal lymph node showing caseating granulomatous inflammation with anthracosis and parenchymal anthracosis were detected. The diagnosis of anthracosis and mediastinal tuberculous lymphadenitis was made and the patients put on antituberculous treatment. But she was un- fortunately died in the second month of the treatment because of the abdominal complication of gastric adenocarcinoma operation.
Key Words: Hoarseness, vocal cord paralysis, mediastinal tuberculous lymphadenitis, pulmonary anthracosis.
platelet: 493.000/mm3, ESR: 60 mm/hour, total protein: 8.4 mg/dL, albumin: 5.2 mg/dL and all other biochemical parameters were in normal li- mits. The Mauntoux test was strongly positive for tuberculosis (20 mm). Her chest X-ray revealed left hilar calcification consistent with old tubercu- losis (Figure 1). Computed tomography (CT) of the neck was normal, but thorax CT revealed a few millimetric pulmonary nodules and multiple lymphadenopathy which were localized pretrac- heal, prevascular and hilar regions (Figure 2). Fi- beroptic bronchoscopy revealed left vocal cord paralysis, and diffuse bronchial inflammatory ap- pearance accompanied by anthracotic plaques localized on the multiple lobar orifices. Bronchial lavage was negative for acid fast bacilli and nega- tive for malignancy. The histopathological exami- nation of transbronchial lung biopsy revealed in- terstitial fibrosis and anthracosis, but no granulo-
ma formation or malignancy. Thereafter, the Lo- wenstein-Jensen culture of bronchial lavage re- mained negative. As definite diagnosis could not be reached with these procedures, thoracotomy was performed as a diagnostic procedure. Right paratracheal lymphadenopathy and a 2 mm int- raparenchymal nodule observed during thoraco- tomy were extracted. Histopathological examina- tion of these two materials revealed a fibrotic no- dule surrounded by parenchymal tissue with anthracotic pigmentation and a lymph node sho- wing caseating granulomatous inflammation with anthracosis (Figure 3). In the view of these fin- dings the diagnosis of pulmonary anthracosis and mediastinal tuberculous lymphadenitis was made and patient was put on antituberculous treatment namely, isoniazide, rifampicin, pyrizinamide, and ethambutol along with corticosteroids administe- red in tapering doses. The patient was readmitted to emergency room with the symptoms sugges- ting acute abdomen such as stomach ache, na- usea and vomiting on the second month of the treatment. Histopathological examination of en- doscopic biopsy revealed antral gastric adeno- carcinoma. She was unfortunately died after a short time from the diagnosis due to postoperati- ve complication of total gastrectomy.
DISCUSSION
Anthracosis is mostly seen in coal workers due to accumulation of coal dust in lung, but it can also occur due to exposure to air pollution and biomass or cigarette smoke (1-5). Anthracosis often causes intrapulmonary lymphadenopathy but rarely mediastinal mass or lymphadeno- pathy (6). Beside this, intrathoracic lymph no- des are often anthracotic in elderly persons (11).
In the course of erosion into the bronchus car- bon particles in the lymph node may penetrate through the bronchial wall and the bronchial mucosa may be colored. On the other hand, ne- ither accumulation of anthracotic pigment from air pollutants nor smoke induces focal bronchial abnormality, because carbon is inert and elicits little or no fibrosis. Intrabronchial perforation of tuberculous lymphadenitis is the other well- known cause of anthracotic pigmentation in the bronchial mucosa (12,13). In the study of Chung et al., the patients showing only anthra- cotic pigmentation on bronchial mucosa without Figure 1. The patients’ chest X-ray showing left hilar
calcification.
Figure 2. Thorax CT revealed multiple mediastinal lymphadenopathy without peripheral rim enhace- ment and low attenuation.
bronchial stenosis were excluded, and the cha- racteristic features of the patients with anthraco- fibrosis were described as follows (8):
1. Preponderance of older female patients, 2. No association with pneumoconiosis or smo- king,
3. Chief complaint of cough and dyspnea witho- ut constitutional symptoms,
4. Segmental or lobar consolidation on simple chest radiographs,
5. Abnormalities of bronchial airways with pe- ribronchial cuffs of soft tissue or surrounding lymph nodes on chest CT,
6. Most frequent involvement of the right middle lobe bronchus,
7. Active tuberculous infection demonstrated in
> 60% of the patients.
In our case, she had no history of occupational dust exposure, but air pollution is rampant and biomass usage is highly prevalent in Isparta, a rural town in Lakes region of Turkey where she used to live. In bronchoscopic examination we did not find any sign of endobronchial tubercu- losis or antracofibrosis. The pathological fin- dings revelaed paranchymal anthracosis but there was no evidence indicating malignancy or tuberculosis. So, it was assumed that bronchos- copic findings might be due to air pollution or bi- omass usage.
The next step in the differential diagnosis of me- diastinal lymphadenopaties is mediastinoscopy.
Instead of mediastinoscopy, thoracotomy was performed for excisional biopsy because medi- astinoscopy can not be performed in our center.
The histopathological examination of biopsies sho- wed tuberculous lymphadenitis with anthracosis.
Tuberculous lymphadenitis is one of the most common forms of extrapulmonary tuberculosis and the first lymphoid tissues encountered du- ring spread are presumably hilar and mediastinal lymph nodes (14). However, mediastinal lymph nodes are rarely reported at the site of tuberculous lymphadenitis and it is also very rare in adults (14).
Clinical conditions are quite variable depending on the affected structures in patients with medi- astinal tuberculosis. Hoarseness resulting from involvement of RLN, dysphagia due to compres- sion of esophagus, and typical manifestations of superior vena cava syndrome resulting from in- volvement of superior vena cava can be seen (9,10,14-17).
In developed countries mediastinal lymphade- nopathy with vocal cord dysfunction is generally related to malignant process (15). Tuberculosis is rarely reported in the etiopathogenesis of ho- arseness, although it is frequently seen in the developing countries like ours (9). Entrapment in scar tissue, traction by fibrosis, or compressi- on with enlarged lymphadenopathy lodged in the prevascular or aortopulmonary window sho- uld be discussed in the pathology of paralysis of RLN. Fowler and Hetzel suggested that direct spread of infection from perforated lymph node abscess damage the RLN rather than mechani- cal factors alone (15). Functional damage of Figure 4. A caseating granuloma surrounding with
anthracotic pigmentation is seen in high power (HE, x200).
Figure 3. Histopathological examination of mediasti- nal lymphadenopathy revealed caseating granuloma- tous inflammation with anthracosis (HE, x40).
RLN caused by direct spread of the infection from caseous necrosis in the mediastinum is ir- reversible, but it is complete reversible with an- tituberculous treatment if it has been resulted from compression (10). Although it is suggested that compression of enlarged lymph nodes is the cause of RLN involvement, we couldn’t evaluate any response to the antituberculous therapy be- cause of the early death of our patient.
As the detection of asid fast basilli in micros- copy and culture of the material other than spu- tum is very low; the diagnosis of mediastinal tu- berculous lymphadenitis is quite difficult. The Mountoux test is found positive in 90% of pati- ents with tuberculous lymphadenitis, as in our case. About 30% of chest X-rays show abnor- mality consistent with sequelaand the involve- ment of right paratracheal and tracheobronchial lymph nodes are frequent (14). After injection of contrast medium, central low attenuation and peripheral rim enhancement of nodes larger than 2 cm in diameter is highly suggestive for active tuberculous lymphadenitis (18-20). Simi- lar to the anthracofibrotic patients of Chung et al., multiple mediastinal lymphadenopathies we- re detected in our case without low attenuation and rim enhacement (8). The radiological ab- sence of low attenuation and rim enhancement may be due to the deposition of the coal dust in lymph nodes.
In conclusion, we decided to present this case report because;
1. Hoarseness is an unusual presentation in the tuberculous lympadenitis,
2. In contrast to anthracofibrosis, bronchial anthracosis and tuberculous lymphadenitis were found coincidentally,
3. Both tuberculosis and anthracosis was found in the same lymph node, and
4. Typical radiological image of active tubercu- lous lymphadenitis was not detected.
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