ORIGINAL ARTICLE
The Clinical Respiratory Journal
Concurrent central nervous system and endobronchial tuberculosis mimicking a metastatic lung cancer Fatma Ciftci1, Naoto Shimbori2 and Demet Karnak1 1 Department of Chest Disease, Ankara University School of Medicine, Ankara, Turkey 2 Department of Internal Medicine, Ankara University School of Medicine, Ankara, Turkey
Abstract Background and Aims: Early diagnosis and treatment of central nervous system tuberculosis are important because of high mortality and morbidity. Airways must be carefully evaluated in differential diagnosis. Methods: We present a rare case of intracranial-endobronchial tuberculosis mimicking lung cancer with brain metastasis. Results: A vegetative mass lesion, confirmed as necrotic granulomatous inflammation, originating from the entrance of the right upper lobe entry, was coagulated and extracted by argon plasma coagulation and cryotherapy to prevent permanent upper lobe atelectasis. Mycobacterium tuberculosis complex was detected in bronchoscopic material. A four-drug initial anti-tuberculous treatment regimen was given in 2 months. The upper lobe atelectasis was resolved at the fourth month of therapy despite upper lobar bronchus patency. The patient was doing well and completed the two-drug maintenance therapy of 7 months with complete resolution. Conclusion: In such cases, tissue diagnosis should be achieved as early as possible and anti-tuberculous treatment was commenced along with advanced interventional techniques. Please cite this paper as: Ciftci F, Shimbori N and Karnak D. Concurrent central nervous system and endobronchial tuberculosis mimicking a metastatic lung cancer. Clin Respir J 2015; ••: ••–••. DOI:10.1111/crj.12311.
Key words bronchoscopy – central nervous system tuberculosis – endobronchial tuberculosis – tuberculoma Correspondence Demet Karnak, MD, Department of Chest Disease, Ankara University School of Medicine, Cebeci, 06100 Ankara, Turkey. Tel: +903125956572 Fax: +903123190046 email: [email protected]; [email protected] Received: 20 August 2014 Revision requested: 11 May 2015 Accepted: 21 April 2015 DOI:10.1111/crj.12311 Authorship and contributorship Fatma Ciftci performed the literature search, data collection, manuscript preparation and review of the manuscript. Naoto Shimbori performed the manuscript preparation and review of the manuscript. Demet Karnak performed the study design, data collection, manuscript preparation and review of manuscript. Ethics Written informed consent was obtained from the patient for publication of this case report and accompanying images. Conflict of interest The authors have stated explicitly that there are no conflicts of interest in connection with this article.
Introduction Tuberculosis (TB) is a prevalent disease in developing countries. Tuberculosis with central nervous system
The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd
involvement (CNS-TB) is uncommon and associated with high morbidity and mortality rates, even in adequately treated patients (1, 2). CNS-TB usually presents as tuberculous meningoencephalitis, and rarely
1
CNS-TB and EBTB
presents as tuberculoma. Although CNS-TB is treatable, delayed diagnosis is associated with severe morbidity (3). Drugs are indicated in patients with diffuse brain tissue involvement; however, tuberculoma is a welldefined mass lesion, allowing relatively straightforward surgical resection. In addition, surgical resection allows for a histopathological diagnosis, which is necessary for any intramedullary mass (4, 5). Endobronchial tuberculosis (EBTB) is a serious disease that is frequently complicated with bronchial stenosis, and it may simulate a bronchial asthma, foreign body aspiration or bronchogenic carcinoma (6, 7). Chest radiography (8), computed tomography (CT) and pulmonary function test also have been allocated in detecting EBTB and differentiating it from the other diseases. EBTB causes problems in both making a diagnosis and following up the endobronchial lesion during the antituberculous treatment. Frequently, the diagnosis and follow-up of EBTB can be managed by bronchoscopy. This form of TB should be immediately subjected to antibiotic treatment to avoid serious complications. Bronchoscopic interventional methods such as argon plasma coagulation, cryotherapy or laser application can be preferred to relieve endobronchial stenosis. We wish to bring attention to the following case of CNS-TB and EBTB mimicking stage IV lung cancer with brain metastasis.
Case presentation A 27-year-old woman with familial Mediterranean fever (FMF) presented to neurosurgery clinic with 1-month duration of persistent headache emerging in her right temporal region and weight loss. She was on colchicine in 1.5 mg/day for years. Questioning of complaints revealed that vomiting and double vision. There was papilledema on fundoscopic examination. Cranial X-ray revealed a mass lesion at the right parietal region, compressing bony structures (Fig. 1A). These findings were considered to be secondary to the compressive effect of the mass and elevated intracranial pressure. She was scheduled on an emergent debulking surgery. Preoperative chest X-ray revealed a right hilar lesion as well as a pneumonic consolidation extending from the right hilus to periphery (Fig. 1B). Chest CT showed a 2–2.5 cm sized necrotic right hilar lymphadenopathy observed along with other enlarged lymph nodes. In addition, a 3-cm soft tissue mass constricted right upper lobe bronchus causing a pneumonic consolidation with pleural nodules in the right lung (Fig. 1C).
2
Ciftci et al.
Thoracic surgery department considered this patient as inoperable lung carcinoma with cranial metastases according to the radiologic evaluation. She underwent emergent right parietal craniotomy and had a total tumor excision. The excised mass consisting of dura and bony tissue samples was reported as suppurative necrotizing granulomatous inflammation and meningoencephalitis. The patient was referred to our clinic for differential diagnoses of lung lesion. She had fever (39°C) and cachexia (body mass index 13.6) at initial physical examination with remarkable coarse crackles, bronchial sound and localized sonor rhonchi at the right upper lung side. Laboratory findings revealed hemoglobin level 10, 2 g/dL, white blood cell count 5500/mL and thrombocyte count 443 000/mL. Blood chemistry was in normal limits except for elevated erythrocyte sedimentation rate (90 mm/h) and C-reactive protein level (22.5 mg/L). She had severe proteinuria (300 mg/dL) most probably because of FMF-related renal amyloidosis. A tuberculin skin test was positive with 18 mm sized induration. A Bacillus Calmette–Guerin vaccination scar was observed on her left shoulder. Sputum acidfast bacteria staining was negative. Fiberoptic bronchoscopy showed a vegetative mass lesion originating from the entrance of the right upper lobe, and obstructing right main bronchus by nearly 50% (Fig. 2). Bronchial brush, fine needle aspiration and lesion biopsy were performed from the occluded right upper lobe entry. Histopathologic examination showed as a necrotic granulomatous inflammation compatible with TB infection. The tumoral mass was coagulated and extracted by argon plasma coagulation and cryotherapy, respectively, to open the main bronchus and prevent permanent upper lobe atelectasis in two separate sessions. Right main bronchus opening was achieved at the end. In the meanwhile, Mycobacterium tuberculosis complex was cultured in an automated radiometric system for mycobacterial growth in culture method on bronchial lavage and the biopsied endobronchial mass. A four-drug initial anti-tuberculous treatment regimen (isoniazid, rifampicin, pyrazinamide and ethambutol) and systemic prednisolone therapy were commenced at a dose of (30 mg/d), diminishing 5 mg weekly until discontinuing. The upper lobe atelectasis resolved at fourth month of therapy despite upper lobar bronchus patency (Fig. 1D). She was doing well and completed the therapy of the nine months with complete resolution.
The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd
Ciftci et al.
CNS-TB and EBTB
(A)
(C)
(B)
(D)
Figure 1. (A) Cranial X-ray shows a lesion at the right parietal region. (B) Chest X-ray shows a right hilar lesion and a pneumonic consolidation at the right lung. (C) Computed tomography shows a necrotic right hilar lymphadenopathy and a soft tissue mass constricted right upper lobe bronchus causing a pneumonic consolidation with pleural nodules in the right lung. (D) Chest X-ray shows resolved consolidation at fourth month of therapy.
Discussion TB is highly prevalent in developing countries. However, intracranial involvement is rare and usually a part of miliary TB. CNS-TB is more common among immunosuppressed individuals such as those with acquired immunodeficiency syndrome (AIDS), alcoholism, diabetes mellitus, malignancy, chronic renal diseases and malnutrition (1, 9). CNS involvement occurs in 2%–15% of all patients with TB and in 10% of those with AIDS-related TB. Synchronous extraneural TB is reported in up to 50% of cases, and may be an important clue to the diagnosis of CNS-TB (1, 2, 10). CNS-TB is the most serious type of systemic TB because of its high mortality rate, common neurological complications and sequelae formation.
The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd
CNS-TB manifests itself primarily as tuberculous meningitis and less commonly as tubercular encephalitis and/or an intracranial tuberculoma. Tuberculomas often occur in the absence of meningitis. However, they may also co-occur with meningitis, as in this case. They more commonly arise as solitary lesions, but multiple tuberculomas can been seen as well (11). EBTB was first described by Richard Morten in 1698 (12). It is a rare and life-threatening disease with serious complications such as bronchial obstruction (13). Long-term tracheobronchial stenosis may cause pulmonary complications such as repeated pulmonary infection, atelectasis, bronchiectasis and honeycomb lung. All of the above conditions may be associated with reduced lung function and physical capacity. In 3
CNS-TB and EBTB
Ciftci et al.
(A)
(B)
(C)
(D)
(E)
(F)
Figure 2. (A) A necrotizing vegetative lesion consistent with endobronchial tuberculosis is seen, which completely obstructs right upper lobe bifurcation carina and upper lobe. (B) The lesion can be passed with forceps. (C) The obstructing lesion is removed by freezing it with cryoextraction. (D) Capillary hemorrhage following cryoextraction is controlled with argon plasma coagulation. (E) Debris are cleaned up with a hot biopsy forceps. (F) Upper lobe patency is provided, albeit to a low extent.
extreme conditions, even death can be seen secondary to respiratory failure or asphyxia (14). Our case was a 27-year-old female with comorbidities as renal amyloidosis and proteinuria due to FMF leading to malnutrition. Protein malnutrition impairs host defense against TB (15). To our best knowledge of the literature, this case illustrated for the first time mistaken with malignancy and treated by interventional bronchoscopy along with antituberculous therapy. At present, the pathogenesis of EBTB is not entirely clear. Possible mechanisms include direct mucosal implantation of bacilli originating from cavitary lesions of parenchyma, from infected lymph nodes which erode through the tracheobronchial tree, lymphatic or hematogenous dissemination from parenchymal lesions located in peribronchial regions (16–19). In our case, the cause
4
was most likely a direct extension of infectious paratracheal lymph nodes into the bronchi. The bronchoscopic appearance of EBTB is closely related to the pathological changes and can be classified into seven subtypes: nonspecific bronchitic (tracheobronchial mucosa is only mildly swollen and/or hyperemic), edematous-hyperemic (tracheobronchial mucosa is severely swollen and hyperemic), actively caseating (tracheobronchial mucosa is swollen, hyperemic and covered with a large amount of whitish cheese-like material), granular (tracheobronchial mucosa appears severely inflamed and there are scattered rice-like nodules), ulcerative (ulcerated tracheobronchial mucosa), tumorous (hyperplastic focal tissue shapes, intraluminal mass-like tumor) and fibrostenotic (tracheobronchial lumen narrows due to fibrous hyperplasia and contractures) (20–22). Our
The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd
Ciftci et al.
case was the tumorous type with ulceration and whitish granules. In active EBTB, the most important goal of treatment is eradication of tubercle bacilli. For treatment, the endobronchial approach may provide bronchial patency in addition to the classical approach. This approach may allow delayed symptom onset and patient relief. Argon plasma coagulation, cryotherapy and sometimes metallic stents are techniques available for the bronchoscopic treatment of this type of nonmalignant tracheobronchial stenosis (23). CNS-TB and EBTB were rarely seen together and can mimic lung cancer with brain metastasis. In such cases, tissue diagnosis should be achieved as early as possible and anti-tuberculous treatment was commenced along with advanced interventional techniques.
References 1. Kelly JD, Teeter LD, Graviss EA, Tweardy DJ. Intracranial tuberculomas in adults: a report of twelve consecutive patients in Houston, Texas. Scand J Infect Dis. 2011;43: 785–91. 2. Li H, Liu W, You C. Central nervous system tuberculoma. J Clin Neurosci. 2012;19(5): 691–5. 3. Prabhakar S, Thuss UA. Central nervous system tuberculosis. Neurol India. 1997;45: 132–40. 4. DeAngelis LM. Intracranial tuberculoma: case report and review of the literature. Neurology. 1981;31(9): 1133–6. 5. Gupta RK, Gupta S, Kumar S. MRI in intraspinal tuberculosis. Neuroradiology. 1994;36(1): 39–43. 6. Hoheisel G, Chan BK, Chan CH, Chan KS, Teschler H, Costabel U. Endobronchial tuberculosis: diagnostic features and therapeutic outcome. Respir Med. 1994;88(8): 593–7. 7. Matthews JI, Matarese SL, Carpenter JL. Endobronchial tuberculosis simulating lung cancer. Chest. 1984;86(4): 642–4. 8. Lee JH, Park SS, Lee DH, Shin DH, Yang SC, Yoo BM. Endobronchial tuberculosis, clinical and bronchoscopic features in 121 cases. Chest. 1992;102(4): 990–4. 9. Chou PS, Liu CK, Lin RT, Lai CL, Chao AC. Central nervous system tuberculosis: a forgotten diagnosis. Neurologist. 2012;18(4): 219–22.
The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd
CNS-TB and EBTB
10. Bathla G, Khandelwal G, Maller VG, Gupta A. Manifestations of cerebral tuberculosis. Singapore Med J. 2011;52(2): 124–30. 11. Cegielski JP, McMurray DN. The relationship between malnutrition and tuberculosis: evidence from studies in humans and experimental animals. Int J Tuberc Lung Dis. 2004;8(3): 286–98. 12. Hudson EH. Respiratory tuberculosis: clinical diagnosis. In: Heaf ERG, editor. Symposium on Tuberculosis. London, Cassel and Co, 1957: 321–464. 13. Albert RK, Petty TL. Endobronchial tuberculosis progressing to bronchial stenosis. Chest. 1976;70(4): 537–9. 14. Ucar Y, Sozener ZC, Karnak D. Endotracheal tuberculosis with obstruction. Southeast Asian J Trop Med Public Health. 2010;41(3): 602–7. 15. Hood ML. A narrative review of recent progress in understanding the relationship between tuberculosis and protein energy malnutrition. Eur J Clin Nutr. 2013;67(11): 1122–8. 16. Davidson PT. Tuberculosis; new views of on old disease. N Engl J Med. 1985;312(23): 1514–5. 17. Smith LS, Schillaci RF, Sarlin RF. Endobronchial tuberculosis: serial fiberoptic bronchoscopy and natural history. Chest. 1987;91(5): 644–7. 18. Kashyap S, Mohapatra PR, Saini V. Endobronchial tuberculosis. Indian J Chest Dis Allied Sci. 2003;45(4): 247–56. 19. Steinfort DP, Smallwood D, Antippa P, Irving LB. Endobronchial extension of granulomatous lymphadenitis in an HIV-positive man with immune reconstitution syndrome. Respirology. 2009;14(7): 1064–6. 20. Chung HS, Lee JH. Bronchoscopic, radiologic and pulmonary function evaluation of endobronchial tuberculosis. Respirology. 2000;5(4): 411–7. 21. Chung HS, Lee JH. Bronchoscopic assessment of the evolution of endobronchial tuberculosis. Chest. 2000;117(2): 385–92. 22. Cakir E, Uyan ZS, Oktem S, Karakoç F, Ersu R, Karadag˘ B, Dag˘lı E. Flexible bronchoscopy for diagnosis and follow up of childhood endobronchial tuberculosis. Pediatr Infect Dis J. 2008;27(9): 783–7. 23. Marasso A, Gallo E, Massaglia GM, Onoscuri M, Bernardi V. Cryosurgery in bronchoscopic treatment of tracheobronchial stenosis. Indications, limits, personal experience. Chest. 1993;103(2): 472–4.
5
The Clinical Respiratory Journal
Concurrent central nervous system and endobronchial tuberculosis mimicking a metastatic lung cancer Fatma Ciftci1, Naoto Shimbori2 and Demet Karnak1 1 Department of Chest Disease, Ankara University School of Medicine, Ankara, Turkey 2 Department of Internal Medicine, Ankara University School of Medicine, Ankara, Turkey
Abstract Background and Aims: Early diagnosis and treatment of central nervous system tuberculosis are important because of high mortality and morbidity. Airways must be carefully evaluated in differential diagnosis. Methods: We present a rare case of intracranial-endobronchial tuberculosis mimicking lung cancer with brain metastasis. Results: A vegetative mass lesion, confirmed as necrotic granulomatous inflammation, originating from the entrance of the right upper lobe entry, was coagulated and extracted by argon plasma coagulation and cryotherapy to prevent permanent upper lobe atelectasis. Mycobacterium tuberculosis complex was detected in bronchoscopic material. A four-drug initial anti-tuberculous treatment regimen was given in 2 months. The upper lobe atelectasis was resolved at the fourth month of therapy despite upper lobar bronchus patency. The patient was doing well and completed the two-drug maintenance therapy of 7 months with complete resolution. Conclusion: In such cases, tissue diagnosis should be achieved as early as possible and anti-tuberculous treatment was commenced along with advanced interventional techniques. Please cite this paper as: Ciftci F, Shimbori N and Karnak D. Concurrent central nervous system and endobronchial tuberculosis mimicking a metastatic lung cancer. Clin Respir J 2015; ••: ••–••. DOI:10.1111/crj.12311.
Key words bronchoscopy – central nervous system tuberculosis – endobronchial tuberculosis – tuberculoma Correspondence Demet Karnak, MD, Department of Chest Disease, Ankara University School of Medicine, Cebeci, 06100 Ankara, Turkey. Tel: +903125956572 Fax: +903123190046 email: [email protected]; [email protected] Received: 20 August 2014 Revision requested: 11 May 2015 Accepted: 21 April 2015 DOI:10.1111/crj.12311 Authorship and contributorship Fatma Ciftci performed the literature search, data collection, manuscript preparation and review of the manuscript. Naoto Shimbori performed the manuscript preparation and review of the manuscript. Demet Karnak performed the study design, data collection, manuscript preparation and review of manuscript. Ethics Written informed consent was obtained from the patient for publication of this case report and accompanying images. Conflict of interest The authors have stated explicitly that there are no conflicts of interest in connection with this article.
Introduction Tuberculosis (TB) is a prevalent disease in developing countries. Tuberculosis with central nervous system
The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd
involvement (CNS-TB) is uncommon and associated with high morbidity and mortality rates, even in adequately treated patients (1, 2). CNS-TB usually presents as tuberculous meningoencephalitis, and rarely
1
CNS-TB and EBTB
presents as tuberculoma. Although CNS-TB is treatable, delayed diagnosis is associated with severe morbidity (3). Drugs are indicated in patients with diffuse brain tissue involvement; however, tuberculoma is a welldefined mass lesion, allowing relatively straightforward surgical resection. In addition, surgical resection allows for a histopathological diagnosis, which is necessary for any intramedullary mass (4, 5). Endobronchial tuberculosis (EBTB) is a serious disease that is frequently complicated with bronchial stenosis, and it may simulate a bronchial asthma, foreign body aspiration or bronchogenic carcinoma (6, 7). Chest radiography (8), computed tomography (CT) and pulmonary function test also have been allocated in detecting EBTB and differentiating it from the other diseases. EBTB causes problems in both making a diagnosis and following up the endobronchial lesion during the antituberculous treatment. Frequently, the diagnosis and follow-up of EBTB can be managed by bronchoscopy. This form of TB should be immediately subjected to antibiotic treatment to avoid serious complications. Bronchoscopic interventional methods such as argon plasma coagulation, cryotherapy or laser application can be preferred to relieve endobronchial stenosis. We wish to bring attention to the following case of CNS-TB and EBTB mimicking stage IV lung cancer with brain metastasis.
Case presentation A 27-year-old woman with familial Mediterranean fever (FMF) presented to neurosurgery clinic with 1-month duration of persistent headache emerging in her right temporal region and weight loss. She was on colchicine in 1.5 mg/day for years. Questioning of complaints revealed that vomiting and double vision. There was papilledema on fundoscopic examination. Cranial X-ray revealed a mass lesion at the right parietal region, compressing bony structures (Fig. 1A). These findings were considered to be secondary to the compressive effect of the mass and elevated intracranial pressure. She was scheduled on an emergent debulking surgery. Preoperative chest X-ray revealed a right hilar lesion as well as a pneumonic consolidation extending from the right hilus to periphery (Fig. 1B). Chest CT showed a 2–2.5 cm sized necrotic right hilar lymphadenopathy observed along with other enlarged lymph nodes. In addition, a 3-cm soft tissue mass constricted right upper lobe bronchus causing a pneumonic consolidation with pleural nodules in the right lung (Fig. 1C).
2
Ciftci et al.
Thoracic surgery department considered this patient as inoperable lung carcinoma with cranial metastases according to the radiologic evaluation. She underwent emergent right parietal craniotomy and had a total tumor excision. The excised mass consisting of dura and bony tissue samples was reported as suppurative necrotizing granulomatous inflammation and meningoencephalitis. The patient was referred to our clinic for differential diagnoses of lung lesion. She had fever (39°C) and cachexia (body mass index 13.6) at initial physical examination with remarkable coarse crackles, bronchial sound and localized sonor rhonchi at the right upper lung side. Laboratory findings revealed hemoglobin level 10, 2 g/dL, white blood cell count 5500/mL and thrombocyte count 443 000/mL. Blood chemistry was in normal limits except for elevated erythrocyte sedimentation rate (90 mm/h) and C-reactive protein level (22.5 mg/L). She had severe proteinuria (300 mg/dL) most probably because of FMF-related renal amyloidosis. A tuberculin skin test was positive with 18 mm sized induration. A Bacillus Calmette–Guerin vaccination scar was observed on her left shoulder. Sputum acidfast bacteria staining was negative. Fiberoptic bronchoscopy showed a vegetative mass lesion originating from the entrance of the right upper lobe, and obstructing right main bronchus by nearly 50% (Fig. 2). Bronchial brush, fine needle aspiration and lesion biopsy were performed from the occluded right upper lobe entry. Histopathologic examination showed as a necrotic granulomatous inflammation compatible with TB infection. The tumoral mass was coagulated and extracted by argon plasma coagulation and cryotherapy, respectively, to open the main bronchus and prevent permanent upper lobe atelectasis in two separate sessions. Right main bronchus opening was achieved at the end. In the meanwhile, Mycobacterium tuberculosis complex was cultured in an automated radiometric system for mycobacterial growth in culture method on bronchial lavage and the biopsied endobronchial mass. A four-drug initial anti-tuberculous treatment regimen (isoniazid, rifampicin, pyrazinamide and ethambutol) and systemic prednisolone therapy were commenced at a dose of (30 mg/d), diminishing 5 mg weekly until discontinuing. The upper lobe atelectasis resolved at fourth month of therapy despite upper lobar bronchus patency (Fig. 1D). She was doing well and completed the therapy of the nine months with complete resolution.
The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd
Ciftci et al.
CNS-TB and EBTB
(A)
(C)
(B)
(D)
Figure 1. (A) Cranial X-ray shows a lesion at the right parietal region. (B) Chest X-ray shows a right hilar lesion and a pneumonic consolidation at the right lung. (C) Computed tomography shows a necrotic right hilar lymphadenopathy and a soft tissue mass constricted right upper lobe bronchus causing a pneumonic consolidation with pleural nodules in the right lung. (D) Chest X-ray shows resolved consolidation at fourth month of therapy.
Discussion TB is highly prevalent in developing countries. However, intracranial involvement is rare and usually a part of miliary TB. CNS-TB is more common among immunosuppressed individuals such as those with acquired immunodeficiency syndrome (AIDS), alcoholism, diabetes mellitus, malignancy, chronic renal diseases and malnutrition (1, 9). CNS involvement occurs in 2%–15% of all patients with TB and in 10% of those with AIDS-related TB. Synchronous extraneural TB is reported in up to 50% of cases, and may be an important clue to the diagnosis of CNS-TB (1, 2, 10). CNS-TB is the most serious type of systemic TB because of its high mortality rate, common neurological complications and sequelae formation.
The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd
CNS-TB manifests itself primarily as tuberculous meningitis and less commonly as tubercular encephalitis and/or an intracranial tuberculoma. Tuberculomas often occur in the absence of meningitis. However, they may also co-occur with meningitis, as in this case. They more commonly arise as solitary lesions, but multiple tuberculomas can been seen as well (11). EBTB was first described by Richard Morten in 1698 (12). It is a rare and life-threatening disease with serious complications such as bronchial obstruction (13). Long-term tracheobronchial stenosis may cause pulmonary complications such as repeated pulmonary infection, atelectasis, bronchiectasis and honeycomb lung. All of the above conditions may be associated with reduced lung function and physical capacity. In 3
CNS-TB and EBTB
Ciftci et al.
(A)
(B)
(C)
(D)
(E)
(F)
Figure 2. (A) A necrotizing vegetative lesion consistent with endobronchial tuberculosis is seen, which completely obstructs right upper lobe bifurcation carina and upper lobe. (B) The lesion can be passed with forceps. (C) The obstructing lesion is removed by freezing it with cryoextraction. (D) Capillary hemorrhage following cryoextraction is controlled with argon plasma coagulation. (E) Debris are cleaned up with a hot biopsy forceps. (F) Upper lobe patency is provided, albeit to a low extent.
extreme conditions, even death can be seen secondary to respiratory failure or asphyxia (14). Our case was a 27-year-old female with comorbidities as renal amyloidosis and proteinuria due to FMF leading to malnutrition. Protein malnutrition impairs host defense against TB (15). To our best knowledge of the literature, this case illustrated for the first time mistaken with malignancy and treated by interventional bronchoscopy along with antituberculous therapy. At present, the pathogenesis of EBTB is not entirely clear. Possible mechanisms include direct mucosal implantation of bacilli originating from cavitary lesions of parenchyma, from infected lymph nodes which erode through the tracheobronchial tree, lymphatic or hematogenous dissemination from parenchymal lesions located in peribronchial regions (16–19). In our case, the cause
4
was most likely a direct extension of infectious paratracheal lymph nodes into the bronchi. The bronchoscopic appearance of EBTB is closely related to the pathological changes and can be classified into seven subtypes: nonspecific bronchitic (tracheobronchial mucosa is only mildly swollen and/or hyperemic), edematous-hyperemic (tracheobronchial mucosa is severely swollen and hyperemic), actively caseating (tracheobronchial mucosa is swollen, hyperemic and covered with a large amount of whitish cheese-like material), granular (tracheobronchial mucosa appears severely inflamed and there are scattered rice-like nodules), ulcerative (ulcerated tracheobronchial mucosa), tumorous (hyperplastic focal tissue shapes, intraluminal mass-like tumor) and fibrostenotic (tracheobronchial lumen narrows due to fibrous hyperplasia and contractures) (20–22). Our
The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd
Ciftci et al.
case was the tumorous type with ulceration and whitish granules. In active EBTB, the most important goal of treatment is eradication of tubercle bacilli. For treatment, the endobronchial approach may provide bronchial patency in addition to the classical approach. This approach may allow delayed symptom onset and patient relief. Argon plasma coagulation, cryotherapy and sometimes metallic stents are techniques available for the bronchoscopic treatment of this type of nonmalignant tracheobronchial stenosis (23). CNS-TB and EBTB were rarely seen together and can mimic lung cancer with brain metastasis. In such cases, tissue diagnosis should be achieved as early as possible and anti-tuberculous treatment was commenced along with advanced interventional techniques.
References 1. Kelly JD, Teeter LD, Graviss EA, Tweardy DJ. Intracranial tuberculomas in adults: a report of twelve consecutive patients in Houston, Texas. Scand J Infect Dis. 2011;43: 785–91. 2. Li H, Liu W, You C. Central nervous system tuberculoma. J Clin Neurosci. 2012;19(5): 691–5. 3. Prabhakar S, Thuss UA. Central nervous system tuberculosis. Neurol India. 1997;45: 132–40. 4. DeAngelis LM. Intracranial tuberculoma: case report and review of the literature. Neurology. 1981;31(9): 1133–6. 5. Gupta RK, Gupta S, Kumar S. MRI in intraspinal tuberculosis. Neuroradiology. 1994;36(1): 39–43. 6. Hoheisel G, Chan BK, Chan CH, Chan KS, Teschler H, Costabel U. Endobronchial tuberculosis: diagnostic features and therapeutic outcome. Respir Med. 1994;88(8): 593–7. 7. Matthews JI, Matarese SL, Carpenter JL. Endobronchial tuberculosis simulating lung cancer. Chest. 1984;86(4): 642–4. 8. Lee JH, Park SS, Lee DH, Shin DH, Yang SC, Yoo BM. Endobronchial tuberculosis, clinical and bronchoscopic features in 121 cases. Chest. 1992;102(4): 990–4. 9. Chou PS, Liu CK, Lin RT, Lai CL, Chao AC. Central nervous system tuberculosis: a forgotten diagnosis. Neurologist. 2012;18(4): 219–22.
The Clinical Respiratory Journal (2015) • ISSN 1752-6981 © 2015 John Wiley & Sons Ltd
CNS-TB and EBTB
10. Bathla G, Khandelwal G, Maller VG, Gupta A. Manifestations of cerebral tuberculosis. Singapore Med J. 2011;52(2): 124–30. 11. Cegielski JP, McMurray DN. The relationship between malnutrition and tuberculosis: evidence from studies in humans and experimental animals. Int J Tuberc Lung Dis. 2004;8(3): 286–98. 12. Hudson EH. Respiratory tuberculosis: clinical diagnosis. In: Heaf ERG, editor. Symposium on Tuberculosis. London, Cassel and Co, 1957: 321–464. 13. Albert RK, Petty TL. Endobronchial tuberculosis progressing to bronchial stenosis. Chest. 1976;70(4): 537–9. 14. Ucar Y, Sozener ZC, Karnak D. Endotracheal tuberculosis with obstruction. Southeast Asian J Trop Med Public Health. 2010;41(3): 602–7. 15. Hood ML. A narrative review of recent progress in understanding the relationship between tuberculosis and protein energy malnutrition. Eur J Clin Nutr. 2013;67(11): 1122–8. 16. Davidson PT. Tuberculosis; new views of on old disease. N Engl J Med. 1985;312(23): 1514–5. 17. Smith LS, Schillaci RF, Sarlin RF. Endobronchial tuberculosis: serial fiberoptic bronchoscopy and natural history. Chest. 1987;91(5): 644–7. 18. Kashyap S, Mohapatra PR, Saini V. Endobronchial tuberculosis. Indian J Chest Dis Allied Sci. 2003;45(4): 247–56. 19. Steinfort DP, Smallwood D, Antippa P, Irving LB. Endobronchial extension of granulomatous lymphadenitis in an HIV-positive man with immune reconstitution syndrome. Respirology. 2009;14(7): 1064–6. 20. Chung HS, Lee JH. Bronchoscopic, radiologic and pulmonary function evaluation of endobronchial tuberculosis. Respirology. 2000;5(4): 411–7. 21. Chung HS, Lee JH. Bronchoscopic assessment of the evolution of endobronchial tuberculosis. Chest. 2000;117(2): 385–92. 22. Cakir E, Uyan ZS, Oktem S, Karakoç F, Ersu R, Karadag˘ B, Dag˘lı E. Flexible bronchoscopy for diagnosis and follow up of childhood endobronchial tuberculosis. Pediatr Infect Dis J. 2008;27(9): 783–7. 23. Marasso A, Gallo E, Massaglia GM, Onoscuri M, Bernardi V. Cryosurgery in bronchoscopic treatment of tracheobronchial stenosis. Indications, limits, personal experience. Chest. 1993;103(2): 472–4.
5
