Vol.2, No.9, 495-498 (2013) Case Reports in Clinical Medicine
http://dx.doi.org/10.4236/crcm.2013.29129
Pulmonary tuberculosis presenting with aortic
pseudoaneurysm, diffuse alveolar hemorrhage, and
cardiomyopathy: Is there an intriguing relationship?*
Min Shen1, Xuejun Zeng2#, Xingrong Liu3, Bao Liu4, Baotong Zhou5, Xue Lin6, Ran Tian6
1Department of Rheumatology, Peking Union Medical College Hospital (PUMCH), Chinese Academy of Medical Sciences (CAMS)
& Peking Union Medical College (PUMC), Key Laboratory of Rheumatology and Clinical Immunology, Ministry of Education,
Beijing, China
2Department of General Internal Medicine, PUMCH, CAMS & PUMC, Beijing, China;
#Corresponding Author: zxuejun@yahoo.com
3Department of Cardiac Surgery, PUMCH, CAMS & PUMC, Beijing, China
4Department of Vascular Surgery, PUMCH, CAMS & PUMC, Beijing, China
5Department of Infectious Diseases, PUMCH, CAMS & PUMC, Beijing, China
6Department of Cardiology, PUMCH, CAMS & PUMC, Beijing, China
Received 29 October 2013; revised 27 November 2013; accepted 10 December 2013
Copyright © 2013 Min Shen et al. This is an open access article distributed under the Creative Commons Attribution License, which
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ABSTRACT
A 22-year-old Ch inese man presented with fever,
cough, hoarseness, neck pain, acute heart fail-
ure and hemoptysis. Pulmonary tuberculosis
was proved by sputum culture. Chest imaging
showed an aortic arch pseudoaneurysm and
bilateral ground glass opacities. Echocardi-
ography confirmed cardiomyopathy. With anti-
TB drugs, high-dose prednisone and surgery,
the life of this patient was successfully pro-
longed for more than four months. The con-
comitant disorders of aortic pseudoaneurysm,
alveolar hemorrhage and cardiomyopathy in
pulmonary tuberculosis are intriguing. We pos-
tulate that immune-mediated small vessel vas-
culitis triggered by pathogens plays an impor-
tant role in the pathogenesis of this disease,
rather than a direct TB infection.
Keywords: Pseudoaneurysm; Vasculitis; Thoracic
Aorta
1. INTRODUCTION
Tuberculous pseudoaneurysm is a rare but fatal condi-
tion that is invariably indicative of disseminated tuber-
culosis (TB), which is usually diagnosed with evidence
of TB infection at other positions and fever and persis-
tent pain, besides para-aortic mass [1]. TB is rarely asso-
ciated to diffuse alveolar hemorrhage (DAH), and to our
knowledge, pulmonary TB has been reported only twice
as the cause of DAH [2].
Here, we report a culture-proven pulmonary TB patient
concomitantly presenting with aortic pseudoaneurysm,
DAH and cardiomyopathy, which has not been previ-
ously reported. The intriguing relationship among pseu-
doaneurysm, DAH, cardiomyopathy and TB will be dis-
cussed in this report.
2. CASE PRESENTATION
A 22-year-old Chinese man was admitted to our hos-
pital, complaining of fever and cough for more than one
month, aggravated in the recent 10 days. He also had
fatigue, anorexia, and weight loss, without night sweat,
rash, or arthralgia. Ten days before admission, he began
to suffer from lower limb pain, and was unable to walk.
He experienced progressive shortness of breath, hoarse-
ness, cough productive of blood-tinged sputum, palpita-
tion, oppressive pain of left side of the neck, and left
upper extremity weakness. He was healthy before this
admission. One of his classmates was diagnosed with
pulmonary TB previous year. His mother had a history of
pulmonary TB when she was five. He denied smoking
and drug abuse.
*Competing interests: The authors declare that they have no competing
interests.
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M. Shen et al. / Case Reports in Clinical Medicine 2 (2013) 495-498
496
On admission he looked distressed, with a heart rate of
128 bpm, bilateral blood pressure of 140/82 mm Hg, and
oxygen saturation of 92% under normal conditions.
Physical examination revealed diminished breath sounds
and rales in the right lower lung. Neurological examina-
tion was normal. Laboratory results showed a WBC
count of 5.3 × 109/L (4.0 - 10.0 × 109/L), hemoglobin
126 g/L (120 - 160 g/L), and platelet count of 374 ×
109/L (100 – 300 × 109/L). Urinalysis and renal, hepatic,
and coagulation profiles were normal. Serum investiga-
tions found a raised erythrocyte sedimentation rate (ESR)
and C-reactive protein (CRP). Antinuclear antibodies,
antiphospholipid antibodies and antineutrophil cytoplas-
mic antibodies were negative. He tested negative for the
purified protein derivative of tuberculin (PPD), but his
results of Mycobacterium tuberculosis (MTB)-specific
IFN-γ release assays were as high as 3620 SFCs/106
PBMC. Bronchoscopy was conducted with normal find-
ings. Multiple sputum sample smears found no evidence
for bacterial, mycobacterial, or fungal organisms.
His condition worsened at night on the admission day,
and he presented with severe cough, and orthopnea with
hemoptysis. His pulse rate was 170/min, blood pressure
150/100 mm Hg, respiratory rate 35/min, and his oxygen
saturation was only 90% under reservoir oxygen mask
with velocity of flow 10 L/min. Chest examination re-
vealed widespread rales of bilateral lungs. Laboratory
testing showed an obvious drop of hemoglobin level
from 126 g/L to 101 g/L, and elevation of brain natri-
uretic peptide (BNP) (1441 ng/L; reference value 0 - 100
ng/L), while cardiac enzymes were normal. Chest CT
scan (Figure 1) showed an aortic arch pseudoaneurysm
with a 1.2 × 3.0 cm defect in the lateral wall and mural
thrombus. Meanwhile, high-resolution CT revealed bi-
lateral areas of ground glass opacities. Echocardiography
showed a decreased cardiac systolic function, suggestive
of cardiomyopathy with an ejection fraction of only 48%.
Sputum cultures grew Mycobacterium tuberculosis
after his admission. The patient was treated with a stan-
dard therapy with rifampicin, isoniazid, ethambutol,
pyrazinamide, and moxifloxacin. In addition, corticos-
teroids were given on the same day because vasculitis
was suspected. His symptoms resolved in the first week
while chest CT cleared in about 10 days (Figure 1). His
left ventricular ejection fraction increased to 60%. Two
weeks later, he suddenly developed severe pain in the
chest and lower extremities with high blood pressure,
while the blood pressure of lower limbs could not be
measured. Repeat CT revealed a further increase in the
size of the pseudoaneurysm (Figure 1). The patient un-
derwent an emergency surgery, with exclusion of the
pseudoaneurysm. The pathology of the tissue of the aor-
tic pseudoaneurysm revealed a creeping rupture of the
media of the wall, and focal inflammatory cell infiltra-
(a)(b)
(c)(d)
Figure 1. (a) High-resolution CT reveals bilateral ground glass
opacities and pleural effusion. (b) Longitudinal contrast-en-
hanced CT scan shows a pseudoaneurysm arising from the
aortic arch, with mural thrombus. CT scan also shows a 1.2 ×
3.0 cm defect in the lateral wall of the aortic arch (arrow), and
the pseudoaneurysm compresses the adjacent aortic arch, left
pulmonary artery, and left main bronchus. (c) High-resolution
CT reveals that the lungs get clear after 10 days therapy of anti-
TB drugs and high-dose steroids. (d) Longitudinal contrast-
enhanced CT scan performed three weeks later (after image
shown in A) reveals a further increase in the size of the pseu-
doaneurysm, with the defect in the lateral wall of the aortic
arch increasing to 1.5 × 3.7 cm (arrow).
tion at the adventitia, without caseous necrosis. In addi-
tion, the culture of the tissue was negative for any organ-
isms. The anti-tubercular chemotherapy was continued,
with corticosteroids tapered gradually. After 19 weeks of
the operation, the patient had sudden onset hematemesis
and hypovolemic shock, and died soon thereafter. The
postmortem could not be performed.
3. DISCUSSION
A patient who developed aortic pseudoaneurysm,
DAH, cardiomyopathy, and pulmonary TB is described
in this report. Other infectious, malignant, or immu-
nological etiologies for the development of aortic pseu-
doaneurysm, DAH, and cardiomyopathy were excluded
after extensive studies. We therefore think that these dis-
orders were the complications of TB infection. The con-
comitant appearance of all the unusual complications in
one patient has not been reported in literature.
So far, TB as the cause of pseudoaneurysm has been
only occasionally reported, with less than 50 cases since
1945 in the literature in English [1,3-7]. Typical clinical
scenarios of tuberculous aortic pseudoaneurysm include
evidence of TB infection elsewhere in the body, with 1 or
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M. Shen et al. / Case Reports in Clinical Medicine 2 (2013) 495-498 497
more of 3 presentations: fever and persistent pain related
to the location of the pseudoaneurysm, dysphagia, or
hoarseness; evidence of massive bleeding; or pulsative,
rapidly expanding para-aortic mass [1]. In our patient,
pseudoaneurysm with pulmonary TB should raise the
suspicion of tuberculous aortic pseudoaneurysm. But our
case was unusual because neither anti-fast bacilli nor
caseous necrosis was detected from the tissue of the aor-
tic pseudoaneurysm. Therefore, the cause of pseudoaneu-
rysm in our patient might have been secondary to vascu-
litis which was strongly related to the TB infection, as
could be seen in the relation between TB and Takayasu
arteritis [8], or that between TB and Behcet disease [9].
In our patient, the diagnosis of DAH relied on clinical
(hemoptysis and dyspnea), laboratory (rapid drop of he-
moglobin), and typical radiological findings. The possi-
ble mechanisms for TB associated DAH include infec-
tion related diffuse alveolar damage, immune-mediated
process, and antiphospholipid antibodies [2,10]. Coinci-
dentally, cardiomyopathy was also confirmed by echo-
cardiography. The causal link between TB, DAH and
cardiomyopathy was strongly supported by the prompt
clinical response to anti-TB therapy and corticosteroids.
Indeed, the clinical outcomes claimed that immune-me-
diated damage caused by TB infection might have played
an important role in the pathogenesis of DAH and car-
diac involvement in this patient.
Considering the above findings, aortic pseudoaneu-
rysm, DAH and cardiomyopathy in this TB patient were
most likely caused by immune-mediated vasculitis rather
than direct TB infection, though we did not find a first-
hand evidence of vasculitis. The patient should have
been treated with anti-TB drugs and steroids, coupled
with surgery as soon as possible. Unfortunately, the case
was complicated with DAH and cardiomyopathy at the
time of admission, because of which definitive surgical
treatment could not be instituted on time. Emergency
operation thereafter helped extend his life for more than
four months, but he eventually expired, most probably
owing to the perforation contained in the exclusion
pseudoaneurysm into the gastrointestinal tract.
To our knowledge, this is the first reported case of ac-
tive pulmonary TB complicated with aortic aneurysm,
DAH, and cardiomyopathy in an immune-competent
patient. However, because of a lack of previously pub-
lished experience on diagnosis and therapy, we were
confronted with enormous difficulties. With prompt com-
mencement of anti-TB therapy, high-dose prednisone,
and surgical treatment, it is possible to offer the best
chance for a cure and constitute the only way to salvage
patients struck with this disease. Medical management
alone or surgical management alone is not adequate. As
the prevalence of TB is rising worldwide, we hope this
report will increase alertness in physicians to the com-
plications of TB infection.
4. CONSENT
Written informed consent was obtained from the fami-
lies of the patient for publication of this case report and
accompanying images. A copy of the written consent is
available for review by the Editor-in-Chief of this journal.
5. AUTHORS’ CONTRIBUTIONS
MS was involved in the conception of the report, lit-
erature review, manuscript writing and submission. XZ
was involved in the conception of the report, manuscript
critique and review. XL and BL performed the surgery
and involved in the manuscript critique and review. BZ,
XL and RT were involved in the diagnosis and treatment
course, manuscript critique and review. All authors read
and approved the final manuscript.
6. ACKNOWLEDGEMENTS
This work was supported by the Clinical Research Funds of Chinese
Medical Association (grant number 12040760376), and the Youth Re-
search Funds of Peking Union Medical College Hospital (grant number
2010103).
REFERENCES
[1] Kon, A.S., Grumach, A.S., Colombo, A.L., et al, (2008)
Guidelines in cryptococcosis. Revista da Sociedade Bra-
sileira de Medicina Tropical, 41, 524-544.
[2] Patz Jr., E.F. and Goodman, P.C. (1992) Pulmonary
cryptococcosis. Journal of Thoracic Imaging, 7, 51-55.
http://dx.doi.org/10.1097/00005382-199209000-00008
[3] Severo, C.B., Gazzoni, A.F. and Severo, L.C. (2009) Pul-
monary cryptococcosis. Jornal Brasileiro de Pneumologia,
35, 1136-1144.
http://dx.doi.org/10.1590/S1806-37132009001100012
[4] Silva, A.C.G., Marchiori, E., Souza Jr., A. and Irion, K.L.
(2003) Criptococose pulmonar: Aspectos na tomografia
computadorizada. Radiologia Brasileira, 36, 277-282.
http://dx.doi.org/10.1590/S0100-39842003000500005
[5] Guimarães, M.D., Marchiori, E., Meirelles, G.S.P., et al.
(2013) Fungal infection mimicking pulmonary malign-
nancy: Clinical and radiological characteristics. Lung,
191, 655-662.
http://dx.doi.org/10.1007/s00408-013-9506-0
[6] Fong, I.A., Glória, C., Mesquita, I. and Diogo, N. (1999)
Pulmonary cryptococcosis in an immunocompetent pa-
tient. A case report and a review. Revista Portuguesa de
Pneumologia, 4, 419-426.
[7] Barbosa, A.T.F., Colares, F.A., Gusmão, E.S., Barros,
A.A., Cordeiro, C.G. and Andrade, M.C.T. (2006) Iso-
lated pulmonary cryptococcosis in an immunocompetent
patient. Jornal Brasileiro de Pneumologia, 32, 470-480.
http://dx.doi.org/10.1590/S1806-37132006000500016
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LIST OF ABBREVIATIONS
DAH: diffuse alveolar hemorrhage;
TB: tuberculosis;
ESR: erythrocyte sedimentation rate;
CRP: C-reactive protein;
BNP: brain natriuretic peptide.