Bilateral spontaneous pneumothorax in a patient with miliary
In addition to oxygen therapy, treatment with a combination of
isoniazid, rifampicin, ethambutol, and pyrazinamide was started on
the third day of hospitalization. On the fifth day of
antituberculous treatment, her fever had not yet decreased, and
methylprednisolone (1 mg/kg) was added to the therapy. Multiple
blood cultures were negative. No pathogens were identified in
aerobic cultures of sputum or urine. Wright and Rose Bengal
agglutination tests for brucellosis were negative. The serum levels
of CA-125 and CA-15.3 were high, but gynecological examination and
thyroid and mammary ultrasonography showed no signs of malignancy.
No fiberoptic bronchoscopy or other invasive procedures were
performed because of respiratory failure. On the 38th day of
hospitalization, she complained of pleuritic chest pain, and chest
CT revealed a left-sided pneumothorax (Figure 2). A tube
thoracostomy was placed, but after 1 week she developed a new
pneumothorax on the other side (Figure 3). The patient was managed
with bilateral tube thoracostomy, and both lungs were expanded. Her
high temperature, which was occasionally subfebrile, continued for
2 months in spite of systemic corticotherapy. In the fifth month of
therapy, the tumor markers (CA-125, CA-15.3) returned to normal
levels. The treatment (antituberculosis drugs and corticosteroids)
was continued for 12 months, but the miliary shadows did not
disappear. Her forced vital capacity (FVC) was 55%, her forced
expiratory volume in 1 second (FEV1
) was 58%, and her
/FVC was 93% in pulmonary function tests. Diffusion
capacity was 65%, but the patient was not a candidate for an
invasive diagnostic procedure at this time.
Miliary pattern and pneumothorax are rare radiological features in
pulmonary tuberculosis. Their incidences are nearly 1.3% and 1.5%,
However, the coincidence of pneumothorax
on miliary tuberculosis is a very rare event. We found only 9
well-documented cases in the English literature.2-7
age distribution of these patients showed adult predominance (Table
1), which supported the findings of other studies.8,9
Table 1 also illustrates that the male/female ratio was 4:6.
However, this rate was 3:1 in all causes of secondary spontaneous
The pathogenesis of pneumothorax in miliary tuberculosis is
unclear, but the following mechanisms can be considered: caseation
or necrosis of subpleural miliary nodules and their subsequent
rupture can cause pneumothorax. On the other hand, acute miliary
dissemination may lead to emphysematous changes. This mechanism may
explain the bilateral, simultaneous, and/or recurrent
pneumothoraces.2,4 Despite this knowledge, it is not
clear why a pneumothorax complicating miliary tuberculosis is
It has been noted that surgical pleurectomy should be attempted
early in simultaneous bilateral secondary spontaneous
pneumothorax.5 In miliary tuberculosis, open thoracotomy
should not be considered until the patient has received
antituberculous therapy for at least several weeks. The initial
treatment for nearly every patient with a secondary spontaneous
pneumothorax should be tube thoracostomy.
In this patient, at the end of the twelfth month of
antituberculous therapy with corticotherapy, the miliary pattern
was unchanged and her diffusion capacity was reduced, despite the
presence of good performance status. These findings clearly
suggested an interstitial lung disease, such as histiocytosis X or
Langerhans cell his-tiocytosis. Pulmonary histiocytosis X is an
uncommon, smoking-related, interstitial lung disease that primarily
affects young adults. Spontaneous pneumothorax, which may be
recurrent, is a recognized feature of this disease and likely
results from the destruction of lung parenchyma with associated
cysts or nodules (with or without cavitation). Prior to a biopsy
procedure, a high-resolution chest CT can be helpful in the
diagnostic evaluation. The natural history is variable, with some
patients experiencing spontaneous remission of symptoms and others
progressing to end-stage fibrotic lung disease. There is no clear
benefit for either corticosteroids or cytotoxic
The possibility of a histiocytosis X should be considered in a
young adult with a miliary pattern that is unresponsive to a long
duration of antituberculosis drugs and corticosteroids,
particularly in the absence of neurologic findings. Tuberculosis
can accompany an interstitial lung disease that is characterized by
a miliary pattern. In such a condition, fever may not respond to
corticosteroid therapy. Finally, tumor markers may be used in the
follow-up of tuberculosis.
- Aktogu S, Yorgancioglu A, Cirak K, et al. Clinical spectrum of
pulmonary and pleural tuberculosis: a report of 5,480 cases.Eur
Respir J.1996;9:2031-2035. Comment in:Eur Respir J.
- Peiken AS, Lamberta F, Seriff NS. Bilateral recurrent
pneumothoraces: A rare complication of miliary tuberculosis. Am Rev
- Narang RK, Kumar S, Gupta A. Pneumothorax and pneumomediastinum
complicating acute miliary tuberculosis.
- Chandra KS, Prasad AS, Prasad CE, et al. Recurrent
pneumothoraces in miliary tuberculosis. Trop Geogr Med.
- Graf-Deuel E, Knoblauch A. Simultaneous bilateral spontaneous
pneumothorax. Chest. 1994;105:1142-1146.
- Wammanda RD, Ameh EA, Ali FU. Bilateral pneumothorax
complicating miliary tuberculosis in children: Case report and
review of the literature. Ann Trop Paediatr. 2003;23:149-152.
- Mert A, Bilir M, Akman C, et al. Spontaneous pneumothorax: A
rare complication of miliary tuberculosis. Ann Thorac Cardiovasc
- Kim JH, Langston AA, Gallis HA. Miliary tuberculosis:
Epidemiology, clinical manifestations, diagnosis, and outcome. Rev
Infect Dis.1990; 12:583-590. Comment in: Rev Infect Dis.
- Maartens G, Willcox PA, Benatar SR. Miliary tuberculosis: Rapid
diagnosis, hematologic abnormalities, and outcome in 109 treated
adults. Am J Med. 1990;89:291-296.
- Melton LJ 3rd, Hepper NG, Offord KP. Incidence of spontaneous
pneumothorax in Olmsted County, Minnesota: 1950 to 1974. Am Rev
Respir Dis. 1979;120:1379-1382.
- Mendez JL, Nadrous HF, Vassallo R, et al. Pneumothorax in
pulmonary Langerhans cell histiocytosis. Chest.
- Vassallo R, Ryu JH, Schroeder DR, et al. Clinical outcomes of
pulmonary Langerhans'-cell histiocytosis in adults. N Engl J Med.