Journal of Surgical Case Reports, 2024, 5, rjae365

[Link]
Case Report

Case Report
Pulmonary tuberculoma-induced cyst formation leading
to repeated pneumothorax: a case report
Takaaki Suwa1 , Nobutaka Kawamoto 1,
*, Shunsuke Morita1 , Hiroshi Hasegawa2 , Junichi Zaitsu3 , Keizo Misumi1

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1 Department of Thoracic Surgery, JA Hiroshima General Hospital, 1-3-3 Jigozen, Hatsukaichi, Hiroshima 738-8503, Japan
2 Department of Respiratory Medicine, JA Hiroshima General Hospital, 1-3-3 Jigozen, Hatsukaichi, Hiroshima 738-8503, Japan
3 Department of Pathology, JA Hiroshima General Hospital, 1-3-3 Jigozen, Hatsukaichi, Hiroshima 738-8503, Japan

*Corresponding author. Department of Thoracic Surgery, JA Hiroshima General Hospital, 1-3-3 Jigozen, Hatsukaichi, Hiroshima 738-8503, Japan.
E-mail: kawamotonobutaka@[Link]

Abstract
Most cases of secondary spontaneous pneumothorax in patients with active pulmonary tuberculosis are caused by rupturing of the
visceral pleura caused by Mycobacterium tuberculosis. The check-valve airway mechanism in the lungs is generally involved in the
formation of pulmonary cysts, which often cause spontaneous pneumothorax. Herein, we describe a rare case of repeated spontaneous
pneumothorax suspected to have been caused by pulmonary cyst formation as a result of a tuberculoma. The patient was a man with a
family history of pulmonary tuberculosis. Pulmonary cysts were gradually enlarged on the peripheral side of a lung mass in the upper
lobe of the patient’s right lung, who experienced two spontaneous pneumothoraxes in the area. Exploratory surgery was performed to
diagnose the lung mass and treat the pneumothorax, resulting in a final diagnosis of pulmonary tuberculoma. A check-valve mechanism
caused by the pulmonary tuberculoma was suspected based on the patient’s clinical course.

Keywords: check-valve mechanism; pneumothorax; pulmonary cyst; pulmonary tuberculoma

considered to have formed through a check-valve mechanism

Introduction caused by pulmonary tuberculoma. To our knowledge, this is the
Spontaneous pneumothorax can be classified into primary and first report of pulmonary tuberculoma resulting in pulmonary
secondary types [1]. Secondary spontaneous pneumothorax is cyst formation.
considered a complication of an underlying pulmonary disease
such as chronic obstructive pulmonary disease, cystic lung dis-
ease, malignancy, pulmonary infections, or interstitial lung dis-
Case report
eases [2]. Secondary spontaneous pneumothorax develops in 1%– The patient, a 75-year-old man, had a history of type 2 dia-
2% of patients with active pulmonary tuberculosis [3], and most betes mellitus, dyslipidaemia, hypertension, hyperuricaemia, and
cases are caused by rupture of the visceral pleura as a result of alcohol-related chronic pancreatitis and was obese (body mass
Mycobacterium tuberculosis infection [4]. index, 35.1 kg/m2 ). He had quit smoking at 60 years of age,
The check-valve mechanism of the airway is key to the for- with a 60-pack-year smoking history. He had a family history of
mation of pulmonary cysts, which in turn lead to spontaneous pulmonary tuberculosis on his father’s side.
pneumothorax, wherein air becomes progressively trapped in During the computed tomography (CT) scan of the 62-year-old
alveoli that are distal to the obstruction [5]. Most cases of pul- patient, physicians did not point out infiltrating shadows in the
monary cyst formation associated with lung masses are later right upper lobe caused by suspected pulmonary tuberculosis.
diagnosed as cases of lung cancer [6]. In rare cases, obstructive Therefore, a chest CT scan was not performed until the patient
bronchiolitis caused by an infectious pulmonary disease can give developed a right pneumothorax at age 70. At 70 years old,
rise to a check-valve phenomenon that results in the forma- the patient developed pneumothorax in his right lung; a CT
tion of pulmonary cysts on the peripheral side of the affected scan revealed a 3.2 cm mass with satellite lesions in the right
lung [7]. upper lobe, as well as pulmonary cysts on the peripheral side
Herein, we report a surgical case of repeated secondary of the mass (Fig. 1). At this point, a retrospective review of the
spontaneous pneumothorax, wherein pulmonary cysts were patient’s earlier CT images (at age 62 years) confirmed that there

Received: April 22, 2024. Accepted: May 13, 2024

Published by Oxford University Press and JSCR Publishing Ltd. © The Author(s) 2024.
This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ([Link]
licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For
commercial re-use, please contact [Link]@[Link]
2 | Suwa et al.

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Figure 1. CT imaging findings; at age 62, shadows suggesting pulmonary infiltration were present in the upper lobe of the patient’s right lung, but no
pulmonary cysts were observed; at age 70, the patient developed spontaneous pneumothorax in his right lung; another scan revealed a 3.2 cm lung
mass with satellite lesions in the upper lobe of the right lung, as well as pulmonary cysts on the peripheral side of the mass; at age 75, the patient
experienced a relapse of the spontaneous pneumothorax in the same lung.

Figure 2. CT images of the intervening period between the initial and relapsed pneumothorax; no enlargement of the lung mass was observed, but
gradual enlargement of the pulmonary cysts was apparent.

were some shadows indicative of pulmonary infiltration in the pulmonary cysts were excised (Fig. 3A and B). The lung mass
upper lobe of the right lung, although pulmonary cysts were not was diagnosed as a granuloma with necrosis through analysis of
visible. The pulmonary cysts were considered to have caused frozen surgical biopsy sections, and a polymerase chain reaction
the patient’s pneumothorax, which resolved following chest (PCR) test for tuberculosis was positive. The operative time was
drainage. A bronchoscopy was performed after the pneumothorax 134 min, and the estimated blood loss was 15 ml. No postoperative
improved, with brushing cytology and bronchoalveolar lavage air leakage was observed, and the patient’s chest drain tube was
cytology revealing no malignant cells. An acid-fast bacilli culture removed on postoperative Day 2. He was discharged on postopera-
of the patient’s bronchoalveolar lavage f luid and the T-cell spot tive Day 12, following respiratory rehabilitation due to his obesity.
test for tuberculosis infection ([Link]) were both negative. The timeline of the patient’s clinical course is presented in Fig. 4.
An 18 F-f luorodeoxyglucose positron emission tomography/CT Macroscopic findings revealed bronchioles surrounded by a
scan showed a maximum standard uptake value (SUVmax) of pale-yellow lung mass, with pulmonary cysts on the peripheral
1.8 in the lung mass. Since the lung mass was not suspected to side of the mass (Fig. 5A). Histopathological findings indicated
be aggressively malignant [8], careful follow-up was performed, that the mass was a caseous granuloma with multinucleated
involving another CT scan. The lung mass was not enlarged; Langhans giant cells and lymphocyte accumulation (Fig. 5B). This
however, some growth of the pulmonary cysts was observed was diagnosed as a pulmonary tuberculoma, based on the PCR
(Fig. 2). results. Lymphocyte infiltration and interstitial tissue fibrosis
At 75 years old, the patient’s right spontaneous pneumothorax associated with chronic inf lammation were observed around the
relapsed (Fig. 1). Exploratory surgery was performed to diagnose bronchioles (Fig. 5C). Neither bronchial obstruction nor evidence
the lung mass and treat the pneumothorax. The lung mass and of malignancy was observed.
 Pulmonary tuberculoma-induced cyst formation | 3

Figure 3. Surgical findings; (A) pulmonary cysts were observed in the upper lobe of the patient’s right lung; (B) the lung mass and pulmonary cysts
were removed using a surgical stapler; RUL, right upper lobe.

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Figure 4. Timeline of the patient’s clinical course; CT, computed tomography.

One month postoperatively, the patient was started on isoni- satellite lesions are observed, even if bronchoscopy or [Link]
azid, rifampicin, and ethambutol. No pneumothorax recurrence results are negative.
was observed at a 9-month postoperative follow-up. Most cases of pulmonary cyst formation associated with lung
masses result from lung cancer [6], with infectious pulmonary dis-
eases representing an exceedingly rare cause [7]. In our case, there
were initially no pulmonary cysts in the upper lobe of the patient’s
Discussion right lung. When a pulmonary tuberculoma was identified via CT,
Tuberculosis is a global health concern. Although the number pulmonary cysts on the peripheral side of the mass were also
of patients with tuberculosis in Japan is decreasing, 8.2 persons observed for the first time. These cysts then enlarged over time,
per 100 000 population are expected to develop it in 2022 [9]. T- suggesting that they were caused by a check-valve mechanism in
[Link] is used to detect latent tuberculosis infection, with a sen- the bronchioles that were surrounded by the pulmonary tubercu-
sitivity of 81%–88% [10, 11]. Therefore, some false-negative results loma. Surgical treatment to prevent pneumothorax and diagnose
do occur. Pulmonary tuberculoma occurs in ∼9% of patients with the lung mass may be advisable in cases of enlarged pulmonary
pulmonary tuberculosis [12]. The CT findings of pulmonary tuber- cysts associated with lung masses.
culoma often show a lung mass with satellite lesions [13]. How- A major limitation of this case is the difficulty of conclusively
ever, differentiating pulmonary tuberculoma from lung cancer demonstrating the suspected check-valve mechanism behind the
via clinical and imaging findings is challenging [14]. Pulmonary patient’s cysts, based on histopathological and CT findings alone.
tuberculoma should be considered whenever lung masses with Reviews of similar case reports may help to clarify this hypothesis.
4 | Suwa et al.

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Figure 5. Macroscopic and histopathological findings; (A) a pale-yellow lung mass surrounding the bronchioles (dotted lines) and pulmonary cysts
(arrows) on the peripheral side of the mass were observed; (B) a caseous granuloma with multinucleated Langhans giant cells (arrow) and
accumulation of lymphocytes was observed; this was diagnosed as a pulmonary tuberculoma, after considering the result of a PCR test;
(C) lymphocyte infiltration and fibrosis of the interstitial tissue associated with chronic inf lammation were observed around the bronchioles (arrows);
neither bronchial obstruction caused by the lung mass nor emphysematous changes in the background lung were observed.

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Conflict of interest statement [Link]
6. Snoeckx A, Reyntiens P, Carp L, et al. Diagnostic and clinical
None.
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