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Respir Med Case Rep
Respir Med Case Rep
Respiratory Medicine Case Reports
2213-0071
Elsevier

S2213-0071(24)00116-3
10.1016/j.rmcr.2024.102093
102093
Case Report
Unilateral acute respiratory distress syndrome appearing contralateral to lung cancer
Sokai Akihiko sokai.med@gmail.com
a⁎
Iwata Toshiyuki a
Ikeo Satoshi a
Hayashi Yasuyuki a
Sakai Yuki a
Yasuda Naoaki a
Tasato Miki a
Shibuya Shinsuke b
Nishimura Takashi a
a Department of Respiratory Medicine, Kyoto Katsura Hospital, 17 Yamadahirao-cho, Nishikyo-ku, Kyoto, 615-8256, Japan
b Department of Pathology, Kyoto Katsura Hospital, 17 Yamadahirao-cho, Nishikyo-ku, Kyoto, 615-8256, Japan
⁎ Corresponding author. sokai.med@gmail.com
18 8 2024
2024
18 8 2024
51 10209330 5 2024
8 8 2024
17 8 2024
© 2024 The Authors
2024
https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
An 83-year-old woman was suspected to have lung cancer in the right lung. However, diffuse opacities only appeared in the left lung, and she was urgently hospitalized due to severe respiratory failure. The opacities in the left lung deteriorated, and she died despite treatments including antibiotics and steroids. An autopsy revealed pleomorphic carcinoma in the right upper lobe, stenosis of the right pulmonary artery due to compression of the right hilar lymph nodes, and diffuse alveolar damage (DAD) throughout the left lobes. Acute respiratory distress syndrome (ARDS), of which a histological feature is DAD, consists of bilateral opacities in the lungs according to the definition. Unilateral ARDS is extremely rare and has reportedly developed in patients with unilateral pulmonary artery agenesis. The unilateral absence of pulmonary perfusion might be involved in the pathogenesis of unilateral ARDS. In patients with lung cancer, compression of the pulmonary artery may result in unilateral ARDS.

Keywords

Acute respiratory distress syndrome
Lung cancer
Pulmonary artery
Unilateral
Abbreviations

ARDS acute respiratory distress syndrome

DAD diffuse alveolar damage
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pmc1 Introduction

Acute respiratory distress syndrome (ARDS) consists of bilateral opacities in the lungs according to the definition [1]; therefore, unilateral ARDS is an extremely rare clinical entity. In most cases, ARDS cases appearing to be unilateral actually have bilateral opacities on computed tomography or develop into bilateral later [2]. Unilateral pulmonary artery agenesis has been reported to develop into unilateral ARDS [[3], [4], [5]], while other unilateral ARDS cases have rarely been reported.

2 Case presentation

An 83-year-old woman who had no particular medical history was referred to our hospital for a mass in the right upper lung field (Fig. 1A). She had a one-month history of fatigue, appetite loss, and body weight loss. She also had dyspnea and wheezing for several days, while her oxygen saturation was 95 % on room air. Computed tomography revealed a tumor in the right upper lobe and enlarged right hilar and mediastinal lymph nodes compressing the right bronchus; moreover, slight ground-glass opacities were detected in the left lower lobe (Fig. 1B and C). Examinations for diagnosis and staging were scheduled.Fig. 1 (A) A chest X-ray taken at the first visit shows a mass in the right upper field. (B and C) Computed tomography reveals a mass in the right upper lobe, enlarged mediastinal lymph nodes compressing the right bronchus (yellow arrowhead), and slight ground glass opacities in the left lower lobe (white arrowheads). (D) A chest X-ray taken in the emergency room the day after the first visit shows deteriorating infiltrates only in the left field. (E) A chest X-ray taken four days after the first visit shows more deteriorating infiltrates only in the left field than did a chest X-ray on admission. (For interpretation of the references to colour in this figure legend, the reader is referred to the Web version of this article.)

Fig. 1

On the day following the first visit, however, the patient presented to the emergency department with hypercapnic respiratory failure. The patient had a breathing effort, and her oxygen saturation was 87 %, with 10 L per minute of oxygen provided by a face mask. Her arterial carbon dioxide pressure was 72.2 mmHg. Pulmonary examination revealed rhonchi in the right lower lobe and crackles in the left lung field. A chest X-ray image showed diffuse opacities in the left lung (Fig. 1D). Examinations, including echocardiography, revealed no evidence of heart failure or pulmonary embolism. Noninvasive positive pressure ventilation and multiple antibiotics were immediately introduced. Betamethasone was also administered to improve airway constriction. However, even with those treatments, the patient's respiratory condition continued to deteriorate, as indicated by the persistent opacities observed in the left lung (Fig. 1E). The patient passed away five days after admission, and an autopsy was performed.

The autopsy revealed pleomorphic carcinoma, including adenocarcinoma and spindle cell carcinoma, in the right upper lobe (Fig. 2A). Furthermore, pulmonary edema, hyaline membranes, and infiltrating leukocytes were present throughout the opposite left lobes, which indicated diffuse alveolar damage (DAD) and bronchopneumonia (Fig. 2B and C). No histopathological findings suggesting ARDS were detected in the right lobes. The autopsy also revealed no findings suggesting tumorous invasion or thrombus of the pulmonary artery, but stenosis of the right pulmonary artery caused by compression of right hilar lymph nodes was observed (Fig. 2D).Fig. 2 (A) Pathological analysis of the tumor reveals pleomorphic carcinoma, which consisted of adenocarcinoma and spindle cell carcinoma (H&E staining, x400). (B and C) Pathological analysis of the left lung reveals pulmonary edema, hyaline membranes, and infiltrating leukocytes throughout the left lobes (H&E staining, ×20 (B) and ×200 (C)). (D) Macroscopic findings reveal stenosis (red arrowheads) of the right pulmonary artery (PA) compressed by enlarged right hilar and mediastinal lymph nodes (LNs). (For interpretation of the references to colour in this figure legend, the reader is referred to the Web version of this article.)

Fig. 2

3 Discussion

ARDS consists of bilateral opacities in the lungs according to the definition [1]; therefore, unilateral ARDS is an extremely rare clinical entity. In the present case, stenosis of the right pulmonary artery was not detected on unenhanced computed tomography, but the macroscopic findings revealed that the enlarged right hilar and mediastinal lymph nodes compressed the right bronchus and the right pulmonary artery. DAD, which is a histological feature of ARDS, was histopathologically confirmed to be only unilateral, which derived the possibility that the unilateral absence or decrease of lung perfusion might induce contralateral unilateral ARDS.

ARDS is a well-recognized condition of acute-onset noncardiogenic hypoxemia with a preceding underlying disease or trauma and is characterized pathologically by DAD. In patients with lung cancer, acute hypoxemia, including ARDS and drug-induced interstitial lung disease, often results from interventions such as pulmonary resection and chemotherapy. Similar to the present case, reports of ARDS in patients with untreated lung cancer are limited. An ARDS patient with untreated lung cancer was also reported to have pleomorphic carcinoma [6]. One possible pathogenesis of ARDS is the release of mediators such as cytokines; therefore, pleomorphic carcinoma, which releases a variety of cytokines, may be likely to induce ARDS.

The radiological features of ARDS include bilateral infiltrates, even if they are asymmetrical. However, patients with ARDS sometimes exhibit unilateral infiltrates. Several factors that could warrant the assessment of unilateral ARDS are considered [2]. For example, chest X-ray images show that infiltrates, which are bilateral on computed tomography, appear unilateral. Some patients with unilateral infiltrates experience the progression of ARDS with bilateral infiltrates. Evaluation with portable chest radiographs, which are poor in quality, is undependable. The quality of the device for the assessment and the temporal heterogeneity may cause bilateral ARDS to be underestimated as unilateral ARDS. Therefore, there are only a few cases of true unilateral ARDS, and reports of unilateral ARDS are limited. Unilateral pulmonary opacities are sometimes caused by the treatment of lung cancer, whereas unilateral ARDS that is not related to treatment has not been reported in patients with lung cancer. In the present case, computed tomography could not be performed at admission due to severe respiratory failure, while the diagnosis of unilateral ARDS could be made pathologically.

Several cases have been reported that could be considered true unilateral ARDS rather than underestimated unilateral ARDS; these cases suggest a potential cause of unilateral ARDS. Unilateral pulmonary artery agenesis is a rare vascular malformation in which unilateral ARDS develops due to the absence of perfusion in the contralateral lung [[3], [4], [5]]. In the present case, there were no histological findings suggesting tumorous invasion of the pulmonary artery or arterial obstruction, and unenhanced computed tomography did not reveal obvious compression of the right pulmonary artery. However, macroscopic findings revealed stenosis of the right pulmonary artery, and it was possible that there was impaired perfusion of the compressed right pulmonary artery as well as the right bronchus compressed by the enlarged right hilar and mediastinal lymph nodes. External compression might be followed by the unilateral absence of lung perfusion and unilateral ARDS. A study evaluating the difference between ARDS and unilateral infiltrate in patients with severe acute respiratory failure requiring management with mechanical ventilation found that the extent of the spread of infiltrates had a greater impact on prognosis than ARDS or unilateral infiltrates, and there was no fundamental difference in management between patients with ARDS and those with unilateral infiltrates [7]. However, most patients with unilateral infiltrates are likely to have normal pulmonary perfusion in the contralateral lung, whereas contralateral pulmonary perfusion would likely be deficient in patients with true unilateral ARDS. Thus, the pathogenesis of these two conditions might be quite different. Whether unilateral ARDS has a different prognosis from bilateral ARDS and whether there are differences in the management of both conditions, including mechanical ventilation, is still unknown.

Cardiogenic pulmonary edema caused mainly by cardiac dysfunction and fluid overload in the lungs also usually causes bilateral opacities, whereas asymmetric or unilateral pulmonary edema has also been reported. In several cases, the patients developed unilateral pulmonary edema with the contralateral absence of pulmonary perfusion [8,9]. Thus, the balance of pulmonary perfusion is considered related to the distribution of ARDS and pulmonary edema. Unilateral ARDS or pulmonary edema should be suspected in patients with lung cancer when contralateral infiltrates appear and computed tomography shows a compressed pulmonary artery.

The unilateral absence or decrease of pulmonary perfusion might be involved in the pathogenesis of unilateral ARDS. In the present case, the diagnosis of unilateral ARDS was made based on the pathological finding of unilateral DAD; a decrease in unilateral pulmonary perfusion was suspected but not confirmed. Further studies on the relationship between pulmonary perfusion and unilateral ARDS that does not involve unilateral pulmonary artery agenesis are needed. It should be recognized in patients with lung cancer, in which compression of the pulmonary artery may result in unilateral ARDS and lead to death in a few days.

Funding

None.

Informed consent

Written consent for publication was obtained from the patient's family.

CRediT authorship contribution statement

Akihiko Sokai: Writing – review & editing, Writing – original draft, Visualization. Toshiyuki Iwata: Writing – review & editing. Satoshi Ikeo: Writing – review & editing. Yasuyuki Hayashi: Writing – review & editing. Yuki Sakai: Writing – review & editing. Naoaki Yasuda: Writing – review & editing. Miki Tasato: Writing – review & editing. Shinsuke Shibuya: Writing – review & editing, Visualization. Takashi Nishimura: Writing – review & editing.

Declaration of competing interest

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
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