A case of adenocarcinoma presenting with cystic lesion and recurrent pneumothoraces

Abstract

Background

In this paper, a rare case is reported, where the patient is a 74-year-old man. He suffered from recurrent pneumothorax within half a year and experienced a relapse after receiving conservative treatments.

Case presentation

Diagnostic workup revealed a cystic lesion in the right middle lobe, which has been interpreted as a bulla during the initial chest CT scan. Due to recurrent pneumothorax and poor response to the conservative treatments, the patient underwent bullectomy and pleurodesis. The pathology showed that the wall of the cystic lesion was invasive adenocarcinoma.

Conclusions

This case highlights the importance of monitoring cystic lesions in the lungs, especially in patients with a history of smoking and emphysema.

Background

Thin-walled cystic lung cancer refers to lung cancer with cystic lumen originating from the cystic wall, which lacks corresponding clinical symptoms and auxiliary examination manifestations, and is difficult to diagnose in early stage, easy to be misdiagnosed and missed, and unfavourable to early treatment. Some studies have found that thin-walled cystic-cavity lung adenocarcinomas develop with intra- and extra-cavitary nodules and changes in the thickness of the cystic cavity wall [1,2,3,4,5]. In this paper, a case of 74-year-old patient with thin-walled cystic lung adenocarcinoma discovered by accident is presented.

Case presentation

The patient is a 74-year-old man, who visited the Affiliated Jinhua Hospital, Zhejiang University School of Medicine for treatment on December, 2023 due to Repeated coughing and wheezing for 10 years, which became aggravated in the last 2 days. The patient has a history of hypertension for more than 5 years and takes nifedipine extended-release tablets (20 mg po qd) to control his blood pressure. He has a history of smoking and had been consuming 1 pack/day for more than 30 years, and quit smoking 2 years ago. He also has a history of drinking and has been drinking alcohol about 50%, 50 ml/day for 30 years.

The patient was diagnosed with chronic bronchitis with emphysema 10 years ago, the exact treatment was unknown, and the symptoms were recurrent. The patient visited the Emergency Department of this hospital 6 months ago (on June, 2023) due to pain in the right side of the chest for 1 day. An chest CT scan suggested chronic bronchitis in both lungs and emphysema accompanied by a bulla (Fig. 1), and the patient was given an anti-infective therapy, he received medical treatment however was not responsive.

Fig. 1

Chest CT (First CT, showing emphysema with cystic lesions)

Three months ago (September, 2023), the patient was readmitted to the hospital due to cough accompanied by chest tightness for 4 days. A chest CT scan suggested hydropneumothorax in the right side (with a compression ratio of approximately 20%), chronic bronchitis in both lungs, and emphysema accompanied by a bulla (Fig. 2), so the patient was given oxygen, anti-infection treatment and other symptomatic and supportive treatments. After the treatments, a chest CT scan taken on October, 2023 suggested reduction of hydropneumothorax in the right side (with a compression ratio of less than 10%), as well as chronic bronchitis in both lungs and emphysema accompanied by a bulla (Fig. 3). In view of the mildness and stability of pneumothorax, the patient was discharged for improvement and advised to have follow-up visits.

Fig. 2

Chest CT (2nd CT, showing emphysema with cystic lesions with 20% pneumothorax on the right side)

Fig. 3

chest CT (3rd CT, showing emphysema with cystic lesions with right-sided < 10% pneumothorax)

On December, 2023, the patient was admitted to the hospital due to a recurrence of symptoms. His vital signs included a body temperature of 36.5 °C, a respiratory rate of 18 times/min, a pulse rate of 85 beats/min, a blood pressure of 135/76mmhg, and a blood oxygen saturation of 93% (when breathing air). A chest CT scan suggested increase in hydropneumothorax in the right side (a compression ratio of approximately 20%), chronic bronchitis in both lungs and emphysema accompanied by a bulla (Fig. 4), and the patient was admitted in view of pneumothorax in the right side. The test results of his complete blood count, liver function, renal function and blood tumor markers were all within the normal ranges, and a chest CT scan suggested no significant enlargement of the cystic airspace. Because of the small amount of pneumothorax, he did not undergo pleurodesis during the whole process and was treated conservatively with oxygen, and because of the recurrent pneumothorax, we chose to have an multidisciplinary discussions among doctors from the Imaging Department, Department of Pulmonary and Critical Care Medicine and Department of Cardiothoracic Surgery. After discussions, as well as adequate communications with the patient, the patient’s family members, and thoracic surgeons, the patient received bullectomy in the right lung and pleurodesis on December, 2023 in view of the poor effect of the conservative treatments. General anesthesia with tracheal intubation, intermittent two-lung ventilation and one-lung ventilation during surgery. Three chest drains were placed postoperatively. Intraoperatively, at the end of the procedure, check was carried out to ensure that there is no air leakage from the residual surface of the lungs and that there is no post-operative hemorrhage in the thoracic cavity. The pathological examination result showed that the wall of the cystic lesion in the free pulmonary bulla tissue was invasive acinar and papillary adenocarcinoma. Considering the patient’s inability to tolerate unilateral lung ventilation during surgery and the patient’s poor lung function, the patient was operated with a right middle lung wedge resection; three chest drains were placed postoperatively, and the patient was followed up with a 1-week postoperative follow-up and a repeat chest X-ray. Subsequently, we advised the patient to improve genetic testing and further treatments such as chemotherapy, which the patient declined, then the patient was lost to follow-up. An immunohistochemical analysis showed: CK-P + elastic fiber (+) with no breakthrough (Figs. 5 and 6).

Fig. 4

Chest CT (4th CT, showing emphysema with cystic lesions with 20% pneumothorax on the right side)

Fig. 5

Pathological HE staining

Fig. 6

Pathological CK-P + elastic fiber (no breakthrough)

Discussion

Lung cancer is one of the known causes of spontaneous pneumothorax (SPx). Spontaneous pneumothorax (SPx) can occur in primary or metastatic lesions; metastatic diseases such as sarcomas are more likely to lead to spontaneous pneumothorax than primary lung cancer [1]. Spontaneous pneumothorax as the first symptom of primary lung cancer SPx has been widely reported as the first symptom of primary lung cancer. In non-small cell lung cancer and small cell lung cancer, SPx has been widely reported as the first symptom of primary lung cancer [2,3,4,5]. 1.8% of patients with spontaneous pneumothorax were found to have lung cancer in one study [6], and pneumothorax was found in 0.32% of patients with primary lung cancer in another study [7]. However, there are not many reports of lung cancer found in the manifestation of cystic changes with recurrent pneumothorax.

At the beginning of the pneumothorax in our patient, we just thought that the patient had a pneumothorax caused by chronic bronchitis with emphysema, and because the pneumothorax was not very large, we started to treat it conservatively and did not notice the changes in the patient’s cystic cavity, and with the occurrence of the patient’s pneumothorax for the second time, we still did not show more attention to this cystic cavity because after conservative treatment, the result was not good, and to the cardiothoracic surgical treatment was done for the treatment of pneumothorax, and his pulmonary adenocarcinoma was found incidentally at the time of surgery. So clinically, we need to pay attention to such patients and changes in their cystic cavity type of presentation.

Cystic lung cancer is a special type of lung cancer that mainly involves a thin-walled cystic airspace or a thin-walled cystic airspace surrounding a solid lesion in terms of imaging. Very little data on the incidence of lung cancer related to cystic diseases are available. According to a report, such cases merely account for approximately 1-3.7% of non-small cell lung cancer cases [8]. Reports on cystic lung cancer have been increasing, most of which were related to adenocarcinoma; however, other types of cancer have also been reported [9,10,11]. Pneumothorax is a rare manifestation of primary lung cancer³. The incidence of pneumothorax in patients with primary lung cancer is approximately 0.32% [8]. However, pneumothorax may be an initial symptom of lung cancer, and the causes of pneumothorax and cyst formation in primary lung cancer are still unclear. According to literature, avascular necrosis, obstructive bronchiectasis, and air containing new biological spaces can be possible mechanisms of the formation of cystic lesions [7, 9, 11]. Some studies also showed that the one-way valve mechanism leads to cystic dilatation and air trapping in the distal bronchus, subsequently causing increased tension in a cyst and its rupture, which may be one of the causes of the complications of pneumothorax in cystic lung cancer [8, 12, 13].

There are limited data on the incidence of lung cancer related to cystic diseases [9]. In previous lung cancer screenings, malignant tumors related to cystic lesions were considered one of the reasons for missed diagnoses of lung cancer [10]. In some studies, cystic lesions in the lungs were focused on [8], and a classification system based on morphology was established, which includes 4 types: in Type I, a nodule is located outside the cystic airspace; in Type II is, a nodule originates from the cyst wall and grows into the cystic airspace; in Type III, the cyst wall shows circular thickening; in Type IV, there are multilocular cystic changes with solid or non-solid components. However, the above classification is merely CT-based morphological classification and cannot provide prognostic information.

Conclusion

In this paper, we described a case of cystic adenocarcinoma presenting with recurrent pneumothorax, which may be the first manifestation of lung cancer. Therefore, this case highlights the necessity and importance of a broad differential diagnosis. Timely examination and appropriate follow-up visits are critical to avoiding delays in cancer diagnosis and treatment, and it is necessary to recognize that pneumothorax may be an early complication of primary lung cancer.

We should also notice that in patients with cystic lesions combined with pneumothorax, we need to be vigilant for the presence of lung cancer.