301 cases of pulmonary hamartoma confirmed by pathology in The First Affiliated Hospital of Soochow University from July 2000 to October 2019 were retrospectively reviewed, including 13(13/301, 4%) cases of endobronchial hamartoma. The patients included 11 males and 2 females, aged 48-74 years, with an average age of(63±9) years. All 13 cases of EH were single lesion.
Among the 13 patients, eight patients were found incidentally during health examination or examination for another reasons, including CT and bronchoscopy. Three of the 13 patients with EH are asymptomatic. For the remaining 10 patients had atypical symptoms: cough in 6 cases, chest tightness in 4 cases, chest pain in 1 case, fever in 2 cases and hemoptysis in 2 cases.
There were 3 patients having underlying respiratory diseases: one had chronic obstructive pulmonary disease(COPD), one had allergic alveoli, and one had interstitial lung disease and died of respiratory failure during hospitalization finally. Other basic diseases include hypertension in 5 cases, diabetes in 2 cases and renal carcinoma in 1 case. Six patients had a history of smoking and one had a history of exposing to dusts.
The location of EH
The location of these tumors are as follows: one was found in the right upper lobe bronchus, one in the right middle lobe bronchus, three in the right lower lobe bronchus, one in the right intermediate bronchus, three in the left upper lobe bronchus, two in the left lower lobe bronchus and two in the left main bronchus.
The following image features of 13 cases are obtained from image and image report records.
Chest radiograph findings were abnormal in 3 of the 11 patients(excepting the 2 patients with allergic alveolitis and Interstitial lung disease), main performance is obstructive pneumonia.
Chest CT showed obstructive pneumonia in 10 of the 13 patients. However, in our study, many cases may be poorly demonstrated or may not be seen at all on chest CT. After our careful review of CT, among 13 patients with EH, visible endobronchial tumor were present in 7 patients, retrospectively. Among these 7 cases, however, before the diagnosis of EH, only 3 cases were diagnosed with endobronchial tumor by radiologists. In addition, two case was misdiagnosed as lung cancer according to CT.
Among 7 visible endobronchial tumors on CT, areas of focal fat were observed in 4 lesions. Calcification can be seen in 3 lesions of all 13 cases. Contrast-enhanced CT was performed in 3 cases, which showed no enhancement in 1 case, slight enhancement in 1 case, obvious heterogeneous enhancement in 1 case. (Figure 1)
All cases received bronchoscopy. We recorded 10 cases of bronchoscopy images, as follows.
Bronchoscopy showed mass with visible stalk during bronchoscopy in 5 of 10 cases. But we believe that mass with stalk is much more than 5 cases, because some EH obstruct the airway severely, we may not be able to see the stalk of EH of the posterior airway after the obstructed site. Seven cases showed Smooth or regular surface under bronchoscopy, three cases showed irregular surface. For mucosa characteristics: Surface with a lot of necrosis tissues in 1 case, pale mucosa in 2 cases, increased redness mucosa in 2 cases and similar to normal mucosa were present in 5 patients, of which 1 case rich in blood vessels.(Figure 2)
Seven patients were treated with bronchoscopic techniques, all of them were treated via flexible bronchoscopy.
Three patients received bronchoscopic treatments more than once: One case(N1) failed in the first interventional treatment with snare via flexible bronchoscope due to difficulty in exposing the stalk of the mass, and then switched to argon plasma coagulation(APC) therapy. Residual tumors was found in the second bronchoscopy, APC was performed the second time. Four months later, bronchoscopy showed that the EH was completely removed, but hyperemia of the mucosa. CT examination showed no recurrence 16 months later.
One case(N2)’s EH was removed partially with snare via flexible bronchoscopy the first time. So a second intervention, rigid bronchoscopy was performed initially, but we found it was difficult to insert rigid bronchoscopy due to patient’s narrow oropharynx. Then we switched to flexible bronchoscopy via laryngeal mask airway(LMA). Using APC combined with cryotherapy, the mass was removed completely, taken out by wire basket.
One case(N3) received bronchoscopic intervention 4 times. The mass was cauterized with APC the first time, APC and cryotherapy were used at the second time due to recurrence, and cryotherapy was used the third time. Scar stenosis was found during the fourth bronchoscopy, then dilated by endoscopic biopsy forceps.
For the remaining 4 patients, both of them were treated with snare electrocautery via flexible bronchoscopy, and then mass root was treated with APC or cryotherapy, or combination of the two.
There were no major complications in these 7 cases, except a little bleeding in some patients and one patient who developed slight pneumomediastinum and pneumothorax, successfully recovered with simple oxygen therapy.
Six patients received pulmonary lobectomy via thoracotomy. All patients who underwent surgery received bronchoscope biopsy and brush biopsy in the initial bronchoscopy. However, no definite diagnosis of EH was given.
One of them appeared intolerance and sharp increased blood pressure during bronchoscopy, though under adequate local anesthesia with lidocaine. In addition, bronchoscopy showed the mass with a lot of necrotic, which was highly suspected of malignancy. So we gave up the interventional treatment through bronchoscope, and switched to surgery.
For the remaining 5 patients, CT scan showed moderate or severe irreversible pulmonary damage which require pulmonary lobectomy or enlargement of hilar or mediastinal lymph node, which cannot rule out malignancy lymphatic metastasis. The enlargement of hilar and mediastinal lymph node finally confirmed as chronic inflammation by surgical pathology.
Chylothorax occurred in 1 case, and hydrothorax became clear and significantly reduced after 5 days of drainage. No other complications were observed.
The clinical characteristics of 13 cases of endobronchial hamartoma are summarized in Table 1.
Follow-up CT scan was performed in 7 of 13 patients(mean 47 months, range 17-69 months), which revealed no recurrence in all 7 patients. Unfortunately, follow-up bronchoscopy was not performed except two cases(N1, N3) mentioned above, which spanned a longer time period for bronchoscopy(4 months and 5 months in total ).