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ORIGINAL ARTICLE
Year : 2016  |  Volume : 12  |  Issue : 5  |  Page : 116-119

Outcomes of surgical treatments of pulmonary hamartoma


Department of Thoracic Surgery, Chinese PLA General Hospital, Beijing 100853, China

Date of Web Publication7-Oct-2016

Correspondence Address:
Tao Wang
Department of Thoracic Surgery, Chinese PLA General Hospital, Beijing 100853
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.191620

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 > Abstract 

Objective: To retrospectively analyze the results of surgical resection in patients with pulmonary hamartoma (PH) in the Peoples Liberation Army (PLA) General Hospital during the past 30 years.
Materials and Methods: We retrospectively enrolled 226 patients with PH who underwent surgical resection in the PLA General Hospital between January 1980 and January 2010, including tumors of pulmonary parenchyma (n = 216) and endobronchial tumors (n = 10). The type of operation include tumor enucleation (n = 103), wedge resection (n = 103), and lobectomy (n = 20). The postoperative follow-up time was 5–22 years.
Results: Only one postoperative death occurred. The intraoperative blood loss and the drainage volume within 24 postoperative hours were significantly less in patients underwent lateral thoracotomy or video-assisted thoracic surgery (VATS) than those of patients underwent conventional incision. None of the patients experienced regional recurrence.
Conclusion: We recommended lateral thoracotomy or VATS for the patients whose PH could not be confirmed preoperatively, and tumor enucleation or wedge resection was recommended.

Keywords: Outcome, pulmonary hamartoma, surgical procedures


How to cite this article:
Wang T, Liu Y. Outcomes of surgical treatments of pulmonary hamartoma. J Can Res Ther 2016;12:116-9

How to cite this URL:
Wang T, Liu Y. Outcomes of surgical treatments of pulmonary hamartoma. J Can Res Ther [serial online] 2016 [cited 2018 Nov 16];12:116-9. Available from: http://www.cancerjournal.net/text.asp?2016/12/5/116/191620


 > Introduction Top


Pulmonary hamartoma (PH), although comparatively rare in clinical practice of thoracic surgery, is the most common pulmonary benign tumor. Although the medical imaging technology has developed rapidly over the past 30 years, surgical resection and pathological examination are still the important methods for PH diagnosis. The operative indication and surgical procedure for PH are still very controversial. In the present study, we analyzed the characteristics and follow-up results of patients with PH who underwent surgical resection between 1980 and 2010 in the Peoples Liberation Army (PLA) General Hospital.


 > Materials and Methods Top


The general information

A total of 226 patients with PH who underwent surgical resection and diagnosed by pathological examination in the Department of Thoracic Surgery of the PLA General Hospital between January 1980 and January 2010 were enrolled. The follow-up time was more than 5 years (5–22 years).

This cohort included 141 males (62.4%) and 85 females (37.6%), and the sex ratio (male/female) was 1.66:1. The average age was 54.3 ± 11.1 (range 19–79), and 213 patients (94.2%) aged between 30 and 70 years old. Eighty patients (35.4%) were admitted with positive symptoms, including varying degrees of cough, sputum, chest pain, bloody sputum, or fever. Among them, 10 patients with intraluminal hamartoma showed different degree of obstructive pneumonia symptoms (1 case suffering from gastric carcinoma situ simultaneously). Another 146 asymptomatic patients were detected by chest X-ray and computed tomography (CT) examination. Sixteen patients were diagnosed with other tumors (3 esophageal cancers, 2 stomach cancers, 1 B type thymoma, and 10 lung cancers), whose PH were detected during preoperative examinations. One hundred and twenty patients had tumors in the left lung, and 106 patients had tumors in the right lung.

Methods

All the patients underwent chest X-ray and CT examination preoperatively. There were 216 tumors of pulmonary parenchyma and 10 endobronchial tumors. The most common image for tumors of pulmonary parenchyma was shallow lobulated, high-density shadow with a clear boundary. Fifty-two patients had calcification (23.0%), only five of them had typical popcorn-like calcification, the rest 47 cases had scattered spot or sheet-like calcification. The endobronchial PH showed the imaging findings of obstructive pulmonary emphysema and obstructive pneumonia. According to the preoperative chest X-ray and CT examination, the primary lung cancer could not be ruled out in 45 patients, and the metastatic lung cancer could not be ruled out in 17 patients with other malignant tumors. The rest of 164 patients were considered to be benign lung tumors, and 22 of them were diagnosed as hamartoma according to fat density. Eight patients (diameter 0.8–3.8 cm) who underwent positron emission tomography (PET) examination without obvious lesions were diagnosed as benign tumors.

The interval time between diagnosis and surgery: 15 patients were 15–172 months, and the tumors had obvious increased in 8 of 15 patients; the remaining 121 patients were 1–12 months without tumor changing. The average maximum diameter of tumor was 2.1 ± 1.3 cm (0.5–12.0 cm). Thirty-two patients had tumors larger than 3 cm, 21 patients had tumors smaller than 1 cm, and the remaining 173 patients had tumors with diameter from 1 to 3 cm.


 > Results Top


Surgical procedure

There were 216 patients had tumors of pulmonary parenchyma, who underwent surgical procedure including tumor enucleation (n = 99), pulmonary wedge resection (n = 102), and lobectomy (n = 15). Three of them had synchronous lung cancer in the same lobe. Ten patients with intraluminal tumor underwent lobectomy (n = 5), pulmonary wedge resection (n = 1), tracheostomy (n = 1), and bronchoscope tumorectomy (n = 3).

Operation approach

For 216 patients with tumors of pulmonary parenchyma, conventional incision was performed in 96 cases, including 56 cases of posterolateral incision, 27 cases of rib removed and rib bed incision, and 13 cases of anterolateral incision; lateral thoracotomy were performed in 58 cases;[1] video-assisted thoracic surgery (VATS) were performed in 61 cases, and longitudinal sternotomy incision was performed in one case who had synchronous thymic carcinoma, and the right middle lobe wedge resection was performed simultaneously [Table 1].
Table 1: Surgical procedure and operation approach of 226 pulmonary parenchyma pulmonary hamartoma

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Outcomes

The operating time, intraoperative blood loss, the drainage volume within 24 postoperative hours, and drainage duration were analyzed in 200 cases with pulmonary parenchymal tumors, 15 cases with other malignant tumors and one case underwent longitudinal sternotomy incision were excluded in this analysis. The results showed that the amount of intraoperative blood loss were significantly less in lateral thoracotomy group and VATS group than that of conventional incision group (P < 0.001). The drainage volume within 24 postoperative hours was significantly less in VATS group than that of conventional incision group (P = 0.001). The drainage volume within 24 postoperative hours was less in lateral thoracotomy group (average 189 ml) than that of conventional incision group (average 232 ml), however, the difference was not statistically significant. No significant difference of operation time and drainage duration was found in lateral thoracotomy group or VATS group when compared with conventional incision group. In addition, no significant difference of four parameters was found between lateral thoracotomy group and VATS group [Table 2].
Table 2: Differences among three surgical approaches

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There was no operative death in all 226 cases. The tumor location of 3 cases could not be determined during the VATS; therefore, the operation was expanded to lateral thoracotomy. The 200–600 ml of blood were transfused postoperatively in 5 cases, including 2 cases of esophageal carcinoma, one case of anemia, and 2 cases of severe adhesion. Two cases underwent secondary surgery to stop bleeding caused by hemothorax. One patient died in the next day after operation caused by pulmonary embolism (confirmed by autopsy).

Follow-up

The postoperative follow-up time ranged from 5 to 22 years, and the end-point of the follow-up was January 2015. Fourteen of the 17 cases with other malignant tumors died of tumor metastasis; the other 3 cases were alive more than 3 years during follow-up. All remaining cases were alive, and no hamartoma recurrence or metastasis was found.


 > Discussion Top


Khouri et al.[2] reported that PH accounted for about 4% of solitary pulmonary nodules, and was the most common benign lung tumor. The pathogenesis of PH is still controversial now. In this study, we found that the size of PH in eight cases increased during the long-term follow-up, which indicated the essence of tumor.

PH occurs in any part of lungs while intrapulmonary PH is the most common type, and accounted for 95.6% (216/226) in our study. Endobronchial PHs are relatively rare type and accounted for 4.4% (10/226) in our study. Rare types, such as multiple original PH,[3] cystic PH [4] and endotracheal diffuse PH [5] were not detected in our study. In this cohort, there was no significant difference of PH location (106 in left lung, 120 in right lung), which is consistent with the previous reports.

PH can occur at any age and more often found in middle-aged people. In our study, 94.2% of cases were 30–70 years old, the average age was 51.3 years old, which are consistent with the previous reports.[6] In our study, 64.6% of PH were found during physical examination, and PH grow slowly, and few young patients underwent CT examination regularly, so the exact time of PH formed was unknown in our study.

It was reported that the incidence of PH in male and female was 2–4:1.[6] In our study, the sex ratio (male:female) was 1.66:1, and reduced over time: 3.17:1 (1980–1990), 2.60:1 (1990–2000), and 1.30:1 (2000–2010). The decrease of the ration might correlate with the development of economy and the increased awareness of the physical examination.

Ninety percent of PH in chest X-ray showed peripheral solitary lung nodules. The tumor size was reported to be 1–2 cm in general. In our study, 76.5% of tumors were 1–3 cm, and the maximum one was 12.0 cm in the right lower lobe and was resected by lobectomy.

It was reported that calcification could be seen in 20–30% of PH. In our study, 23.0% tumors have calcification in CT examination, and 5 of them showed typical popcorn-like calcification.

The preoperative diagnosis of PH mainly based on medical history and image examination, and the atypical patients were often misdiagnosed with tuberculosis or lung cancer. PH in patients with synchronous malignant tumor was often misdiagnosed as lung metastatic cancer, which led to giving up the surgery. Bronchoscopy examination and biopsy had no diagnostic value for peripheral PH; however, it was diagnosis and treatment method for endobronchial PH with obstructive pulmonary disease. In our study, 3 cases of endobronchial PH were successfully removed using bronchoscope, which avoided the general anesthesia for patients. Hamper et al. reported that percutaneous lung biopsy got the diagnostic information in 85% PH patients.[7] However, percutaneous lung biopsy led to pneumothorax in 50% patients underwent examination because PH was relatively tough. In our study, 3 cases underwent CT-guided percutaneous lung biopsy preoperatively, one cases got confirmed diagnosis pathologically, and pneumothorax appeared in one case, therefore, percutaneous lung biopsy should be selected carefully in patients with PH. Fluorodeoxyglucose-PET (FDG-PET) examination can provide accurate information for PH diagnosis; however, it is expensive. Eight cases in our study were accurately diagnosed as benign tumor preoperatively.

For the PH patients who had a clear preoperative diagnosis, just follow-up observation without surgical resection could be recommended for them. However, if the diagnosis could not be confirmed, surgical resection should be performed for PH patients as: (1) It is difficult to distinguish most of PH from lung cancer, tuberculosis, or other tumors; (2) some patients with PH have obvious respiratory symptoms, and PH will progress gradually; (3) most patients have a great psychological burden owing to unclear diagnosis; (4) although there is no conclusive evidence, there are some reports suggest benign PH become malignant transformation.[8],[9]

Lateral thoracotomy and VATS should be recommended for PH patients, as the intraoperative blood loss, drainage volume with postoperative 24 h and drainage duration is significantly less or shorter than that of conventional incision. These two kinds of minimally invasive resection have their own advantages. For the deeper and smaller PH, it is difficult to locate tumors using VATS. Moreover, VATS takes more operation time than lateral thoracotomy, although the difference was not statistically significant in our study. In addition, considering the radiation exposure and higher cost of preoperative or intraoperative CT-guided hooks positioning, we prefer to extend the operation incision slightly (7–10 cm) to lateral thoracotomy and then palpation positioning.

In principle, normal lung tissue should be minimal resected as far as possible, local tumor excision should be first recommended. Usually, PH locates in lung parenchyma and can moved back and forth, and tumor can be squeezed to the lung surface or subpleural area and be removed by cutting the visceral pleura and small lung tissue. If the PH is fixed in the lung parenchyma, pulmonary wedge resection, segmentectomy or lobectomy can be chosen depending on the circumstances, but pneumonectomy should be avoided. For endobronchial PH, enucleation should be the first choice. If the distal lung tissue is normal, bronchial incision tumor excision or bronchial sleeve resection are feasible. When irreversible changes appeared in the distal lung tissue, lung segment or lobe resection can be considered. With the advancement in endobronchial operating equipment recent years, feasibility of removing the tumor under bronchoscope should be evaluated first for PH. In our study, 3 patients underwent bronchoscopic resection avoided the thoracic anesthesia surgery and had good long-term results. No PH recurrence or another primary PH were found in our study.

Above all, it is difficult for a considerable part of PH to have a confirmed diagnosis preoperatively; surgery is necessary for them. For patients who do not want to receive surgery, FDG-PET examination provides more accurate information for discriminating benign from a malignant tumor. Lateral thoracotomy or VATS is first recommended for PH excision.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
 > References Top

1.
Athanassiadi K, Kakaris S, Theakos N, Skottis I. Muscle-sparing versus posterolateral thoracotomy: A prospective study. Eur J Cardiothorac Surg 2007;31:496-9.  Back to cited text no. 1
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2.
Khouri NF, Meziane MA, Zerhouni EA, Fishman EK, Siegelman SS. The solitary pulmonary nodule. Assessment, diagnosis, and management. Chest 1987;91:128-33.  Back to cited text no. 2
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Bennett LL, Lesar MS, Tellis CJ. Multiple calcified chondrohamartomas of the lung: CT appearance. J Comput Assist Tomogr 1985;9:180-2.  Back to cited text no. 3
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Miura K, Hori T, Yoshizawa K, Hamaguchi N, Morita J. Cystic pulmonary hamartoma. Ann Thorac Surg 1990;49:828-9.  Back to cited text no. 4
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Minami Y, Iijima T, Yamamoto T, Morishita Y, Terashima H, Onizuka M, et al. Diffuse pulmonary hamartoma: A case report. Pathol Res Pract 2005;200:813-6.  Back to cited text no. 5
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Liu Y, Chen G, Qiu X, Xu S, Wu Y, Liu R, et al. Intrapulmonary unicentric Castleman disease mimicking peripheral pulmonary malignancy. Thoracic Cancer 2014;5:576-80.  Back to cited text no. 6
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Hamper UM, Khouri NF, Stitik FP, Siegelman SS. Pulmonary hamartoma: Diagnosis by transthoracic needle-aspiration biopsy. Radiology 1985;155:15-8.  Back to cited text no. 7
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van den Bosch JM, Wagenaar SS, Corrin B, Elbers JR, Knaepen PJ, Westermann CJ. Mesenchymoma of the lung (so called hamartoma): A review of 154 parenchymal and endobronchial cases. Thorax 1987;42:790-3.  Back to cited text no. 8
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Basile A, Gregoris A, Antoci B, Romanelli M. Malignant change in a benign pulmonary hamartoma. Thorax 1989;44:232-3.  Back to cited text no. 9
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