hamartoma by electrocautery and
cryotherapy via flexible bronchoscopy
Sedat ALTIN1, Levent DALAR2, Levent KARASULU1, Erdoğan ÇETİNKAYA1, Sinem TİMUR1, Nur SOLMAZER1
1 Yedikule Göğüs Hastalıkları ve Göğüs Cerrahisi Eğitim ve Araştırma Hastanesi, İstanbul, 2 Bozüyük Devlet Hastanesi, Bozüyük, Bilecik.
ÖZET
Dev bir endobronşiyal hamartomun fiberoptik bronkoskop kullanılarak elektrokoter ve kriyoterapi ile tedavisi
Endobronşiyal hamartomlar tüm hamartom olgularının %3-10’unu kapsayan nadir tümörlerdir. Bu yazıda fiberoptik flek- sibl bronkoskop aracılığıyla elektrokoter ve kriyoterapi kullanılarak tamamen rezeke edilen bir endobronşiyal hamartom olgusu tartışıldı. Egzersiz dispnesi, göğüs ağrısı ve hemoptizi yakınmalarıyla başvuran 71 yaşındaki erkek olgunun bilgi- sayarlı toraks tomografisinde trakea lümeni alt ucunu ve sol ana bronşu oblitere eden kitle izlendi. Tanısal bronkoskopik incelemesinde sol ana bronşu tama yakın tıkayan geniş polipoid kitle görüldü. Lezyon solunum hareketleriyle sağ bronş sistemine protrude oluyordu. Elektrokoterin snare probu kullanılarak kitle dört parça halinde eksize edildi ve tümörün kay- naklandığı bazal bölüme kriyoterapi uygulandı. On gün ve üç ay sonra yapılan bronkoskopik kontrolde tüm bronş siste- minin tamamen açık olduğu görüldü.Tümörün patolojik tanısı fibrovasküler komponent içeren hamartom olarak kondu.
Sonuç olarak; malignite şüphesi taşımayan endobronşiyal hamartom olgularında endoskopik tedavi yaklaşımları güvenli ve minimal invazivdir. Bu nedenle öncelikli tedavi yaklaşımı olarak değerlendirilmelidir.
Anahtar Kelimeler: Endobronşiyal hamartom, endobronşiyal tedavi, elektrokoter, kriyoterapi.
Yazışma Adresi (Address for Correspondence):
Dr. Levent DALAR, Bozüyük Devlet Hastanesi B Binası Göğüs Hastalıkları Kliniği, Bozüyük 11300 BİLECİK - TURKEY
e-mail: leventdalar@gmail.com
Most tumors of tracheobroncial tree are malig- nant. Less than 1% of lung tumors are benign and of these, hamartomas are most common.
Endobronchially located hamartomas are rare and account for only for 1.4% of all pulmonary hamartomas in a recent analysis (1). Endob- ronchial hamartomas arise from major bronchi.
They are composed of mixture of bronchial components and they are described by the prin- cipal bronchial component (chondromatous, li- pomatous or mixed mesenchymal). Frequently, cartilaginous elements predominate in most of them, but a few may contain predominantly adi- pose tissue or bone (2). The diagnosis may be suggested by endoscopic aspects. Computed tomography (CT) may reveal endobronchial tu- mor with fat density (3). Due to their benign na- ture endobronchial hamartomas are excised via bronchotomy. Another effective and easily per- formed technique is cryotherapy. Endobronchial laser therapy can also be used. If irreversible lung damage has occurred because of chronic
obstruction and suppuration, pulmonary resecti- on may be indicated (4,5).
CASE REPORT
A 71-year-old male presented to our clinic with progressive dispnea on exertion, chest pain and hemoptysis. He has nonproductive cough and dispnea for 5 years but for the last a few months he also has hemoptysis. There is not any speci- alty in his history except a 50 packet/years of smoking history. Biochemical analysis is in nor- mal ranges. There was a mass lesion on his tho- rax CT which is planned because of revealing volume lost in left lung and atelectasis in the left lower lobe in his postero-anterior chest X-ray (Figure 1). Thorax CT scan of the patient reve- aled a mass lesion just prior to the carina most- ly obliterating tracheal lumen and by extension nearly totally obliterating left main bronchus, ca- using volume loss in the left lung and hyperinf- lation. Consolidation, acinary and tree-in-bud appearance in the left lung lingular segment. Wi- SUMMARY
Resection of giant endobronchial hamartoma by electrocautery and cryotherapy via flexible bronchoscopy
Sedat ALTIN1, Levent DALAR2, Levent KARASULU1, Erdoğan ÇETİNKAYA1, Sinem TİMUR1, Nur SOLMAZER1
1 Yedikule Chest Disease and Chest Surgery Education and Research Hospital, İstanbul, Turkey, 2 Bozüyük State Hospital, Bozüyük, Bilecik, Turkey.
Endobronchial hamartoma is a rare tumor which is 3-10% of all hamartomas. We presented a case of endobronchial hamar- toma that was resected and ablated with electrocautery and cryotherapy by fiberoptic bronchoscopy. A 71-year-old male, presented with progressive dispnea on exertion, chest pain and hemoptysis. Chest computed tomography (CT) scan of the patient revealed a mass lesion mostly obliterating tracheal lumen and left main bronchus. An initial bronchoscopy showed a large polypoid lesion within the left mainstem bronchus. Polipoid lesion protrudes to the right with respiration. Interven- tional bronchoscopy with snare probe of the electrocautery resected the polyp in 4 pieces followed by cryotherapy of the base. Pathology of the resected lesion diagnosed as bronchial hamartoma with fibrovasculer component. Control bronchos- copy applied 10 days later revealed patency of all airways. The majority of hamartomas is parenchymal and is often loca- ted in the periphery of the lung, unlike our patient who had a more central lesion. Where a firm diagnosis is made preope- ratively, surgical removal may not be necessary unless symptoms are present, or tumor expansion is noted. Our case de- monstrates resection of an endobronchial hamartoma by snare probe of the electrocautery and with cryotherapy. Electro- cautery and crytherapy may present an alternative therapy for resection of selected benign endobronchial benign tumors.
Key Words: Endobronchial hamartoma, endobronchial therapy, electrocautery, cryotherapy.
despread bronchiectatic changes observed in the left lower lobe. Dilated bronchus filled with mucus was observed at the level of basal seg- ments. There is not any abnormality in his physical examination and evident obstruction was seen in his respiratory function tests. Bronc- hoscopy showed a large polypoid lesion covered by a smooth mucosa located at the lower part of the trachea obstructing the left mainstem bronc- hus and with respiration moving towards the right bronchus. Pathology of the biopsy material revealed compatible with lipoma or hamartoma.
Polypoid lesion with smooth mucosa protrudes from the left main bronchus and changes psition to right main bronchus with inspiration. Videob- ronchoscopy with snare probe of the loop electro- cautery resected the polyp in 4 pieces (Figure 2).
Left main bronchus cleaned up totally. The ori- gin of the lesion was observed at the left lower
lobe. In the second séance residue in the entran- ce of the left lower lobe resected with snare pro- be of the loop electrocautery followed by cryot- herapy of the base remained in the anterior seg- ment. Definite pathological diagnosis assigned as hamartoma (Figure 3). In the control bronc- hoscopy done 10 days later no tumor remnant was seen and patency of all lower lobe segments was revealed. The patient’s dispnea resolved and increase in the lung function tests (FVC and FEV1values) observed.
DISCUSSION
The term hamartomas was coined by Albrecht in 1904 to describe tumor-like malformations resulting from a presumptive developmental ab- normality. In 1934 Goldsworthy applied this term to benign tumors located in the lung that Figure 1. Chest X-rays of the patient, before (A) and after (B) the resection.
Figure 2. The resected giant hamartoma.
Figure 3. The cytologic examination view of the polyp (HE, x100).
A B
were composed predominantly of a combination of fat and cartilage. Other authors have propo- sed that the hamartoma represents a true neop- lasm instead of a developmental abnormality.
Cytogenetic studies have identified chromoso- mal bands of recombination located at positions 6p21 and 14q24, supporting the idea that ha- martomas represent mesenchymal clonal neop- lasms (6).
Pulmonary hamartoma is a rare benign tumor.
The endobronchial location of the benign ha- martoma is a rarer occurrence. Their incidence changes between 0.025% and 0.32% according to different necropsy studies. Arrigoni et al re- viewed 130 benign lung tumors and 77% were hamartomas. Only 3% of these hamartomas were endobronchial (7). According to Minisian, thoracotomy was performed to 3450 patients for pulmonary neoplasms. Of these 33 of them had hamartoma. Only two of the hamartomas were endobronchial (8). In a 1950 study of lung carcinoma, bronchial adenoma and lung hamartoma, Le Roux found 3000 patients with lung carcinomas, 40 with bronchial adenomas, and 27 with lung hamartomas of which only 8%
were endobronchial (9). Other series estimate the incidence of endobronchial hamartomas between 10% and 20% of all lung hamartomas (6). In a recent analysis with the largest series, endobronchially located hamartomas are rare and account for only 1.4% of all pulmonary ha- martomas (1).
Intraparenchimal and also endobronchial tu- mors take origin from primitive bronchial me- senchymal tissue which has the capacity to dif- ferentiate toward multiple mature mesenchymal components (2). Hamartomas, intrapulmonary and endobronchial, contain cartilage, fat, fibro- us tissue, and an epithelial component. Endob- ronchial lesions have relatively more fat than parenchymal lesions (3,10). Cartilaginous ele- ments predominate but a few may contain pre- dominantly adipose tissue or bone (11). In our patient hamartoma was composed mainly of fatty tissue.
The mean age of the patient at presentation was between the sixth and seventh decades as has be-
en found in previous series, with male predomi- nance (6). Male to female ratio was 3-5/1 (12).
Endobronchial hamartoma is a special form of intrapulmonary hamartomas by originating from the larger bronchus but obstructs the bronchiole by growing toward the lumen. Endoscopic featu- res that suggest the presence of a hamartoma were the presence of an exophytic or polypoidal mass with a smooth well-limited surface and without signs of submucosal infiltration. The ret- rospective series of Cosio et al. demonstrates that endobronchial hamartomas in comparison with previously described parenchymal hamar- tomas are frequently asymptomatic and for this reason treatment frequently indicated (6). As they can be polypoid or pedunculated they can cause cough, dispnea, and fever which were symptoms of endobronchial lesions (1,4). On presentation, the most frequent clinical symp- toms were recurrent respiratory infections or obstructive pneumonia and hemoptysis with or without other respiratory complaints such as co- ugh or dyspnea (6).
Main CT finding of the endobronchial hamarto- ma is endobronchial mass with or without the company of obstructive pneumonia or atelecta- sis. CT can be useful when the lesion contains an abundance of fat. Proto reported two cases with parenchymal hamartoma that showed fat attenuation areas on CT scans (13). Also Ahn et al. reported three patients with endobronchial hamartoma that have fat attenuation areas (6).
For hamartoma CT findings of a homogenous mass with fat density and no enhancing contrast are considered diagnostic (11).
Malign transformation and tendency to invasion was rare although reported for some cases. Pe- ripherically localized hamartomas can not be se- en bronchoscopically, for the diagnosis fine ne- edle aspiration biopsy offers a valuable alterna- tive to diagnostic thoracotomy in peripheral ha- martomas with a diagnostic yield of up to 85%.
For the treatment of endobronchial hamartomas which are benign lesions endoscopic treatment approach must be the first alternative. Lobecto- mies and even pneumonectomies have been
performed for the suspicion of malignancy. Due to their benign nature endobronchial hamarto- mas should initially be treated endoscopically.
Nevertheless if the tumor is large or lung behind the tumor is destroyed surgical resection has to be considered (11). In the case of localized en- dobronchial hamartomas bronchoscopic resec- tion is a frequently used method. In a 36 cases study, Cosio et al. made laser together with bronchoscopy to 17 cases and from these 36, thoracotomy was needed only for 5 of them. In two of the cases forceps resection with fiberop- tic bronchoscopy was done (6).
Although laser and mechanical resection with ri- gid bronchoscopy were the basic approach, for some selected cases fiberoptic bronchoscopy and electrocautery can be safe and easily appli- ed treatment modalities. For the removal of the residue, tumor cryotherapy, is a perfect choice without any risk for stricture, stenosis or any ot- her complication (5). Kaya and colleagues have been successfully treated a case with endob- ronchial hamartoma using electrocautery (14).
Horio and colleagues successfully excised a pe- dunculated hamartoma located at the right middle bronchus of a 61 years old man using snare probe of the electrocautery (15). Likewise there are examples of endobronchial hamarto- mas treated using rigid bronchoscopy and laser (16-19). In one case segmentectomy was per- formed for the suspicion of malignity (17). Our case is an example of successfully treated giant endobronchial hamartoma by electrocautery and cryotherapy via flexible bronchoscopy.
In conclusion, endobronchial hamartomas are benign neoplasms of the tracheobronchial tree that potentially can be dangerous due to bronc- hial obstruction or bleeding. Endoscopic treat- ment with flexible bronchoscope and electroca- utery and cryotherapy provides an excellent outcome when performed by expert hands. The- refore such treatment should be recommended in highly symptomatic patients or in those pati- ents with significant bronchial obstructions.
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