Endobronchial Hamartoma*

Borja G. Cosı́o, MD; Victoria Villena, MD; Jose Echave-Sustaeta, MD;

Eduardo de Miguel, MD; Jose Alfaro, MD; Luis Hernandez, MD; and
Teresa Sotelo, MD

Objectives: To describe clinical, endoscopic, radiographic, and follow-up characteristics of a

series of patients in whom endobronchial hamartoma (EH) had been diagnosed.
Methods: Retrospective study of all cases of hamartoma diagnosed by bronchial biopsy between
1974 and 1997 in a tertiary referral hospital in Madrid, Spain.
Results: EH was diagnosed 47 patients during the study period. Four patients were excluded from
the study because no clinical history was available. We analyzed the cases of 43 patients (37 men
and 6 women), with a mean (ⴞ SD) age of 62 ⴞ 12 years. Seven patients had a concurrent lung
neoplasm, and the EH was an incidental endoscopic finding. Among the other 36 patients, 31 had
a new onset of respiratory symptoms, most commonly, recurrent respiratory infections in 16
patients (44%) and hemoptysis in a further 12 patients (33.4%). Chest radiograph findings were
abnormal in 38 of 43 patients. At bronchoscopy, the lesions were equally distributed throughout
the right and left lungs with no clear lobar predilection. Endobronchial obstruction was evident
in 26 patients (72.2%) without concurrent neoplasm, 17 of whom underwent resection with a rigid
bronchoscope and laser, with total resolution in 13 patients. Partial resolution was achieved in
four patients, two of whom needed a second endoscopic procedure. Five patients were treated
with open lung surgery. Clinical and endoscopic follow-up was performed in 23 patients at 1 to
73 months (mean, 17 months), and recurrence was found in 4 patients.
Conclusion: EH frequently produces respiratory complaints and radiographic abnormalities.
Patients with endobronchial obstructions had satisfactory responses to endoscopic therapy.
(CHEST 2002; 122:202–205)

Key words: benign lung neoplasm; endobronchial; hamartoma

Abbreviation: EH ⫽ endobronchial hamartoma

T he1904termto describe
hamartomas was coined by Albrecht in
tumor-like malformations result-
1 benign lung tumors, with an incidence between
ing from a presumptive developmental abnormality. In
1934, Goldsworthy2 applied this term to benign tumors
located in the lung that were composed predominantly
of a combination of fat and cartilage. Other authors
have proposed that the hamartoma represents a true
neoplasm instead of a developmental abnormality.3
Cytogenetic studies have identified chromosomal
bands of recombination located at positions 6p21 and
14q24, supporting the idea that hamartomas represent
mesenchymal clonal neoplasms.4
Lung hamartomas are the most common form of
*From the Respiratory Department (Drs. Cosı́o, Villena, Echave-
Sustaeta, de Miguel, Alfaro, and Hernandez) and the Pathology
Department (Dr. Sotelo), Hospital 12 de Octubre, Madrid,
Spain.
Presented at the European Respiratory Society Congress in
Madrid, October 1999.
Manuscript received November 14, 2000; revision accepted
December 24, 2001.
Correspondence to: B. G. Cosı́o, MD, Servicio de Neumologı́a,
Hospital 12 de Octubre, Cra De Andalucı́a Km 5,400, 28041
Madrid, Spain; e-mail: b.cosio@ic.ac.uk
0.025% and 0.32%5,6 according to different necropsy
studies. In large clinical series of patients with lung
hamartomas, most patients are asymptomatic at the
time of diagnosis, and the hamartoma is a radiographic
finding. Endobronchial hamartomas (EHs) have a low
frequency in all these studies, and their characteristics
are poorly described. In the largest review series that
has been published (n ⫽ 215),7 only 1.4% of hamarto-
mas had an endobronchial location, the remainder
being located within the parenchyma. Other series
estimate the incidence of EHs between 10% and 20%
of all lung hamartomas.8 –10 This is the first review
specifically addressing the characteristics of EHs.
The aim of this study is to analyze the clinical,
radiographic, and endoscopic characteristics of the
EHs, and to discuss the outcome of treated cases in
this the largest series described.
Materials and Methods
The Bronchoscopy Unit of the 12 de Octubre Hospital, a
tertiary hospital with 1,800 beds, performed about 31,000 bron-

202 Clinical Investigations

choscopy procedures between 1974 and 1997. Since 1980, this
hospital has served as a referral center for endoscopic therapy of
endobronchial lesions by rigid bronchoscope and laser therapy.
In this study, we retrospectively analyzed the cases of all
patients with bronchial biopsy-proven EH during a period of 23
years. The Bronchoscopy Unit database was analyzed in order to
identify the patients in whom EHs had been diagnosed by
bronchial biopsy. Pathology department files were reviewed to
confirm the diagnosis. Once the patients were identified and the
diagnosis was confirmed, the clinical history, radiology findings,
and endoscopic follow-up procedures were reviewed. The follow-
ing variables were studied: age at diagnosis; gender; symptoms at
diagnosis; indication for bronchoscopy; medical history with
special emphasis on previous or concurrent neoplasm; radio-
graphic abnormalities; endobronchial location of the hamarto-
mas; endoscopic appearance; pathologic characteristics; grade of
bronchial obstruction; therapy, if any; clinical and endoscopic
follow-up; and recurrence.
The indication for therapy was based on the presence of severe
symptoms such as hemoptysis or recurrent obstructive pneumo-
nia. The selection of therapy depended on the size and site of the
obstruction and the technical applicability of each technique.
Surgery has not been performed in the management of EH in
this center since 1980 due to the highly successful outcomes
achieved with bronchoscopy. The rigid bronchoscopy procedures
included mechanical resection of the hamartoma and the appli-
cation of an Nd-YAG laser to its borders when necessary due to
insufficient resection or excessive bleeding.
In the evaluation of the endoscopic therapy, the reestablish-
ment of the bronchial lumen to ⬎ 80% of the original lumen
diameter with radiologic resolution was considered to be success-
ful therapy. In all cases, successful endobronchial therapy was
accompanied by radiologic resolution as well. Partial success was
considered when the bronchial lumen was reestablished between
50% and 80% without complete radiologic resolution. Recur-
rence was considered to have occurred when growth of the lesion
was observed with progression of the obstruction in ⬎ 20% of the
previously reported lumen diameter.
Table 1—Indication for Bronchoscopy
Indication No. of Patients (%)
Recurrent pneumonia 16 (37.2)
Hemoptysis 10 (23.3)
Radiographic abnormalities
Volume loss 5 (11.6)
Others 10 (23.3)
Persistent cough 1 (2.3)
Endoscopic finding in a review procedure 1 (2.3)
tree are shown in Figure 1. There was not a predom-
inant location for EHs in our series.
Endoscopic features that suggested 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 (Fig 2).
At biopsy, spongiform consistency was always de-
scribed.
The endoscopic appearance of the lesion later
diagnosed as an hamartoma was described as sugges-
tive of benign neoplasm in all 43 cases. The histo-
logic pattern most frequently described was that of
chondroid hamartoma in 16 cases (37.2%) and lipoid
hamartoma in 13 cases (30.2%). A lipoid hamartoma
is shown in detail in Figure 3. In the other 14
samples, no predominant cellular component was
identified.

Results
During the study period, 47 EHs were diagnosed
in the Hospital 12 de Octubre. Four patients were
excluded because their clinical histories were not
available. Forty-three patients were studied (37 men
and 6 women), with a mean (⫾ SD) age of 62 ⫾ 12
years (range, 29 to 81 years). Only nine patients were
asymptomatic at the time of diagnosis. On presenta-
tion, the most frequent clinical symptoms were
recurrent respiratory infections or obstructive pneu-
monia in 16 patients (37%) and hemoptysis in 14
patients (32%), with or without other respiratory
complaints such as cough or dyspnea. Indications for
bronchoscopy (Table 1) in these patients can be
classified into the following three groups: (1) bron-
chial symptoms such as persistent cough or hemop-
tysis; (2) radiographic abnormalities such as volume Figure 1. Distribution of the 43 EHs within the tracheobron-
loss, solitary pulmonary nodule, interstitial pattern, chial tree as follows: trachea, 4; right main bronchus, 2; left main
or recurrent alveolar infiltrates; and (3) recurrent bronchus, 6; intermediate bronchus, 3; middle lobe bronchus, 3;
right upper lobe bronchus, 8; left upper lobe bronchus, 8; left
pneumonia. upper lobe bronchus, 3; right lower lobe bronchus, 3; and left
The locations of the EHs in the tracheobronchial lower lobe bronchus, 3.

www.chestjournal.org CHEST / 122 / 1 / JULY, 2002 203


Figure 2. Endoscopic appearance of EH.
In seven patients, the EH was an endoscopic
finding during the assessment of a concurrent lung
neoplasm (squamous carcinoma, three patients; ad-
enocarcinoma, one patient; large cell carcinoma, one
patient; oat cell carcinoma, one patient; and lym-
phoma, one patient). In only one case was the
hamartoma in the same lung as the neoplasm, albeit
in a different lobe.
Of the 36 patients without concurrent lung neo-
plasms, 31 (86.1%) had the onset of new respiratory
complaints at the time of diagnosis. The most com-
mon complaint, recurrent respiratory infections, was
present in 16 patients (44.4%), while hemoptysis was
present in 12 patients (33.4%) [Table 2]. Chest
radiography findings for this group were normal in
just five patients (13.9%). The radiographic abnor-
malities most frequently observed were alveolar in-
filtrates (11 patients; 30.6%), atelectasis or volume
loss (9 patients; 25%), or both (8 patients; 22.2%).
Other radiographic patterns were observed in three
patients (8.3%) in association with non-neoplastic
Figure 3. Lipoid hamartoma. (hematoxylin-eosin, original
⫻400). Components shown in detail are adipoid and mesenchy-
mal tissue.
204
Table 2—Symptoms at Diagnosis
Clinical Presentation No. of Patients (%)
Obstructive pneumonia 16 (44.4)
Hemoptysis 6 (16.7)
Cough and hemoptysis 4 (11.1)
Cough 2 (5.6)
Dyspnea and hemoptysis 2 (5.6)
Dyspnea 1 (2.8)
Asymptomatic 5 (13.9)
parenchymal disease (benign solitary pulmonary
nodules, two patients; and interstitial disease, one
patient).
EHs produced a degree of bronchial obstruction
in 26 of the 36 patients with no concurrent tumoral
lung disease related to a tumor (tracheal obstruction,
1 patient; main bronchus obstruction, 5 patients;
lobar bronchus obstruction, 10 patients; and seg-
mental bronchus obstruction, 10 patients). Of the 36
patients with EHs but no concurrent malignant
neoplasms, 24 underwent treatment that was aimed
at eliminating the EH (Table 3). The therapeutic
method most frequently used was rigid bronchos-
copy with or without laser application. Five patients
were treated surgically, with three undergoing bron-
chotomies and two undergoing lobectomies.
Endoscopic reevaluation was performed in 22 of
the 24 patients that were treated. The treatment was
considered to be successful in 18 patients (81.8%),
and partially successful in 4 patients (18.2%). Of the
17 patients treated with rigid bronchoscopy, the
treatment was considered to be successful in 13 and
partially successful in 4. There was an endoscopic
follow-up in 15 of the 17 patients treated by this
method, during a mean period of 16.2 months
(range, 1 to 72 months). In this group, recurrence
was observed in four patients, and a second endo-
scopic procedure was performed in three of them.
Discussion
The endobronchial location of the benign hamar-
toma is a rare occurrence. In a 1950 study of lung
carcinoma, bronchial adenoma, and lung hamar-
toma, Le Roux11 found 3,000 patients with lung
Table 3—Management of EHs
Treatment No. of Patients (%)
Rigid bronchoscopy and laser 17 (47.2)
Thoracotomy 5 (13.9)
Fiberoptic bronchoscopy with forceps 2 (5.6)
resection
No treatment 12 (33.3)
Clinical Investigations
carcinomas, 40 with bronchial adenomas, and 27
with lung hamartomas, of which only 8% were EHs.
In the largest published series of pulmonary hamar-
tomas, Gjevre et al7 analyzed 215 cases of hamar-
toma, of which only 1.4% were located endobronchi-
ally; the authors concluded that the hamartoma was
a benign neoplasm and frequently asymptomatic.
Our retrospective series demonstrates that EHs, in
comparison with previously described parenchymal
hamartomas, are frequently symptomatic, and for
this reason treatment is frequently indicated. The
mean age of the patient at presentation was between
the sixth and seventh decades, as has been found in
previous series,7 with male predominance.
In the population of the study by Gjevre et al,7
most of the patients were asymptomatic, and the
hamartoma was an incidental finding. In our series,
80% of the patients had at least one respiratory
complaint. The clinical features most frequently
found were those of hemoptysis and obstructive
pneumonia. This likely can be explained by the fact
that we are a reference center for endoscopic ther-
apy in Spain and, surely, we are not seeing the
patients without severe clinical manifestations. The
high incidence of radiographic abnormalities is sim-
ilar to those in previous reports.9
We found no cases of multiple hamartoma, and no
association between parenchymal hamartoma and
EH, as previously has been reported.12 Lung hamar-
tomas are benign neoplasms with very low risk of
malignancy and a low rate of recurrence. We have
not found any case of malignancy, although we have
observed the growth of benign hamartomas in four
patients during follow-up, three of whom had to
undergo a second endoscopic procedure. Although
there was concurrent tumor-related lung disease in
seven patients, we could not find any association
between hamartoma and tumor location.
The management of EHs must be individualized
according to the characteristics of each patient and
each hamartoma. In our experience, endoscopic
treatment with rigid bronchoscopy and laser is a
good therapeutic choice in the symptomatic patient
or in the patient with serious radiographic abnormal-
ities. This technique leads to the control of symp-
toms and might avoid the risk of thoracotomy.13
www.chestjournal.org
In conclusion, EHs are benign neoplasms of the
tracheobronchial tree that potentially can be danger-
ous due to bronchial obstruction or bleeding. Endo-
scopic treatment with rigid bronchoscopy and lasers
provides an excellent outcome when performed by
expert hands. Therefore, such treatment should be
recommended in highly symptomatic patients or in
those patients with significant bronchial obstruc-
tions.
ACKNOWLEDGMENT: The authors gratefully acknowledge
Dr. Robert Stirling for his help in editing this manuscript.
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