Case Report
Endobronchial lipoma, an extremely rare benign tumour of the lung, mimicking asthma bronchiale M. Rooijakkers1, J. Wynants2, J. Aumann2 1
Resident Pneumology, University Hospitals Leuven, Belgium, 2Department of Pneumology, Jessa Ziekenhuis, Hasselt, Belgium Endobronchial lipomas are extremely rare benign tumours of the lung. Their clinical presentation mimics that of obstructive lung diseases such as asthma and chronic obstructive pulmonary disease (COPD), leading to a delay in diagnosis and errors in treatment. Therefore, making precise diagnosis may be challenging. We report a case of a 63-year-old man with paroxysmal attacks of dyspnea, non-productive cough, and wheezing, initially suspect for adult onset asthma, but with a final diagnosis of endobronchial lipoma.
Keywords: endobronchial lipoma, rare benign tumour, mimicking asthma, malignancy, endoscopic resection
Introduction Endobronchial lipoma are rare benign tumours and account for only 0.1–0.5% of all benign tumours of the lung and endobronchial tree. They can easily leads to a misdiagnosis of asthma, chronic obstructive pulmonary disease (COPD), or malignancy because of their obstructive character. Therefore, accurate diagnosis of this insidious entity is essential since tumour removal is the only right treatment. We report a case of a 63-year-old patient with an endobronchial lipoma who had previously been treated for adult onset asthma. Inadequate response to appropriate asthma therapy led to suspicion of airway obstruction, which was confirmed by means of CT scan and subsequent bronchoscopy.
Case History A 63-year-old patient was referred for paroxysmal attacks of dyspnea coupled with a non-productive cough and wheezing. He had a history of hypercholesterolemia, arterial hypertension, and a humerus fracture on the right sight. His medication was Omeprazole (OmeprazoleH), Atorvastatine (LipitorH), citalopram hydrobromide (CitalopramH), and zolpidem hemitartrate (ZolpidemH). His respiratory problems started after an upper respiratory tract infection a few weeks before presentation in the outpatient clinic. During that period, the patient had been submitted to treatment Correspondence to: M Rooijakkers, Godtschalckstraat 5 bus 04/03, 8400 Oostende, Belgium. Email: [email protected]
74
ß Acta Clinica Belgica 2014 DOI 10.1179/0001551213Z.00000000010
with bronchodilators and antibiotic therapy, which evoked only a partial response. He never noticed fever or haemoptysis. He had smoked 20 cigarettes per day until 20 years ago and there was no family history of asthma. Physical examination revealed decreased respiratory sounds and diffuse end-expiratory wheezing with a peripheral oxygen saturation level of 96% without receiving any supplemental oxygen. Lung function data demonstrated FEV1 80% and FEV1/VC 68%. Skin test for classical allergens was negative. Based on this clinical data, the suspicion of adult onset asthma arose and formoterol fumarate 6 mg plus beclometason dipropionate 100 mg (InuvairH) was started, with at that time a good response, leading to less dyspnea, an FEV1/VC above 70%, and complete disappearance of end-expiratory wheezing on physical examination. Five months later, the patient presented again at the outpatient clinic with repeating complaints of dyspnea on exertion and wheezing, at this moment, both inspiratory and expiratory. Spirometry showed increasing signs of airway obstruction (FEV1/VC 62%). Conventional chest radiographs showed a consolidation in the left upper lobe (lingula) and additional CT scan was performed. This revealed an endoluminal mass, 10610 mm in size, in the left main bronchus in close proximity of the entrance of the lingular part of the left upper lobe without evidence of tumour extension into the bronchial wall (Fig. 1). Flexible bronchoscopy confirmed the presence of a
Acta Clinica Belgica
2014
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NO.
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Rooijakkers et al.
Figure 1 Endoluminal mass (10 x 10 mm) in the left main bronchus in close proximity of the entrance of the lingular part of the left upper lobe without evidence of tumour extension into the bronchial wall.
pedunculated polypoid lesion with a smooth surface in the left main bronchus. Histopathological examination of the biopsies showed submucosal welldifferentiated benign fat tissue covered with normal respiratory epithelium, and thus, a diagnosis of endobronchial lipoma was established. Subsequently, the patient was referred to the Interventional Pulmonary Department of the University Hospitals Leuven for endoscopic resection. Successful lasering of the endobronchial lipoma was performed with complete disappearance of the respiratory complaints and normal spirometry values.
Discussion Endobronchial lipoma are extremely rare benign tumours and account for only 0.1–0.5% of all benign tumours of the lung and endobronchial tree.1–3 The majority are located in the first three subdivisions of the tracheobronchial tree with a preference of the right lung1,2 and transbronchial extension is rare.2 They are more seen in men in the fifth or sixth decade of life, especially in those with obesity and smoking.1,3 Symptoms occur late and depend on the degree of airway obstruction. The most prominent complaint is cough, occurring in 86% of cases.1 Other symptoms include dyspnea, chest pain, wheeze, recurrent fever, and pneumonia. Consequently, these low-grade respiratory symptoms can easily lead to a misdiagnosis of asthma or COPD. Haemoptysis is uncommon due to the avascular nature of these tumour, but it can occasionally occur as a result of post-obstructive infection.1–3 Sometimes they remain asymptomatic and detected through abnormal conventional radiographs or at autopsy.2
Bronchial lipoma
Macroscopically, lipomas appear as a pedunculated or sessile yellow-grey polypoid lesion with a smooth surface.1,3 Histologically, they originate from submucosal or interstitial adipose tissue and are lined by normal respiratory epithelium.1–3 Conventional chest radiography shows non-specific post-obstructive changes such as atelectasis, pneumonia, and even bronchiectasis in 80%, but is generally non-diagnostic.1–3 The imaging modality of choice for establishing the diagnosis of lipoma is CT or MRI because of their high specificity and sensitivity of detecting these fat density lesions.1–3 A homogenous lesion with low attenuation around 2100 Hounsfield unit (Hu) on CT is suggestive for a lipoma.3 However, the gold standard for diagnosis is bronchoscopy, as it identifies the location of the lipoma and facilitates collection of tissue for histopathology.1–3 Note that transbronchial biopsies are frequently inadequate for diagnosis since the capsule of the lipoma is pliable and covered with normal respiratory epithelium and thus, complete resection is needed to confirm diagnosis.1–3 Endoscopic resection, with preference of rigid endoscopic techniques as it results in complete resection of the lipoma with preservation of lung parenchyma, should be considered as the first choice of treatment.1–4 The use of neodymium–yttrium– aluminium–garnet (Nd : YAG) laser, electrocautery, argon plasma coagulation, and cryotherapy has also been described as alternative therapeutic modalities.1,4 Nevertheless, surgical resection can be indicated in the following circumstances: (1) possibility of malignancy; (2) presence of peripheral lung parenchym damage due to long-term atelectasis or pneumonia; (3) transbronchial growth or subpleural lipomateus disease; or (4) expected technical difficulties during the endoscopic resection due to multidirectional growth of the tumour.1–4 In conclusion, clinicians should be aware of this extremely rare and benign entity mimicking malignancy, asthma, or COPD since early diagnosis and endoscopic resection help to prevent irreversible lung damage.
References 1 Irani F, Kumar B, Reddy P, Narwal-Chadha R, Kasmani R, Tita J. An endobronchial lipoma mimicking asthma and malignancy. Prim Care Respir J. 2010;19(3):281–3. 2 Muraoka M, Oka T, Akamine S, Nagayasu T, Iseki M, Suyama N, et al. Endobronchial lipoma: review of 64 cases reported in Japan. Chest. 2003;123(1):293–6. 3 Cao D, Sun Y, Yang S. Endobronchial lipoma: an unusual cause of bronchial obstruction. Case Report Med. 2011;2011: 1–4. 4 Gao H, Ding X, Wei D, Cheng P, Su X, Liu H, et al. Endoscopic management of benign tracheobronchial tumors. J Thorac Dis. 2011;3(4):255–61.
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Endobronchial lipoma, an extremely rare benign tumour of the lung, mimicking asthma bronchiale M. Rooijakkers1, J. Wynants2, J. Aumann2 1
Resident Pneumology, University Hospitals Leuven, Belgium, 2Department of Pneumology, Jessa Ziekenhuis, Hasselt, Belgium Endobronchial lipomas are extremely rare benign tumours of the lung. Their clinical presentation mimics that of obstructive lung diseases such as asthma and chronic obstructive pulmonary disease (COPD), leading to a delay in diagnosis and errors in treatment. Therefore, making precise diagnosis may be challenging. We report a case of a 63-year-old man with paroxysmal attacks of dyspnea, non-productive cough, and wheezing, initially suspect for adult onset asthma, but with a final diagnosis of endobronchial lipoma.
Keywords: endobronchial lipoma, rare benign tumour, mimicking asthma, malignancy, endoscopic resection
Introduction Endobronchial lipoma are rare benign tumours and account for only 0.1–0.5% of all benign tumours of the lung and endobronchial tree. They can easily leads to a misdiagnosis of asthma, chronic obstructive pulmonary disease (COPD), or malignancy because of their obstructive character. Therefore, accurate diagnosis of this insidious entity is essential since tumour removal is the only right treatment. We report a case of a 63-year-old patient with an endobronchial lipoma who had previously been treated for adult onset asthma. Inadequate response to appropriate asthma therapy led to suspicion of airway obstruction, which was confirmed by means of CT scan and subsequent bronchoscopy.
Case History A 63-year-old patient was referred for paroxysmal attacks of dyspnea coupled with a non-productive cough and wheezing. He had a history of hypercholesterolemia, arterial hypertension, and a humerus fracture on the right sight. His medication was Omeprazole (OmeprazoleH), Atorvastatine (LipitorH), citalopram hydrobromide (CitalopramH), and zolpidem hemitartrate (ZolpidemH). His respiratory problems started after an upper respiratory tract infection a few weeks before presentation in the outpatient clinic. During that period, the patient had been submitted to treatment Correspondence to: M Rooijakkers, Godtschalckstraat 5 bus 04/03, 8400 Oostende, Belgium. Email: [email protected]
74
ß Acta Clinica Belgica 2014 DOI 10.1179/0001551213Z.00000000010
with bronchodilators and antibiotic therapy, which evoked only a partial response. He never noticed fever or haemoptysis. He had smoked 20 cigarettes per day until 20 years ago and there was no family history of asthma. Physical examination revealed decreased respiratory sounds and diffuse end-expiratory wheezing with a peripheral oxygen saturation level of 96% without receiving any supplemental oxygen. Lung function data demonstrated FEV1 80% and FEV1/VC 68%. Skin test for classical allergens was negative. Based on this clinical data, the suspicion of adult onset asthma arose and formoterol fumarate 6 mg plus beclometason dipropionate 100 mg (InuvairH) was started, with at that time a good response, leading to less dyspnea, an FEV1/VC above 70%, and complete disappearance of end-expiratory wheezing on physical examination. Five months later, the patient presented again at the outpatient clinic with repeating complaints of dyspnea on exertion and wheezing, at this moment, both inspiratory and expiratory. Spirometry showed increasing signs of airway obstruction (FEV1/VC 62%). Conventional chest radiographs showed a consolidation in the left upper lobe (lingula) and additional CT scan was performed. This revealed an endoluminal mass, 10610 mm in size, in the left main bronchus in close proximity of the entrance of the lingular part of the left upper lobe without evidence of tumour extension into the bronchial wall (Fig. 1). Flexible bronchoscopy confirmed the presence of a
Acta Clinica Belgica
2014
VOL.
69
NO.
1
Rooijakkers et al.
Figure 1 Endoluminal mass (10 x 10 mm) in the left main bronchus in close proximity of the entrance of the lingular part of the left upper lobe without evidence of tumour extension into the bronchial wall.
pedunculated polypoid lesion with a smooth surface in the left main bronchus. Histopathological examination of the biopsies showed submucosal welldifferentiated benign fat tissue covered with normal respiratory epithelium, and thus, a diagnosis of endobronchial lipoma was established. Subsequently, the patient was referred to the Interventional Pulmonary Department of the University Hospitals Leuven for endoscopic resection. Successful lasering of the endobronchial lipoma was performed with complete disappearance of the respiratory complaints and normal spirometry values.
Discussion Endobronchial lipoma are extremely rare benign tumours and account for only 0.1–0.5% of all benign tumours of the lung and endobronchial tree.1–3 The majority are located in the first three subdivisions of the tracheobronchial tree with a preference of the right lung1,2 and transbronchial extension is rare.2 They are more seen in men in the fifth or sixth decade of life, especially in those with obesity and smoking.1,3 Symptoms occur late and depend on the degree of airway obstruction. The most prominent complaint is cough, occurring in 86% of cases.1 Other symptoms include dyspnea, chest pain, wheeze, recurrent fever, and pneumonia. Consequently, these low-grade respiratory symptoms can easily lead to a misdiagnosis of asthma or COPD. Haemoptysis is uncommon due to the avascular nature of these tumour, but it can occasionally occur as a result of post-obstructive infection.1–3 Sometimes they remain asymptomatic and detected through abnormal conventional radiographs or at autopsy.2
Bronchial lipoma
Macroscopically, lipomas appear as a pedunculated or sessile yellow-grey polypoid lesion with a smooth surface.1,3 Histologically, they originate from submucosal or interstitial adipose tissue and are lined by normal respiratory epithelium.1–3 Conventional chest radiography shows non-specific post-obstructive changes such as atelectasis, pneumonia, and even bronchiectasis in 80%, but is generally non-diagnostic.1–3 The imaging modality of choice for establishing the diagnosis of lipoma is CT or MRI because of their high specificity and sensitivity of detecting these fat density lesions.1–3 A homogenous lesion with low attenuation around 2100 Hounsfield unit (Hu) on CT is suggestive for a lipoma.3 However, the gold standard for diagnosis is bronchoscopy, as it identifies the location of the lipoma and facilitates collection of tissue for histopathology.1–3 Note that transbronchial biopsies are frequently inadequate for diagnosis since the capsule of the lipoma is pliable and covered with normal respiratory epithelium and thus, complete resection is needed to confirm diagnosis.1–3 Endoscopic resection, with preference of rigid endoscopic techniques as it results in complete resection of the lipoma with preservation of lung parenchyma, should be considered as the first choice of treatment.1–4 The use of neodymium–yttrium– aluminium–garnet (Nd : YAG) laser, electrocautery, argon plasma coagulation, and cryotherapy has also been described as alternative therapeutic modalities.1,4 Nevertheless, surgical resection can be indicated in the following circumstances: (1) possibility of malignancy; (2) presence of peripheral lung parenchym damage due to long-term atelectasis or pneumonia; (3) transbronchial growth or subpleural lipomateus disease; or (4) expected technical difficulties during the endoscopic resection due to multidirectional growth of the tumour.1–4 In conclusion, clinicians should be aware of this extremely rare and benign entity mimicking malignancy, asthma, or COPD since early diagnosis and endoscopic resection help to prevent irreversible lung damage.
References 1 Irani F, Kumar B, Reddy P, Narwal-Chadha R, Kasmani R, Tita J. An endobronchial lipoma mimicking asthma and malignancy. Prim Care Respir J. 2010;19(3):281–3. 2 Muraoka M, Oka T, Akamine S, Nagayasu T, Iseki M, Suyama N, et al. Endobronchial lipoma: review of 64 cases reported in Japan. Chest. 2003;123(1):293–6. 3 Cao D, Sun Y, Yang S. Endobronchial lipoma: an unusual cause of bronchial obstruction. Case Report Med. 2011;2011: 1–4. 4 Gao H, Ding X, Wei D, Cheng P, Su X, Liu H, et al. Endoscopic management of benign tracheobronchial tumors. J Thorac Dis. 2011;3(4):255–61.
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