Ann Thorac Surg 2009;88:300-302. doi:10.1016/j.athoracsur.2008.09.067
© 2009 The Society of Thoracic Surgeons
Case Reports
Bronchial Angiolipoma
Lei Jiang, MD,
Yan-li Wang, MD,
Yi-ming Zhou, MD,
Bo-xiong Xie, MD,
Lü Wang, MD,
Jia-an Ding, MD,
Ge-ning Jiang, MD*
Departments of Thoracic Surgery and Pathology, Shanghai Pulmonary Hospital, Shanghai Tongji University, Shanghai, China
Accepted for publication September 22, 2008.
* Address correspondence to Dr Ge-ning Jiang, Department of Thoracic Surgery, Shanghai Pulmonary Hospital, No. 507 Zhengming Rd, Shanghai, 200433, China (Email: jgnwp{at}yahoo.com.cn).
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Abstract
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Angiolipoma occurs preferentially in the extremities and trunk. We present a patient with involvement of the bronchus and describe successful localized resection of the lesion.
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Introduction
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Angiolipoma in the thorax has been infrequently described in the English literature. Kline and colleagues [1] reported the first angiolipoma of the mediastinum in 1990, which was successfully treated with surgical resection. We present a patient with a bronchial angiolipoma.
A 72-year-old man was referred to our department for recurrent dry cough and right-sided obstructive pneumonia. A year before this admission, when he underwent laryngectomy for laryngeal carcinoma, an endobronchial ovoid lesion had already been discovered by bronchoscopy. A thoracic computed tomography (CT) scan had also demonstrated a well-circumscribed nodule (–84.8 HU) which was located in the bronchus intermedius and resulted in partial obstructive atelectasis of the right lower lobe. The patient refused surgical intervention at that time.
A follow-up CT scan 1 year later noted no apparent increase in size (Fig 1). Repeat bronchoscopy revealed a smooth nodule arising from the bronchus intermedius just distal to the right upper lobe bronchus orifice. When the patient coughed, it moved up-and-down in the bronchial lumen (Fig 2). An adequate biopsy specimen could not be obtained because of the nodule's smooth and hard surface. A 18-fluorodeoxyglucose positron emission tomographic whole body scan was negative for the lesion and showed no other suspicious uptake.
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Fig 1. (A) Computed tomographic (CT) scan of the chest on March 9, 2006, demonstrates a nodular opacity in the bronchus intermedius partially covering the opening of the upper bronchus. (B) A CT scan on March 28, 2007, shows the lesion had nearly no apparent changes compared with the prior size.
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Fig 2. (A) Bronchoscopy reveals an ovoid lesion in the bronchus intermedius and (B) the upper lobar orifice is not affected. The arrow indicates the part of the bronchus the pedicle was based on. (RMB = right mainstem bronchus; RUB = right upper bronchus; T = tumor.)
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A bronchial segmental resection of the bronchus intermedius for resection of the lesion was performed through a posterolateral thoracotomy under general anesthesia. The intermediate bronchus was opened transversely just beyond the right upper lobe bronchus. The endobronchial tumor, measuring 2.0 x 1.5 x1.5 cm, was exposed. This well-demarcated, yellowish homogenous tumor was firmly attached to the cartilaginous portion of the bronchus. A 1-cm-long segment of the bronchus intermedius was excised to completely resect the tumor. The periphery of the bronchus intermedius was patent.
After frozen section examination confirmed benignity of the tumor and the absence of a microscopically positive margin, restoration of airway continuity was accomplished by the bronchial end-to-end anastomosis using interrupted 4-0 absorbable suture (Ethicon Inc, Somerville, NJ).
Pathologic examination of excised tumor confirmed the diagnosis of angiolipoma; the pathologic findings showed an encapsuled nodule that was composed of mature fat cells, numerous blood vessels mostly located in the tumor periphery, and marked microthrombosis (Fig 3A). Immunohistochemical stains were positive for S-100, vimentin, and CD34, but were negative for desmin, Ki-67, and p53 (Fig 3B).
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Fig 3. (A) At light microscopy, the tumor was composed of mature fat cells and numerous blood vessels mostly located in the tumor periphery (hematoxylin and eosin stain, original magnification x 200). (B) Blood vessel component of the tumor was positive for CD34 (Envision, original magnification x 400).
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The patient had an uneventful recovery and was discharged on postoperative day 9. At the last follow-up 14 months later, no evidence of recurrence was observed, and the atelectatic lower lobe had totally reexpanded.
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Comment
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Angiolipoma is characterized by lobules of mature lipocytes and densely distributed networks of small and larger blood vessels. It commonly occurs in the subcutaneous tissues of the extremities and trunk in young patients, and other reported locations include the larynx, rectum, esophagus, obturator canal, and spine [2–6]. Very few cases of pulmonary angiomyolipomas that possess the three histopathologic components of adipose tissue, smooth muscle, and blood vessels have previously been reported [7, 8]. The bronchial angiolipoma in this patient was located in the bronchus intermedius.
In summary, this elderly man had recurrent symptoms of respiratory infections, and the presence of a significantly slow-growing endobronchial lesion with fat-like density had been demonstrated by serial thoracic CT scans. From operative view, an ovoid lesion with smooth surfaces but a hard texture was held by a pedicle with its base located on the internal surface of the bronchus intermedius, and the surrounding tissues were not affected. At light microscopy, the component of blood vessels was most peripherally located with prevalent intraluminal formation of hyaline thrombus, whereas the central area of the tumor was predominantly composed of mature lipocytes.
Even though one successful case is not enough to validate an optimal treatment modality, the use of complete surgical resection of the tumor could prove to be a curative treatment of choice and be advisable if the patient can tolerate the procedure. We suggest that bronchoscopic approach should be reserved for those high-risk patients in consideration with a potential of recurrence.
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References
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- Kline ME, Patel BU, Agosti SJ. Noninfiltrating angiolipoma of the mediastinum Radiology 1990;175:737-738.[Abstract/Free Full Text]
- Mesolella M, Di Martino M, Laguardia M, et al. Angiolipoma of the larynx Otolaryngol Head Neck Surg 2007;136:142-143.[Medline]
- Ishizuka M, Nagata H, Takagi K, et al. Rectal angiolipoma diagnosed after surgical resection: a case report World J Gastroenterol 2007;13:467-469.[Medline]
- Jensen EH, Klapman JB, Kelley ST. Angiolipoma of the esophagus: a rare clinical dilemma Dis Esophagus 2006;19:203-207.[Medline]
- Sarikcioglu L, Demirel BM, Ozsoy U, et al. Angiolipoma located inside the obturator canal and supplied by the umbilical artery Ann Anat 2007;189:75-78.[Medline]
- Konya D, Ozgen S, Kurtkaya O, et al. Lumbar spinal angiolipoma: case report and review of the literature Eur Spine J 2006;15:1025-1028.[Medline]
- Marcheix B, Brouchet L, Lamarche Y, et al. Pulmonary angiomyolipoma Ann Thorac Surg 2006;82:1504-1506.[Abstract/Free Full Text]
- Kasuno K, Ueda S, Tanaka A, et al. Pulmonary angiomyolipoma recurring 26 years after nephrectomy for angiomyolipoma: benign clinical course Clin Nephrol 2004;62:469-472.[Medline]
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Abstract
Introduction
