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DOI:10.2214/AJR.05.0827
AJR 2007; 189:W26-W28
© American Roentgen Ray Society

Case Report

Pulmonary Sclerosing Hemangioma Manifesting as a Nodule with Irregular Air Clefts on High-Resolution CT

Hiroshi Takatani1, Kazuto Ashizawa2, Kiyoko Kawai3 and Shigeru Kohno4

1 Department of Internal Medicine, Nagasaki Municipal Hospital, Nagasaki 852-8555, Japan.
2 Department of Radiology and Radiation Oncology, Nagasaki University Graduate School of Biomedical Science and Nagasaki University Hospital, 1-7-1 Sakamoto, Nagasaki 852-8501, Japan.
3 Department of Pathology, Nagasaki Municipal Hospital, Nagasaki, Japan.
4 Department of Internal Medicine, Nagasaki University Hospital, Nagasaki, Japan.

Received May 16, 2005; accepted after revision July 12, 2005.

 
Address correspondence to K. Ashizawa (ashi{at}net.nagasaki-u.ac.jp).

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Keywords: CT • high-resolution CT • lung disease • lung nodules • pulmonary sclerosing hemangioma


Introduction
Top
 Introduction
Case Report
 Discussion
 References
 
Sclerosing hemangioma of the lung is a rare benign neoplasm [1, 2]. Most affected patients are asymptomatic, but some present with hemoptysis, cough, chest pain, dyspnea, and pleurisy. Because there are few findings on radiologic studies that are characteristic of sclerosing hemangioma, it is difficult to diagnose on the basis of imaging. In a few case reports, investigators have mentioned the presence of an air space around a sclerosing hemangioma on chest CT [3–9]. We report a case of pulmonary sclerosing hemangioma with irregular air spaces on high-resolution CT (HRCT) that had been growing like a ground cleft for 6 years.


Case Report
Top
 Introduction
 Case Report
Discussion
 References
 
A 42-year-old woman with hemoptysis was admitted to our hospital for further workup and management of a pulmonary mass in July 2003. The pulmonary mass was first detected on chest radiography when she underwent surgery for carcinoma of the uterine cervix 6 years earlier. The pulmonary mass had been followed up on chest radiography and CT for 6 years. She had smoked 10 cigarettes per day for 16 years but was no longer smoking cigarettes. On admission, physical and laboratory findings were unremarkable. Sputum studies for acid-fast bacilli, Aspergillus organisms, other pathogenic organisms, and malignant cells were negative.

The initial chest radiograph in 1997 revealed a solitary pulmonary mass in the left upper lung field (Fig. 1A). Chest CT performed on the same day showed a well-defined 2.8-cm mass with irregular air spaces in the left upper lobe. Contrast-enhanced CT was not performed, and CT attenuation value was not calculated. An emphysematous area and ground-glass opacity (GGO) in the surrounding lung parenchyma (Fig. 1B) were noted.


Figure 1
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  		Fig. 1A —42-year-old woman with pulmonary sclerosing hemangioma over 6-year follow-up period. Magnified view of initial chest radiograph in 1997 reveals solitary pulmonary mass in left upper lung field.

 

Figure 2
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  		Fig. 1B —42-year-old woman with pulmonary sclerosing hemangioma over 6-year follow-up period. Initial chest CT image in 1997 shows well-defined mass with irregular air spaces (arrowheads) in left upper lobe. Emphysematous change (black arrows) and ground-glass opacity (GGO) (white arrows) are also seen in surrounding lung parenchyma.

 
Follow-up studies performed in 2000 because the patient was experiencing hemoptysis showed that the mass had become slightly larger than it had been at the initial examination and that the air spaces within the mass had spread like a ground cleft (Fig. 1C). HRCT showed that the area of GGO around the mass had expanded. In 2001, a year after the first follow-up studies, the patient was asymptomatic and HRCT showed that the area of GGO had contracted.


Figure 3
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  		Fig. 1C —42-year-old woman with pulmonary sclerosing hemangioma over 6-year follow-up period. High-resolution CT (HRCT) image obtained in 2000 when patient experienced hemoptysis shows increase in area of GGO (arrows) around mass.

 
On admission in 2003 when the patient was again experiencing hemoptysis, chest radiography revealed slight enlargement of the pulmonary mass and an increase in the size of the areas showing emphysematous change and GGO in the surrounding lung parenchyma. HRCT performed on the same day showed that GGO extended to the pleural surface and that emphysematous change around the mass was more prominent (Fig. 1D). Contrast-enhanced CT was not performed during the follow-up period.


Figure 4
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  		Fig. 1D —42-year-old woman with pulmonary sclerosing hemangioma over 6-year follow-up period. HRCT image obtained at admission in 2003 when patient was again experiencing hemoptysis shows air spaces (arrowheads) within mass have spread like ground cleft, area of GGO (white arrows) extends to pleural surface, and emphysematous change (black arrows) around mass is more marked.

 
Fiberoptic bronchoscopy and transbronchial biopsy were performed, but findings for a definitive diagnosis were not obtained. The presumed diagnoses before surgery were aspergilloma and lung cancer.

The patient underwent partial segmentectomy of the left upper lobe. Pathologic examination revealed a gray-red solitary tumor, measuring 3.3 x 3.4 cm, that was located in the lung parenchyma. The tumor was well circumscribed, but it was not encapsulated. A semicircular rim of air space was noted between the periphery of the tumor and the lung parenchyma surrounding the tumor.

Microscopically, the histopathologic specimen showed hemorrhagic and solid patterns with hyalinization. Irregularly enlarged cystic air spaces that were lined by cuboidal epithelial cells were present in the solid proliferation of epithelioid tumor cells (Fig. 1E). Hemosiderin-laden macrophages and emphysematous change with fibrosis were seen in the lung parenchyma surrounding the tumor. These findings confirmed the diagnosis of sclerosing hemangioma.


Figure 5
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  		Fig. 1E —42-year-old woman with pulmonary sclerosing hemangioma over 6-year follow-up period. Histopathologic specimen shows hemorrhagic and solid pattern with hyalinization. Irregularly enlarged cystic air spaces lined by cuboidal epithelial cells are present in solid proliferation of epithelioid tumor cells. (H and E, x100)

 


Discussion
Top
 Introduction
 Case Report
 Discussion
References
 
Pulmonary sclerosing hemangioma is a rare benign tumor. It was first described by Liebow and Hubbell in 1956 [1]. It occurs predominantly in young or middle-aged women. Pathologically, it is composed of four major histologic components—namely, solid, papillary, sclerotic, and hemangiomatous components. On the basis of ultrastructural and immunohistochemical findings, it is considered to be a proliferation of type II pneumocytes [2].

Radiologically, the tumor usually presents as a well-defined homogeneous mass on chest radiography and CT and frequently manifests as a markedly contrast-enhancing juxtapleural mass with or without areas of low attenuation and calcification on contrast-enhanced CT [3]. Because there are few characteristic features of pulmonary sclerosing hemangioma on radiologic studies, it is difficult to diagnose preoperatively. The "air meniscus" sign, or the presence of an air-trapping zone, is one of the characteristics of this tumor.

In a few case reports, researchers have described interesting CT findings [4–6]. In these reports, investigators proposed possible mechanisms of air-space development. These include, first, proliferation and hyalinization of undifferentiated alveolar mesenchymal cells that wrap around the bronchus and lead to distention of the distal air space [4]; second, contraction of the capsule and tumor, particularly when the tumor is highly cellular and hemorrhagic [4]; third, peritumoral hemorrhage followed by communication with the airway [5, 7]; and, fourth, peritumoral hemorrhage and tumor contraction [8].

The distinguishing characteristics of pulmonary sclerosing hemangioma in our patient are air spaces within the tumor and emphysematous change and GGO around the tumor. Over a 6-year follow-up period, the air spaces had slowly spread like a ground cleft. The area of GGO increased when the patient experienced hemoptysis and decreased when she was asymptomatic. The size of the area showing emphysematous change had also gradually increased. Sakamoto et al. [9] reported that a "check-valve" effect is an important mechanism in the formation of peritumoral emphysematous change.

Although we did not recognize the communication between the tumor and bronchus in our patient, we hypothesize that repeated hemorrhage from the tumor in surrounding lung parenchyma resulted in the formation of irregular air spaces. The presence of many hemosiderin-laden macrophages in the air spaces suggests that peritumoral bleeding had occurred. We also hypothesize that fibrous change due to peritumoral hemorrhage made a check-valve mechanism that led to an increase in the size of the area showing emphysematous change.

The differential diagnosis for the air meniscus sign includes infectious diseases, such as aspergilloma, tuberculoma, echinococcal cyst, and lung abscess, and neoplastic diseases, such as lung cancer and hamartoma. A tuberculous cavity with a Rasmussen aneurysm is also considered. In our patient, however, the so-called "air meniscus sign" and air spaces within the tumor were seen on HRCT. Emphysematous change and GGO around the tumor were also seen. Although these findings are not common, knowledge of them is useful to correctly diagnose pulmonary sclerosing hemangioma.


Acknowledgments
 
We thank Keiji Inoue, Kazunori Minami, Yuji Ishimatsu, and Yasumasa Doutsu for their useful work and discussions, and we thank Rashid Hashmi for his assistance in the preparation of this manuscript.


References
Top
 Introduction
 Case Report
 Discussion
 References
 

  1. Liebow AA, Hubbell DS. Sclerosing hemangioma (histiocytoma, xanthoma) of the lung. Cancer 1956;9 : 53-75[CrossRef][Medline]
  2. Nagata N, Dairaku M, Ishida T, Sueishi K, Tanaka K. Sclerosing hemangioma of the lung: immunohistochemical characterization of its origin as related to surfactant apoprotein. Cancer1985; 55:116 -123[CrossRef][Medline]
  3. Im JG, Kim WH, Han MC, et al. Sclerosing hemangioma of the lung and interlobar fissures: CT findings. J Comput Assist Tomogr 1994; 18:34 -38[Medline]
  4. Bahk YW, Shinn KS, Choi BS. The air meniscus sign in sclerosing hemangioma of the lung. Radiology 1978;128 : 27-29[Abstract]
  5. Nam JE, Ryu YH, Cho SH, et al. Air-trapping zone surrounding sclerosing hemangioma of the lung. J Comput Assist Tomogr 2002; 26:358 -361[CrossRef][Medline]
  6. Cheung YC, Ng SH, Chang JW, Tan CF, Huang SE, Yu CT. Histopathological and CT features of pulmonary sclerosing hemangiomas. Clin Radiol 2003;58 : 630-635[CrossRef][Medline]
  7. Sagara Y, Hayashi K, Shiraishi Y, et al. The pulmonary air meniscus sign in a case of sclerosing hemangioma [in Japanese]. Nihon Kyobu Shikkan Gakkai Zasshi 1994;32 : 774-777[Medline]
  8. Matsuyama W, Hirotsu Y, Mizoguchi A, Iwami F, Kawabata M, Osame M. Pulmonary sclerosing hemangioma with specific CT findings [in Japanese]. Nihon Kokyuki Gakkai Zasshi 1998;36 : 564-567[Medline]
  9. Sakamoto I, Tomiyama N, Sugita A, Miyagawa M. A case of sclerosing hemangioma surrounded by emphysematous change. Radiat Med 2004; 22:123 -125[Medline]

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