Yonago Acta medica 2008;51:21–24

Bilateral Multiple Pulmonary Sclerosing Hemangioma in aYoung Male Patient
Kenichiro Miyoshi, Hiroshige Nakamura*, Ken Miwa*, Yoshin Adachi*, ShinjiFujioka*, Yuji Taniguchi* and Yasushi Horie†
Center for Clinical Residency Program, *Division of General Thoracic Surgery and †Pathology
Division, Tottori University Hospital, Yonago 683-8504 Japan

The patient was a 24-year-old male who visited our University Hospital complaining ofchest pain, and chest computed tomography revealed multiple bilateral nodules. Thechest pain disappeared almost immediately, but the tumor underwent no changes during
the 3 years of follow-up observations. We used a thoracoscope to perform a partial lungresection of a nodule that reached a maximum diameter of 9 mm for the purpose of obtaining a definite diagnosis. From the pathological findings, the patient was diagnosed
to have pulmonary sclerosing hemangioma in which circular tumor cells lacking nuclear
atypia rose to papillary hyperplasia. The mindbomb homolog-1 positive rate (MIB-1index) of the tumor cells was less than 1%, and it is believed to have a poor proliferation
activity. Pulmonary sclerosing hemangioma is predominantly found in cases of middleaged female patients and occurs unilaterally. Cases of bilateral multiple forms in young
males are extremely rare. Some cases of enlargement, metastasis and relapse have also
been reported, so in the future, careful follow-up is required.
Key words: bilateral multiple lung nodule; sclerosing hemangioma; video-assisted thoracic surgery; young maleboth lungs. There were no differences between
the right and left nodules, but they were mostly
found in the lower lobe, and their interiors wereeven and showed no calcification. No contrast
effects were observed (Figs. 1A and B). The
chest pain disappeared almost immediately, butthere were no changes in the number or the sizeof the nodules during the 3 years of follow-up
observation. All inflammatory reactions and tumor markers showed normal ranges in the blood
examinations, and the patient tested negative
for aspergillus antigen and cryptococcal antigen
analyses. Upon obtaining the patient’s informed
consent, we performed surgery to obtain a definite diagnosis, because the patient was still young
and we were unable to rule out the possibility ofCases of pulmonary sclerosing hemangioma arecommonly found to occur unilaterally in middleaged females. In this study, we experienced an
extremely rare case of bilateral multiple pulmonary sclerosing hemangioma in a young male,
and we hereby present our results, together with anumber of bibliographical considerations.

Patient Report
The patient was a 24-year-old male. By chest
computed tomographic scan performed to address
his major complaint of chest pain, well-circumscribed multiple small circular nodules with amaximum diameter of 9 mm were observed in

Abbreviation: MIB-1 index, mindbomb homolog-1 positive rate

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K. Miyoshi et al.

BAFig. 1. Chest computed tomographic scan reveals well-circumscribed multiple small circular nodules (arrows) inboth lungs (A). They are mostly found in the lower lobe (A). A nodule with the maximum diameter of 9 mm (arrow)is observed in the right middle lobe (B).

the histopathological findings, the patient wasdiagnosed with pulmonary sclerosing hemangioma, because the circular tumor cells that hadpoor nuclear atypia rose to papillary hyperplasia(Figs. 3A and B). In an immunohistochemical
test, thyroid transcription factor-1 and surfactant
aproprotein A tested positive, and the mindbomb
homolog-1 positive rate (MIB-1 index) was lessthan 1%. No significant lesion has been observed,
with respect to the size and the number of the remaining tumors, 6 months after surgery.

a malignant tumor or an infectious lesion. Thoracoscopic observations identified multiple small
nodules with the same appearance that were 3 to5 mm in size immediately under the pleural membrane, so using a thoracoscope, we performed thepartial lung resection of a nodule with the largest
diameter, located in the right median lobe. The
resected tumor was 9 8 mm in size with a flat,smooth surface and a solid interior (Fig. 2). FromDiscussion
Pulmonary sclerosing hemangioma was firstreported by Liebow and Hubble in 1956. The
frequency of the occurrence thereof accounted
for approximately 1% of all pulmonary tumors,
following hamartoma among the benign tumors,
but more than 80% of all cases occur in female
patients who are mostly in their 40s (Iyoda et al.,2004). The majority of these cases are asymptomatic, but coughing, chest pain and bloody
sputum are also observed in symptomatic cases(Kaltzenstein et al., 1983; Iyoda et al., 2004). There
are various views regarding the histogenesis such

Fig. 2. The resected tumor (arrow) in the right middle
lobe is 9 8 mm in size with a flat, smooth surface and asolid interior.

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Multiple pulmonary sclerosing hemangioma

Fig. 3. Pathological examination shows circular tumor cells with poor nuclear atypia rising to papillary hyperplasia
with hematoxylin and eosin staining of low power field (A: bar = 500 µm) and high power field (B: bar = 50 µm).

as mesothelial origin (Kaltzenstein et al., 1983),
hystiocyte origin (Nagata et al., 1985) or alveolar
epithelium origin (Yousem et al., 1988). This casealso tested positive for thyroid transcription factor-1 and surfactant aproprotein A, and the findings could be defined as an alveolar epithelial tumor (Chan and Chan, 2000). The incidence rateof multiple occurrences is very low, accounting
for approximately 5% of all occurrences (Liebowand Hubble, 1956; Noguchi et al., 1986). Hypotheses regarding cases of multiple occurrences include the multicentric occurrence hypothesis andthe intrapulmonary metastasis hypothesis, but nospecific conclusion has yet been reached (Noguchiet al., 1986; Hanaoka et al., 2005). In Japan, there
have been 29 cases of multiple occurrences, ofwhich only 5 cases were of male patients (Inoueet al., 2007). This is the 6th case of a multiple occurrence found in a male in Japan, but we believethat this is the 1st case of a bilateral occurrence ina patient in his 20s. It has been reported duringfollow-up after a resection that pulmonary sclerosing hemangioma can occasionally lead to therapid enlargement and metastasis of lymph nodes
and relapse (Tanaka et al.,1986; Katakura et al.,2005). Jungraithmayr et al. (2006) also describedthat sclerosing hemangioma of the lung is a benign tumor with potential for malignancy. The
MIB-1 index of the tumor cells in this case wasless than 1%, and it is believed to have a low proliferation activity (Filiz et al., 2007). However,this was a case of early onset: we therefore needto perform careful follow-up examinations in thefuture.
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Received December 21, 2007; accepted January 11, 2008
Corresponding author: Hiroshige Nakamura, MD24