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Case Report
Respiration 1994;61:109-112
Masahiro Nomura''
Yutaka Nakayaa
Ken Saito*
Hirokazu Miyoshi“
Fumiko Kixhia
Shingo Hibinoa
Tetsuya Saijyoa
Susumu Ito"
Koji Nakagawab
Hideki Nakanishib
Hiroaki Nagaeh
Noriyuki Todab
Shinji Tanaka*1
Hiroshi Hciradab
Kazuya Matsumotob
Tadashi Hasegawac
Hemopneumothorax Secondary to
Multiple Cavitary Metastasis in
Angiosarcoma of the Scalp
Second Department o f Internal Medicine,
Department o f Dermatology and
First Department o f Pathology,
School o f Medicine. The University o f
Tokushima, Japan
Abstract
Key Words
Angiosarcoma
Lung tumor
Cavitary metastasis
Hemopneumothorax
We report a case o f hemopneumothorax secondary to multiple cavitary
metastasis in the angiosarcoma o f the scalp in an 86-year-old woman, who
died o f respiratory failure. At autopsy, multiple cavities were found in both
lungs. Histologic specimen o f the cavitary metastasis of the lung showed that
tumor cells proliferated forming several tubular spaces and these tubular spac­
es seemed to communicate with the central cyst. These findings suggested
that imperfect vessel-like structures of the cavitary metastasis are likely to
break down and finally grow up to large thin-walled cavities.
Angiosarcoma is a relatively rare malignant tumor
originating in vessels, and its prognosis is reported to be
extremely poor 11-3]. An angiosarcoma is frequently ac­
companied with pulmonary metastasis, but thin-walled
cavernous pulmonary metastases are extremely rare |4],
We report here an autopsy case with angiosarcoma o f the
scalp, who died of respiratory failure caused by hemo­
pneumothorax due to pleural infiltration o f angiosarcoma
and metastases to the pulmonary cavity.
Case Report
An 86-year-okl woman was admitted to the hospital because of
erythematous nodule on her forehead in 1988. Histological exami­
nation disclosed angiosarcoma o f the right scalp. Tumor was ex-
Received:
March I. 1993
Accepted alter revision
October I. 1993
eised and skin was grafted. An X-ray film o f the chest showed no
cystic pulmonary disease at the time o f operation. On March 1991.
erythematous tumor reappeared near the operative scar and tumor
was excised again. On November 1991. she was hospitalized in our
department because o f severe dyspnea. X-ray film o f the chest
showed multiple thin-walled cavitary lesions and pleural effusion in
both sides (fig. 1). Body temperature was 36.5 ° C . blood pressure
was 110/70 mm H g. heart rate was 90 per min. Heart sound was
clear but the breath sounds were decreased bilaterally. Surfacial
lymph node could not be palpated. Chest C T scan image showed
multiple cavities and hydropneumothorax. Thoracic drainage and
continuous aspiration were performed immediately al ter admission.
The pleural effusion was bloody and cloudy and its specific gravity
was 1.032. The number o f cells was 7.680/3. protein 3.589 mg/dl.
glucose 109 mg/dl. and LD H 1,095 IU/I. Malignant cells were not
observed, but tumor markers (C'EA and CA125) showed higher
values, suspicious o f pleural infiltration o f angiosarcoma. After
both lungs were expanded by the continuous thoracic drainage,
pleural adhesion (OK-432) was carried out. but air leak could not
be prevented. She died o f respiratory failure on December 26. 1991.
Masahiro Nomura. M l)
O 1994 S. Knrger A G . Basel
The Second Department o f Internal Medicine
School of Medicine
Tlie University o f Tokushima
(X)25- 7931/94/0612-0109
$ 5.00/0
2-50 Kuramolo cho. Tokushima. T70 (Japan)
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Introduction
Biopsy Findings o f the Primary Head Skin Lesion at the
Operation
Extended proliferation o f intradennal tumor with bleeding was
observed along with ulceration o f the head skin. In the corium.
epithelioid tumor cells contained abundant alveoli diffusively prop-
agated. forming irregular blood vessels. Heterotypic mitosis was
frequently encountered.
A ntopsy Findings
In the head skin, malignant cells o f angiosarcoma W'ere not
found. Cystic lesions were found in both lungs (fig. 2). Several
metastatic lesions could be observed in upper, middle and lower
right lobes and lower left lobe. In addition, congestive lesions
were found over both lungs.
Autopsy Histological Findings
Spindle cells proliferated solitary in the primary head skin le­
sion. but proliferated in slit-like shape in the metastatic lesions in
lungs (fig. 3). forming clear boundary to the surrounding normal
tissue around cystic space. In the cavities, small amounts o f exudate
tinted with blood were observed. The density o f tumor cells was
I It)
Nomura et al.
Fig. 3. Photomicrograph showing metastatic lung tumor. The
density o f tumor cells is high and they proliferated forming several
tubular spaces. These tubular spaces seem to communicate w ith the
central cyst. a x i l ) , b 660.
Cavitary Metastasis
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Fig. 1. Multiple thin-walled cavitary lesions (arrows) and
pleural effusion are seen in both sides.
Fig. 2. Cystic lesion o f the lung.
high and they proliferated forming several tubular spaces. These
tubular spaces seemed to communicate with the central cyst. On the
relatively large tubes, the tumor cells lined the wall so as to form
epithelioid endothelium. Factor V III staining o f the tumor cells
determined by PAP method was negative.
Autopsy findings were as follows: ( I ) angiosarcoma o f skin me­
tastasis. both lungs: (2) bronchopneumonia: (3) pulmonary edema:
(4) pulmonary congestion; (5) hypertrophy o f left ventricle; (6) re­
tention cysts o f kidney; (7) paraovarian cyst.
Discussion
Angiosarcoma is a malignant tumor that originates
from endothelial cells of the lymph or blood vessels, and
occurs most frequently in the head and face skin o f the
aged [5, 6|. Recurrence and lung metastasis appear at an
early stage and the prognosis is very poor [71.
This disease is so far reported as 'angiosarcoma',
hemangioendothelioma’ , ‘ malignant angioendothelioma’
or ‘ lymphangiosarcoma’ . It was difficult to determine
whether angiosarcoma originates from a blood vessel or
from a lymphatic vessel. However, in recent years, intra­
cellular Weibel-Palade’s granules [8. 9J. factor VIII in
cells by immunofluorescence technique [10] and the PAP
method have been developed as the specific markers for
endothelial cells o f blood vessels. In our case, the factor
VIII o f tumor cell by the PAP method was negative, but
this does not rule out the possibilities o f blood vessel en­
dothelium as origin 1111.
Pulmonary metastasis forming the cavity o f an angio­
sarcoma has often been reported 112. 13]. There have
been few reports o f the cases, in which hemopneumothorax due to cavitating pulmonary metastases was com­
bined with pleural infiltration and only reported by Cardozo et al. [14|. Starshak 115] and Lanese et al. [4]. It is
suggested that our case was accompanied with metastatic
pleural infiltration because of the remarkable increase in
tumor marker in spite of no malignant cells in the pleural
exudation.
The frequency o f cavity formation in primary lung
cancer is 20-30% 116. 17]. and moreover, the squamous
cell carcinoma of the head and neck frequently produces
cavitary metastasis in the lung, but that in metastatic lung
cancer is very rare. Dodd and Boyle 118] and Honma et
al. 1171 reported that its frequency is 4 and 1%, respec­
tively. The primary organs causing metastatic lung cancer
are often colon and rectum, and the mechanism o f patho­
genesis developing to the cavity is tumor necrosis [17].
The various pathogenesis o f thin-walled cavity forma­
tion were so far reported, but definite conclusions remain
unclear. Dines et al. 119] and Ohno et al. [20] reported
that the mechanism o f development of pneumothorax in
metastatic sarcoma was a rupture o f peripheral necrotic
tumor into a bronchus and the pleural space, creating
bronchopleural fistula. Moreover, they reported the other
possibility that tumor embolus with infarction o f the
lung formed the air leak. Peabody et al. [16] and Crow
and Brogdon [211have also suggested that cavitating pul­
monary metastases could arise in a lung cyst.
With regard to the mechanism o f cavity formation in
our case, we can postulate additional pathogenesis. Ne­
crotic substances did not exist in the thin-walled cavities,
and the chest radiograph at the first operation did not re­
veal any cystic lesion. The histopathological findings in
autopsy disclosed that the spindle cells proliferated solid­
ly in the primary lesion o f the head skin, but on the other
hand the slit-like proliferation was observed in the meta­
static lesion o f the lung. Although these malignant cells
have a tendency to form vessels, the lining o f these ves­
sels was incomplete. These imperfect vessel-like struc­
tures could break down, coalesce, and form the larger
thin-walled cavities observed in the study.
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