European Journal of Radiology Extra 61 (2007) 15–18
Alveolar soft part sarcoma of the sphincter ani externus:
Control of vaginal bleeding by recurrent embolisations
Michael Grundbichler
a
, Andreas Leithner
b,*
, Horst Rupert Portugaller
c
,
Markus Hochegger
b
, Reinhard Windhager
b
a
Institute of Physiology, Paracelsus Private Medical University, Salzburg, Austria
b
Department of Orthopedic Surgery, Medical University Graz, Auenbruggerplatz 5-7, A-8036 Graz, Austria
c
Department of Radiology, Medical University Graz, Graz, Austria
Received 3 July 2006; received in revised form 15 October 2006; accepted 16 October 2006
Abstract
Alveolar soft part sarcoma is a rare malignancy mainly localized in the deep soft tissue of the extremities. This is the first report of an
alveolar soft part sarcoma originating from the sphincter ani externus. A 27-year-old pregnant woman presented with a tumour located in
the right ischiorectal fossa which had rapidly enlarged during pregnancy 6 years after first detection. Admitted to our hospital, she presented
with a continuous strong vaginal bleeding, multiple pulmonal metastases and an inoperable local tumour mass. Palliative chemotherapy was
unsuccessful, control of local bleeding was only possible with multiple embolisations. The patient died 43 months after initial diagnosis. Our
intention is to point out the remarkable origin, a rapid tumour growth during pregnancy, and the possibility of controlling massive tumour
bleeding by embolisation.
© 2006 Elsevier Ireland Ltd. All rights reserved.
Keywords: Alveolar soft part sarcoma; Pregnancy; Embolisation
1. Introduction
Alveolar soft part sarcoma is an uncommon soft tissue
tumour. It was first named as an own entity by Christopherson
et al. [1] in 1952. It occurs at any age but is most com-
mon between 15 and 35 years with a female predominance
before the age of 30 [2]. It is a slow growing, highly vas-
cularized, usually painless and therefore easily overlooked
tumour [2–5]. It is malignant and there is no benign coun-
terpart [5]. In adults the typical localisation is in the deep
soft tissue of the extremities particularly in the anterior thigh
[2,4,6–8]. Early metastasis mainly to the lung or brain is a
characteristic feature [2,3,6,8]. To our knowledge this is the
first report of an alveolar soft part sarcoma originating from
the sphincter ani externus.
*
Corresponding author. Tel.: +43 316 385 81899; fax: +43 316 385 4806.
E-mail address: andreas.leithner@meduni-graz.at (A. Leithner).
2. Case report
In 1992, a 21-year-old woman first recognized a small
paraanal knot which she did not care about. Six years later
in November 1998, the knot had suddenly enlarged dur-
ing pregnancy. The tumour located in the right ischiorectal
fossa was diagnosed by chance by a local gynaecologist at
an antepartum examination in the 23rd week of gravidity.
Inzisional biopsy showed a malign highly vascularized mes-
enchymal neoplasm. Preoperative sonography displayed up
to 4 mm dilated vessels filled with tumour thrombi partic-
ularly in the cranial part. A microscopically intralesional
local excision (R1) was performed at a local centre. The
tumour originated from the sphincter ani externus with a
diameter of 6 cm. Histological and immunohistochemical
findings proved an alveolar soft part sarcoma. After child-
birth by caesarean section in April 1999 X-ray presented
metastases to the lung. Subsequent a high dose chemother-
apy using Epirubicin (Farmorubicin®, Pfizer Pharma Gmbh,
1571-4675/$ – see front matter © 2006 Elsevier Ireland Ltd. All rights reserved.
doi:10.1016/j.ejrex.2006.10.008