Asian J Androl 2007; 9 ( ): –
Tel: +86-21-5492-2824; Fax: +86-21-5492-2825; Shanghai, China
© 2007, Asian Journal of Andrology, Shanghai Institute of Materia Medica, Chinese Academy of Sciences. All rights reserved.
Scrotal aggressive angiomyxoma mimicking inguinal hernia
Chia-Chang Wu1,4, Stephen Shei-Dei Yang1,3, Daniel T. H. Chin2, Cheng-Hsing Hsieh1, Yu-Mei Hsueh4,
1Department of Urology, 2 Department of Pathology, Buddhist Tzu Chi General Hospital, Taipei Branch, Taiwan, China
3Medical College of Tzu Chi University, Hualian, Taiwan, China
4Department of Public Health, School of Medicine, Taipei Medical University, Taipei, Taiwan, China
. . Letters to the Editor . .
ww w.asiaandro.c om
I am Yao-Chou Tsai, from Department of Urology,
Buddhist Tzu Chi General Hospital, Taipei Branch, Taipei,
Taiwan, China. I write to you to present a case report of
scrotal aggressive angiomyoma.
Aggressive angiomyxoma (AAM) is a rare, locally
infiltrative mesenchymal benign neoplasm with a predi-
lection for involving the female pelvis, perineum, vagina
and cervix. Its occurrence in men is much less frequent
and only scattered case reports have been reported. To
our knowledge, only 43 cases of AAM occurring in men
have been reported in the English-language literature .
AAM in the male inguinal area can present as an inguinal
mass that is usually diagnosed and treated as inguinal
hernia [2, 3]. Herein, we report an unusual case of a 40-
year-old man with an AAM involving the scrotum, who
underwent a herniorrhaphy at a prior hospital. The AAM
was not removed. To our knowledge, this is the first
documented case in which scrotal AAM was missed
during surgical exploration for inguinal hernia.
A 40-year-old man complained of a right scrotal mass,
which had grown slowly over the previous10 years. No
local pain or urinary tract symptoms were noted. The
scrotal mass was initially diagnosed as an inguinal hernia
and a herniorrhaphy was performed at a prior hospital.
However, the scrotal mass still remained in situ after the
operation. Therefore, he came to our hospital for fur-
On physical examination, the right scrotal mass was
elastic, non-tender, non-transilluminated and not reducible.
Scrotal ultrasonography revealed a homogeneous,
hypoechoic, extratesticular, extraepididymal, well-demar-
cated mass lesion. No calcification was present. Pelvic
computed tomography (CT) demonstrated a 5.0 × 5.1 ×
5.4 cm heterogeneous mass in the right scrotum with scro-
tal wall thickening. The testicle was displaced cephaladly.
Chest X-ray radiograph revealed no significant abnormal
finding. Laboratory studies were normal for blood count,
urine analysis, renal function, liver function, serum alpha-
fetoprotein, beta-human chorionic gonadotropin and lac-
tic dehydrogenase. The clinical impression was a
paratesticular tumor and a wide excision was planned.
In surgery, an incision through a previous hernior-
rhaphy wound was done and a large tumor was found
within the spermatic fascia (Figure 1). The boundary of
the tumor was well circumscribed and away from the
right testicle and epididymis (Figure 2). The cut surface
of the tumor was yellowish and gelatinous, and no ne-
crosis or cystic changes were present. The frozen sec-
tion of the tumor showed a loose myxoid and fibrous
background containing blood vessels of variable caliber.
Correspondence to: Dr Yao-Chou Tsai, Department of Urology, Buddhist Tzu Chi General Hospital, Taipei Branch, 289 Jianguo Road,
Xindian city, Taipei, Taiwan, China.
Tel: +886-2-6628-9779 ext. 2238 Fax: +886-2-6628-9009 E-mail: email@example.com
Received 2007-01-04 Accepted 2007-03-10
Scrotal aggressive angiomyxoma
The evenly distributed stromal cells were wavy, spindle
or stellate in shape and had delicate cytoplasmic processes.
Mitoses and cellular pleomorphism were lacking
(Figure 3). The findings are consistent with AAM. The
tumor was excised completely and the patient was dis-
charged on the first postoperative day. The patient has
remained free of local recurrence for 12 months since
Since Steeper and Rosai  first described nine cases
of aggressive angiomyxoma in female adults in 1983, its
incidence has increased year by year. Male AAM tends
to be an asymptomatic round mass lesion that is some-
times initially diagnosed as an inguinal hernia. Previously
reported AAM that have been found incidentally during
herniorrhaphy have been resected smoothly with satis-
factory postoperative results [2, 3]. However, in clinical
practice, it is possible that a scrotal AAM is missed if a
concurrent inguinal hernia exists. To avoid this, detailed
preoperative physical examination and confirmative tu-
mor removal is necessary.
Preoperative imaging studies do not always provide
a correct diagnosis because the tumor usually shows an
invasive and hypervascular picture that makes the tumor
seem malignant . Gray-scale ultrasonography de-
monstrates a hypoechoic, homogeneous and well-demar-
cated mass without significant flow using color Doppler
ultrasonography. On CT, AAM is hypodense relative to
muscle and hypoattenuating with specific swirling inter-
nal architecture on enhanced scans. The tumor has high
signal intensity using T2-weighted magnetic resonance
imaging. The swirling internal architecture of lower in-
tensity after intravenous contrast enhancement is also
seen within the high signal tumor.
Grossly, AAM is yellowish in color and usually well
demarcated and encapsulated. The cut surface demon-
Figure 3. Microscopically, various sized vessels (arrow) with
spindled or stellate-shaped tumor cells were scattered in the loose
myxoid stroma. Scale bar = 50 µm (H&E, ×200).
Figure 2. Gross findings during operation. Thick arrow indicated
AAM that was separated from right testicle and epididymis (thin
Figure 1. The tumor was well enclosed with right testis by a fascia
continued from spermatic cord.
Asian J Androl 2007; 9 ( ): – Download full-text
Tel: +86-21-5492-2824; Fax: +86-21-5492-2825; Shanghai, China
strates gelatinous and glistening material with delicate,
white, fibrous strands. Microscopically, AAM demon-
strates stellate and spindle-shaped cells in a loose myxoid
stroma without mitosis or nuclear atypia. Vascularization
is abundant, and small to large thick-walled vessels to-
gether with collagen fibrils are scattered in the hypocellular
Aggressive angiomyxoma should be distinguished from
other benign and low local recurrent potential lesions such
as intramuscular myxoma, myxoid neurofibroma, myxoid
or spindle cell lipoma, superficial angiomyxoma and
angiomyofibroblastoma. The malignant tumors with
widespread metastasis potential should also be consid-
ered a differential diagnosis (the myxoid variants of
liposarcoma, malignant fibrous histiocystoma and em-
bryonal/botryoid rhabdomyosarcoma) .
Characteristically, AAM is locally aggressive and in-
filtrative without metastasizing potential, with the excep-
tion of two cases of death reported by Blandamura et al.
 and Siassi et al. . Its local recurrence rate is ap-
proximately 37% and average time of recurrence is
9 months to 14 years postoperatively [2, 4].
In conclusion, AAM should be considered when an
usually soft tissue mass is found during physical examina-
tion or surgical correction for inguinal hernia. Because of
its highly recurrent characteristics, wide excision of the
tumor and strict postoperative follow up is recommended.
1Idrees MT, Hoch BL, Wang BY, Unger PD. Aggressive
angiomyxoma of male genital region. Report of 4 cases with
immunohistochemical evaluation including hormone receptor
status. Ann Diagn Pathol 2006; 10: 197–204.
Murakami S, Ishiga N, Sho T, Sakai K, Fujii Y, Yamamoto Y,
et al. Aggressive angiomyxoma occurring in the scrotum: re-
port of a case. Surg Today 1996; 26: 289–91.
Clatch RJ, Drake WK, Gonzalez JG. Aggressive angiomyxoma
in men. A report of two cases associated with inguinal hernias.
Arch Pathol Lab Med 1993; 117: 911–3.
Steeper TA, Rosai J. Aggressive angiomyxoma of the female
pelvis and perineum. Report of nine cases of a distinctive type
of gynecologic soft-tissue neoplasm. Am J Surg Pathol 1983;
Chihara Y, Fujimoto K, Takada S, Hirayama A, Cho M, Yoshida
K, et al. Aggressive angiomyxoma in the scrotum expressing
androgen and progesterone receptors. Int J Urol 2003; 10:
Iezzoni JC, Fechner RE, Wong LS, Rosai J. Aggressive
angiomyxoma in males. A report of four cases. Am J Clin
Pathol 1995; 104: 391–6.
Blandamura S, Cruz J, Faure Vergara L, Machado Puerto I,
Ninfo V. Aggressive angiomyxoma: a second case of metasta-
sis with patient’s death. Hum Pathol 2003; 34: 1072–4.
Siassi RM, Papadopoulos T, Matzel KE, Siassi RM,
Papadopoulos T, Matzel KE. Metastasizing aggressive
angiomyxoma. N Eng J Med 1999; 341: 1772.
Edited by Sae-Chul Kim