A Lal

A Yarci

Amr A Soliman

Bassma ElSabaa

D Stutterecker

F Yildiz

G Scutiero

Hassan Sallam

HW Lin

KA Kho

MM Zaitoon

N Fasih

Noha Hassan

O Abulafia

PJ Maskery

RA Sayer

SF Cramer

Tarek Ezzat

V Poliquin

VC Buttram Jr

WD Kang

Journal of Medical Case Reports

English

PubMed

Crossref

Degenerated huge retroperitoneal leiomyoma presenting with sonographic features mimicking a large uterine leiomyoma in an infertile woman with a history of myomectomy: a case report

Springer - Publisher Connector

CASE REPORT Open Access
Degenerated huge retroperitoneal leiomyoma
presenting with sonographic features mimicking
a large uterine leiomyoma in an infertile woman
with a history of myomectomy: a case report
Amr A Soliman1*, Bassma ElSabaa2, Noha Hassan1, Hassan Sallam1 and Tarek Ezzat3,4
Abstract
Introduction: Retroperitoneal leiomyomata are rare. They are either mistaken preoperatively for malignant
retroperitoneal tumors or dealt with as cases of subserous leiomyomata that turn out intraoperatively to be huge
retroperitoneal masses of unknown nature.
Case presentation: We report the case of a 46-year-old nulligravid female patient of Middle Eastern ethnicity who
presented to our university hospital with lower abdominal as well as pelvic pain along with a bloated sensation.
She also reported noticing an unusual increase in her abdominal girth. These symptoms developed over the
previous two months. Preoperative investigation by means of an ultrasound suggested a degenerated subserous
huge uterine leiomyoma. An abdominal hysterectomy was planned. Intraoperatively, a normal sized uterus was
found, the surface of which was studded with multiple variable sized pedunculated subserous leiomyomata.
Another huge retroperitoneal soft to firm mass was found extending from her left pelvic wall to the level of her
spleen, with no connections to her uterus. The mass was excised and a histopathological examination revealed a
degenerated leiomyoma.
Conclusion: Some unusually located extra-uterine leiomyomata have been reported; retroperitoneal leiomyoma
being among them. The origin of such tumors is still obscure; a parasitic origin as well as Müllerian cell rests or
smooth muscle cells in the retroperitoneal vessels wall have been suggested. An ‘iatrogenic’ origin for such
growths is also a possible theory. The origin of uncommonly located leiomyomata is an unexplored issue that
merits more investigation.
Introduction
Retroperitoneal leiomyomata are a rare occurrence.
Kang et al. found only 46 reported cases during their
PubMed search up to October 2008 [1]. We updated
this search using the keywords ‘extra-uterine’, ‘leio-
myoma’ and ‘retroperitoneal leiomyoma’ up to Septem-
ber 2011 to find only four more reported cases in that
time period [2-5]. This rarity makes it an unexpected
incident that is either mistaken preoperatively for a ret-
roperitoneal mass which could be malignant or, as in
our case, dealt with as a huge leiomyoma that presents
intraoperatively as a huge retroperitoneal mass of
unknown nature.
Case presentation
The currently reported case is of a 46-year-old nulligra-
vid female patient, of Middle Eastern ethnicity, who pre-
sented to the outpatient Gynaecology Clinic of our
hospital one year ago. Her complaints included mild
pelvic pain requiring the intake of non-steroidal anti-
inflammatory drugs once a day two days a week at
most. She also complained of pelvic heaviness combined
with a gastrointestinal bloating sensation that required
the daily intake of antiflatulent medication, which was
not helpful in soothing the complaint. She had a past
medical history of primary infertility for 20 years. She
* Correspondence: amr_soliman@alexmed.edu.eg
1Department of Obstetrics and Gynaecology, University of Alexandria, Egypt
Full list of author information is available at the end of the article
Soliman et al. Journal of Medical Case Reports 2011, 5:578
http://www.jmedicalcasereports.com/content/5/1/578 JOURNAL OF MEDICAL
CASE REPORTS
© 2011 Soliman et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons
Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in
any medium, provided the original work is properly cited.
also had an abdominal myomectomy through a low-
transverse abdominal incision 17 years earlier. A clinical
examination revealed a huge pelvi-abdominal mass
extending up to the level of her xiphisternum. A com-
bined abdominal and vaginal ultrasound (US) revealed
the presence of multiple leiomyomata with a huge sub-
serous leiomyoma showing evidence of degeneration
and extending to the level of the left lobe of her liver
(Figure 1). There was no evidence of ureteric obstruc-
tion or renal pelvic ectasia as shown by a renal US. For
an abdominal US we used a curvilinear probe with a fre-
quency of 5 MHz, while for the transvaginal US a 7.5
MHz probe was used. The long history of the mass and
its slow growth rate constituted a low index of suspicion
of a uterine sarcoma.
No further imaging investigations were requested due
to the highly suggestive characteristics of the mass on
US; accordingly the clinical diagnosis of a huge subser-
ous uterine leiomyoma was made. An abdominal hyster-
ectomy via a lower midline incision with left
periumbilical extension was performed. Intraoperatively,
the uterus was found to be of normal size with numer-
ous variable sized pedunculated subserous leiomyomata
diffusely attached to its surface (Figure 2).
The huge degenerated leiomyoma previously deli-
neated on sonography (Figure 3) turned out to be a ret-
roperitoneal mass extending from the left side of her
pelvis through the infundibulopelvic ligament upwards
to the lower border of her spleen, with no connections
with the leiomyomata-studded uterus. The mass dis-
placed her mesosigmoid and her descending colon
medially and even the root of the mesentry was dis-
placed towards the midline. General surgeons were
involved; they dissected the mass from its retroperito-
neal vascular connections. The mass was in close proxi-
mity to her descending colon which was reflected
medially in order to gain better access to the mass. The
mass was crossing her left kidney anteriorly to the level
of her spleen. Dissection in this area was very meticu-
lous due to the proximity to the tail of her pancreas.
The hysterectomy specimen and the huge retroperito-
neal mass were sent for pathological examination. The
Figure 1 US and Doppler examination of the leiomyoma. US examination showing a heterogeneous echotexture of the mass and presence
of multiple echo free areas, Doppler blood flow indices in and around the huge leiomyoma recorded a resistance index of 0.61 and pulsatility
index of 0.96
Soliman et al. Journal of Medical Case Reports 2011, 5:578
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Figure 2 Uterine fibroids. Normal sized uterus with multiple subserous fibroids.
Figure 3 Retroperitoneal leiomyoma. Large 18 cm × 23 cm retroperitoneal fibroid completely separable from the uterine fibroids. (A)
Intraoperatively and (B) after resection.
Soliman et al. Journal of Medical Case Reports 2011, 5:578
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postoperative course of our patient was very smooth and
she was safely discharged on the fourth postoperative
day. She returned ten days later for wound care which
showed very good healing.
The histopathologic examination revealed a non infil-
trative growth with scant mitotic activity (one mitotic
figure per 10 high power field) with no atypia, thus con-
firming the benign leiomyomatous nature of this huge
retroperitoneal growth, with evidence of hyaline degen-
eration (Figure 4)
Discussion
Uterine leiomyoma is the most common benign gyneco-
logical tumor affecting as many as 25% of women in the
reproductive age group [6], and is present in about 80%
of all hysterectomy specimens [7]. In addition to the tra-
ditional patterns of leiomyomatous growth in the uterus,
some unusual extra-uterine growth presentations are
mentioned in the literature; benign metastasizing leio-
myoma, disseminated peritoneal leiomyomatosis, intrave-
nous leiomyomatosis, parasitic leiomyomata and
retroperitoneal growth [8]. The incidence of retroperito-
neal leiomyomata is quite low, and it is even lower for
those extending to or originating in the abdomen. Of the
reported retroperitoneal leiomyomata, 73% are located in
the pelvis [9]. Most of the published case reports diag-
nosed the cases clinically as retroperitoneal growths with
high suspicion of malignancy without suspecting their
leiomyomatous nature [10-15]. The origin of such
tumors is a puzzling issue with much scientific debate.
Poliquin and coworkers observed a 40% association of
retroperitoneal leiomyomas with uterine counterparts or
a history of hysterectomy due to uterine leiomyomata [9].
Zaitoon suggested the parasitic theory for such tumor
growth [10] while Stutterecker et al. claimed that Müller-
ian cell rests or smooth muscle cells in the retroperito-
neal vessels wall are the putative origin [12]. Kho and
Nezhat proposed an ‘iatrogenic’ origin for such growths
while analyzing a case series of extra-uterine leiomyo-
mata, mostly of retroperitoneal or intraperitoneal loca-
tion with no visible connection to the uterus. They found
out that 83% of their case series had previous abdominal
operations, and 67% had myomectomies, most of them
via laparoscopy with morcellation [16]. In our reported
case, although the iatrogenic theory for such leiomyo-
mata could explain the growth of the numerous pedun-
culated subserosal leiomyomata scattered on the uterine
surface (Figure 2), it cannot explain the growth of the ret-
roperitoneal mass which was completely separable from
the uterus and seated deep in the posterior abdominal
wall peritoneum. The concept suggested by Stutterecker
et al. is the more accepted explanation for such a location
for the growth of a leiomyoma [12].
Conclusion
Thorough radiographic imaging of sonographically diag-
nosed leiomyomata is important, especially for those
which are large in size or present in an uncommon
location. Several theories have been postulated regarding
the origin of retroperitoneal leiomyomata; however, the
exact etiology is still an unexplored issue that merits
more investigation.
Consent
Written informed consent was obtained from the patient
for publication of this case report and any accompanying
Figure 4 Histological examination of the leiomyoma. The retroperitoneal mass showed a whorled (fascicular) pattern of smooth muscle
bundles separated by well vascularized connective tissue; smooth muscle cells were elongated with eosinophilic or occasional fibrillar cytoplasm
and distinct cell membranes. (A) The growth was not infiltrative and contained thick walled arteries throughout and cleft-like spaces. (B) Areas
of hyaline degeneration were also seen.
Soliman et al. Journal of Medical Case Reports 2011, 5:578
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images. A copy of the written consent is available for
review by the Editor in-Chief of this journal.
Author details
1Department of Obstetrics and Gynaecology, University of Alexandria, Egypt.
2Department of Pathology, University of Alexandria, Egypt. 3Department of
Surgery, University of Alexandria, Egypt. 4Division of Surgery and
Interventional Science, University College London, London, UK.
Authors’ contributions
AS wrote the manuscript and was directly involved in the patient care. BE
performed the histological examination, diagnosis and the photo acquisition.
NH was directly involved in the patient care and contributed to the review
of the literature. HS was involved in the manuscript idea and drafting. TE
provided general surgical input and critical reading of the manuscript. All
authors read and approved the final manuscript.
Competing interests
The authors declare that they have no competing interests.
Received: 23 July 2011 Accepted: 16 December 2011
Published: 16 December 2011
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doi:10.1186/1752-1947-5-578
Cite this article as: Soliman et al.: Degenerated huge retroperitoneal
leiomyoma presenting with sonographic features mimicking a large
uterine leiomyoma in an infertile woman with a history of
myomectomy: a case report. Journal of Medical Case Reports 2011 5:578.
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Soliman et al. Journal of Medical Case Reports 2011, 5:578
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Amundsen CL: Giant pelvic retroperitoneal leiomyoma arising from the rectal wall. Obstet Gynecol

Benign metastasizing leiomyoma of external iliac vessels: an unusual case report.

Cheong ML: Pelvic retroperitoneal leiomyoma.

Guldur ME: A rare cause of difficulty with sexual intercourse: large retroperitoneal leiomyoma.

HS: Three isolated, large retroperitoneal leiomyomas. Arch Gynecol Obstet

Khandelwal N: A huge renal capsular leiomyoma mimicking retroperitoneal sarcoma.

Leiomyoma of the space of Retzius: a report of 2 cases.

Leiomyomas beyond the uterus: unusual locations, rare manifestations. Radiographics

Parasitic myomas. Obstet Gynecol

Patel A: The frequency of uterine leiomyomas.

Reiter RC: Uterine leiomyomata: etiology, symptomatology, and management. Fertil Steril

Retroperitoneal leiomyomatosis following hysterectomy. Proc R Soc Med

Retroperitoneal parasitic leiomyoma causing unilateral ureteral obstruction.

Ultrasonographic and magnetic resonance imaging findings of a large asymptomatic retroperitoneal pelvic leiomyoma.

Vaginal vault leiomyoma: 25 years after total abdominal hysterectomy.

Vilos GA: Epidemiology, presentation, and management of retroperitoneal leiomyomata: systematic literature review and case report.

http://www.pubmedcentral.nih.gov/articlerender.fcgi?artid=3259091

Degenerated huge retroperitoneal leiomyoma presenting with sonographic features mimicking a large uterine leiomyoma in an infertile woman with a history of myomectomy: a case report

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