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An Unusual Case of Uterine FibroidI Adibah, K Norhayati, KC Liew
Department of Obstetrics and Gynaecology, School of Medical Sciences, Universiti Sains Malaysia, 16150 Kubang Kerian, Kelantan, Malaysia
Introduction and Objective
The case
The case continues……
Mayer-Rokitansky-Küster-Hauser (MRKH)
syndrome is an uncommon congenital anomaly,
affecting 1 in 4000 to 5000 of women. It is
characterized by vaginal aplasia with normal
external genitalia, absence of fibromuscular
remnants of the uterus with normally developed
fallopian tubes and ovaries.
The presence of solid pelvic mass in patients with
MRKH is difficult to examine, especially when no
vaginal reconstruction has been performed. An
imaging technique may provide further information
about the origin but may also be misleading. By
describing the uncommon association between
uterine fibroid and MRKH may make gynaecologist
to be aware of its possibility in dealing with pelvic
mass in MRKH cases.
A 51 year old Malay lady was referred to our centre
for further management of primary amenorrhoea. Her
previous gynaecologic history included the diagnosis
of MRKH made by another service hospital, when
she was 18 years of age. She had not undergone any
vaginal operation apart from her diagnostic
laparoscopy. She was advised to come back to the
hospital when she got married which occurred 33
years later.
The patient managed to have a consummative
sexual intercourse. However, she had some
climacteric symptoms.
Physical examination revealed that the secondary
sexual characteristics were well developed, and the
external genitalia were normal. She had a functional
vagina with a length of 4cm achieved through
sexual activity, an absence of cervix, and a palpable
right pelvic mass.
The serum level of oestrogen was normal; however
the levels of follicle-stimulating hormone and
luteinizing hormone were within menopausal range.
The intravenous pyelogram showed duplex right
hydroureters with mild hydronephrosis and a single
left hydroureter. Transvaginal scan revealed a solid
pelvic mass, which could be ovarian in origin. A
rudimentary uterus and an atrophic ovary
measuring 2cm x 1cm were visualized on the left
side. CT scan of the pelvic identified a solid right
ovarian mass measuring 5cm x 6cm.
The patient underwent a laparotomy. The uterus
was substituted by 2 bilateral remnants, of which
the one on the right side showed multiple uterine
fibroids. The remnants were completely separated.
Both fallopian tubes were normal and both ovaries
were noted atrophic.
The case continues…..
Right fallopian tube
Fibroid s
Remnants of the uterus
Myomectomy of the uterine fibroids was performed,
leaving the uterine remnant to the size of 2cm x 1 cm.
The patient was discharged well on the third
postoperative day. Histopathology examination of the
fibroids confirmed the diagnosis.
Discussion
References
Patients with MRKH syndrome have normally
developed ovaries and fallopian tubes, and many of
them have two uterine remnants. These uterine
remnants consist of fibromuscular tissue, which
may be the target tissue for ovarian steroids, from
which tumours like leiomyomas can originate.
A pelvic mass found in MRKH patient needs to be
thoroughly evaluated. As illustrated in this case,
imaging technique may give some value but may
also mislead the physician to the origin of the mass.
A better way to evaluate such case is by performing
diagnostic laparoscopy, provided that the risk of
developing malignancy is low.
In conclusion, although the development of uterine
fibroid is rare in MRKH patients, it needs to be
included in the differential diagnosis of pelvic mass
in these patients.
1. Capraro VJ, Gallego MB. Vaginal agenesis. Am J Obstet Gynecol 1976;124:98-107
2. Efthimios Deligeoroglou, Antonios Kontoravdis, Evangelos Makrakis, Panagiotis Christopoulos, Apastolos Kountouris and George Creatsas. Development of leiomyomas on the uterine mremnants of two women with Mayer-Rokitansky-Küster-Hauser syndrome. Fertil Steril 2004;81:1385-1387
3. Faber M, Stein A, Adashi E. Rokitanski-Küster-Hauser syndrome. Obstet Gynecol; 51 (supp 11): 70-3
4. Marta Lamarca, Ricardo Navario, Maria Eugenia Ballesteros, Salvador Garecia-Agurire, Maria Pilarconte and Jose Antonio Dugue. Leiomyomas in both uterine remnants in a woman with the Mayer-Rokitansky-Küster-Hauser syndrome. Fertil Steril 2009;91:931 e13-e15
5. Metzger DA, Massad LS, Piseitelli JT. Leiomyoma in a mullerian remnant. A case report. J Reprod Med 1988;33:246-8
Right ovary