DOI:
Review
Torsion of a uterine leiomyoma – a rare cause of hemoperitoneum;
a case report and review of the literature
Antonia Mihaela Levai, Ioana Cristina Rotar, Daniel Mureșan
1st Obstetrics and Gynecology Department, Emergency County Hospital, “Iuliu Hațieganu” University of Medicine and Pharmacy, Cluj Napoca, Romania
Received 03.11.2018 Accepted 11.12.2018 Med Ultrason
2019, Vol. 21, No 1, 77-82
Corresponding author: Ioana Cristina Rotar
1st Obstetrics and Gynecology Department 3-5 Clinicilor street,
400006 Cluj Napoca, Romania E-mail: [email protected]
Introduction
Uterine fibroids, also known as myomas or leiomyo- mas, represent the commonest tumor in women of repro- ductive age being encountered in approximately 50-60%
of women [1-3]. The most accurate term is leiomyoma, the tumor being the result of a monoclonal cell prolifera- tion that originates from the uterine smooth muscle tissue and its connective tissue under the influence of ovarian steroids especially progesterone and local factors [1,2].
Fibroids can remain asymptomatic during the reproduc- tive life being discovered by routine ultrasound (US) or in about 30-40% of casesare diagnosed due to their chronic complications [2].
Fibroid location represents the key factor for the oc- currence of a particular complication. Many fibroid clas- sifications are available; the one from FIGO (Interna- tional Federation of Gynecology and Obstetrics), based on myoma location correlates best with tumor symptoms and treatment [4]. Intracavitary fibroids can determine heavy menstrual bleeding and therefore iron deficiency anemia, but also infertility, while subserosal fibroids can determine pelvic pressure, chronic pelvic pain, urinary or gastrointestinal symptoms or even hydronephrosis [2,5].
The relationship between fibroids and infertility is com- plex; this type of benign uterine tumors could interfere with fertility through many mechanisms: alteration of endometrial function, increased uterine contractility, im- paired endometrial and myometrial blood flow but also alteration of local paracrine factors that can interfere with blastocyst implantation [2].
Extremely rare fibroids can be involved in acute com- plications such as thromboembolism, acute torsion of subserosal pedunculated leiomyoma, acute urinary reten- tion and renal failure, acute pain caused by red degenera- tion during pregnancy, acute vaginal or intra-peritoneal Abstract
Uterine leiomyomas are common benign uterine tumors but by contrast, their acute complications are very rare. We present an unusual case of 38-year-old woman that came to the emergency department with acute abdominal pain. The ultra- sound revealed hemoperitoneum, a uterus with two intramural fibroids and a tender inhomogeneous pelvic mass that seems to connect with the uterus. Computer tomographic (CT) examination raised the suspicion of a degenerated fibroid and hemo- peritoneum. Emergency laparotomy was performed: the hemoperitoneum was determined by a degenerated fundal subserosal fibroid. Myomectomy was subsequently carried out. Even though this condition is extremely rare, every clinician has to bear in mind that acute fibroid complications can be a potential cause of acute abdominal pain that requires immediate surgery. The imagistic tools, ultrasound and CT are extremely helpful for the diagnosis.
Due to its relative rarity in the second part of the article we have performed a review of the existing literature regarding the acute complications of fibroid torsion.
Keywords: leiomyoma; torsion; hemoperitoneum; fibroid degeneration; acute abdomen
DOI: 10.11152/mu-1784
hemorrhage, mesenteric vein thrombosis and intestinal gangrene [4].
The management of uterine fibroids depends on tu- mor characteristic: number, size, location of the fibroids but also on patient age and future fertility plans [2]. Sur- gical therapy is represented by myomectomy or hysterec- tomy by various approaches: hysteroscopy, laparotomy, laparoscopy or even vaginal surgery. Uterine artery em- bolization is an alternative to the surgical treatment.
In the following section, we aim to present a rare case of hemoperitoneum due to fibroid degeneration. In the second part of the article we performed a literature analysis regarding imagistic diagnosis and management of fibroids that determined a hemoperitoneum
Case report
A 38-year-old nulliparous woman, with no gyneco- logic or medical known conditions, presented at the emer- gency room for severe abdominal pain progressively ag- gravated in the last two days, despite self-administration of various antalgic and anti-inflammatory drugs. On the day of admission, besides lower abdominal pain, she also presented nausea, dizziness and intense weakness. The patient reported regular menstrual cycles, her last men- strual period being a week ago. She denied intercourse during the last week. The last gynecological examination had been carried out about two years ago and was within normal limits. On physical examination, the vital signs were within normal limits blood pressure 128/84 mmHg, heart rate 76 bpm and temperature of 36.5° C. Abdominal examination showed generalized tenderness and positive Blumberg’s sign. Bimanual examination revealed a mobile slightly increased uterus and a tender pelvic mass, about 6 cm in size thatcould be mobilized together with the uterus.
Hemoglobin concentration was initially 13.6 g/dL and two hours later 12.8 g/dL with leukocytosis (16.2×109/L).
The urine pregnancy test was negative.
US of the pelvis showed uterus into anteversion and anteflexion, with two intramural (FIGO 4) fibroids measuring 1.6/1.7 cm and 2.7/3.3 cm and a pelvic mass besides the uterus measuring 6.9/5.8 cm (fig 1a,b). Both ovaries were normal. Moderate free pelvic fluid extend- ing into the pouches of Douglas and Morrison was noted.
The repeated US examination after 3 hours revealed the above-mentioned pelvic mass of slightly increased di- mensions 8.86/6.22 cm and an increased quantity of free fluid in the peritoneal cavity.
Our initial differential diagnosis was twisted adnexa, ruptured ectopic pregnancy, hemorrhagic corpus luteum or follicular cyst, endometriosis, ruptured ovarian cyst, torsional ovarian fibroma, ruptured pyosalpinx or tubo- ovarian abscess and even appendicitis.
The computer scan (CT) of the abdomen and pelvis showed a 64/88/68 mm mass arising from the fundal wall of the uterus with a vascular pedicle suggesting a fibroid (fig 1c,d).
Due to the detection of hemoperitoneum on paracen- tesis, corroborated with clinical and imagistic examina- tions, immediate surgery was performed. Intraopera- tively, approximately 400 mL of hemoperitoneum was found originating from the degenerated fundal pediculate fibroid measuring 8 cm, a tumor that has been removed by a myomectomy.
The postoperative recovery was without any incident.
Histopathological examination of the mass showed an in- farcted and torsion leiomyoma measuring. No increase in mitotic activity has been reported.
The patient was discharged on the fourth day after surgery in the absence of any complication. At 6 weeks postoperative visit the abdominal scar was completely healed, and the ultrasound revealed the presence of the above-mentioned fibroids.
At the scheduled 6-month post-surgery visit, the pa- tient had a viable 6 weeks intrauterine pregnancy.
Fig 1. a) Pelvic ultrasound B mode showing a pelvic tumor related to the uterus and free fluid in the abdominal cavity; b) The pelvic mass was highly suggestive for a uterine fibroid; the lump being in close contact with the uterus (CFM mode); c) CT image suggest- ing a uterine fibroid, transverse section; d) CT image, sagital section
Discussions
Spontaneous hemoperitoneum related to fibroids are extremely rare conditions; around 100 cases being re- ported in the literature [6]. We have performed a search of the literature using the term leiomyoma/fibroids and hemoperitoneum and we found 25 cases reported in the last 10 years (between January 2008 and January 2018) (Table I).
In most of the cases, the condition occurred in women of reproductive age; the mean age of the patients being 35.5 years, ranging from 22 to 62 years. However, Sale- hi et al [31] reported a case of a 15-year-old adolescent with a 9.5/8.4/10.7 cm heterogeneous, hypoattenuating, symptomatic solid adnexal mass that resulted in being a degenerative uterine leiomyoma. Several other cases in the puerperal period, in perimenopause or even in post- menopause have been reported [18,26].
The majority of complicated leiomyomas were sub- serosal (92%), among them, 44% being pedunculated.
Only one author reported a huge leiomyoma that encom- passed the entire uterine corpus. In the latter case, the he- moperitoneum occurred due to the rupture of the dilated superficial vein [24].
The reported leiomyomas complicated with hemop- eritoneum varied in size from medium to large dimen- sions from 4 to16.3 cm with a median diameter around 11 cm.
The exact etiopathogenetic mechanisms that deter- mine the occurrence of hemoperitoneum in a patient with uterine fibroids are, in some cases, difficult to be identi- fied, the most frequently identified being the rupture of serosal veins or arteries, avulsion, torsion, rupture of a degenerated fibroid.
A precise hemoperitoneum determining cause was re- ported in several patients: venous congestion, increased abdominal pressure, trauma, rapid growth or degenera- tion of the fibroid [3,6]. Our patient denied trauma, in- tense physical activity or intercourse during the last week. She also denies any hormonal treatment or use of oral contraceptives. The only mechanism that we can as- sume is the rapid growth of a fibroid in a young patient, which overcame its possibilities of vascularization and determined degeneration, later torsion and then rupture of the uterine artery.
Horowitz et al [32] described a case of a 48-year-old woman with a pulsatile bleeding superficial artery, lo- cated on the serosal surface of a 14 cm fundal myoma. In this situation, the authors supposed that intraperitoneal hemorrhage could be caused by the underlying pressure exerted by the growing myoma on the walls of a uterine vessel or by the uterine contractions during menstrua-
tion that may distend the blood vessel to the breaking point. Moreover, Lotterman et al [7] described a 28-year- old nulliparous woman that presented hemoperitoneum resulting from surface vein rupture of a large fibroid weighing more than 1300 g. They assumed that the rup- ture likely occurred after a bowel movement and the in- creased venous congestion led to vessel rupture in their patient. Schwartz et al [26] concluded that the mecha- nism responsible for the degeneration and the rupture of a known fibroid in the case of a 53-year-old woman could be related to decreasing levels of estrogen and proges- terone in combination with menstrual cycle irregularity.
Rarely, a trauma was depicted in the recent past: Es- trade-Huchon et al [11] presented the case of a 46-year- old woman with acute abdominal pain while jogging.
Laparoscopy revealed internal bleeding from an avulsed subserosal leiomyoma. In this case abdominal trauma could be responsible for the leiomyoma rupture. By con- trast another case of spontaneously avulsion of a uterine leiomyomata in the absence of any trauma was also re- ported by Pachy et al [10].
Interesting, Swarray-Deen et al [30] described the case of a 43-year-old multiparous woman with massive intra-abdominal hemorrhage due to a 10 cm subserosal fibroid with a ruptured capsule, 2 days after a spontane- ous vaginal delivery. It was a leiomyoma with cystic de- generation. The authors concluded that the degenerative process and the contractions during labor could have pre- cipitated the rupture. Moreover, Tan at al [33] reported a similar case but 9 weeks postpartum. In the latter two cases the hormonal changes during postpartum could have determined the degenerative changes and increased the fragility of the tumor.
Several authors [14,21,30,34] reported acute abdo- men syndrome after fibroid degenerescence. Three fac- tors have been identified as potential determining for subserosal myoma torsion: the presence of a long and thin pedicle; the size (larger the fibroids less likely to untwist) and the relationship between the myoma and adjacent viscera (the uterine fundus, intestine and pelvic sidewall) [21]. Twisted fibromas causes venous stasis and later on, edema and congestion that will compromise the arterial blood supply. Later hemorrhagic necrosis and gangrene can appear.
Preoperative diagnosis was difficult due to the rare- ness of the pathology (in our service it was the first case reported in 30 years) and the absence of any medical history (normal gynecologic examination 2 years prior presentation). Imaging techniques (US, CT) helped us to formulate a preoperative diagnosis that was definite- ly confirmed by the laparotomy which allowed also the treatment (myomectomy).
Table I. Analysis of Pubmed available articles on fibroids and hemoperitoneum (accessed on August 1st 2018)
Authors Patient
age (years)
Type of
leiomyoma Tumor size
(cm/kg) Cause of
hemorrhage Treatment
Lotterman et al (2008) [7] 28 pedunculated,
fundal 16/13/10
(1.3 kg) surface vein bleeding myomectomy Ihama et al (2008) [8] 28 subserosal, fundal 10/10/10 rupture of a subserosal
vein an autopsy case
Su et al (2008) [9] 36 subserosal, fundal 15/15/14 dilated vein rupture myomectomy Pachy et al (2009) [10] 25 pedunculated
anterior 14/10/6 avulsion myomectomy
Estrade-Huchon (2010) [11] 46 pedunculated,
fundal 4 cm avulsion hysterectomy
Prior et al (2010) [12] 35 pedunculated,
fundal 23/10/17
(3.9 kg) rupture of serosal veins myomectomy Kasum et al (2010) [13] 37 subserosal, fundal,
15 weeks pregnancy
8.5/6.5 ruptured vessel myomectomy
Foissac et al (2011) [14] 62 pedunculated,
fundal 16/23/14 torsion myomectomy
Toquero et al (2012) [15] 43 subserosal not mentioned fibroid laceration hysterectomy Chen et at (2013) [16] 22 subserosal 13/13/13 rupture of a superficial
UA laparoscopic
myomectomy Fontarensky et al (two cases)
(2013) [17] 47 pedunculated,
fundal 10/9/7 no arterial bleeding embolization of
UA, scheduled hysterectomy Fontarensky et al (two cases)
(2013) [17] 44 intramural, anterior 14/14/8.5 no arterial bleeding embolization of UA, scheduled hysterectomy
Alharbi et al (2013) [18] 55 subserosal fundal 15/12/7 perforation hysterectomy
Ymeleet al (2013) [19] 46 pedunculated,
fundal 22/22/22
(5.5kg) ruptured varices hysterectomy
Hicks et al (2014) [20] 33 pedunculated 7.3/6.1/6.5 avulsion myomectomy
Nigam et al (2014) [21] 42 pedunculated,
fundal 9.2/11.7/5.2 torsion myomectomy
Seet et al (2014) [22] 55 subserosal, fundal 7.3/10/12 ruptured degenerative
fibroid hysterectomy
Peng et al (2015) [23] 39 subserosal,
posterior 8/9/9 spontaneous laceration hysterectomy Aydin et al (2015) [24] 31 kugel myoma
(large leiomyoma which encom- passed the whole uterine corpus)
like a 14-16 weeks gesta- tional uterus
rupture of the dilated
superficial veins UA ligation failed, hysterectomy
Gulati et al (2016) [25] 29 pedunculated 14/19/10 ruptured large serosal
vessel myomectomy
Schwartz et al (2017) [26] 53 subserosal, fundal 8.8/7.3/8.3 ruptured degenerated hysterectomy Mizrahi et al (2017) [27] 39 pedunculated,
fundal 10/10/10 arterial vessel arising
from a right UA myomectomy and ligation of a right UA Tajima et al (2015) [28] 54 subserosal, pos-
terior 6.5/6/5.5 ruptured arterial
aneurysm myomectomy
Jenayah et al (2017) [29] 37 subserosal, fundal 10/11 ruptured dilated vein myomectomy Swarray-Deen et al (2017) [30] 43 subserosal, fundal 10/8 ruptured capsule myomectomy UA – uterine arteries
Tsai et al [35] argue that clinical examination together with US is sufficient for a preliminary diagnosis, advo- cating that no additional advanced radiographic imag- ing technique is necessary. They justified their option by the fact that emergency surgery must be performed as early as possible to avoid worsening of patient sta- tus and avoid consumptive coagulopathy. In our case we considered that the CT scan was helpful and added no significant delay to the patient status, but the use of a CT scan should be considered individualized based also on local resources.
Treatment aims to stop the bleeding and to preserve the uterus, if possible, especially in young women. My- omectomy was reported in 56% of cases (for details see table I); in the rest of the cases, a hysterectomy was per- formed [11,15,17-19,22-24]. In only two cases, uterine artery embolization was the first line of treatment for patient hemodynamic stabilization followed by a hyster- ectomy [17].
The immediate intervention is mandatory; the de- layed diagnosis and management being associated with increased morbidity and even mortality – one of the re- ported cases was an autopsy case of a 28 years old wom- an [8].
Mizrahi et al [27] described the necessity of the liga- tion of a right uterine artery because of an arterial vessel arising from a right uterine artery with an active bleeding.
Fontarensky et al [17] described the cases of two nul- liparous patients with voluminous uterine fibroids com- plicated by intraperitoneal hemorrhage. The CT showed no arterial blush suggesting active intraperitoneal ex- travasation and no adnexal mass bleeding. The authors decided to carry out the embolization of the uterine ar- tery, as an emergency therapeutic management, to ensure hemodynamic stabilization before scheduled surgery.
Laparoscopic exploration was performed during the fol- lowing days, but due to the volume of the fibroids the procedure was converted to laparotomy. Moreover, Take- da et al [36] performed gasless laparo-endoscopic single- site myomectomy with in-bag manual extraction for a pe- dunculated subserosal myoma with a torsion measuring 55/41 mm, without hemoperitoneum.
Conclusions
Hemoperitoneum caused by a uterine fibroid is an extremely rare condition; therefore, it has to be consid- ered as a potential diagnosis after ruling out other more frequent causes of intraperitoneal bleeding in reproduc- tive age women. Imagistic techniquesare extremely use- ful for the diagnosis, US being mandatory. CT can be useful, when available, if the patient is hemodynamically
stable. CT can not only evidence the lesion but also the exclusion of other intra-abdominal and extragenital pa- thologies. Whenever a fibroid is present in a patient with acute abdominal pain, the rupture of the fibroid feeding vessel should be evaluated. Surgery, regardless of the ap- proach, confirms the diagnosis and allows the immediate bleeding arrest and appropriate treatment (myomectomy, hysterectomy).
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