Laparotomic myomectomy of a spontaneous perforated leiomyoma mimicking pseudomyxoma peritonei at 27 weeks of pregnancy

Summary

Objective: We report a rare case of acute abdomen pain in 27 weeks´ gestation caused by a perforated leiomyoma mimicking pseudomyxoma peritonei on magnetic resonance imaging.

Subject: Case report.

Setting: 1st Department of Gynaecology and Obstetrics. Faculty of Medicine, Comenius University in Bratislava, Slovakia.

Case report: In our reported case report of perforated leiomyoma mimicking pseudomyxoma peritonei on magnetic resonance imaging. Successful myomectomy was done, and the pregnancy continued with good outcome. At week 40, the patient underwent caesarean section.

Conclusion: Uterine fibroids in pregnancy can lead to severe complications. Their spontaneous rupture in pregnancy is very rare. To manage acute abdominal pain in pregnancy a good diagnostic must be performed, and surgical treatment should be carefully considered.

Keywords:

myomectomy – pregnancy – perforated leiomyoma – magnetic resonance imaging – ultrasound

INTRODUCTION

Uterine leiomyomas are benign, common tumors affecting many women [14]. About 0.3% to 2.6% of them are estimated to occur during pregnancy. Although most of them in pregnancy present asymptomatic, about 10% result in severe pregnancy complications [8]. These complications are either associated with pregnancy or not. The typical reported pregnancy complications are early pregnancy loss, premature labor, cervical incompetence, abnormal presentation of the fetus, fetal death, periphlebitis, infertility, cesarean delivery, placental abruption, preeclampsia, intrauterine fetal growth restriction or preterm premature rupture of membranes. Other complications of uterus myomatosus which can cause acute abdomen are uterine torsion, hemoperitoneum, enormous increase in size causing cystic degeneration or hydronephrosis, acute urinary retention, myoma rupture or pyomyoma [12].

CASE DESCRIPTION

42-year-old primiparous woman in 26+0 week of gestation was referred to our tertiary perinatal center from lower type hospital due to acute abdominal pain localized to left mesogastrium. Clinical examination upon admission revealed normal nontender uterus bigger than expected for gestation age with solid, painful resistance about 10×8 cm in size. A laboratory investigation showed high inflammatory markers (C-reactive protein 68 mg/l, procalcitonin 0.592 μg/l), high thrombocytosis (701×109/l), high coagulation markers (fibrinogen 8.83 g/l, D-dimer 7898 μg/l), normocytic anemia (hemoglobin 92 g/l) and positive oncomarkers (Ca-125 113.70 U/mL, ROMA index 15.6%). A transabdominal ultrasonographic scan revealed subserous anterior site myoma mostly on left side measuring 13×9 cm in diameter on thick 5 cm wide implantation base with a 2 cm perforation opening with absent color flow (figure 1, 2). The abdominal collection of leaking fluid from perforation opening was visualized in front of uterus. Magnetic resonance imaging (MRI) revealed solid tumor with central necrosis in the upper part of abdominal cavity, prominent from the anterior wall of uterus and intraperitoneal accumulation of a clear gelatinous ascites with septations without air-fluid levels. The finding was concluded as pseudomyoma peritonei of unknown origin likely secondary myoma changes of myoma or peritonei (figure 3, 4).

As tumor necrosis was suggested the patient was treated with double intravenous combination of antibiotics – ampicillin 4 g and gentamicine 240 mg daily dose and nadropadine 0.4 ml subcutaneously. For the acute abdominal pain resistant to analgesics she was indicated for exploratory laparotomic surgery. The operation was performed under general anesthesia in 26+4 week of gestation followed up by antenatal steroids for fetal pulmonary maturation. Laparotomy revealed inflammatory thickened parietal peritoneum of anterior abdominal wall with many fibrine accretion and membranous adhesions to uterus and small intestine. Uterus presented size VI, regular in shape with anterior site myoma of 9×5 cm with perforation opening of 2 cm in diameter (figure 5, 6). Myomectomy and adhesiolysis were performed. Histological examination has revealed leiomyoma with secondary changes without evident signs of malignity with present mild atypical cells. The coagulation markers decreased (Tro 578×109/l, fibrinogen 8.8g/l, D-dimer 6315 μg/l). Because of myasthenia gravis no uterosedatives neither magnesium sulfate was administrated. Further progression of the pregnancy was without any other adverse complications. The girl newborn (weight 2750 g, height 51 cm and Apgar score 10/10) with umbilical cord twice tightened around the neck and with polyhydramnion was delivered by an elective caesarean section at 39+2 week of gestation. During the caesarean section the second myomectomy was performed, hemostasis was achieved. Woman has survived without any other complication and consequences.

DISCUSSION

Uterine leiomyomas in pregnancy usually present asymptomatic or may lead to pregnancy complications. Our patient was referred to our department due to developed symptomatology of acute abdominal pain in 27th gestational weeks. The information that she was diagnosed with uterine leiomyoma in eighth week of pregnancy was absent and she had mentioned it only after further deeper investigation. We received the MRI conclusion, with pseudomyxoma peritonei of unknown origin likely secondary changes of myoma or of peritonei, before our further examination was done. Regarding the MRI finding the previous interpretation of pseudomyxoma peritonei can be considered correct due to intraperitoneal fluid of clear gelatinous character with septations without air-fluid levels. It also did not have a character of hemorrhagic or pyogenic content. Pseudomyxoma peritonei can be caused by different pathology, including subserous fibroid, fibroid torsion, infarct or necrosis. In the time of MRI examination, the anamnestic information about fibroid diagnosed in 1st trimester was missing as well as ultrasonography suspicion for central necrosis of myoma with disruption. Upon admission to our department a transabdominal ultrasonographic scan confirmed the pregnancy and revealed subserous anterior site myoma and a laboratory investigation upon admission showed severe coagulopathy with inflammation. According to diagnostic work-up the myoma with spontaneous rupture was supposed. MRI is very helpful method to be used as complementary especially in unclear diagnostic situations.

To identify case reports which were previously reported, PubMed using a combination of key words: “myomectomy”, “myoma”, “fibroids” and “pregnancy” were searched. We focused on case reports of myoma necrosis and perforation during pregnancy or puerperium. Eight cases of myoma necrosis and one case of spontaneous myoma perforation in pregnancy were reported by Makar, 1989. Spontaneous perforation of a necrotizing leiomyoma is reported very rare. Makar reported that degenerating myoma should be treated conservative with analgesics, ice-packs and bed-rest. He considered surgical exploration to be mandatory when the general condition deteriorates and general peritonitis with spiking high fever and marked leucocytosis or massive blood loss occurs. His article was published in 1989 and that time he reported that myomectomy should be contra-indicated during pregnancy, as it has a high morbidity and mortality due to massive bleeding, high risk of fulminant infection and abortion. That time the exploratory laparotomy without myomectomy in pregnancy was done but in five days a spontaneous abortion occurred [6].

In now-days literature, the main conditions leading to myoma surgery in pregnancy which were previously reported were due to uterine fibroid torsion, abnormal myoma size, necrosis and resultant inflammatory peritoneal reaction. According to the actual literature the surgical intervention should be considered if acute abdomen symptoms persist after 72 h of pharmacological therapy [1]. As our patient had acute abdominal pain localized in left mesogastrium, non-responding to analgesics we decided for exploratory laparotomy. As the operation finding was difficult to assess it was necessary to be prepared for acute caesarean section. After prenatal consultation with a neonatologist it was decided to promote the fetal lung maturation with dexamethasone.

Surgical management of acute abdominal pain caused by uterus myomatosus includes myomectomy [13], done by laparoscopic, laparotomic or even vaginal way. Abdominal operations in general are performed in around 2% of all pregnancies [3]. As the subserous myoma in our case was on anterior side of uterus, myomectomy was easily performed. The post-operative management did not include preventive tocolysis. It was contraindicated due to patient’s primary illness myasthenia gravis. Myoma rupture in pregnancy could be prevented by uterine artery embolization in women with future maternity plans [10]. Referring to literature, 92% of patients after surgical treatment in pregnancy are without complications [5]. The complication reported after myomectomy in literature are: uterus rupture during or after labor [7, 9] full term abdominal pregnancy [2], septic necrosis of myometrium [11], etc. In our case the patient struggled with hepatopathy which started about one week after surgical procedure. We supposed that etiology of hepatopathy being multifactorial consisting of anesthetic lesion in combination with intrahepatic cholestasis of pregnant women. We consider that the polyhydramnion founded at 36 weeks of gestation, was idiopathic. Despite the increasing trend in caesarean section [4] our patient was indicated for elective surgery because of previous uterine scar.

CONCLUSION

Surgery during pregnancy is a challenge but it has its place when necessary. Benefits of decided treatment should always overcome risks. Many patients undergo surgery in pregnancy and most of them are without complications. This case is a rare and very interesting due to its efficient outcome for mother and her child.

MUDr. Eva Korňanová

1st Department of Gynaecology and Obstetrics

Faculty of Medicine

Comenius University

Antolská 11

851 07 Bratislava

Slovakia

e-mail: eva.kornanova@gmail.com