Resection of a Giant Ovarian Mucinous Cystadenoma with Intraoperative Drainage

Abstract

Background: Giant ovarian tumors are now rarely encountered, because of the wide availability of medical care, and most cases are benign. Case: A rare case of a giant ovarian cystadenoma in a 41-year-old woman, with such severe abdominal distension that she could not walk, is reported. Her abdominal girth was 141 cm. The patient was 151 cm tall and weighed 92.6 kg. Computed tomography (CT) showed a large, multilocular, cystic tumor that occupied the whole abdomen. The preoperative diagnosis was malignant ovarian tumor or ovarian cystadenoma. Results: On laparotomy, gradual intraoperative drainage of the tumor was first performed over 2 hours, and then total hysterectomy with bilateral salpingo-oophorectomy was performed successfully. Pathologic findings confirmed that the tumor was a mucinous cystadenoma. Conclusions: There is a lack of evidence on how to manage such giant tumors in the perioperative period; however, in the present case, intraoperative drainage and tumor resection were safely performed without complications. (J GYNECOL SURG 28:215)

Introduction

Giant ovarian tumors are now rarely encountered, because of the wide availability of medical care. The largest ovarian tumor ever reported weighed 149 kg and was removed in 1905.¹ Most cases are benign, but some cases of low– malignant-potential tumors or adenocarcinomas have been reported.^2–7 Their management, especially whether and when to drain the cysts, has been discussed over the years, but there is no clear consensus. Complications associated with the removal of giant ovarian tumors have been described, including massive bleeding or re-expansion pulmonary edema.^4,5,7,9–12 To the authors' best knowledge, 22 cases of giant ovarian tumor of >40 kg have been reported since 1970 (Table 1).^2–23 A giant ovarian mucinous cystadenoma that underwent successful intraoperative drainage over 2 hours is reported, along with a review of the literature.

Table 1.

Reported Cases of Giant Ovarian Tumors Weighing More than 40 kg (>88 lb)

No	Author	Year	Age (years)	Tumor weight (kg)	Histopathologic diagnosis	Pre- or intraoperative drainage (duration)	Bleeding (mL)	Perioperative complications	Outcomes
1	Bezjian et al.¹⁷	1971	79	54.9	Pseudomucinous cystadenoma	Pre (6 h)+intra	ND	None	Alive
2	Hunter¹⁰	1980	54	71.1	Mucinous cystadenoma	Pre (1 day)	ND	Massive bleeding, peritonitis	Alive
3	Drife and Trotter⁹	1981	44	62.5	Mucinous cystadenoma	Pre (5 days)+intra	ND	Pulmonary edema	Alive
4	Buller et al.²	1982	30	80.3	Mucinous cystadenoma of low malignancy	Pre+intra	ND	None	Alive
5	Morrison and Morgan¹¹	1987	58	120	Mucinous cystadenoma	Pre+intra	10,000	None	Alive
6	Matory et al.³	1989	65	51.4	Mucinous cystadenocarcinoma	Pre+intra	ND	None	Alive
7	Vipond et al.¹⁸	1991	37	83	Pseudomucinous cystadenoma with teratoma	Pre (10 days)+intra	ND	None	Alive
8	Zamora-Garzal et al.¹⁹	1992	22	70	Serous cystadenoma	Pre (7 days)+intra	ND	None	Alive
9	Sunthrasaj et al.¹²	1993	37	64	Mucinous cystadenoma	Pre (3 days)+intra	4,000	Massive bleeding, SSI, pneumonia	Alive
10	Tait et al.²⁰	1997	63	70.9	Mucinous cystadenoma	Whole	None	None	Alive
11	O'Hanlan¹³	1994	35	134.7	Mucinous cystadenoma with teratoma	Whole	1,000	None	Alive
12	Nishiyama and Hanaoka¹⁴	1997	58	47	Mucinous cystadenoma	Intra	1,800	None	Alive
13	Kim et al.¹⁵	1999	21	62	Mucinous cystadenoma	Pre (11 days)	2,200	None	Alive
14	Yusuf et al.²¹	2000	45	64	Serous cystadenoma	Whole	ND	None	Alive
15	Koshiba et al.⁴	2003	33	55	Cystadenocarcinoma with low malignancy	Pre (days)	5,300	None	Alive
16	Manchanda and Kriplani ⁵	2005	40	74	Well-differentiated mucinous adenocarcinoma	Pre+intra	3,000	None	Alive
17	Nakamura et al.⁶	2005	51	50.7	Mucinous cystadenocarcinoma	Intra	ND	Atelectasis	Dead
18	Einenkel et al.⁸	2006	57	49	Mucinous cystadenoma	Intra	ND	None	Alive
19	Yanazume et al.⁷	2007	30	100	Mucinous cystadenocarcinoma	Intra	18,900	Massive bleeding	Dead
20	Ueda et al.¹⁶	2008	34	86.5	Serous cystadenoma	Intra (8 h)	459	None	Alive
21	Hackethal et al.²²	2009	60	60	Mucinous cystadenoma	Whole	ND	None	Alive
22	Fobe et al.²³	2010	24	59	Mucosal-serosal cystadenoma	Whole	ND	None	Alive

ND, not described; SSI, Surgical site infection; Pre, preoperative; Intra, intraoperative.

Case

A 41-year-old nulliparous female was referred to Yoshida General Hospital with a 9-year history of increasing abdominal girth. In addition, she had become almost totally immobilized for 1 month and could not stand from the supine position because of worsening dyspnea. She was 151 cm tall and weighed 92.6 kg. Physical examination revealed gross abdominal distension and abdominal surface collateral veins. The laboratory data showed that CA-125 was elevated up to 150.3 U/mL. The chest radiograph showed bilateral diaphragmatic elevation and diminished lung fields (Fig. 1). Computed tomography (CT) showed a large, multilocular, cystic tumor that occupied the entire abdomen (Fig. 2). On abdominal and transvaginal ultrasonography (US), the multilocular tumor seemed to be derived from the ovary, and a solid protruding structure was detected in the cyst (Fig. 3). The patient could not be placed in the supine position because of aggravation of dyspnea (Fig. 4). Therefore, she was anesthetized in a reverse Trendelenburg position with a left lateral tilt, to decrease the pressure on the lungs, improve oxygenation, and prevent supine hypotension. On laparotomy, an attempt was made to remove the mass as a whole, to avoid rupture causing malignant cell spillage into the abdominal cavity, but there were too many dense peritoneal adhesions. Intraoperative drainage was then performed over 2 hours to shrink the tumor, while intensely monitoring the patient's vital signs; the total drainage amount was 41 L. Subsequently, tumor removal, hysterectomy, and bilateral salpingo-oophorectomy were performed successfully. The cyst had a diameter of 45 cm and weighed 3.5 kg (Fig. 5). On pathology, a single-layer columnar epithelium with mucin-containing cytoplasm and uniform basally arranged nuclei was seen (Fig. 6). The final pathologic diagnosis of the giant tumor was mucinous cystadenoma. The patient was finally discharged from the hospital 2 months after the surgery without any complications, when she was capable of walking on her own. The rehabilitation period was prolonged because she had been unable to walk for so long. Her body mass index (BMI) on discharge was 19 kg/m².

FIG. 1.

Chest radiograph demonstrates bilateral diaphragmatic elevation with diminished lung fields.

FIG. 2.

Computed tomography scan shows a huge multilocular cystic mass that occupies the entire peritoneal cavity.

FIG. 3.

Transabdominal ultrasound shows the tumor with a protruding structure in the cystic wall.

FIG. 4.

The patient at operation, showing the markedly distended abdomen with greatly dilated subcutaneous veins.

FIG. 5.

The removed ovarian cyst measured 45 cm in diameter.

FIG. 6.

Microscopic pathology of the tumor showing a single layer of columnar epithelium with mucin-containing cytoplasm and uniform, basally arranged nuclei (hematoxylin/eosin).

Discussion

Recent advances in the healthcare system and patient education have decreased the frequency of giant ovarian tumors. Giant ovarian tumors are benign tumors, mainly serous or mucinous cystadenomas, but some cases of low–malignancy-potential tumors and adenocarcinomas have been reported.^2–7 The largest ovarian tumor, reported in 1970, weighed 149 kg.¹ There have been 22 resected giant ovarian tumor cases of >40 kg since then (Table 1).^2–23 Special attention must be directed to their perioperative management, especially during cyst drainage, to avoid associated complications.

Table 1 shows the details of the reported perioperative management, complications, and clinicopathologic data of patients with giant ovarian tumors weighing>40 kg. The tumor weights ranged from 47 to 134.7 kg, with a mean weight of 71.4 kg. The histopathologic diagnoses were benign tumors in 16 cases (72.7%) and malignant tumors, including low-potential malignant tumors and mucinous cystadenocarcinomas, in 6 cases (27.2%). The highest intraoperative bleeding amount was 18,900 mL, which caused disseminated intravascular coagulation and death.⁷ Complications associated with cyst drainage were: massive bleeding in 9 cases (36.3%);^{4,5,7,10–15} repeat surgery resulting from peritonitis and massive bleeding in 2 cases (9.0%);^9,10 re-expansion pulmonary edema, which needed intensive monitoring of respiratory management, in 1 case (4.5%);⁹ and pulmonary dysfunction, such as pneumonia and atelectasis, in 2 cases (9%).^6,12

The main advantage of drainage is to shrink the cyst to a more manageable size, facilitating subsequent surgery. This makes it safe to perform the operation by widening the working space. Einenkel et al. reviewed the literature about whether and when to perform drainage, and concluded that it was better to avoid drainage, because of the risk of cell spillage that would worsen the prognosis.⁸ However, late recurrence caused by disseminated carcinoma cells has not been reported so far. Furthermore, the advantage of preoperative drainage is prevention of pulmonary edema and improvement of the pulmonary and circulatory systems through slow reduction in tumor size. One must be careful when using long-term tube drainage to avoid problems such as infection, peritoneal dissemination, and sudden massive body fluid loss from the drainage hole. High pressure inside the tumor would make it especially difficult to stop the discharge once it begins.

Intraoperative drainage was useful to prevent massive bleeding and cell spillage into the peritoneum in the present case. Dense adhesions, the size of the tumor, and the associated small working space make it difficult to stop bleeding once it starts. Preoperative drainage was performed safely in all cases, but there was concern about the possibility of infection, as well as pulmonary and circulatory dysfunction caused by tube movement, in the present case. The reported complications did not differ depending on whether pre- or intraoperative drainage or their combination was performed. The important point is to ensure the availability of sufficient blood for transfusions in case of bleeding, and to monitor the pulmonary and circulatory systems carefully in the perioperative period.

Various complications associated with drainage have been reported. Re-expansion pulmonary edema has been described in association with cyst drainage.^9,11 The patient should be protected against pulmonary edema because of rapid re-expansion of collapsed lungs and fluid shifting during surgery. Pulmonary edema may also occur after removal of the tumor because of the sudden re-expansion of a chronically collapsed lung as a result of compression by the elevated abdomen. Once this occurs, it sometimes requires ventilation.

The safe management of such cases requires a multidisciplinary team approach to manage the many challenges that may occur as a result of potential severe cardiovascular and respiratory complications.⁸ The massive tumor makes it difficult to induce general anesthesia, because the enlarged abdomen decreases the lungs and compresses the circulatory system.

Conclusions

In conclusion, there is a lack of evidence on how to manage such giant tumors in the perioperative period. The advantages and disadvantages of pre- or intraoperative cyst drainage need to be acknowledged to help decide whether to perform pre- or intraoperative drainage. Providing intensive care to monitor the pulmonary and circulatory systems is important, because of the enormous effect of the tumor itself, which can result in their dysfunction before and after surgery. In the present case, intraoperative drainage and tumor resection were safely performed without complications.

Footnotes

Disclosure Statement

No competing financial conflicts exist.

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