- Case report
- Open Access
Can ovaries be preserved after an ovarian arteriovenous disconnection? One case report and a review of surgical treatment using Da Vinci robots for aggressive ovarian fibromatosis
- Jun Ying†1,
- Jiawen Feng†1,
- Jinghui Hu1,
- Shuo Wang1,
- Peilin Han1,
- Yujie Huang1,
- Wei Zhao1 and
- Jianhua Qian1Email author
© The Author(s). 2019
- Received: 1 April 2019
- Accepted: 24 May 2019
- Published: 7 June 2019
The ovary is an important organ of the female reproductive system, which produces oocytes and secretes reproductive hormones. Ovaries have complex dual blood supplies with their blood supply being the core component to protect and ensure ovarian function. Ovarian preservation surgery often encounters problems related to whether or not to preserve ovarian vessels on the affected side.
This study reports on the case of a 30-year-old female patient with the retroperitoneal fibromatosis that had a history of uterine leiomyoma. During the operation, the ovarian arteries and veins were separated according to what was found during the procedure. A postoperative examination demonstrated good function and morphology of the ovary.
A thorough review of academic journals combined with our collection of clinical data was conducted, which confirmed the double blood supply source to the ovaries. As a result of this exploration, a new surgical method is being proposed that is designed to protect the ovaries. By conducting this new procedure, the patient’s disease was not only halted and ultimately cured, but results demonstrate that the method was also able to retain the shape and function of the ovary. The postoperative satisfaction of the patient was significantly improved.
- Aggressive fibromatosis
- Ovarian vessels
- Ovary protection
Considered to be of the most important reproductive organs of women, the ovary has two main functions: producing germ cells and secreting endocrine hormones. It is worth mentioning that ovaries have a complex blood supply system. This is largely due to ovarian arteries and uterine arteries, which are required to support ovarian functions . Common diseases in women often affect the blood supply of ovarian arteries, such as adnexal torsion and pelvic masses. Malignant tumors of the reproductive system are especially common in elderly women . Normally, when an ovarian artery is twisted or invaded by a mass, a surgeon will choose to perform a procedure to rectify the problem and remove the affected ovary at the same time. This study is reporting on a case of aggressive fibromatosis involving an ovarian artery, in order to demonstrate a new and alternative ovarian protection procedure.
Aggressive fibromatosis is a benign tumor composed of differentiated fibroblastic cells with locally aggressive features. However, it does not metastasize . Fibroblastic cells with bands of collagen and an ill-defined cytoplasm rarely show mitosis . It has a low incidence and only accounts for about 0.03% of all tumors. Relevant literature reports that the annual average incidence of the disease in the general population is about 0.002–0.004‰. Most of these incidences are sporadic cases, occurring mainly in women, and especially in patients aged 10–40 years [5, 6]. At present, surgery is the primary method of treatment with the standard surgical goal to completely resect the lesion of negative margins [7, 8]. Aggressive fibromatosis occurring in the posterior peritoneum can invade peripheral blood vessels and nerves. How to deal with the complexities regarding ovarian blood vessels has become a problem worthy of further investigation.
During the course of current procedure method, the invaded blood vessels and the organs they supply are usually removed. However, in this study, in order to completely resect the tumor, a procedure was adopted that cut off the ovarian blood vessels on the affected side to protect the ovary. This is a new and innovative procedure that has been designed to preserve the ovary.
According to available literature, aggressive fibromatosis is a rare tumor of monoclonal fibroblastic proliferation, which is characterized by variable and complicated clinical processes [3, 4], These are divided into three categories depending on their anatomical location: extra-abdominal (60%), abdominal wall (25%), and intra-abdominal (8–15%) . The Intra-abdominal AF often occurs in the small intestinal mesentery, but it can also occur in the posterior peritoneum, ileocolic mesentery, gastrocolic ligament and greater omentum. The diagnosis can be confirmed by additional examinations, such as an MRI, which is the method of choice when fibromatosis and recurrence are suspected , as well as pathology and immunohistochemistry [11, 12]. Presently, surgery is the main and most effective method of treatment, with the standard surgical goal to completely excise the lesion with negative surgical margins [7, 8]. The postoperative recurrence rate is 13–68% and the recurrence generally occurs more than 2 years after the initial surgery . Multiple recurrences may lead to a wider range of lesions, and even endangering life further by its invasion of important organs. It is also likely that the recurrence and mortality rate after a second operation will also increase significantly. A retrospective, multicenter analysis showed that the effect of the laparoscopic surgery (LS) and a robot-assisted laparoscopic (RALS) approach for the treatment of early-stage ovarian cancer is comparable to existing treatments  To fully expose the anatomical structure of the lesion, a RALS approach was selected. In this study, the mass was completely removed, and the margin of incision was negative, which reached the required standards for operating procedures. There were no recurrences detected in the 18 months after the operation, and all of the patient’s organs affected by the tumors were able to demonstrate good morphology and function.
It is worth noting that this mass invaded the right infundibulopelvic ligament and the right ovary arteries and veins. The right ovary’s arteries and veins were removed during the operation. However, the function and morphology of the right ovary was not significantly or negatively affected after the operation. This led the researchers to further explore the anatomical structure of ovarian arteries and veins, as well as its blood supply system. Relevant anatomical data has reported that ovaries have a double blood supply system, which comes from the ovarian artery (OA) and the ovarian branch of the uterine artery . The origin of the ovarian artery is relatively constant and regular. A total of 95.8% of the OA originates from the anterolateral part of the abdominal aorta. The uterine artery originates from the branch of the anterior trunk of the internal iliac artery . There are four known models related to the ovarian blood supply. Type I: the main ovarian artery is anastomosed with the ovarian branch of the uterine artery at the ovarian hilum, and both are supplied with blood equally, which accounts for 72.5%. Type II: the main ovarian artery and the ovarian branch of the uterine artery form a loop shape, and both are supplied with blood equally, which accounts for 13.7%. Type III: the ovarian branch of the uterine artery is anastomosed with a small branch of the ovarian artery, with the blood supply of the ovary mainly supplied by the uterine artery, which accounts for 10%. Type IV: the ovarian artery and the uterine artery are anastomosed at the uterine end of the fallopian tube, and the blood supply of the ovary is mainly supplied by the ovarian artery, which accounts for 3.7% . Such statistics demonstrate that only 3.7% of cases rely mainly on ovarian arteries for their blood supply. This provides a theoretical basis for the surgical approach proposed in this study, with the primary objective to preserve and avoid the removal of the ovaries.
This theory of ovarian vascular anatomy can also be applied to the treatment of another disease. Ovarian tumor pedicle torsion is a very common gynecological acute abdomen. Over recent years, a new surgical method of ovarian preservation has been developed. A ligation of the ovarian arteries and veins is conducted, but not cut off, with the reversed appendages repositioned and the ovarian cysts removed . This approach not only retains the morphology and function of the ovary, but it also blocks the channel of thrombus shedding, making the thrombus remain in the ovary vessels, and it avoids the occurrence of a pulmonary artery embolism. It also blocks the blood supply from the ovarian arteries, with the uterine artery and collateral vessels needed for a continued blood supply. This type of ovarian tumor generates a lot of collateral circulation before the procedure, which is a certain source of blood supply for ovaries. Therefore, several months after the initial procedure and during a follow-up examination, the appearance and function of the patient’s ovaries were found to be postive. Huang et al.  analyzed the surgical method applied during 42 cases of ovarian cyst pedicle torsion. Their findings determined that when compared with traditional surgical methods of ovarian resection, ovarian preservation surgical methods have distinct advantages for postoperative endocrine function recovery, and it has additional safety and effectiveness outcomes . Taskin et al.  established the mouse model with uterine adnexal torsion in 1998. They found that the ovary arteriovenous occlusion in mice caused ischemia for 12–24 h. Even after 36 h, and although the ovaries were a blue-black color, the retained ovaries still had normal functions. This outcome demonstrated the possibility of the ovary’s survival even after cutting off the ovarian arteries and veins, from the perspective of histology and biochemistry . However, Lagana et al.  emphasized a pathological process to maintain the circulation of the ovary after detorsion, deteriorates the tissue injury and leads to a pathologic process called ischemia/reperfusion (I/R) injury. This is characterized by oxidative stress. It is suggested that researchers should develop shared protocols for clinical use and application . Sipahi et al.  conducted a controlled trial to demonstrate that remote ischemic modulation (RIC) can alleviate ovarian ischemia/reperfusion injuries in rats. These studies have contributed to laying the theoretical foundation for the success of this proposed procedure.
A thorough exploration of existing literature and an in-depth analysis of this case, has confirmed the existence of a double blood supply to the ovaries. In this study, a new surgical procedure was proposed to treat aggressive ovarian fibromatosis. The procedure proposes that when a pelvic tumor invades the ovarian arteries and veins, the ovarian arteries and veins are cut and divided, in order to retain ovarian function. Due to the unique blood supply system of the ovaries and special conditions related to the disease, the procedure that formed the basis of this study, was able to successfully remove the tumors on the premise of preserving the ovaries. Such a ground-breaking surgical method needs to be confirmed for a wider application based on research and shared protocols, indicators, contradictions and specific conditions related to the patient. Only then will practitioners have the confidence to determine whether the procedure is effective and feasible.
The authors have no funding to declare.
YJ and FJW collected the clinical data and drafted the manuscript, including the revision. YJ, HJH and WS participated in the surgical treatment. HPL and HYJ reviewed the manuscript. FJW and ZW consulted the relevant literature. All authors read and approved the final manuscript.
Ethics approval and consent to participate
Subjects (or their parents or guardians) have given their written informed consent. This study was approved by the Ethics Committee of the First Affiliated Hospital, ZheJiang University School of Medicine.
Consent for publication
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
The authors declare that they have no competing interests.
Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
- Ouyang Z, Liu P, Yu Y, Chen C, Song X, Liang B, et al. Role of ovarian artery-to-uterine artery anastomoses in uterine artery embolization: initial anatomic and radiologic studies. Surg Radiol Anat. 2012;34(8):737–41. https://doi.org/10.1007/s00276-011-0883-x.View ArticlePubMedGoogle Scholar
- Vitale SG, Capriglione S, Zito G, Lopez S, Gulino FA, Di Guardo F, et al. Management of endometrial, ovarian and cervical cancer in the elderly: current approach to a challenging condition. Arch Gynecol Obstet. 2019;299(2):299–315. https://doi.org/10.1007/s00404-018-5006-z.View ArticlePubMedGoogle Scholar
- Campara Z, Spasic A, Aleksic P, Milev B. An Aggressive Retroperitoneal Fibromatosis. Medical Archives. 2016;70(2):154–7. https://doi.org/10.5455/medarh.2016.70.154-157.View ArticlePubMedPubMed CentralGoogle Scholar
- Tankshali R, Srivastava S, Anshuman K, Shah M. An unusual presentation of aggressive fibromatosis (retroperitoneal desmoid tumour) in pelvic cavity presented as obstructed labour. J Indian Med Assoc. 2011;109(8):589–91.PubMedGoogle Scholar
- Otero S, Moskovic EC, Strauss DC, Benson C, Miah AB, Thway K, et al. Desmoid-type fibromatosis. Clin Radiol. 2015;70(9):1038–45. https://doi.org/10.1016/j.crad.2015.04.015.View ArticlePubMedGoogle Scholar
- Reitamo JJ, Hayry P, Nykyri E, Saxen E. The desmoid tumor. I. Incidence, sex-, age- and anatomical distribution in the Finnish population. Am J Clin Pathol. 1982;77(6):665–73.View ArticleGoogle Scholar
- Ndzengue A, Mora M, Iqbal S, Becher R, Kchao S, Rosal Z, et al. Not every recurrent pelvic mass in a female is a leiomyoma. Am J Med Sci. 2013;345(1):72–4. https://doi.org/10.1097/MAJ.0b013e318262dc0d.View ArticlePubMedGoogle Scholar
- Jenayah AA, Bettaieb H, Saoudi S, Gharsa A, Sfar E, Boudaya F, et al. Desmoid tumors: clinical features and treatment options: a case report and a review of literature. Pan Afr Med J. 2015;21:93. https://doi.org/10.11604/pamj.2015.21.93.7037.View ArticlePubMedPubMed CentralGoogle Scholar
- Ono H, Hori K, Tashima L, Tsuruta T, Nakatsuka SI, Ito K. A case of retroperitoneal desmoid-type fibromatosis that involved the unilateral ureter after gynaecologic surgery. Int J Surg Case Rep. 2018;47:30–3. https://doi.org/10.1016/j.ijscr.2018.03.039.View ArticlePubMedPubMed CentralGoogle Scholar
- Stankiewicz A, Jeyadevan NN. Fibromatosis involving pelvic floor muscles. BJR Case Rep. 2016;2(3):20150239. https://doi.org/10.1259/bjrcr.20150239.View ArticlePubMedPubMed CentralGoogle Scholar
- Montoriol PF, Mons A, Da Ines D, Bourdel N, Tixier L, Garcier JM. Fibrous tumours of the ovary: aetiologies and MRI features. Clin Radiol. 2013;68(12):1276–83. https://doi.org/10.1016/j.crad.2013.07.005.View ArticlePubMedGoogle Scholar
- Nakanishi K, Shida D, Tsukamoto S, Ochiai H, Mazaki J, Taniguchi H, et al. Multiple rapidly growing desmoid tumors that were difficult to distinguish from recurrence of rectal cancer. World J Surg Oncol. 2017;15(1):180. https://doi.org/10.1186/s12957-017-1248-7.View ArticlePubMedPubMed CentralGoogle Scholar
- Chang CW, Wang TE, Chang WH, Yang TL, Chen CK, Hung YC, et al. Unusual presentation of desmoid tumor in the small intestine: a case report. Med Oncol. 2011;28(1):159–62. https://doi.org/10.1007/s12032-010-9429-z.View ArticlePubMedGoogle Scholar
- Bellia A, Vitale SG, Lagana AS, Cannone F, Houvenaeghel G, Rua S, et al. Feasibility and surgical outcomes of conventional and robot-assisted laparoscopy for early-stage ovarian cancer: a retrospective, multicenter analysis. Arch Gynecol Obstet. 2016;294(3):615–22. https://doi.org/10.1007/s00404-016-4087-9.View ArticlePubMedGoogle Scholar
- Chen C, Huang R, Liu P, Ouyang Z, Guo H, Tang L, et al. Construction and clinical significance of normal uterine arterial vascular network models. Gynecol Obstet Investig. 2010;69(1):14–9. https://doi.org/10.1159/000245941.View ArticleGoogle Scholar
- Gocmen A, Karaca M, Sari A. Conservative laparoscopic approach to adnexal torsion. Arch Gynecol Obstet. 2008;277(6):535–8. https://doi.org/10.1007/s00404-007-0501-7.View ArticlePubMedGoogle Scholar
- Huang C, Hong MK, Chu TY, Ding DC. A retrospective study of surgical treatment and outcome among women with adnexal torsion in eastern Taiwan from 2010 to 2015. PeerJ. 2018;6:e5995. https://doi.org/10.7717/peerj.5995.View ArticlePubMedPubMed CentralGoogle Scholar
- Taskin O, Birincioglu M, Aydin A, Buhur A, Burak F, Yilmaz I, et al. The effects of twisted ischaemic adnexa managed by detorsion on ovarian viability and histology: an ischaemia-reperfusion rodent model. Hum Reprod. 1998;13(1O):2823–7.View ArticleGoogle Scholar
- Lagana AS, Sofo V, Salmeri FM, Palmara VI, Triolo O, Terzic MM, et al. Oxidative stress during ovarian torsion in pediatric and adolescent patients: changing the perspective of the disease. Int J Fertil Steril. 2016;9(4):416–23.PubMedGoogle Scholar
- Sipahi M, Gunaydin M, Kesicioglu T, Usta M, Yavuz BT, Tomruk C. A new approach to prevent ischemia/reperfusion injury in a rat model: remote ischemic conditioning. Arch Gynecol Obstet. 2019. https://doi.org/10.1007/s00404-019-05149-1.View ArticleGoogle Scholar