Journal of Obstetrics and Gynaecology

ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20

Haemoperitoneum: An extremely rare complication of leiomyoma Ç. Aydin, H. Şen Selim, S. Eriş & Y. Yalçin To cite this article: Ç. Aydin, H. Şen Selim, S. Eriş & Y. Yalçin (2015) Haemoperitoneum: An extremely rare complication of leiomyoma, Journal of Obstetrics and Gynaecology, 35:1, 109-110, DOI: 10.3109/01443615.2014.937680 To link to this article: http://dx.doi.org/10.3109/01443615.2014.937680

Published online: 24 Jul 2014.

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Gynaecology Case Reports and the subsequent analysis indicated that all samples were benign. Further pathological examination showed the following: mucinous adenocarcinoma and AM of the cervix (Figure 1b, c), and mucinous cystadenoma of the right ovary (Figure 1d). CEA, CDX2, CD20 and CD7 immunochemical staining was performed. Positron emission tomography–computed tomography (PET–CT) was performed and showed no abnormal findings. The final diagnosis was: ‘Stage Ib well-differentiated mucinous adenocarcinoma and adenoma malignum of the cervix’. The patient had an uneventful postoperative course, underwent radiotherapy following surgery and had a 6-month disease-free survival.

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Discussion Profuse watery vaginal discharge and abnormal vaginal bleeding are the most common symptoms observed in AM (Chang Jie et al. 2008; Lim et al. 2012). Ultrasonographic features include: cervical enlargement, multiloccular cyctic masses with solid components and homogeneously hypoechoic fluid (Chang Jie et al. 2008; Lim et al. 2012). Ultrasound imaging with Doppler examination was reported to be more efficient and accurate in the evaluation of such lesions. On Doppler examination, AM shows intralesional vascularity. However, the pseudocapsule of an intramural cervical myoma usually shows an arc of vascularity observed as a ‘peripheral rim’ and benign multilocular lesions, such as multiple nabothian cysts, show no intralesional vascularity (Park et al. 2011). CT findings are often nonspecific. Magnetic resonance imaging findings suggestive of AM include multiple irregular cystic lesions, which mimic benign nabothian cysts. These lesions often occur with solid components located within the deep cervical stroma (Doi et al. 1997; Itoh et al. 2000; Okamoto et al. 2003). In our case, the only complaint from the patient was profuse watery discharge. Neither ultrasonography nor CT revealed a specific finding that may be compatible with AM. Diagnosing AM preoperatively is difficult if the physician does not examine the patient with particular consideration of AM. Because of its deep locating in the endocervical stroma and its rarity, determining an accurate cytological diagnosis for AM is difficult. Several studies have concluded that a Pap smear is not as important for detecting AM (Kaminski and Norris 1983; Gilks et al. 1989). In our case, the final surgical pathology confirmed the diagnosis of ‘well-differentiated mucinous adenocarcinoma and adenoma malignum of the cervix’. The final diagnosis was made after surgery was performed based on a misdiagnosis. An association exists between AM and ovarian mucinous adenocarcinoma. Additionally, the possibility of coincidental mucinous tumours of the ovaries was suggested. In our case, AM and right ovarian mucinous adenoma were diagnosed coincidentally. The prognosis has been differentially reported in various studies (Gilks et al. 1989; Silverberg and Hurt 1975). Treatment of AM is similar to that of endocervical adenocarcinomas and surgery is the standard treatment modality (Lim et al. 2012; Chang et al. 2008). In our case, the patient underwent simple hysterectomy and bilateral salpingo-oophorectomy. Depending on the benign findings of frozen section analysis, a more extensive surgical procedure was not necessary. Further pathological examination of the cervix from the hysterectomy specimen showed mucinous adenocarcinoma and AM of the cervix. The patient was referred for medical oncology, and radiation oncology consultation and radiotherapy was planned. In conclusion, precise diagnosis of adenoma malignum before histopathological examination of the specimen may be difficult.

Acknowledgements We would like to thank Textcheck (English Editing Service). Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.

References Chang Jie ZS, Zhou H, Liang J-X et al. 2008. Clinical analysis of minimal deviation adenocarcinoma of the cervix, a report of five cases. Chinese Journal of Cancer 27:1310–1314.

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Doi T, Yamashita Y, Yasunaga T et al. 1997. Adenoma malignum: MR imaging and pathologic study. Radiology 204:39–42. Gilks CB, Young RH, Aguirre P et al. 1989. Adenoma malignum (minimal deviation adenocarcinoma) of the uterine cervix. A clinicopathological and immunohistochemical analysis of 26 cases. American Journal of Surgical Pathology 13:717–729. Itoh K, Toki T, Shiohara S et al. 2000. A comparative analysis of cross sectional imaging techniques in minimal deviation adenocarcinoma of the uterine cervix. British Journal of Obstetrics and Gynaecology 107:1158–1163. Kaminski PF, Norris HJ. 1983. Minimal deviation carcinoma (adenoma malignum) of the cervix. International Journal of Gynecological Pathology 2:141–152. Lim KT, Lee IH, Kim TJ et al. 2012. Adenoma malignum of the uterine cervix: Clinicopathologic analysis of 18 cases. Kaohsiung Journal of Medical Sciences 28:161–164. Okamoto Y, Tanaka YO, Nishida M et al. 2003. MR imaging of the uterine cervix: imaging-pathologic correlation. Radiographics 23:425–445; quiz 534–535. Park SB, Moon MH, Hong SR et al. 2011. Adenoma malignum of the uterine cervix: ultrasonographic findings in 11 patients. Ultrasound in Obstetrics and Gynecology, 38:716–721. Silverberg SG, Hurt WG. 1975. Minimal deviation adenocarcinoma (‘adenoma malignum’) of the cervix: a reappraisal. American Journal of Obstetrics and Gynecology 121:971–975.

Haemoperitoneum: An extremely rare complication of leiomyoma Ç. Aydin, H. Şen Selim, S. Eriş & Y. Yalçin Gynecology and Obstetrics Department of Izmir Ataturk Training and Research Hospital, Izmir, Turkey DOI: 10.3109/01443615.2014.937680 Correspondence: Ç. Aydın, Gynecology and Obstetrics Department of Ataturk Training and Research Hospital, Basin Sitesi, Yesilyurt, 35360 Izmır, Turkey. E-mail: [email protected]

Case report A 31-year-old nulliparous woman presented to our hospital on the first day of her menstrual cycle complaining of abdominal pain. The patient had no history of traumatic or violent coitus. She had been using an oral contraceptive pill for over a year. Upon physical examination, she was afebrile and very pale, with a pulse of 110 b.p.m. and an arterial blood pressure of 80/50 mmHg. Her abdomen was distended with generalised tenderness. A pelvic examination revealed a mass consistent with a 14–16 week gestational uterus. Her serum β-hCG level was ⬍ 2 IU and her haemoglobin level was 8.52 g/dl. Computerised tomography (CT) and ultrasonography revealed a uterine leiomyoma and intraperitoneal haemorrhage. The patient became haemodynamically unstable; therefore, an exploratory laparotomy was performed, during which approximately 3,000 ml of blood was found in the peritoneal cavity. The patient required 4 U of packed red blood cells during surgery. Pelvic exploration identified a large leiomyoma 14–16 weeks in size, which encompassed the whole uterine corpus; this is known traditionally as a Kugel myoma. Multiple abnormal varicose vessels were found on the surface of the leiomyoma (Figure 1). The haemorrhage was caused by rupture of the dilated superficial veins of the leiomyoma. No other sources of bleeding were found. The bleeding remained uncontrolled after suture of the ruptured vessels and a subsequent uterine artery ligation. As conservative surgical methods were unsuccessful and the patient remained haemodynamically unstable, a subtotal hysterectomy without bilateral salpingo-oophorectomy was necessary immediately in this emergency situation. Histopathological evaluation of the leiomyoma revealed uniform smooth muscle fibres with multiple areas of degeneration but without cellular atypia or mitotic activity. The postoperative course was uneventful, except that the patient developed sub-ileus 4 days after surgery. At 9 days after surgery, the patient was discharged from hospital in good clinical condition.

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Figure 1. Pelvic exploration identified a large leiomyoma 14–16 weeks in size. There were multiple abnormal varicose vessels on the surface of the leiomyoma.

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Discussion Haemoperitoneum in a woman of reproductive age is usually caused by an ectopic pregnancy, rupture or torsion of an ovarian cyst, or by non-gynaecological reasons. Bleeding from the superficial vessels of a leiomyoma is rare; fewer than 100 cases have been reported in the literature (Wong et al. 2005). Bleeding is the result of trauma or torsion in most cases, and spontaneous rupture of a superficial vein is extremely rare (Akahira et al. 1998; Jain et al. 2004). Several theories have been formulated to explain the cause of a spontaneous rupture of a uterine vessel. One such theory states that, in a leiomyoma ⬎ 10 cm in diameter, tears in the overlying vessels may be caused by the pressure exerted by the growing leiomyoma (Mattison and Yeh 1980). Alternatively, a uterine blood vessel may be distended and, as a result, torn by uterine contractions during menstruation, which may have been the case in the current patient (Mattison and Yeh 1980). Menstruation and pregnancy may both increase congestion of the superficial veins of a leiomyoma. Hormonal therapy may be a risk factor for uterine bleeding. Indeed, a related case was reported by Tang et al. (2008) who wrote, ‘a post-menopausal woman on hormone-replacement therapy presented with massive intra-abdominal haemorrhage due to bleeding from fibroids’. Our patient had been using an oral contraceptive pill for over a year. Preoperative diagnosis of haemoperitoneum in a patient in an emergency condition is difficult; in most cases, the diagnosis is made only during the laparotomy. Common initial diagnoses include acute abdominal and intra-abdominal bleeding from an unknown source. Ultrasonography, magnetic resonance imaging (MRI) and CT are all modalities that may help to diagnose haemoperitoneum. However, such techniques can only identify a leiomyoma with a large mass and/ or fluid in the peritoneal cavity; these diagnostic methods cannot determine the exact origin of the bleeding. Contrast-enhanced 2- and 3-dimensional sonography (Liu et al. 2002) and three-dimensional contrast-enhanced MRI (Weishaupt et al. 2000) are both state-of-theart modalities for the accurate diagnosis of intra-abdominal bleeding; however, a sufficiently quick performance of these techniques may be difficult.

The management of haemoperitoneum is supportive and surgical. Blood transfusions and intravenous fluids should be given. Exploratory laparotomy should be performed as soon as possible to allow diagnosis and definitive treatment to begin. Haemostatic suturing, myomectomy or hysterectomy may be used to control the bleeding; however, myomectomy may be difficult, or impossible, to perform. The decision to perform a hysterectomy should take the woman’s age and her desire to have children into consideration (Lotterman 2008). In the present case, performance of a myomectomy was impossible because the uterine corpus was globally and symmetrically enlarged (Kugel myoma); therefore, a subtotal hysterectomy was performed. The specimen included a partial upper segment of the cervix, and the lesion included a minimal cervical extension. On microscopic evaluation, spindle cell smooth muscle proliferation was observed as intersected fascicles. Some areas showed hyalinisation and degenerative changes. Conservative myomectomy may be attempted in similar cases – with the exception of Kugel myomas – if the location of the leiomyoma is appropriate and future fertility is desired. To conclude, emergency physicians and gynaecologists should be aware that women with fibroids who present with an acute abdomen may be suffering from haemoperitoneum, although the condition is rare. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.

References Akahira J, Ito K, Nakamura R et al. 1998. Massive intraperitoneal hemorrhage and hypovolemic shock due to rupture of a coronary vessel of a uterine leiomyoma: a report of two cases. Tohoku Journal of Experimental Medicine 185:217–222. Jain P, Pradhan P, Cietak KA et al. 2004. Acute abdomen following spontaneous variceal rupture overlying uterine leiomyoma. Journal of Obstetrics and Gynaecology 24:589. Liu JB, Merton DA, Goldberg BB et al. 2002. Contrast-enhanced two- and threedimensional sonography for evaluation of intra-abdominal hemorrhage. Journal of Ultrasound in Medicine 21:161–169. Lotterman S.2008. Massive hemoperitoneum resulting from spontaneous rupture of uterine leiomyoma. American Journal of Emergency Medicine 26:974.e1–e2. Mattison DR, Yeh SY. 1980. Hemoperitoneum from rupture of a uterine vein overlying a leiomyoma. American Journal of Obstetrics and Gynecology 136:415–416. Tang A, Rao S, Cawdell G. 2008. Massive intra-abdominal haemorrhage due to spontaneous bleeding from fibroids in a post-menopausal woman. Journal of Obstetrics and Gynaecology 28:244–245. Weishaupt D, Hetzer FH, Ruehm SG et al. 2000. Three-dimensional contrastenhanced MRI using an intravascular contrast agent for detection of traumatic intra-abdominal hemorrhage and abdominal parenchymal injuries: an experimental study. European Radiology 10:1958–1964. Wong L, Ching TW, Kok TL et al. 2005. Spontaneous hemoperitoneum from a uterine leiomyoma in pregnancy. Acta Obstetricia et Gynecologica Scandinavica 84:1208–1209.

Haemoperitoneum: an extremely rare complication of leiomyoma.

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