Journal of Obstetrics and Gynaecology

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Concealed haematometra causing chronic upper abdominal pain S. Wali, J. Chatterjee, R. Zeegen & R. Smith To cite this article: S. Wali, J. Chatterjee, R. Zeegen & R. Smith (2015) Concealed haematometra causing chronic upper abdominal pain, Journal of Obstetrics and Gynaecology, 35:5, 537-539, DOI: 10.3109/01443615.2014.970148 To link to this article: http://dx.doi.org/10.3109/01443615.2014.970148

Published online: 14 Oct 2015.

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Gynaecology Case Reports  537 (Wheelock et al. 1984). In our case primary cancer in other organs was excluded and the tumour was derived from the Bartholin gland. In general, surgical treatment of the Bartholin gland carcinoma includes modified radical vulvectomy and ipsilateral, if the tumour is less than 20 mm and is not near the midline, or bilateral inguinal lymphadenectomy, should the mass be larger than 20 mm or near the midline (Finan and Barre 2003). In our case, it was decided to perform a left hemivulvectomy and ipsilateral inguinal lymphadenectomy. Primary clear-cell adenocarcinoma of the Bartholin gland has been reported only once before and in that case a wide local excision was performed followed by bilateral inguinal lymphadenectomy (Lim et  al. 2002). The surgical margins were free of disease and no pathological lymph nodes were found. The presence of metastasis in the inguinal lymph nodes is the most important prognostic factor for the Bartholin gland carcinoma (Wydra et al. 2006). Apart from the surgical approach, other treatment modalities have been reported concerning the management of primary Bartholin gland cancer. These include primary chemoradiation or pelvic radiation in selected cases (Massad and De Geest 1999).­

Figure 1. (a) Deep vulvar infiltration by clear-cell adenocarcinoma (Η-ΕX100). (b) The neoplasm has solid and tubular pattern and is composed of cells with abundant clear cytoplasm and eccentric nuclei (H-EX400).

diagnosis was that of clear-cell adenocarcinoma. The left Bartholin gland was enlarged, solid, about 3 centimetres in diameter and not tender, and the woman reported that she had noticed it growing since 6 months. Inguinal examination revealed no clinically apparent enlargement of the inguinal nodes. A computerised tomography scan of the abdomen and the pelvis, however, revealed an enlarged left inguinal node of 8 millimetres in diameter. The patient was subjected to left hemivulvectomy and left inguinal lymph node dissection. Macroscopically, a well-circumscribed, grey-white, firm–to-hard tumour, measuring a maximum of 2.2 cm in diameter, was observed in the anatomical site of Bartholin gland. Histologically, tumour proved to be a clear-cell adenocarcinoma with tubulocystic and solid patterns, consisting mainly of polygonal cells with abundant clear cytoplasm (Figure 1a and b). Hobnail cells were also noted. Remnants of Bartholin gland were found in the vicinity of the tumour, feature compatible with Bartholin’s origin. Post-operative course was uncomplicated and the woman was discharged from the hospital after thirteen days. The patient did not receive any adjuvant therapy and had a follow-up every six months. There has been no recurrence for three years and three months after treatment. During the initial follow-up period it was necessary for the patient to attend three psychotherapy sessions.

Discussion

The median age of the patients with primary carcinoma of the Bartholin gland is 57 years with a range of 14–85 years, and 38.3% were below 50 years of age, according to an analysis of 90 cases (Leuchter et  al. 1982). Furthermore, it is reported that the overall incidence of Bartholin gland carcinoma is similar in pre- and postmenopausal women (Visco and Del Priore 1996). Bartholin gland malignancies usually present with a painless tumour located on the posterior half of the vulva, while other symptoms might be present; therefore, a mass especially in a post-menopausal woman, located in the Bartholin gland should raise suspicions for malignancy (Finan and Barre 2003). However, due to the rarity of the condition and the fact that an enlargement in the region is often attributed to other causes, diagnosis might be delayed (Wydra et al. 2006). In order for primary carcinoma of the Bartholin gland to be diagnosed, apart from the exclusion of primary cancer elsewhere, the tumour should involve the area of the gland and there should be an area of histologic transition from normal to neoplastic tissue

Declaration of interest: The authors report no declarations of interest. The authors alone are responsible for the content and writing of the paper.

References

Finan MA, Barre G. 2003. Bartholin’s gland carcinoma, malignant melanoma and other rare tumours of the vulva. Best Practice Research Clinical Obstetrics Gynaecology 17:609–633. Forman D, De Martel C, Lacey CJ, Soerjomataram I, Lortet-Tieulent J, Bruni L et  al. 2012. Global burden of human papillomavirus and related diseases. Vaccine 30(Suppl 5):F12–23. Hwang TL, Hung YC, Chang HW. 2012. Adenoid cystic carcinoma of Bartholin’s gland. Taiwanese Journal of Obstetrics and Gynecology 51:119–20. Leuchter RS, Hacker NF, Voet RL, Berek JS, Townsend, DE, Lagasse LD. 1982. Primary carcinoma of the Bartholin gland: a report of 14 cases and review of the literature. Obstetrics & Gynecology 60:361–368. Lim KC, Thompson IW, Wiener, JJ. 2002. A case of primary clear cell adenocarcinoma of Bartholin’s gland. British Journal of Obstetrics and Gynaecology 109:1305–1307. Massad LS, De Geest K. 1999. Multimodality therapy for carcinoma of the Bartholin gland. Gynecologic Oncology 75:305–307. Ouldamer L, Chraibi Z, Arbion F, Barillot I, Body G. 2013. Bartholin’s gland carcinoma: epidemiology and therapeutic management. Surgical Oncology 22:117–122. Visco AG, Del Priore G. 1996. Postmenopausal bartholin gland enlargement: a hospital-based cancer risk assessment. Obstetrics & Gynecology 87:286–290. Wheelock JB, Goplerud DR, Dunn LJ, Oates JF, 3rd 1984. Primary carcinoma of the Bartholin gland: a report of ten cases. Obstetrics & Gynecology 63: 820–824. Woida FM, Ribeiro-Silva A. 2007. Adenoid cystic carcinoma of the Bartholin gland: an overview. Archives of Pathology & Laboratory Medicine 131: 796–798. Wydra D, Klasa-Mazurkiewicz D, Emerich J, Milczek T. 2006. The problem of accurate initial diagnosis of Bartholin’s gland carcinoma resulting in delayed treatment and aggressive course of the disease. International Journal of Gynecological Cancer 16:1469–1472.

Concealed haematometra causing chronic upper abdominal pain S. Wali1, J. Chatterjee1, R. Zeegen2 & R. Smith1 1Queen Charlotte’s and Chelsea Hospital, Obstetrics and

Gynaecology, London, UK and 2Chelsea and Westminster Hospital, Gastroentrology, London, UK DOI: 10.3109/01443615.2014.970148 Correspondence: Dr Sarah Wali, Queen Charlotte’s and Chelsea Hospital, Obstetrics and Gynaecology, London, UK. Tel:  07544391181. E-mail: Sarah. [email protected]

538  Gynaecology Case Reports

Background

Haematometra is a collection of menstrual blood in the uterus resulting in its distension and causing pain. It can be due to congenital malformations or most commonly secondary to cervical stenosis, the causes of which are numerous and include cervical cancer, infection, endometrial ablation, uterine curettage, cervical cryocoagulation and electrocoagulation, and trauma (childbirths and traumatic instrumental delivery). The aetiologies of upper abdominal pain are not commonly attributed to gynaecological pathology.

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Case report

We present an interesting and unusual clinical presentation of haematometra causing chronic upper abdominal pain in a peri-menopausal woman. A 52-year-old woman with parity of 2 (spontaneous vaginal delivery followed by caesarean section and sterilisation) underwent NovaSure endometrial ablation for menorrhagia. Immediately after surgery she felt her abdomen had become distended and continued to be so for the next 2 years, causing her discomfort and upper abdominal pain described as like ‘being pregnant’. The patient’s symptoms had a huge impact on her quality of life affecting all aspects of her daily routine, general health and sense of wellbeing. She gave no history of vaginal discharge or bleeding, urinary symptoms or abnormal bowel habits. She had occasional hot flushes. She had no significant medical history and no family history of note. She was on clonidine for hot flushes – the medication was initiated a year after her upper abdominal pain had started, so was not linked with the side effects (nausea, vomiting and constipation). Physical examination revealed a healthy non-obese woman. She was found to have substantial abdominal bloating on sitting and standing; however, the rest of her abdominal and pelvic examinations were normal with no palpable masses. She kept a daily morning and evening weight and girth chart for 1 week that showed no evidence of any idiopathic fluid retention syndrome. Given her symptoms, the patient was extensively investigated with a range of blood tests and multiple triple swabs for infection (including Chlamydia trachomatis and Neisseria gonorrhoeae) which were all negative. She had a computerised tomography (CT) scan of her abdomen and pelvis as well as serial ultrasound scans of her abdomen and pelvis over the 2 years (Figure 1), none of which showed any obvious signs for her symptoms and no sign of haematometra. She had several invasive diagnostic procedures including upper and lower gastrointestinal endoscopes (normal), a hysteroscopy (unable to enter the uterus due to cervical stenosis and an obliterated endometrial cavity) and a laparoscopy (hydrosalpinx bound in minor adhesions which was incised and drained). Following this extensive work up and lack of evidence for an organic cause of her pain, she was diagnosed with irritable bowel syndrome and offered several options to help with the pain including simple analgesia and hypnotherapy. However, the patient continued to be distraught by her symptoms and notwithstanding that she had

upper abdominal pain, she was offered a hysterectomy. During her laparoscopically assisted vaginal hysterectomy (LAVH) and bilateral salpingoopherectomy, it was found that she had a large central haematometra that was causing her uterus to be under much tension. During the LAVH, no peri-hepatic or omental adhesions were present. Histology of the specimen showed that the endometrial cavity was layered by haemorrhagic endometrium. Microscopically, there was a markedly atrophic endometrium without recognisable glands and focally replaced by haemosiderin-laden macrophages and chronic inflammatory cells. Following hysterectomy the patient’s upper abdominal pain resolved and her quality of life improved. She is still free of pain at 6 months after operation.

Discussion

Amenorrhoea rates for the NovaSure procedure range from 30% to 75% (Gimpelson 2014) and complete occlusion of the upper genital tract was found in 85.7% of women who underwent microwave endometrial ablation (Tawfeek et al. 2006). After endometrial ablation, our patient had amenorrhoea, which was the desired effect of the procedure. It was difficult to understand the connection between the procedure and her symptoms of upper abdominal pain and bloating. She had, however, a stenosis of her cervix that had led to gradual accumulation of menstrual blood resulting in haematometra. Consequently, she experienced chronic upper abdominal pain. The female internal reproductive organs share similar visceral innervation to parts of the hindgut as well as the lower ileum through the lumbar and pelvic splanchnic nerves (Hightower and Roberts 1981). Because of this shared pathway, differentiating between pain of gynaecological and gastrointestinal origin can be challenging due to referred visceral pain. In our patient’s case, it is possible that neuronal damage behind the endometrial ablation caused aberrant spinal pathways giving her the impression that pathology was in the upper abdomen when in fact the problem was in the pelvis. Another hypothesis for pelvic pain referring to the upper abdomen is that occasionally the omentum can be lying low in the abdomen and in the presence of adhesions (e.g., after a caesarean section or laparoscopic procedures) can be attached to the uterine body and this could stretch, pulling on the stomach and transverse colon to give discomfort and pain in the upper abdomen which is continuous and unrelated to bowel action. This mechanism was, however, excluded as a cause of pain in our patient during the time of laparoscopy which showed no omental adhesions. Unfortunately in our patient’s case, imaging modalities (Ultrasound and CT scan of the abdomen and pelvis) failed to detect the haematometra which was discovered at the time of her hysterectomy. Could further diagnostic imaging, for example a pelvic MRI, have prevented the prolonged period of frustration and multiple investigations for a patient who was found to have an organic cause for her pain? Despite the unusual location of the pain, the temporal association with endometrial ablation and the procedure’s known complications should have resulted in a quick decision to perform hysterectomy or drain the heamatometra by dilating the stenosed cervix and breaking down the adhesions. This might reveal the underlying pathology and avoid chronic morbidity as seen in this case.

Conclusion

Figure 1. USS 2013: Small uterus (6.6  3.8  3.2 cm) with fibroids (1.7 cm). The endometrium is thin (2.9 mm) and well defined.

Haematometra secondary to cervical stenosis, a known complication of endometrial ablation, is a common entity that should be considered in the differential diagnosis of chronic upper abdominal pain in women who have had the procedure. The diagnosis may be easily overlooked because gynaecologic pathology is not a common differential for upper abdominal pain so increased suspicion of this entity is essential. The fact that this patient had NovaSure ablation and the inflammatory changes found in the uterus possibly contributed to the imaging modalities that are not being accurate in diagnosing the pathology. We think that CT imaging of the pelvic organs are not always reliable (Walker et al. 2011) and an MRI pelvis would have probably provided better information about this patient’s uterus.

Gynaecology Case Reports  539 Declaration of interest: The authors report no declarations of interest. The authors alone are responsible for the content and writing of the paper.­

References

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Gimpelson R. 2014. Ten-year literature review of global endometrial ablation with the NovaSure® device. International Journal of Women’s Health 6:269–280. Hightower NC, Roberts JW. 1981. Acute and chronic lower abdominal pain of enterologic origin in chronic pelvic pain. In: Renaer MR, editor. Chronic pelvic pain in women. New York: Springer–Verlag. p 110–137. Tawfeek S, Sholapurkar S, Sharp N. 2006. Incidence of upper genital tract occlusion following microwave endometrial ablation (MEA). British Journal of Obstetrics and Gynaecology 113:958–960. Walker DK, Salibian RA, Salibian AD, Belen KM, Palmer SL. 2011. Overlooked diseases of the vagina: a directed anatomic pathalogic approach for imaging assessment. Radiographics 31:1583–1598.

Intravascular leiomyomatosis: A case report and review of the literature A. J. Simon1, W. R. Parry-Smith2, C. W. E. Redman3, M. Kodampur3, R. Todd3, C. Satur4 & R. Morgan5 1School of Medicine, Keele University Medical School, Keele University, Stoke-on-Trent, UK 2Department of Emergency, University Hospital of North Staffordshire, The Royal Infirmary, Staffordshire, Stoke-onTrent, UK, 3Department of Gynaecology, University Hospital of North Staffordshire, City General Hospital, Staffordshire, Stoke-onTrent, UK, 4Cardiothoracic Surgery, University Hospital of North Staffordshire, City General Hospital, Staffordshire, Stoke-on-Trent, UK, and 5Vascular Surgery, University Hospital of North Staffordshire, City General Hospital, Staffordshire, Stoke-on-Trent, UK

DOI: 10.3109/01443615.2014.978847 Correspondence: Mr Andrew James Simon, Fourth Year Medical Student, School of Medicine, Keele University Medical School, David Weatherall Building, Keele University, Stoke-on-Trent, ST5 5BG, UK. Tel: 07869375257. E-mail: [email protected]

Introduction

Intravascular leiomyomatosis (IVL) is a rare condition in which histologically benign smooth muscle neoplasms arising from the intrauterine vessels infiltrate and invade systemic veins, including the inferior vena cava (IVC) (Cho et  al. 2011). These tumours can further extend to the right heart and into the pulmonary arterial system, causing a pulmonary embolism (Fasih et  al. 2008). This is called intracardiac leiomyomatosis and may result in the development of cardiac symptoms, a murmur, syncope and even death (Lou et al. 2011). To correctly diagnose IVL pre-operatively, comprehensive imaging using computerised tomography (CT) and magnetic resonance imaging (MRI), and possibly other modalities, need to be incorporated (Pan et  al. 2013). Treatment is surgical, with excision of the primary uterine mass and often cardiotomy to remove any intracardiac lesion; though, unusually, we present a case without the need for cardiotomy, a staged approach or cardiothoracic surgery (Lam et al. 2003).

Case report

A previously fit 54-year-old woman presented to her general practitioner (GP) complaining of increasing dyspnoea of three-week duration. The patient had no significant medical history and had undergone menopause at age of 51 years. Her main co-morbidities were obsessive–compulsive disorder and a body mass index (BMI) of 42. On examination, the patient was noted to be dyspnoeic at rest and a pansystolic murmur was audible over the cardiac apex. Significant bilateral leg and sacral oedema were noted. Auscultation of the lungs was unremarkable. An echocardiogram demonstrated a mass in the IVC with extension to the right side of the heart and prolapse of the mass through the tricuspid valve. Subsequent CT scan of the chest, abdomen and

pelvis revealed a hypervascular, partly calcified uterine lesion, with axial dimensions of 22  10 cm with tumour thrombus extending through the right ovarian vein into the IVC and into the right ventricle (Figure 1a). Bilateral hydronephrosis, worse on the left was noted. Biochemistry confirmed renal impairment with an estimated glomerular filtration rate (eGFR) of 10 ml/min, urea of 27 mmol/l and creatinine of 413 mmol/l. A left-sided nephrostomy was inserted, although antegrade stenting was unsuccessful due to obstruction of the ureter by the mass. An MRI of the heart revealed a large tubular mass ascending the IVC, entering the right atrium and prolapsing through the tricuspid valve and into the right ventricle (Figure 1b). Pelvic MRI confirmed a large abdominopelvic solid mass involving the right uterine fundus (Figure 1c). Intermediate T2-weighted signal was noted, suggesting a carcinoma or sarcoma, rather than a benign pathology. CA-125 was raised at 139 (normal range: 0–35 u/ml). The endometrium and lymph nodes were normal. Following discussion at the gynaecology multi-disciplinary team meeting, an ultrasound-guided biopsy was performed, and the patient was commenced on prophylactic low-molecular-weight heparin. A retrograde left ureteric stent was inserted and renal function returned to normal. Biopsy revealed fragments of a vascular, benign spindle cell tumour, with cigar-shaped nuclei and a small eosinophilic cytoplasm. There was no evidence of necrosis, significant mitotic activity or pleomorphism. Immunochemistry was strongly positive for actin and desmin and positive for B-cell lymphoma 2 (BCL-2). There was also strong positivity for oestrogen and progesterone receptors. There was negative staining with S-100 and CD34 and CD10 showed weak focal positivity. The MIB-1 stain showed an extremely low proliferative index (less than 1%). The provisional diagnosis at this stage was of a fibroid uterus with benign intravascular leiomyomatosis. Further multidisciplinary meetings involving gynaecological oncologists, cardiothoracic and vascular surgeons decided on performing an initial exploratory laparotomy to assess the pelvic and abdominal disease, proceeding to remove the vascular/cardiac disease if the pelvic disease was resectable. To reduce tumour bulk the patient was started on a gonadotropin-releasing hormone analogue and an aromatase inhibitor for three months; however, repeat preoperative imaging revealed only a small reduction in the size of the mass. At laparotomy, the findings were of a large uterine mass (20 cm craniocaudal dimensions) with adherence to surrounding small bowel mesentery and omentum (Figure 1d). Large, tumour-filled plexi of veins were noted, extending from the right side of the uterus into the uterine vessels and the common iliac vein. After initial adhesiolysis, a total abdominal hysterectomy and bilateral salpingo-oophorectomy were performed. The right common iliac vein and common iliac artery were delineated and the vena cava was dissected up to the level of the right renal vein. The IVC was noted to be distended and a nonadherent mass could be felt within. Control of the IVC was obtained digitally in its infra-renal portion and control of the iliac vein distally was achieved, clear of the right iliac fossa mass. Venotomy over the area of the most significant adhesion to the right lateral wall of the distal IVC was performed. The intra caval extension of the tumour was free of any attachment barring the 2  3-cm-diameter anchor point within the most distal IVC and right iliac vein. Gentle traction on the IVC extension of the tumour allowed easy delivery through the venotomy. Cardiac echo at the time of manipulation confirmed movement of the mass within the right atrium towards the superior VC–IVC junction when gentle traction was applied distally. Complete removal of the IVC and cardiac mass were achieved without difficulty.

Discussion

Intravascular leiomyomatosis usually occurs from co-existing or previously resected uterine leiomyomas. Due to its rarity, the diagnosis of IVL is typically delayed. Pre-operative misdiagnosis is common, and means that many patients initially receive inappropriate treatment. Removal of the intra-cardiac mass by traction and avoiding

Concealed haematometra causing chronic upper abdominal pain.

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