Journal of Obstetrics and Gynaecology
ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20
An unusual ovarian cyst N. Rambocas & D. Murphy To cite this article: N. Rambocas & D. Murphy (2014) An unusual ovarian cyst, Journal of Obstetrics and Gynaecology, 34:5, 453-454 To link to this article: http://dx.doi.org/10.3109/01443615.2014.902039
Published online: 11 Apr 2014.
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Gynaecology Case Reports 453 The patient presented with partner discomfort on intercourse on first erosion and with a stone in the vagina the second time. The lack of discharge and bleeding is not unusual with late erosion, which can be relatively asymptomatic (Ward and Hilton 2008), whereas discharge and bleeding tend to be associated with early erosion (Kobashi and Govier 2003). It is important however, to realise that the clinical presentation of mesh complications can vary and infected mesh can present late with discharge, even if it is not associated with mesh erosion (Ismail 2007). The erosion described here is also notable, in that it persisted and recurred. There is only one case report in the literature describing recurrent vaginal mesh erosion after tension-free vaginal tape (TVT) sling insertion, which was associated with actinomyces infection, which was identified from histology of the excised vaginal edges on recovering the exposed mesh (Bafghi et al. 2005). Histological examination was not performed in the case described here, but this is worth doing if she develops any recurrence in the future, or in similar cases, to guide the use of antibiotics. The recurrence after burial of mesh erosion also raises questions as to whether this approach is better than excision or not. This is an area that can be addressed by a large multicentre randomised controlled trial, given the small number of cases likely to be encountered in individual units. (A national registry, such as the British Society of Urogynaecology surgical database: www.bsug.net, may be helpful in this respect.) Stone formation is a recognised complication of mid-urethral tape mesh erosion into the bladder, as a result of urinary calcium deposition on the eroding mesh (Mustafa and Wadie 2007). However, this is the first time stone formation has been reported for mesh erosion into the vagina and is likely to be the result of urine backflow into the vagina as the patient passed urine. The recurrent erosion actually must have been there for some time before presentation, for the stone to form and reach 1 cm size. For this reason, there is a point in following them up until complete healing of the erosion, to ensure they do not develop such encrustation, which might get larger with time and cause ulceration and fistula formation.
Acknowledgements The authors are grateful to Mrs Margaret Wiliams and Mrs Lorna Keyes for their help with case notes retrieval. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Bafghi A, Colomb F, Vandenbos F et al. 2005. Recurrent vaginal erosion of tension free vaginal tape due to Actinomyces infection. British Journal of Obstetrics and Gynaecology 112:1661–1662. Ismail SI. 2007. Mesh infection without erosion after ObTape sling insertion: a diagnostic challenge. International Urogynecology Journal 18:1115–1118. Kobashi KC, Govier FE. 2003. Management of vaginal erosion of polypropylene mesh slings. Journal of Urology 169:2242–2243. Mustafa M, Wadie BS. 2007. Bladder erosion of tension-free vaginal tape presented as vesical stone; management and review of literature. International Urology Nephrology 39:453–455. Ward K, Hilton P; on behalf of the UK and Ireland TVT Trial Group. 2008. Tension-free vaginal tape versus colposuspension for primary urodynamic stress incontinence: 5-year follow-up. British Journal of Obstetrics and Gynaecology 115:226–233.
Case report A 68-year-old woman presented to the cardiologist with a twelvemonth history of leg swelling, shortness of breath and fatigue. She also complained of abdominal pains, altered bowel habits, weight loss and some episodes of generalised pruritus and flushing. On examination, she had a raised jugular venous pressure, bilateral pedal oedema and various cardiac murmurs. Due to the cardiac findings, an echocardiogram was done, which showed marked tricuspid and pulmonary regurgitation, suggestive of carcinoid heart disease. The left side of the heart was unremarkable. Abdominal and pelvic ultrasound scans were ordered, together with a 24 h urine collection for 5 hydroxyindoleacetic acid (5HIAA), which was markedly elevated. The abdominal ultrasound scan was normal. However, in the pelvis, there was an abnormal looking solid cystic mass measuring 10 ⫻ 15 cm with Doppler blood flow. A pelvic CT scan showed this mass to be arising from the ovary, with no evidence of metastatic disease. It was hypothesised that this was a pelvic tumour secreting high levels of serotonin resulting in carcinoid heart disease. She was started on Octreotide treatment. She was referred to the gynaecological oncology team where her operation was scheduled to be performed with cardiology and intensive care support. At laparotomy, she had a normal uterus and right ovary. However, there was a left ovarian mass with ascites, marked pelvic varices and severe retroperitoneal fibrosis. As a result of this, the left tube and ovary were removed and biopsies were taken. Close inspection of the large and small bowels, together with the appendix revealed these to be normal. She had a long postoperative recovery period, with intensive care support. About seven months later, due to the valvular heart damage, she had tricuspid and pulmonary valve replacements. The histology of the left ovary showed a tumour which was composed of small, round darkly staining cells arranged into islands and acini structures (i.e. an insular growth pattern). There was no mitotic activity and anaplasia. The serosal surface of the ovary was intact. There was also a cystic component of the ovary, which contained keratinous debris and hair shaft fragments, which suggested that the cyst was dermoid in nature (Figure 1). Immunohistochemistry showed the tumour cells to express the neuroendocrine markers CD56, chromogranin and synaptophysin. The appearance was suggestive of a carcinoid tumour within a mature cystic teratoma, rather than a metastatic carcinoid tumour. The biopsies confirmed fibrotic connective tissue but no malignancy.
Discussion Carcinoid tumours are rare tumours of the neuroendocrine system that most commonly occur in the gut. Only 0.3% of carcinoid tumours
An unusual ovarian cyst N. Rambocas & D. Murphy Department of Gynaecological Oncology, New Cross Hospital, Wolverhampton, UK DOI: 10.3109/01443615.2014.902039 Correspondence: N. Rambocas, Department of Gynaecological Oncology, New Cross Hospital, Wednesfield Road, Wolverhampton, Wolverhampton WV10 0QP, UK. E-mail: [email protected]
Figure 1. Left ovary with the tumour.
Downloaded by [York University Libraries] at 04:04 06 November 2015
454
Gynaecology Case Reports
are ovarian in origin (Mordi and Bridges 2011). There are different types of carcinoid tumours of the ovary and they include the insular (most common), strumal and mucinous types. The majority of ovarian carcinoids occur in association with mature cystic teratoma, as with our case. These tumours tend to occur in the postmenopausal age group. Primary ovarian carcinoids tend to be unilateral, while the metastatic carcinoids are nearly always bilateral (Talerman 1984). Immunohistochemistry markers, such as chromogranin and synaptophysin are commonly used for the detection of carcinoid tumours. Cytokeratin 7 (CK7) can also be used but is positive in only 24% of tumours. CD56 can also be used but it is not very specific or sensitive enough for the carcinoid tumours (Zhao et al. 2007). Only the insular type is associated with the carcinoid syndrome (Talerman 1984). About two-thirds of patients with carcinoid syndrome have evidence of carcinoid heart disease (Sabatini et al. 2000). If heart failure develops at the same time as the presence of an ovarian tumour, removal of the tumour will improve the cardiac function. However, cardiac disease can still occur, despite tumour removal (Mordi and Bridges 2011). Carcinoid heart disease is rare and tends to affect the right heart valves. The characteristic findings are endocardial plaques of fibrous tissue that can involve the tricuspid and pulmonary valves, cardiac chambers, pulmonary artery and coronary sinus. The fibrous tissue in the plaques distorts the valves leading to either stenosis, regurgitation or both. The preferential right heart involvement is most likely related to inactivation of the vasoactive substances by the lungs (Fox and Khattar 2004). About 5–10% of cases have left-sided heart involvement. The three-year mortality rate of carcinoid heart disease is about 31% (Fox and Khattar 2004). In our case, the patient already had
cardiac involvement at the time of diagnosis and despite tumour removal, her cardiac function still deteriorated. The rate of metastases of primary ovarian carcinoids is not very common. In one study by Soga et al. (2000) looking at 329 cases of ovarian carcinoids, a metastatic rate for carcinoids occurring within dermoids was 5.8%. Hence, tumours confined to one ovary can be treated surgically with good overall outcome (Davis et al. 1996). At present, our patient is tumour-free but is still receiving treatment for her cardiac problems. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Davis KP, Hartmann LK, Keeney GL et al. 1996. Primary ovarian carcinoid tumors. Gynecologic Oncology 61:259–265. Fox DJ, Khattar RS. 2004. Carcinoid heart disease: presentation, diagnosis and management. Heart 90:1224–1228. Mordi IR, Bridges A. 2011. A rare case of ovarian carcinoid causing heart failure. Scottish Medical Journal 56:181. Sabatini T, Rozzini R, Morandi GB et al. 2000. Primary carcinoid tumor of the ovary: report of an unusual case. Tumori 86:91–94. Soga J, Osaka M, Yakuwa Y. 2000. Carcinoids of the ovary: an analysis of 329 reported cases. Journal of Experimental and Clinical Cancer Research 19:271–280. Talerman A. 1984. Carcinoid tumors of the ovary. Journal of Cancer Research and Clinical Oncology 107:125–135. Zhao C, Bratthauer GL, Barner R et al. 2007. Comparative analysis of alternative and traditional immunohistochemical markers for the distinction of ovarian Sertoli cell tumor from endometrioid tumors and carcinoid tumor: A study of 160 cases. American Journal of Surgical Pathology 31:255–266.
ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20
An unusual ovarian cyst N. Rambocas & D. Murphy To cite this article: N. Rambocas & D. Murphy (2014) An unusual ovarian cyst, Journal of Obstetrics and Gynaecology, 34:5, 453-454 To link to this article: http://dx.doi.org/10.3109/01443615.2014.902039
Published online: 11 Apr 2014.
Submit your article to this journal
Article views: 47
View related articles
View Crossmark data
Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=ijog20 Download by: [York University Libraries]
Date: 06 November 2015, At: 04:04
Downloaded by [York University Libraries] at 04:04 06 November 2015
Gynaecology Case Reports 453 The patient presented with partner discomfort on intercourse on first erosion and with a stone in the vagina the second time. The lack of discharge and bleeding is not unusual with late erosion, which can be relatively asymptomatic (Ward and Hilton 2008), whereas discharge and bleeding tend to be associated with early erosion (Kobashi and Govier 2003). It is important however, to realise that the clinical presentation of mesh complications can vary and infected mesh can present late with discharge, even if it is not associated with mesh erosion (Ismail 2007). The erosion described here is also notable, in that it persisted and recurred. There is only one case report in the literature describing recurrent vaginal mesh erosion after tension-free vaginal tape (TVT) sling insertion, which was associated with actinomyces infection, which was identified from histology of the excised vaginal edges on recovering the exposed mesh (Bafghi et al. 2005). Histological examination was not performed in the case described here, but this is worth doing if she develops any recurrence in the future, or in similar cases, to guide the use of antibiotics. The recurrence after burial of mesh erosion also raises questions as to whether this approach is better than excision or not. This is an area that can be addressed by a large multicentre randomised controlled trial, given the small number of cases likely to be encountered in individual units. (A national registry, such as the British Society of Urogynaecology surgical database: www.bsug.net, may be helpful in this respect.) Stone formation is a recognised complication of mid-urethral tape mesh erosion into the bladder, as a result of urinary calcium deposition on the eroding mesh (Mustafa and Wadie 2007). However, this is the first time stone formation has been reported for mesh erosion into the vagina and is likely to be the result of urine backflow into the vagina as the patient passed urine. The recurrent erosion actually must have been there for some time before presentation, for the stone to form and reach 1 cm size. For this reason, there is a point in following them up until complete healing of the erosion, to ensure they do not develop such encrustation, which might get larger with time and cause ulceration and fistula formation.
Acknowledgements The authors are grateful to Mrs Margaret Wiliams and Mrs Lorna Keyes for their help with case notes retrieval. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Bafghi A, Colomb F, Vandenbos F et al. 2005. Recurrent vaginal erosion of tension free vaginal tape due to Actinomyces infection. British Journal of Obstetrics and Gynaecology 112:1661–1662. Ismail SI. 2007. Mesh infection without erosion after ObTape sling insertion: a diagnostic challenge. International Urogynecology Journal 18:1115–1118. Kobashi KC, Govier FE. 2003. Management of vaginal erosion of polypropylene mesh slings. Journal of Urology 169:2242–2243. Mustafa M, Wadie BS. 2007. Bladder erosion of tension-free vaginal tape presented as vesical stone; management and review of literature. International Urology Nephrology 39:453–455. Ward K, Hilton P; on behalf of the UK and Ireland TVT Trial Group. 2008. Tension-free vaginal tape versus colposuspension for primary urodynamic stress incontinence: 5-year follow-up. British Journal of Obstetrics and Gynaecology 115:226–233.
Case report A 68-year-old woman presented to the cardiologist with a twelvemonth history of leg swelling, shortness of breath and fatigue. She also complained of abdominal pains, altered bowel habits, weight loss and some episodes of generalised pruritus and flushing. On examination, she had a raised jugular venous pressure, bilateral pedal oedema and various cardiac murmurs. Due to the cardiac findings, an echocardiogram was done, which showed marked tricuspid and pulmonary regurgitation, suggestive of carcinoid heart disease. The left side of the heart was unremarkable. Abdominal and pelvic ultrasound scans were ordered, together with a 24 h urine collection for 5 hydroxyindoleacetic acid (5HIAA), which was markedly elevated. The abdominal ultrasound scan was normal. However, in the pelvis, there was an abnormal looking solid cystic mass measuring 10 ⫻ 15 cm with Doppler blood flow. A pelvic CT scan showed this mass to be arising from the ovary, with no evidence of metastatic disease. It was hypothesised that this was a pelvic tumour secreting high levels of serotonin resulting in carcinoid heart disease. She was started on Octreotide treatment. She was referred to the gynaecological oncology team where her operation was scheduled to be performed with cardiology and intensive care support. At laparotomy, she had a normal uterus and right ovary. However, there was a left ovarian mass with ascites, marked pelvic varices and severe retroperitoneal fibrosis. As a result of this, the left tube and ovary were removed and biopsies were taken. Close inspection of the large and small bowels, together with the appendix revealed these to be normal. She had a long postoperative recovery period, with intensive care support. About seven months later, due to the valvular heart damage, she had tricuspid and pulmonary valve replacements. The histology of the left ovary showed a tumour which was composed of small, round darkly staining cells arranged into islands and acini structures (i.e. an insular growth pattern). There was no mitotic activity and anaplasia. The serosal surface of the ovary was intact. There was also a cystic component of the ovary, which contained keratinous debris and hair shaft fragments, which suggested that the cyst was dermoid in nature (Figure 1). Immunohistochemistry showed the tumour cells to express the neuroendocrine markers CD56, chromogranin and synaptophysin. The appearance was suggestive of a carcinoid tumour within a mature cystic teratoma, rather than a metastatic carcinoid tumour. The biopsies confirmed fibrotic connective tissue but no malignancy.
Discussion Carcinoid tumours are rare tumours of the neuroendocrine system that most commonly occur in the gut. Only 0.3% of carcinoid tumours
An unusual ovarian cyst N. Rambocas & D. Murphy Department of Gynaecological Oncology, New Cross Hospital, Wolverhampton, UK DOI: 10.3109/01443615.2014.902039 Correspondence: N. Rambocas, Department of Gynaecological Oncology, New Cross Hospital, Wednesfield Road, Wolverhampton, Wolverhampton WV10 0QP, UK. E-mail: [email protected]
Figure 1. Left ovary with the tumour.
Downloaded by [York University Libraries] at 04:04 06 November 2015
454
Gynaecology Case Reports
are ovarian in origin (Mordi and Bridges 2011). There are different types of carcinoid tumours of the ovary and they include the insular (most common), strumal and mucinous types. The majority of ovarian carcinoids occur in association with mature cystic teratoma, as with our case. These tumours tend to occur in the postmenopausal age group. Primary ovarian carcinoids tend to be unilateral, while the metastatic carcinoids are nearly always bilateral (Talerman 1984). Immunohistochemistry markers, such as chromogranin and synaptophysin are commonly used for the detection of carcinoid tumours. Cytokeratin 7 (CK7) can also be used but is positive in only 24% of tumours. CD56 can also be used but it is not very specific or sensitive enough for the carcinoid tumours (Zhao et al. 2007). Only the insular type is associated with the carcinoid syndrome (Talerman 1984). About two-thirds of patients with carcinoid syndrome have evidence of carcinoid heart disease (Sabatini et al. 2000). If heart failure develops at the same time as the presence of an ovarian tumour, removal of the tumour will improve the cardiac function. However, cardiac disease can still occur, despite tumour removal (Mordi and Bridges 2011). Carcinoid heart disease is rare and tends to affect the right heart valves. The characteristic findings are endocardial plaques of fibrous tissue that can involve the tricuspid and pulmonary valves, cardiac chambers, pulmonary artery and coronary sinus. The fibrous tissue in the plaques distorts the valves leading to either stenosis, regurgitation or both. The preferential right heart involvement is most likely related to inactivation of the vasoactive substances by the lungs (Fox and Khattar 2004). About 5–10% of cases have left-sided heart involvement. The three-year mortality rate of carcinoid heart disease is about 31% (Fox and Khattar 2004). In our case, the patient already had
cardiac involvement at the time of diagnosis and despite tumour removal, her cardiac function still deteriorated. The rate of metastases of primary ovarian carcinoids is not very common. In one study by Soga et al. (2000) looking at 329 cases of ovarian carcinoids, a metastatic rate for carcinoids occurring within dermoids was 5.8%. Hence, tumours confined to one ovary can be treated surgically with good overall outcome (Davis et al. 1996). At present, our patient is tumour-free but is still receiving treatment for her cardiac problems. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References Davis KP, Hartmann LK, Keeney GL et al. 1996. Primary ovarian carcinoid tumors. Gynecologic Oncology 61:259–265. Fox DJ, Khattar RS. 2004. Carcinoid heart disease: presentation, diagnosis and management. Heart 90:1224–1228. Mordi IR, Bridges A. 2011. A rare case of ovarian carcinoid causing heart failure. Scottish Medical Journal 56:181. Sabatini T, Rozzini R, Morandi GB et al. 2000. Primary carcinoid tumor of the ovary: report of an unusual case. Tumori 86:91–94. Soga J, Osaka M, Yakuwa Y. 2000. Carcinoids of the ovary: an analysis of 329 reported cases. Journal of Experimental and Clinical Cancer Research 19:271–280. Talerman A. 1984. Carcinoid tumors of the ovary. Journal of Cancer Research and Clinical Oncology 107:125–135. Zhao C, Bratthauer GL, Barner R et al. 2007. Comparative analysis of alternative and traditional immunohistochemical markers for the distinction of ovarian Sertoli cell tumor from endometrioid tumors and carcinoid tumor: A study of 160 cases. American Journal of Surgical Pathology 31:255–266.
