Journal of Obstetrics and Gynaecology
ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20
Spontaneous ovarian hyperstimulation mimicking ovarian neoplasm: A rare complication of hypothyroidism S. Rajaram, S. Bhaskaran, P. Aggarwal & N. Goel To cite this article: S. Rajaram, S. Bhaskaran, P. Aggarwal & N. Goel (2015) Spontaneous ovarian hyperstimulation mimicking ovarian neoplasm: A rare complication of hypothyroidism, Journal of Obstetrics and Gynaecology, 35:5, 532-533, DOI: 10.3109/01443615.2014.968112 To link to this article: http://dx.doi.org/10.3109/01443615.2014.968112
Published online: 10 Nov 2014.
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Date: 14 March 2016, At: 19:43
532 Gynaecology Case Reports
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and expertise both in imaging and pathological evaluation (Allen et al. 2006; Moutte et al. 2013). The differential diagnosis is made with other malignant forms of gestational trophoblastic disease, particularly invasive mole and choriocarcinoma but also with benign trophoblastic proliferations, which regress spontaneously (Kim 2003). In contrast to other trophoblastic tumours, PSTT is relatively insensitive to chemotherapy. Surgery remains the primary therapeutic approach in patients presenting with uterine-confined disease, while those with metastatic disease also require chemotherapy (Hyman et al. 2013). There is still a lack of consensus in the literature on the key prognostic factors for PSTT, although the time interval since preceding pregnancy seems to be the most significant independent predictor of survival, with longer times correlating with worst prognoses (Schmid et al. 2009). Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References
Allen SD, Lim AK, Seckl MJ et al. 2006. Radiology of gestational trophoblastic neoplasia. Clinical Radiology 61:301–313. Baergen RN, Rutgers J, Young RH. 2003. Extrauterine lesions of intermediate trophoblast. International Journal of Gynecological Pathology 22:362–367. Hassadia A, Gillespie A, Tidy J et al. 2005. Placental site trophoblastic tumour: clinical features and management. Gynecologic Oncology 99:603–607. Hyman DM, Bakios L, Gualtiere G et al. 2013. Placental site trophoblastic tumour: Analysis of presentation, treatment, and outcome. Gynecologic Oncology 129:58–62. Kim SJ. 2003. Placental site trophoblastic tumour. Best Practice and Research Clinical Obstetrics Gynaecology 17:969–984. Kurman RJ. 1991. The morphology, biology, and pathology of intermediate trophoblast: a look back to the present. Human Pathology 22:847–855. Moutte A, Doret M, Hajri T et al. 2013. Placental site and epithelioid trophoblastic tumours: Diagnostic pitfalls. Gynecologic Oncology 128:568–572. Schmid P, Nagai Y, Agarwal R et al. 2009. Prognostic markers and long-term outcome of placental-site trophoblastic tumours: a retrospective observational study. Lancet 374:48–55. Tang X, Yang F, Jia L et al. 2013. Placental site trophoblastic tumor in the pelvic wall: a case report and review of the literature. Indian Journal of Pathology and Microbiology 56:300–302. Wagner B, Dickey G. 1996. Gestational trophoblastic correlation. Radiographics 16:131–148.
Spontaneous ovarian hyperstimulation mimicking ovarian neoplasm: A rare complication of hypothyroidism
has also been associated with polycystic ovarian syndrome, pregnancy, molar pregnancy and gonadotroph pituitary adenomas. To our knowledge, very few cases of isolated hypothyroidism resulting in ovarian hyperstimulation in adult women have been reported in the literature (Rotmensch and Scommegna 1989; Van Voorhis et al. 1994; Hansen et al. 1997; Taher et al. 2004; Bassam and Ajlouni 2006; Kubota et al. 2008; Shu et al. 2011). Hence, to prevent catastrophic oophorectomies in young women, it is imperative that clinicians should be well informed about this rare but important clinical entity. We report such a case of spontaneous OHSS in primary hypothyroidism mimicking an ovarian neoplasm.
Case report
A 19-year-old unmarried woman (not sexually active) presented with complaints of recurrent lower abdominal pain for 3 months. The pain was colicky and associated with giddiness and syncopal attacks. She had attained menarche at the age of 15 years and her periods had been heavy and irregular since then. On physical examination, her vitals were stable and BMI was 28 kg/m2. She had mild pallor, no clinically apparent thyromegaly and a normal breast examination. Abdominal examination revealed a large cystic non-tender mass of around 20 weeks’ gravid uterus size. A urine pregnancy test was negative and blood investigations including tumour markers revealed mild anaemia with markedly elevated serum inhibin A of 2214.6 pg/ml (normal range, 2–80 pg/ml). Other tumour markers, such as hCG, AFP and CA125 were normal. A thyroid function test was done to evaluate the cause of the menorrhagia, revealed a high serum TSH 150 mU/l and anti-TPO antibodies, 173.8 U. Ultrasound thyroid revealed bilateral atrophic thyroid lobes with two hypoechoic lesions in the right lobe, the largest measuring 9 8 mm, having internal and peripheral vascularity with the possibility of capsular breech. Ultrasound guided FNAC was suggestive of autoimmune thyroiditis, following which Eltroxin 100 mg once daily was started. Ultrasound and computer tomography of the abdomen showed the presence of bilateral large well-defined lobulated cystic lesions of ovaries, with multiple thin septae- right and left measuring 9.5 8.0 cm and 11 8 cm, respectively (Figure 1). With a provisional diagnosis of malignant ovarian tumour, the woman was taken for staging laparotomy. At laparotomy, minimal haemorrhagic fluid and large bilateral multicystic ovaries with smooth surface were present (left, 9 8 cm and right, 8 6 cm). From each ovary, 2–3 cysts were enucleated and sent for histopathology. In view of the benign appearance of tumour intraoperatively and the young age of the patient, a decision to defer further surgery was taken. Histopathology was reported as simple serous cysts. The patient was followed up regularly and the cysts resolved spontaneously after 6 months, with thyroid replacement.
S. Rajaram, S. Bhaskaran, P. Aggarwal & N. Goel Department of Obstetrics and Gynaecology, University College of Medical Sciences, New Delhi, India DOI: 10.3109/01443615.2014.968112 Correspondence: S. Bhaskaran, Department of Obstetrics and Gynaecology, University College of Medical Sciences, DII/18, West Kidwai Nagar, New Delhi 110023, India. E-mail: [email protected]
Introduction
Thyroid disorders are one of the commonest endocrine disorders affecting women. The manifestations of hypothyroidism may range from asymptomatic to frank myxedema. One of the rare presentations of severe hypothyroidism in reproductive age women, is the development of massive ovarian cysts as a result of hyperstimulation of ovaries (OHSS). This can pose a diagnostic dilemma, especially when the cysts are large, complex and associated with elevated tumour markers, masquerading as neoplasm. Spontaneous OHSS
Figure 1. CT scan showing bilateral ovarian cysts (pretreatment).
Gynaecology Case Reports 533
Downloaded by [University of Cincinnati Libraries] at 19:43 14 March 2016
Discussion
Massive ovarian cysts with elevated tumour markers in young women are a common indication of surgical intervention and challenge the treating clinicians, with preservation of ovarian tissue and further reproductive potential. Severe hypothyroidism causing spontaneous hyperstimulated ovaries can present a similar picture and should be kept as a differential diagnosis, while investigating such cases. Spontaneous OHSS may be seen in conditions causing ovarian hyper-responsiveness to circulating FSH or cross-responsiveness of FSH receptors (FSHR) to hormones having a structure similar to FSH, such as hCG or TSH and has been reported in pregnancy, hypothyroidism, PCOS and gonadotropin pituitary adenomas. It is characterised by multicystic ovarian enlargement, often without the systemic features of iatrogenic ovarian hyperstimulation, such as pleural effusion, ascites, haemoconcentration, oliguria, etc. Clinicians often do not consider these possible aetiologies and make the diagnosis of neoplasm leading to surgery, as seen in our case. Van Wyvk Grumbach syndrome, characterised by multicystic ovarian cysts, precocious puberty and juvenile hypothyroidism, was described as early as 1960 and sporadic cases have been reported in adolescents and prepubescent girls since then, hence suggesting the susceptibility of juvenile ovaries to high levels of TSH (Browne et al. 2008). But very few cases of massive ovarian cysts due to hypothyroidism have been reported in adult non-pregnant women between 19 and 26 years of age (n 7), so far (Rotmensch and Scommegna 1989; Van Voorhis et al. 1994; Hansen et al. 1997; Taher et al. 2004; Bassam and Ajlouni 2006; Kubota et al. 2008; Shu et al. 2011). The mechanism for this association is not very clear but it is hypothesised that extremely high levels of TSH lead to activation of FSH receptors, as structures of their receptors are similar. Another explanation could be a possible FSHR gene polymorphism resulting in its hyper-responsiveness to circulating FSH and TSH. The mutations of Asp567Asn, Thr449Ile, Thr449Ala and Ile545Thr were found in women with spontaneous OHSS in pregnancy (Dieterich et al. 2010). Another possibility is that TSH sensitises the ovaries to gonadotropins by stimulating nuclear thyroid receptors in granulosa cells. Previous reported cases had serum TSH 100 mIU/l, as seen in our case too. The fact that thyroxine replacement resulted in the resolution of ovarian cysts, confirms the diagnosis. Also, previous cases have shown an associated raised pituitary gonadotropin, particularly FSH, which was not seen in our patient. Also, the serum inhibin levels were markedly elevated, raising the suspicion of malignancy. However, this could have been due to markedly raised TSH mimicking FSH and stimulating the release of inhibin, which in turn inhibited the FSH, resulting in its low levels. Possibly, this could have been a fallacious report which needed confirmation by repeating the test. None of the previous reported cases have mentioned raised inhibin in their reports. The mainstay of treatment in these cases is thyroid replacement and spontaneous resolution is expected but exploration can be considered in cases of acute emergency due to torsion or rupture of ovarian cysts. If at the time of surgery, neoplasm is suspected, then further management should be guided by frozen section (Shu et al. 2011). Although this syndrome is very rare, failure to recognise it as a differential in adult women presenting with multicystic ovaries, can inadvertently lead to oophorectomy. It is important that not only gynaecologists but also surgeons and physicians, should keep hypothyroidism in mind while dealing with such cases. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References
Bassam T, Ajlouni K. 2006. A case of ovarian enlargement in severe primary hypothyroidism and review of the literature. Annals of Saudi Medicine 26:66–68. Browne LP, Boswell HB, Crotty EJ et al. 2008. Van Wyk and Grumbach syndrome revisited: imaging and clinical findings in pre- and postpubertal girls. Pediatric Radiology 38:538–542. Dieterich M, Bolz M, Reimer T et al. 2010. Two different entities of spontaneous ovarian hyperstimulation in a woman with FSH receptor mutation. Reproductive Biomedicine Online 20:751–758.
Hansen KA, Tho SP, Hanly M et al. 1997. Massive ovarian enlargement in primary hypothyroidism. Fertility and Sterility 67:169–171. Kubota K, Itho M, Kishi H et al. 2008. Primary hypothyroidism presenting as multiple ovarian cysts in an adult woman: a case report. Gynecological Endocrinology 24:586–589. Rotmensch S, Scommegna A. 1989. Spontaneous ovarian hyperstimulation syndrome associated with hypothyroidism. American Journal of Obstetrics and Gynecology 160(5 Part 1):1220–1222. Shu J, Xing L, Zhang L et al. 2011. Ignored adult primary hypothyroidism presenting chiefly with persistent ovarian cysts: a need for increased awareness. Reproductive Biology and Endocrinology 9:119. Taher BM, Ghariabeh RA, Jarrah NS et al. 2004. Spontaneous ovarian hyperstimulation syndrome caused by hypothyroidism in an adult. European Journal of Obstetrics and Gynecology, and Reproductive Biology 112:107–109. Van Voorhis BJ, Neff TW, Syrop CH et al. 1994. Primary hypothyroidism associated with multicystic ovaries and ovarian torsion in an adult. Obstetrics and Gynecology 83(5 Part 2):885–887.
Serous endometrial intraepithelial carcinoma associated with submucosal leiomyomatous polyp: The first reported case M. Yang1,2 & B. Assylbekova1 1Department of Pathology and Laboratory Medicine, UT Health
Science Center at Houston and 2Department of Pathology, LBJ General Hospital, Houston, Texas, USA DOI: 10.3109/01443615.2014.968109
Correspondence: M. Yang, Department of Pathology, LBJ General Hospital, 5656 Kelley Street, 3PA 90.001A-C, Houston, Texas 77026, USA. E-mail: mary.y.yang@ uth.tmc.edu
Introduction
Serous endometrial intraepithelial carcinoma (EIC) is characterised by non-invasive replacement of endometrial surface epithelium and underlying glands with neoplastic cells resembling serous carcinoma cells. The lesion typically shows high proliferative rate on staining for Ki-67 and intense nuclear staining for p53. Serous EIC has been reported in association with endometrial polyps in a number of cases (Soslow et al. 2000; Hui et al. 2005; Trahan et al. 2005; Pathiraja et al. 2013). We report a case of serous EIC associated with a submucosal leiomyomatous polyp.
Case report
A 60-year-old female presented with a 1-year history of postmenopausal bleeding and previous endometrial biopsy (EMB) showing features compatible with EIC. Physical examination revealed normal external genitalia, significant cystocele and a small cervix. Repeated EMB showed benign endometrial polyp. Pelvic computed tomography (CT) scan showed thickened endometrial stripe. Magnetic resonance imaging (MRI) revealed a submucosal fibroid, which projected into the endometrial cavity. Small bilateral ovaries and a normal uterus were found intraoperatively. Hysterectomy and bilateral salpingooophorectomy was performed.
Material and methods
The surgical specimen from the hysterectomy and bilateral salpingooophorectomy was grossly examined. All polyps and endometrium were submitted in entirety for microscopic examination. Immunohistochemistry was performed for p53 (Ventana, Tucson, AZ, mouse monoclonal, DO-7, 1:200 dilution), Ki-67 (Ventana, rabbit monoclonal, 30–9, 1:200 dilution) and oestrogen receptor (ER) (Ventana, rabbit monoclonal, SP1, 1:200 dilution).
Result
Gross examination The surgical specimen consisted of an intact 5.5 5 3 cm hysterectomy specimen with attached cervix, bilateral tubes and ovaries. The
ISSN: 0144-3615 (Print) 1364-6893 (Online) Journal homepage: http://www.tandfonline.com/loi/ijog20
Spontaneous ovarian hyperstimulation mimicking ovarian neoplasm: A rare complication of hypothyroidism S. Rajaram, S. Bhaskaran, P. Aggarwal & N. Goel To cite this article: S. Rajaram, S. Bhaskaran, P. Aggarwal & N. Goel (2015) Spontaneous ovarian hyperstimulation mimicking ovarian neoplasm: A rare complication of hypothyroidism, Journal of Obstetrics and Gynaecology, 35:5, 532-533, DOI: 10.3109/01443615.2014.968112 To link to this article: http://dx.doi.org/10.3109/01443615.2014.968112
Published online: 10 Nov 2014.
Submit your article to this journal
Article views: 24
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: [University of Cincinnati Libraries]
Date: 14 March 2016, At: 19:43
532 Gynaecology Case Reports
Downloaded by [University of Cincinnati Libraries] at 19:43 14 March 2016
and expertise both in imaging and pathological evaluation (Allen et al. 2006; Moutte et al. 2013). The differential diagnosis is made with other malignant forms of gestational trophoblastic disease, particularly invasive mole and choriocarcinoma but also with benign trophoblastic proliferations, which regress spontaneously (Kim 2003). In contrast to other trophoblastic tumours, PSTT is relatively insensitive to chemotherapy. Surgery remains the primary therapeutic approach in patients presenting with uterine-confined disease, while those with metastatic disease also require chemotherapy (Hyman et al. 2013). There is still a lack of consensus in the literature on the key prognostic factors for PSTT, although the time interval since preceding pregnancy seems to be the most significant independent predictor of survival, with longer times correlating with worst prognoses (Schmid et al. 2009). Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References
Allen SD, Lim AK, Seckl MJ et al. 2006. Radiology of gestational trophoblastic neoplasia. Clinical Radiology 61:301–313. Baergen RN, Rutgers J, Young RH. 2003. Extrauterine lesions of intermediate trophoblast. International Journal of Gynecological Pathology 22:362–367. Hassadia A, Gillespie A, Tidy J et al. 2005. Placental site trophoblastic tumour: clinical features and management. Gynecologic Oncology 99:603–607. Hyman DM, Bakios L, Gualtiere G et al. 2013. Placental site trophoblastic tumour: Analysis of presentation, treatment, and outcome. Gynecologic Oncology 129:58–62. Kim SJ. 2003. Placental site trophoblastic tumour. Best Practice and Research Clinical Obstetrics Gynaecology 17:969–984. Kurman RJ. 1991. The morphology, biology, and pathology of intermediate trophoblast: a look back to the present. Human Pathology 22:847–855. Moutte A, Doret M, Hajri T et al. 2013. Placental site and epithelioid trophoblastic tumours: Diagnostic pitfalls. Gynecologic Oncology 128:568–572. Schmid P, Nagai Y, Agarwal R et al. 2009. Prognostic markers and long-term outcome of placental-site trophoblastic tumours: a retrospective observational study. Lancet 374:48–55. Tang X, Yang F, Jia L et al. 2013. Placental site trophoblastic tumor in the pelvic wall: a case report and review of the literature. Indian Journal of Pathology and Microbiology 56:300–302. Wagner B, Dickey G. 1996. Gestational trophoblastic correlation. Radiographics 16:131–148.
Spontaneous ovarian hyperstimulation mimicking ovarian neoplasm: A rare complication of hypothyroidism
has also been associated with polycystic ovarian syndrome, pregnancy, molar pregnancy and gonadotroph pituitary adenomas. To our knowledge, very few cases of isolated hypothyroidism resulting in ovarian hyperstimulation in adult women have been reported in the literature (Rotmensch and Scommegna 1989; Van Voorhis et al. 1994; Hansen et al. 1997; Taher et al. 2004; Bassam and Ajlouni 2006; Kubota et al. 2008; Shu et al. 2011). Hence, to prevent catastrophic oophorectomies in young women, it is imperative that clinicians should be well informed about this rare but important clinical entity. We report such a case of spontaneous OHSS in primary hypothyroidism mimicking an ovarian neoplasm.
Case report
A 19-year-old unmarried woman (not sexually active) presented with complaints of recurrent lower abdominal pain for 3 months. The pain was colicky and associated with giddiness and syncopal attacks. She had attained menarche at the age of 15 years and her periods had been heavy and irregular since then. On physical examination, her vitals were stable and BMI was 28 kg/m2. She had mild pallor, no clinically apparent thyromegaly and a normal breast examination. Abdominal examination revealed a large cystic non-tender mass of around 20 weeks’ gravid uterus size. A urine pregnancy test was negative and blood investigations including tumour markers revealed mild anaemia with markedly elevated serum inhibin A of 2214.6 pg/ml (normal range, 2–80 pg/ml). Other tumour markers, such as hCG, AFP and CA125 were normal. A thyroid function test was done to evaluate the cause of the menorrhagia, revealed a high serum TSH 150 mU/l and anti-TPO antibodies, 173.8 U. Ultrasound thyroid revealed bilateral atrophic thyroid lobes with two hypoechoic lesions in the right lobe, the largest measuring 9 8 mm, having internal and peripheral vascularity with the possibility of capsular breech. Ultrasound guided FNAC was suggestive of autoimmune thyroiditis, following which Eltroxin 100 mg once daily was started. Ultrasound and computer tomography of the abdomen showed the presence of bilateral large well-defined lobulated cystic lesions of ovaries, with multiple thin septae- right and left measuring 9.5 8.0 cm and 11 8 cm, respectively (Figure 1). With a provisional diagnosis of malignant ovarian tumour, the woman was taken for staging laparotomy. At laparotomy, minimal haemorrhagic fluid and large bilateral multicystic ovaries with smooth surface were present (left, 9 8 cm and right, 8 6 cm). From each ovary, 2–3 cysts were enucleated and sent for histopathology. In view of the benign appearance of tumour intraoperatively and the young age of the patient, a decision to defer further surgery was taken. Histopathology was reported as simple serous cysts. The patient was followed up regularly and the cysts resolved spontaneously after 6 months, with thyroid replacement.
S. Rajaram, S. Bhaskaran, P. Aggarwal & N. Goel Department of Obstetrics and Gynaecology, University College of Medical Sciences, New Delhi, India DOI: 10.3109/01443615.2014.968112 Correspondence: S. Bhaskaran, Department of Obstetrics and Gynaecology, University College of Medical Sciences, DII/18, West Kidwai Nagar, New Delhi 110023, India. E-mail: [email protected]
Introduction
Thyroid disorders are one of the commonest endocrine disorders affecting women. The manifestations of hypothyroidism may range from asymptomatic to frank myxedema. One of the rare presentations of severe hypothyroidism in reproductive age women, is the development of massive ovarian cysts as a result of hyperstimulation of ovaries (OHSS). This can pose a diagnostic dilemma, especially when the cysts are large, complex and associated with elevated tumour markers, masquerading as neoplasm. Spontaneous OHSS
Figure 1. CT scan showing bilateral ovarian cysts (pretreatment).
Gynaecology Case Reports 533
Downloaded by [University of Cincinnati Libraries] at 19:43 14 March 2016
Discussion
Massive ovarian cysts with elevated tumour markers in young women are a common indication of surgical intervention and challenge the treating clinicians, with preservation of ovarian tissue and further reproductive potential. Severe hypothyroidism causing spontaneous hyperstimulated ovaries can present a similar picture and should be kept as a differential diagnosis, while investigating such cases. Spontaneous OHSS may be seen in conditions causing ovarian hyper-responsiveness to circulating FSH or cross-responsiveness of FSH receptors (FSHR) to hormones having a structure similar to FSH, such as hCG or TSH and has been reported in pregnancy, hypothyroidism, PCOS and gonadotropin pituitary adenomas. It is characterised by multicystic ovarian enlargement, often without the systemic features of iatrogenic ovarian hyperstimulation, such as pleural effusion, ascites, haemoconcentration, oliguria, etc. Clinicians often do not consider these possible aetiologies and make the diagnosis of neoplasm leading to surgery, as seen in our case. Van Wyvk Grumbach syndrome, characterised by multicystic ovarian cysts, precocious puberty and juvenile hypothyroidism, was described as early as 1960 and sporadic cases have been reported in adolescents and prepubescent girls since then, hence suggesting the susceptibility of juvenile ovaries to high levels of TSH (Browne et al. 2008). But very few cases of massive ovarian cysts due to hypothyroidism have been reported in adult non-pregnant women between 19 and 26 years of age (n 7), so far (Rotmensch and Scommegna 1989; Van Voorhis et al. 1994; Hansen et al. 1997; Taher et al. 2004; Bassam and Ajlouni 2006; Kubota et al. 2008; Shu et al. 2011). The mechanism for this association is not very clear but it is hypothesised that extremely high levels of TSH lead to activation of FSH receptors, as structures of their receptors are similar. Another explanation could be a possible FSHR gene polymorphism resulting in its hyper-responsiveness to circulating FSH and TSH. The mutations of Asp567Asn, Thr449Ile, Thr449Ala and Ile545Thr were found in women with spontaneous OHSS in pregnancy (Dieterich et al. 2010). Another possibility is that TSH sensitises the ovaries to gonadotropins by stimulating nuclear thyroid receptors in granulosa cells. Previous reported cases had serum TSH 100 mIU/l, as seen in our case too. The fact that thyroxine replacement resulted in the resolution of ovarian cysts, confirms the diagnosis. Also, previous cases have shown an associated raised pituitary gonadotropin, particularly FSH, which was not seen in our patient. Also, the serum inhibin levels were markedly elevated, raising the suspicion of malignancy. However, this could have been due to markedly raised TSH mimicking FSH and stimulating the release of inhibin, which in turn inhibited the FSH, resulting in its low levels. Possibly, this could have been a fallacious report which needed confirmation by repeating the test. None of the previous reported cases have mentioned raised inhibin in their reports. The mainstay of treatment in these cases is thyroid replacement and spontaneous resolution is expected but exploration can be considered in cases of acute emergency due to torsion or rupture of ovarian cysts. If at the time of surgery, neoplasm is suspected, then further management should be guided by frozen section (Shu et al. 2011). Although this syndrome is very rare, failure to recognise it as a differential in adult women presenting with multicystic ovaries, can inadvertently lead to oophorectomy. It is important that not only gynaecologists but also surgeons and physicians, should keep hypothyroidism in mind while dealing with such cases. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
References
Bassam T, Ajlouni K. 2006. A case of ovarian enlargement in severe primary hypothyroidism and review of the literature. Annals of Saudi Medicine 26:66–68. Browne LP, Boswell HB, Crotty EJ et al. 2008. Van Wyk and Grumbach syndrome revisited: imaging and clinical findings in pre- and postpubertal girls. Pediatric Radiology 38:538–542. Dieterich M, Bolz M, Reimer T et al. 2010. Two different entities of spontaneous ovarian hyperstimulation in a woman with FSH receptor mutation. Reproductive Biomedicine Online 20:751–758.
Hansen KA, Tho SP, Hanly M et al. 1997. Massive ovarian enlargement in primary hypothyroidism. Fertility and Sterility 67:169–171. Kubota K, Itho M, Kishi H et al. 2008. Primary hypothyroidism presenting as multiple ovarian cysts in an adult woman: a case report. Gynecological Endocrinology 24:586–589. Rotmensch S, Scommegna A. 1989. Spontaneous ovarian hyperstimulation syndrome associated with hypothyroidism. American Journal of Obstetrics and Gynecology 160(5 Part 1):1220–1222. Shu J, Xing L, Zhang L et al. 2011. Ignored adult primary hypothyroidism presenting chiefly with persistent ovarian cysts: a need for increased awareness. Reproductive Biology and Endocrinology 9:119. Taher BM, Ghariabeh RA, Jarrah NS et al. 2004. Spontaneous ovarian hyperstimulation syndrome caused by hypothyroidism in an adult. European Journal of Obstetrics and Gynecology, and Reproductive Biology 112:107–109. Van Voorhis BJ, Neff TW, Syrop CH et al. 1994. Primary hypothyroidism associated with multicystic ovaries and ovarian torsion in an adult. Obstetrics and Gynecology 83(5 Part 2):885–887.
Serous endometrial intraepithelial carcinoma associated with submucosal leiomyomatous polyp: The first reported case M. Yang1,2 & B. Assylbekova1 1Department of Pathology and Laboratory Medicine, UT Health
Science Center at Houston and 2Department of Pathology, LBJ General Hospital, Houston, Texas, USA DOI: 10.3109/01443615.2014.968109
Correspondence: M. Yang, Department of Pathology, LBJ General Hospital, 5656 Kelley Street, 3PA 90.001A-C, Houston, Texas 77026, USA. E-mail: mary.y.yang@ uth.tmc.edu
Introduction
Serous endometrial intraepithelial carcinoma (EIC) is characterised by non-invasive replacement of endometrial surface epithelium and underlying glands with neoplastic cells resembling serous carcinoma cells. The lesion typically shows high proliferative rate on staining for Ki-67 and intense nuclear staining for p53. Serous EIC has been reported in association with endometrial polyps in a number of cases (Soslow et al. 2000; Hui et al. 2005; Trahan et al. 2005; Pathiraja et al. 2013). We report a case of serous EIC associated with a submucosal leiomyomatous polyp.
Case report
A 60-year-old female presented with a 1-year history of postmenopausal bleeding and previous endometrial biopsy (EMB) showing features compatible with EIC. Physical examination revealed normal external genitalia, significant cystocele and a small cervix. Repeated EMB showed benign endometrial polyp. Pelvic computed tomography (CT) scan showed thickened endometrial stripe. Magnetic resonance imaging (MRI) revealed a submucosal fibroid, which projected into the endometrial cavity. Small bilateral ovaries and a normal uterus were found intraoperatively. Hysterectomy and bilateral salpingooophorectomy was performed.
Material and methods
The surgical specimen from the hysterectomy and bilateral salpingooophorectomy was grossly examined. All polyps and endometrium were submitted in entirety for microscopic examination. Immunohistochemistry was performed for p53 (Ventana, Tucson, AZ, mouse monoclonal, DO-7, 1:200 dilution), Ki-67 (Ventana, rabbit monoclonal, 30–9, 1:200 dilution) and oestrogen receptor (ER) (Ventana, rabbit monoclonal, SP1, 1:200 dilution).
Result
Gross examination The surgical specimen consisted of an intact 5.5 5 3 cm hysterectomy specimen with attached cervix, bilateral tubes and ovaries. The
