326
Gynaecology Case Reports
J Obstet Gynaecol Downloaded from informahealthcare.com by Nyu Medical Center on 05/19/15 For personal use only.
Figure 1. (A) Chest CT findings and (B) intraoperative findings under thoracoscopy surgery. Pulmonary nodules (arrow) in left upper lobe were mistaken for pulmonary metastasis from an ovarian carcinoma. The final pathology revealed that the pulmonary nodules were not pulmonary metastasis from an ovarian carcinoma, but benign metastasising leiomyomas.
leiomyomas for the pulmonary nodules (Figure 2). Histological and pathological findings of the pulmonary nodules confirmed uterine myoma. The postoperative course was uneventful and she was discharged on the 9th postoperative day. She is being followed up without any treatment. At present, 24 months after surgery, no new pulmonary lesion or ovarian cancer recurrence has been found.
Discussion Benign metastasising leiomyoma (BML) is the existence of a histologically benign smooth muscle tumour in extrauterine sites, and is a very rare disease in women with a history of benign uterine leiomyoma (Beck et al. 2012; Kim and Han 2012). About 100 cases have been reported since BML was first reported by Steiner in 1939 (Kang et al. 2011; Beck et al. 2012; Kim and Han 2012). Although the lung is the most affected organ, BML can develop in lymph nodes, skin, bones, the retroperitoneum, the heart and the brain (Beck et al. 2012; Bowen et al. 2012; Kang et al. 2011). The aetiology of BML remains unclear, and several hypotheses of the pathogenesis have been proposed: (1) haematogenous spread to extrauterine sites caused by uterine artery embolisation or surgical procedures; (2) malignant behaviour of benign-looking uterine tumours, acting as low-grade malignancy; (3) systemic leiomyomatosis (Beck et al. 2012; Bowen et al. 2012; Kim and Han 2012). Most patients are asymptomatic and diagnosed incidentally on routine image study, and have a history of uterine surgery (Goto et al. 2012). The pulmonary BML size ranges from some millimetres to centimetres. Radiographically, it tends to be well circumscribed and bilateral multiple nodules without contrast enhancement on CT are seen (Goto et al. 2012). It does not have significant metabolic activity on positron emission tomography (PET) scans.
The pathology findings are consistent with benign leiomyomas. Immunohistochemical staining is positive for oestrogen and progesterone receptors, which is suggestive of their uterine origin (Kang et al. 2011). The treatment of BML is still controversial (Kang et al. 2011; Beck et al. 2012; Goto et al. 2012; Kim and Han 2012), and several management options, including close surveillance, hormone therapy and surgical resection, have been performed. The treatment should be different in pulmonary metastasis between benign leiomyoma and other malignancies. In our case, the patient was asymptomatic, and pulmonary nodules were found incidentally during preoperative staging evaluations for ovarian carcinoma. At first, we considered the pulmonary nodules to be metastatic lesions from ovarian carcinoma. If we did not consider the possibility of other pathology or confirm the pathology of these pulmonary lesions, due to various reasons (technically difficult, too small, empirical, etc.), the patient might receive unnecessary treatments, such as a chemotherapy. When a pulmonary metastatic lesion is suspected in a female reproductive organ carcinoma with uterine myoma, we should consider the possibility of benign metastasising leiomyoma to avoid unnecessary treatment. We report a very unusual case in which benign metastasising leiomyoma from a uterine myoma was mistaken for pulmonary metastasis from an ovarian carcinoma. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
Reference Beck MM, Biswas B, D’Souza A et al. 2012. Benign metastasising leiomyoma after hysterectomy and bilateral salpingo-oophorectomy. Hong Kong Medical Journal 18:153–155. Bowen JM, Cates JM, Kash S et al. 2012. Genomic imbalances in benign metastasizing leiomyoma: characterization by conventional karyotypic, fluorescence in situ hybridization, and whole genome SNP array analysis. Cancer Genetics 205:249–254. Goto T, Maeshima A, Akanabe K et al. 2012. Benign metastasizing leiomyoma of the lung. Annals of Thoracic and Cardiovascular Surgery 18:121–124. Kang MW, Kang SK, Yu JH et al. 2011. Benign metastasizing leiomyoma: metastasis to rib and vertebra. Annals of Thoracic and Cardiovascular Surgery 91:924–926. Kim ER, Han KN. 2012. Spontaneous regression of metastatic pulmonary leiomyoma after resection of contralateral metastatic mass. Annals of Thoracic and Cardiovascular Surgery 94:e119–e120.
Endometrial mature cystic teratoma misdiagnosed as submucous fibromyomata Figure 2. Histology of the resected pulmonary nodule. As in uterine leiomyoma, the microscopic findings of the pulmonary lesions showed interacting bundles of spindle-shaped smooth muscle cells with no cellular atypia. The nuclei appear small and round due to the fascicular arrangements. No malignant findings were observed (H&E, ⫻ 100).
Y-L. Wang, J-H. Zhu, J-H. Fu, Y-M. Gao & J-H. Wen First Hospital of Jilin University, Changchun, Jilin, China DOI: 10.3109/01443615.2014.954099
Gynaecology Case Reports 327 Correspondence: J-H. Wen, Department of Gynaecology, First Hospital of Jilin University, 71 Xinmin Street, Changchun 130021, Jilin, China. E-mail: [email protected]
Introduction Mature cystic teratoma (MCT) is a common tumour in the ovary and is often diagnosed by ultrasound examination. However, endometrial MCT is very rare and is difficult to diagnose preoperatively. Thus, the possibility of an endometrial MCT must be considered when examining an endometrial tumour.
J Obstet Gynaecol Downloaded from informahealthcare.com by Nyu Medical Center on 05/19/15 For personal use only.
Case report A 42-year-old woman was admitted to our hospital due to chronic abdominal pain with abnormal uterine bleeding for more than 6 months. Upon physical examination (speculum and bimanual examination), the following was noted: patent vaginal tissue; soft mucosa; smooth cervix; normal sized anteverted uterus; no tenderness; normal uterine appendages; little white secretion without odour. A transvaginal ultrasound examination was conducted, and an intrauterine mass was observed as a 12 ⫻ 10 mm strong echo group (Figure 1A). The mass with a smooth edge was completely localised in the intrauterine cavity. Written consent was obtained from the patient. A hysteroscopy was performed under intravenous anaesthesia. Under hysteroscopy, a mass was observed near the right side wall of the uterine cavity. The mass was about 1.5 ⫻ 2 cm, grey, hard, smooth and with a rich blood supply. Three grey and slightly hard tissues were removed, the biggest was 0.7 ⫻ 1.0 ⫻ 1.5 cm. Tissue histopathology was performed using haematoxylin-eosin (H&E) staining. Histological cross-sectional images showed the tumour to have a duct-like structure surrounded by a large number of adipocytes, with skin and skin appendages (Figure 1B,C). Abnormal uterine bleeding completely disappeared and the menstrual cycle restored to normal after surgery. Follow-up of this patient was carried out at 1, 3, 6 and 12 months; vaginal ultrasound was normal.
Discussion MCT is a common cystic tumour that contains a variety of tissues derived from two or more primary germ cell layers (i.e. ectoderm, mesoderm and endoderm). Teratomas can be classified as mature or immature, based on the presence of immature neuroectodermal elements within the tumour. MCTs are usually found in the ovary. Because endometrial MCT is rarely seen, clinicians can easily overlook it. Endometrial MCT, polyp and submucous fibromyoma have similar clinical symptoms, including abnormal uterine bleeding, pelvic pain, vaginal discharge and enlargement of the uterus. It is therefore difficult to diagnose endometrial MCT preoperatively. In the present case, transvaginal sonography was used but the lesion lacked typical sonographic features. Power Doppler blood flow mapping is a valuable tool for differentiating submucosal fibroids from endometrial polyps (Cil et al. 2010), but cannot identify endometrial MCT and submucous fibromyoma. Using computed tomography (CT) and magnetic resonance imaging (MRI), the clear boundary of the unilocular or multilocular dermoid cystic mass is observable, with varying proportions of a solid component and areas of fat-like density or gross calcifications (Papadia et al. 2007; Seki et al. 2005; Strasser et al. 2002). The major symptom of this patient was chronic abdominal pain with abnormal uterine bleeding for more than 6 months. Because the gynaecological examination was normal and a transvaginal sonogram of the lesion showed a hyperechoic group with distinct margins, our initial diagnosis was submucous fibromyomata (calcification). Endometrial teratoma may be misdiagnosed as uterine polyp (Papadia et al. 2007). Because the preoperative approach for diagnosis of endometrial MCT is not ideal, surgical exploration is a necessary step (Degrate et al. 2012). Complete surgical resection is the definitive treatment, which also enables a conclusive diagnosis (Degrate et al. 2012). Based on the treatment procedures and final pathology report for this patient, we suggest that clinicians should consider MCT as a possibility when treating endometrial lesions. Moreover, serum
Figure 1. (A) Transvaginal sonogram showed that the uterus had normal echogenicity and morphology with a homogeneously thin endometrium (thin arrow) and a hyperechoic endocavitary lesion of 15 mm (thick arrow). The image suggests an endometrial submucous fibromyoma. (B) Histological cross-section showed the duct-like structure of the tumour tissue surrounded by a large number of adipocytes (H&E, ⫻ 40). (C) Histological cross-section showed that skin and skin appendages were visible in the tumour tissue (H&E, ⫻ 40).
alpha-fetoprotein screening can help distinguish benign from malignant teratoma if MCT is diagnosed (Iacono et al. 1993; Papadia et al. 2007).
Acknowledgements We thank Li-Hua Feng at First Hospital of Jilin University for help in the preparation of this paper. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
328
Gynaecology Case Reports
References
J Obstet Gynaecol Downloaded from informahealthcare.com by Nyu Medical Center on 05/19/15 For personal use only.
Cil AP, Tulunay G, Kose MF et al. 2010. Power Doppler properties of endometrial polyps and submucosal fibroids: a preliminary observational study in women with known intracavitary lesions. Ultrasound in Obstetrics and Gynecology 35:233–237. Degrate L, Misani M, Mauri G et al. 2012. Mature cystic teratoma of the pancreas. Case report and review of the literature of a rare pancreatic cystic lesion. Journal of the Pancreas 13:66–72. Iacono C, Zamboni G, Di Marcello R et al. 1993. Dermoid cyst of the head of the pancreas area. International Journal of Pancreatology 14:269–273.
Papadia A, Rutigliani M, Gerbaldo D et al. 2007. Mature cystic teratoma of the uterus presenting as an endometrial polyp. Ultrasound in Obstetrics and Gynecology 29:477–478. Seki M, Ninomiya E, Aruga A et al. 2005. Image-diagnostic features of mature cystic teratomas of the pancreas: report on two cases difficult to diagnose preoperatively. Journal of Hepato-Biliary-Pancreatic Surgery 12:336–340. Strasser G, Kutilek M, Mazal P et al. 2002. Mature teratoma of the pancreas: CT and MR findings. European Radiology 12(Suppl 3):S56–S58.
Gynaecology Case Reports
J Obstet Gynaecol Downloaded from informahealthcare.com by Nyu Medical Center on 05/19/15 For personal use only.
Figure 1. (A) Chest CT findings and (B) intraoperative findings under thoracoscopy surgery. Pulmonary nodules (arrow) in left upper lobe were mistaken for pulmonary metastasis from an ovarian carcinoma. The final pathology revealed that the pulmonary nodules were not pulmonary metastasis from an ovarian carcinoma, but benign metastasising leiomyomas.
leiomyomas for the pulmonary nodules (Figure 2). Histological and pathological findings of the pulmonary nodules confirmed uterine myoma. The postoperative course was uneventful and she was discharged on the 9th postoperative day. She is being followed up without any treatment. At present, 24 months after surgery, no new pulmonary lesion or ovarian cancer recurrence has been found.
Discussion Benign metastasising leiomyoma (BML) is the existence of a histologically benign smooth muscle tumour in extrauterine sites, and is a very rare disease in women with a history of benign uterine leiomyoma (Beck et al. 2012; Kim and Han 2012). About 100 cases have been reported since BML was first reported by Steiner in 1939 (Kang et al. 2011; Beck et al. 2012; Kim and Han 2012). Although the lung is the most affected organ, BML can develop in lymph nodes, skin, bones, the retroperitoneum, the heart and the brain (Beck et al. 2012; Bowen et al. 2012; Kang et al. 2011). The aetiology of BML remains unclear, and several hypotheses of the pathogenesis have been proposed: (1) haematogenous spread to extrauterine sites caused by uterine artery embolisation or surgical procedures; (2) malignant behaviour of benign-looking uterine tumours, acting as low-grade malignancy; (3) systemic leiomyomatosis (Beck et al. 2012; Bowen et al. 2012; Kim and Han 2012). Most patients are asymptomatic and diagnosed incidentally on routine image study, and have a history of uterine surgery (Goto et al. 2012). The pulmonary BML size ranges from some millimetres to centimetres. Radiographically, it tends to be well circumscribed and bilateral multiple nodules without contrast enhancement on CT are seen (Goto et al. 2012). It does not have significant metabolic activity on positron emission tomography (PET) scans.
The pathology findings are consistent with benign leiomyomas. Immunohistochemical staining is positive for oestrogen and progesterone receptors, which is suggestive of their uterine origin (Kang et al. 2011). The treatment of BML is still controversial (Kang et al. 2011; Beck et al. 2012; Goto et al. 2012; Kim and Han 2012), and several management options, including close surveillance, hormone therapy and surgical resection, have been performed. The treatment should be different in pulmonary metastasis between benign leiomyoma and other malignancies. In our case, the patient was asymptomatic, and pulmonary nodules were found incidentally during preoperative staging evaluations for ovarian carcinoma. At first, we considered the pulmonary nodules to be metastatic lesions from ovarian carcinoma. If we did not consider the possibility of other pathology or confirm the pathology of these pulmonary lesions, due to various reasons (technically difficult, too small, empirical, etc.), the patient might receive unnecessary treatments, such as a chemotherapy. When a pulmonary metastatic lesion is suspected in a female reproductive organ carcinoma with uterine myoma, we should consider the possibility of benign metastasising leiomyoma to avoid unnecessary treatment. We report a very unusual case in which benign metastasising leiomyoma from a uterine myoma was mistaken for pulmonary metastasis from an ovarian carcinoma. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
Reference Beck MM, Biswas B, D’Souza A et al. 2012. Benign metastasising leiomyoma after hysterectomy and bilateral salpingo-oophorectomy. Hong Kong Medical Journal 18:153–155. Bowen JM, Cates JM, Kash S et al. 2012. Genomic imbalances in benign metastasizing leiomyoma: characterization by conventional karyotypic, fluorescence in situ hybridization, and whole genome SNP array analysis. Cancer Genetics 205:249–254. Goto T, Maeshima A, Akanabe K et al. 2012. Benign metastasizing leiomyoma of the lung. Annals of Thoracic and Cardiovascular Surgery 18:121–124. Kang MW, Kang SK, Yu JH et al. 2011. Benign metastasizing leiomyoma: metastasis to rib and vertebra. Annals of Thoracic and Cardiovascular Surgery 91:924–926. Kim ER, Han KN. 2012. Spontaneous regression of metastatic pulmonary leiomyoma after resection of contralateral metastatic mass. Annals of Thoracic and Cardiovascular Surgery 94:e119–e120.
Endometrial mature cystic teratoma misdiagnosed as submucous fibromyomata Figure 2. Histology of the resected pulmonary nodule. As in uterine leiomyoma, the microscopic findings of the pulmonary lesions showed interacting bundles of spindle-shaped smooth muscle cells with no cellular atypia. The nuclei appear small and round due to the fascicular arrangements. No malignant findings were observed (H&E, ⫻ 100).
Y-L. Wang, J-H. Zhu, J-H. Fu, Y-M. Gao & J-H. Wen First Hospital of Jilin University, Changchun, Jilin, China DOI: 10.3109/01443615.2014.954099
Gynaecology Case Reports 327 Correspondence: J-H. Wen, Department of Gynaecology, First Hospital of Jilin University, 71 Xinmin Street, Changchun 130021, Jilin, China. E-mail: [email protected]
Introduction Mature cystic teratoma (MCT) is a common tumour in the ovary and is often diagnosed by ultrasound examination. However, endometrial MCT is very rare and is difficult to diagnose preoperatively. Thus, the possibility of an endometrial MCT must be considered when examining an endometrial tumour.
J Obstet Gynaecol Downloaded from informahealthcare.com by Nyu Medical Center on 05/19/15 For personal use only.
Case report A 42-year-old woman was admitted to our hospital due to chronic abdominal pain with abnormal uterine bleeding for more than 6 months. Upon physical examination (speculum and bimanual examination), the following was noted: patent vaginal tissue; soft mucosa; smooth cervix; normal sized anteverted uterus; no tenderness; normal uterine appendages; little white secretion without odour. A transvaginal ultrasound examination was conducted, and an intrauterine mass was observed as a 12 ⫻ 10 mm strong echo group (Figure 1A). The mass with a smooth edge was completely localised in the intrauterine cavity. Written consent was obtained from the patient. A hysteroscopy was performed under intravenous anaesthesia. Under hysteroscopy, a mass was observed near the right side wall of the uterine cavity. The mass was about 1.5 ⫻ 2 cm, grey, hard, smooth and with a rich blood supply. Three grey and slightly hard tissues were removed, the biggest was 0.7 ⫻ 1.0 ⫻ 1.5 cm. Tissue histopathology was performed using haematoxylin-eosin (H&E) staining. Histological cross-sectional images showed the tumour to have a duct-like structure surrounded by a large number of adipocytes, with skin and skin appendages (Figure 1B,C). Abnormal uterine bleeding completely disappeared and the menstrual cycle restored to normal after surgery. Follow-up of this patient was carried out at 1, 3, 6 and 12 months; vaginal ultrasound was normal.
Discussion MCT is a common cystic tumour that contains a variety of tissues derived from two or more primary germ cell layers (i.e. ectoderm, mesoderm and endoderm). Teratomas can be classified as mature or immature, based on the presence of immature neuroectodermal elements within the tumour. MCTs are usually found in the ovary. Because endometrial MCT is rarely seen, clinicians can easily overlook it. Endometrial MCT, polyp and submucous fibromyoma have similar clinical symptoms, including abnormal uterine bleeding, pelvic pain, vaginal discharge and enlargement of the uterus. It is therefore difficult to diagnose endometrial MCT preoperatively. In the present case, transvaginal sonography was used but the lesion lacked typical sonographic features. Power Doppler blood flow mapping is a valuable tool for differentiating submucosal fibroids from endometrial polyps (Cil et al. 2010), but cannot identify endometrial MCT and submucous fibromyoma. Using computed tomography (CT) and magnetic resonance imaging (MRI), the clear boundary of the unilocular or multilocular dermoid cystic mass is observable, with varying proportions of a solid component and areas of fat-like density or gross calcifications (Papadia et al. 2007; Seki et al. 2005; Strasser et al. 2002). The major symptom of this patient was chronic abdominal pain with abnormal uterine bleeding for more than 6 months. Because the gynaecological examination was normal and a transvaginal sonogram of the lesion showed a hyperechoic group with distinct margins, our initial diagnosis was submucous fibromyomata (calcification). Endometrial teratoma may be misdiagnosed as uterine polyp (Papadia et al. 2007). Because the preoperative approach for diagnosis of endometrial MCT is not ideal, surgical exploration is a necessary step (Degrate et al. 2012). Complete surgical resection is the definitive treatment, which also enables a conclusive diagnosis (Degrate et al. 2012). Based on the treatment procedures and final pathology report for this patient, we suggest that clinicians should consider MCT as a possibility when treating endometrial lesions. Moreover, serum
Figure 1. (A) Transvaginal sonogram showed that the uterus had normal echogenicity and morphology with a homogeneously thin endometrium (thin arrow) and a hyperechoic endocavitary lesion of 15 mm (thick arrow). The image suggests an endometrial submucous fibromyoma. (B) Histological cross-section showed the duct-like structure of the tumour tissue surrounded by a large number of adipocytes (H&E, ⫻ 40). (C) Histological cross-section showed that skin and skin appendages were visible in the tumour tissue (H&E, ⫻ 40).
alpha-fetoprotein screening can help distinguish benign from malignant teratoma if MCT is diagnosed (Iacono et al. 1993; Papadia et al. 2007).
Acknowledgements We thank Li-Hua Feng at First Hospital of Jilin University for help in the preparation of this paper. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
328
Gynaecology Case Reports
References
J Obstet Gynaecol Downloaded from informahealthcare.com by Nyu Medical Center on 05/19/15 For personal use only.
Cil AP, Tulunay G, Kose MF et al. 2010. Power Doppler properties of endometrial polyps and submucosal fibroids: a preliminary observational study in women with known intracavitary lesions. Ultrasound in Obstetrics and Gynecology 35:233–237. Degrate L, Misani M, Mauri G et al. 2012. Mature cystic teratoma of the pancreas. Case report and review of the literature of a rare pancreatic cystic lesion. Journal of the Pancreas 13:66–72. Iacono C, Zamboni G, Di Marcello R et al. 1993. Dermoid cyst of the head of the pancreas area. International Journal of Pancreatology 14:269–273.
Papadia A, Rutigliani M, Gerbaldo D et al. 2007. Mature cystic teratoma of the uterus presenting as an endometrial polyp. Ultrasound in Obstetrics and Gynecology 29:477–478. Seki M, Ninomiya E, Aruga A et al. 2005. Image-diagnostic features of mature cystic teratomas of the pancreas: report on two cases difficult to diagnose preoperatively. Journal of Hepato-Biliary-Pancreatic Surgery 12:336–340. Strasser G, Kutilek M, Mazal P et al. 2002. Mature teratoma of the pancreas: CT and MR findings. European Radiology 12(Suppl 3):S56–S58.
