Coexisting Brenner tumor and struma ovarii in the right ovary: case report and review of the literature

A bilateral ovarian tumor composed of mixed Brenner tumor and struma ovarii in the right ovary and mature cystic teratoma in the left ovary, is described. Mixed Brenner tumor and struma ovarii is rare; eight cases are reviewed. In this case, in addition to the typical Brenner tumor and struma ovarii, some nests composed of both Brenner tumor and struma ovarii in one nest were found in the right ovarian tumor. Immunohistochemically, the struma ovarii is stained for thyroglobulin, and Brenner nests showed various degrees of positive stain for thyroglobulin, which is a specific finding. Brenner tumor, in this case, may produce thyroglobulin or have a receptor to thyroglobulin or analog of thyroglobulin. The origin of mixed Brenner tumor and struma ovarii may be germ cell, as described in some literature, or the Brenner tumor may be of a metaplastic nature, although the Brenner tumor is fourfold the size of struma ovarii in the case presented.     

Predominant Brenner Tumor Combined with Struma Ovarii Containing a Papillary Microcarcinoma Associated with Benign Peritoneal Strumosis: Report of a Case and Histologic Features

Brenner tumor and struma ovarii, two uncommon ovarian tumors arising alone or together with dermoid cysts or adenomas, are both rare entities. Both tumors rarely become malignant and rarely metastasize. Few published reports describe coexisting Brenner tumor and malignant struma ovarii. Patients in whom these malignancies coexist only occasionally have peritoneal spreading, strumosis, or a history of thyrotoxicosis. The patient we describe, a 74-year-old woman, presented with a 2 months’ history of lower abdominal pain and episodic intestinal subocclusion due to a complex pelvic mass. The mass consisted predominantly of a Brenner tumor associated with struma ovarii containing a single small island of thyroid tissue that had undergone malignant transformation into a well-differentiated papillary carcinoma and also normal thyroid tissue that had spread to the peritoneum. The patient underwent radical surgical treatment and after 7 years follow-up is disease free.     

Diagnosis of necrotic and degenerate thyroid lesions: value of immunohistochemistry

Aims: Classification of necrotic or degenerate thyroid nodules can be difficult. The aim of this study was to investigate the value of cytokeratins, thyroid-specific markers (TTF-1 and thyroglobulin) and HBME-1 antibodies in such thyroid lesions. METHODS AND RESULTS: Twenty-eight necrotic or degenerate thyroid lesions, including four cervical cystic papillary carcinoma (CPC) metastases, were evaluated with immunohistochemistry for TTF-1, thyroglobulin, HBME-1, AE1&3, Cam5.2, MNF116 and cytokeratin (CK)19. There was loss of TTF-1 staining in all necrotic lesions, with positive staining in degenerate tumour cells of all four metastatic CPCs. Thyroglobulin was retained in 18 lesions. Dual CK19 and HBME-1 expression was seen only in six of seven necrotic papillary thyroid carcinomas and the four metastatic CPCs. Retained immunoreactivity for AE1&3 and Cam5.2 was seen in most necrotic papillary carcinomas (n = 11/11 and n = 10/11, respectively), poorly differentiated carcinomas (n = 2/3 and n = 3/3, respectively) and follicular-patterned areas of anaplastic carcinoma (n = 3/5 and n = 4/5, respectively). Cam5.2 showed spurious staining of macrophages in eight lesions. CONCLUSIONS: Thyroglobulin is useful in establishing the thyroid origin of a necrotic lesion. TTF-1 may be useful for highlighting degenerate tumour cells within metastatic CPCs. Retained expression of CK19 and HBME-1 is seen in necrotic papillary carcinomas. AE1&3 is the most specific and Cam5.2 the most sensitive of the CK cocktails in non-viable thyroid lesions.     

An immunohistochemical study of the rete ovarii and epoophoron

A study to compare the immuno-histochemical profile of the human rete ovarii, and epoophoron, with the Fallopian tube and ovarian surface epithelium was performed with 31 antibodies and antisera. A reaction was present in the epithelial cytoplasm of the rete ovarii and epoophoron of mesonephric origin, for vimentin, GFAP, cytokeratin markers, (AE1/AE3, MNF116; Cam 5.2, 34 beta E12 and for the monospecific antibodies to cytokeratins 7 and 19), heat shock protein 27, in the cell membrane for HBME-1, EMA and in the subepithelial collagen for collagen IV. Reactions were present only in the epithelium in the rete ovarii for EGFR (one case) and CA-125 (four cases). A reaction was present in the epithelium of the epoophoron only for Ber-EP-4 and S100. There was no reaction with antibodies for desmin, neurofilament protein, cytokeratins 20 or 14, actin, calretinin, E-cadherin, C-erb-B2, or CEA (monoclonal and polyclonal reagents). The immuno-histochemical profile of the Fallopian tube was consistent with its para-mesonephric origin and that in the ovarian surface epithelium was consistent with a proposed modified mesothelial origin. This study provides an immunohistochemical profile of these structures with a large panel of commonly available antibodies and antisera, confirming and extending the findings described in previous studies.     

Malignant transformation of exophytic Schneiderian papilloma of the nasal cavity

Schneiderian papilloma (SP) is classified into three types: inverted, oncocytic, and exophytic. Malignant transformation occurs in 10% of SP; most are inverted and oncocytic types. Malignant transformation of polypoid exophytic SP is exceptional; only two cases have been reported in the English literature. A 58-year-old man consulted our hospital because of nasal obstruction. Nasal endoscopy and imaging modalities showed a polyp in the right nasal cavity. Biopsy findings showed compatibility with SP. However, since fludeoxyglucose positron emission tomography (FDG-PET) showed signals, polypectomy was performed. Grossly, the polyp was reddish white and measured 2 cm. Histologically, the polyp consisted of a non-invasive squamous cell carcinoma (SCC) component (70%), a mature squamous component (20%), and Schneiderian epithelium (10%). Vague koilocytosis was present in the SCC component. The three components were arranged in an exophytic papillary pattern. Immunohistochemically, the non-invasive SCC component was positive for cytokeratins (AE1/3, CAM5.2, CK5/6), p63, p53, and Ki67 (labeling 50%). The mature squamous component was positive for cytokeratins (AE1/3, CAM5.2 and CK5/6), p63, and Ki67 (labeling 6%). The Schneiderian component was positive for cytokeratins (AE1/3, CAM5.2, and CK5/6), p63, and Ki-67 (labeling 4%). The tumor was HPV negative in the three components. The polyp was diagnosed as exophytic Schneiderian carcinoma arising from exophytic SP. The patient is now free from tumor 4 years after the operation.     

Sertoli-Leydig cell tumor with heterologous element: a case report and a review of the literature

The patient was a 19-year-old female who presented with a chief complaint of progressive pelvic pain. Preoperative ultrasound of the right ovary revealed an ovarian torsion as the cause of the patient’s progressive pain. Laparoscopy confirmed the torsion and revealed a right ovary measuring 10 cm in greatest diameter. Intraoperative incision into the ovary revealed a simple ovarian cystic mass measuring 3.0 x 1.5 x 0.8 cm. A solid component within the cyst was identified. Histological sections of the cystic mass demonstrated mononuclear and hyperchromatic Sertoli cells with a trabecular growth pattern. Clusters of medium-sized epithelioid cells with abundant eosinophilic cytoplasm consistent with Leydig cells were also identified between the trabeculae of Sertoli cells. In addition, focal areas of intestinal type mucinous epithelium were identified embedded within the trabeculae of Sertoli cells. Immunohistochemical studies revealed that the Sertoli cells were positive for calretinin (bright) while the Leydig cells were positive for calretinin (dim), inhibin, CAM5.2 and AE1&3. CEA showed positivity mainly of the intraluminal contents of the mucinous type intestinal epithelium. The patient had an uneventful post-operative course and was disease-free for 3 years.     

Peptide Tyrosine Tyrosine-positive Ovarian Carcinoid Tumor Arising from a Dermoid Cyst

A case of ovarian carcinoid tumor of the trabecular type is presented. The small carcinoid tumor was found incidentally in a resected dermoid cyst. Histologically, the tumor was surrounded by a smooth muscle layer with focal stromal invasion. Non carcinoid papillary mucosa and a few small glands, simulating the mucosa of the fallopian tube and remnant miillerian glands, respectively, were also present adjacent to the carcinoid tumor and enclosed by the same smooth muscle layer. The tumor cells revealed argyrophilia, and were positive for peptide tyrosine tyrosine (PYY) in the cytoplasm and for CEA in part of the luminal surface. This case was considered to be a rare ovarian carcinoid tumor arising from a dermoid cyst, without association of struma ovarii. PYY may be a potential marker for determining the histogenesis of ovarian carcinoid tumors.     

Infertility: Metastatic ovarian strumosis in an in-vitro fertilization patient

Malignant struma ovarii is a very rare tumour, with considerablecontroversy concerning the necessary histologic features formalignancy. Still more infrequent is the condition termed ‘metastaticovarian strumosis’ or simply ‘benign strumosis orstrumatosis’ and characterized by the presence of peritonealimplants of mature thyroid tissue occurring in struma ovarii.‘Strumosis’ should not be confused with malignancy.Presented is a case of ‘metastatic ovarian strumosis’in a 36-year-old woman with primary infertility who underwentthree in-vitro fertilization (IVF) cycles with ovarian stimulation.She received hormonal treatment for 6 months after her lastIVF because of ‘persistent enlarged ovarian follicles’which were in fact ‘thyroid follicles’.     

Immunohistochemical Study of Cytokeratin Distribution in the Collecting Duct of the Human Kidney

In order to assess the expression of different cytokeratins in the collecting duct cells (CDCs) of the human kidney, three consecutive sections were stained with periodic acid-Schiff, CAM 5.2, and AE-1 (CAM 5.2 recognizes cytokeratins #19,18,8 and AE 1 #19,16,15,14,10 of Moll's catalog.), respectively. By comparing these sections, it was found that most CDCs in the inner medulla were both CAM 5.2 and AE 1 positive, whereas in the outer medulla and cortex, 77% of the CDCs were both CAM 5.2 and AE 1 positive, 15% CAM 5.2 positive and AE 1 negative, 8% both CAM 5.2 and AE-1-negative, and 0.4% CAM 5.2 negative and AE 1 positive. Recent studies have shown that most CDCs express low-molecular weight cytokeratins #7,8,18 and 19(17,18,19,20). Of these cytokeratins, CAM 5.2 recognizes cytokeratins #8,18,19 and AE-1 recognizes cytokeratin #19. Therefore, most CDCs belong to one of the following three major types; 1. Those positive for cytokeratins #8,18 and 19 (CAM 5.2 and AE 1 positive), 2. Those positive for cytokeratins #8 and 18 and negative for #19 (CAM 5.2-positive and AE 1 -negative) and 3. Those negative for cytokeratins #8,18 and 19 (CAM 5.2 and AE 1 negative). A few CAM 5.2 negative and AE 1 positive cells were thought to express high molecular weight cytokeratins. The significance of these various cytokeratin expressions is discussed. Acta Pathol Jpn 41: 516 520, 1991.     

Peptide tyrosine tyrosine-positive ovarian carcinoid tumor arising from a dermoid cyst

A case of ovarian carcinoid tumor of the trabecular type is presented. The small carcinoid tumor was found incidentally in a resected dermoid cyst. Histologically, the tumor was surrounded by a smooth muscle layer with focal stromal invasion. Non-carcinoid papillary mucosa and a few small glands, simulating the mucosa of the fallopian tube and remnant müllerian glands, respectively, were also present adjacent to the carcinoid tumor and enclosed by the same smooth muscle layer. The tumor cells revealed argyrophilia, and were positive for peptide tyrosine tyrosine (PYY) in the cytoplasm and for CEA in part of the luminal surface. This case was considered to be a rare ovarian carcinoid tumor arising from a dermoid cyst, without association of struma ovarii. PYY may be a potential marker for determining the histogenesis of ovarian carcinoid tumors.     

Multicentric contiguous variant of epithelioid angiosarcoma of the bone. A rare variant showing angiotropic spread

Epithelioid angiosarcoma of the bone is a rare tumor and is a diagnostic challenge. Here we present an autopsy case of a 62-year-old man with multifocal osteolytic lesions in the extremities and the pelvis. The initial diagnosis of a tibial biopsy was poorly differentiated adenocarcinoma. On the occasion of autopsy, a fungating thrombotic nodule was found at the anterior wall of the right atrium, and small hemorrhagic infarcts with tumor thrombi were found in the lung. Histologically, the above lesions were identical to the former tibial biopsy and they showed large eosinophilic epithelioid cells with irregular ovoid nuclei and prominent eosinophilic nucleoli. Rare intracytoplasmic lumina were identified. Immunohistochemically, the tumor cells were positive for cytokeratins (CAM5.2 and AE1/AE3), CD31, factor VIII-related antigen, and vimentin. This case showed angiotropic spread of the tumor only to the right atrium and the lung, with no solid mass in other organs. Multicentric epithelioid angiosarcoma of the bone is a pitfall in pathological diagnoses, especially if a strong radiological impression of metastatic carcinoma is provided. Therefore, pathologists should be aware of this rare variant.     

RAS Mutation-Positive Follicular Variant of Papillary Thyroid Carcinoma Arising in a Struma Ovarii

Struma ovarii is an ovarian mature teratoma composed exclusively or predominantly of thyroid tissue. Malignant transformation of struma ovarii is rare and poorly understood, although this process is thought to be similar to carcinogenesis in malignant tumors of differentiated thyroid tissue originating in the thyroid gland. Genetic alterations in the mitogen-activated protein kinase pathway, including mutations of BRAF, RAS, and RET genes, have been implicated in the development of differentiated thyroid carcinoma arising in the thyroid gland. We report here a case with RAS mutation detected in a malignant struma ovarii. The patient is a 38-year-old female who had a 2.4 cm ovarian cyst noted incidentally on a first trimester ultrasound. She proceeded to ovarian cystectomy post-delivery, with pathologic examination detecting a papillary thyroid carcinoma, follicular variant, arising in a cystic teratoma. The tumor was tested for BRAF, RAS, and RET/PTC mutations. HRAS codon 61 mutation was identified. This is the first report of RAS mutation detected in the follicular variant of papillary carcinoma arising in a struma ovarii. It provides evidence that tumors developing in this setting involve molecular mechanisms similar to those implicated in tumors developing in the thyroid gland.     

Imprint cytology of strumal carcinoid of the ovary: A case report with immunocytochemical analysis

Strumal carcinoid is a rare ovarian tumor defined as carcinoid associated with struma ovarii. We report here the second cytological case of strumal carcinoid and performed immunocytochemical analysis for the first time. A 68‐year‐old Japanese female was found to have a solid tumor with small cystic components in the left ovary, and bilateral salpingo‐oophorectomy was performed. The Papanicolaou smear of the imprint cytological specimen of the left ovarian tumor revealed presence of two distinct components. The first component included thyroid follicles, which was composed of flat sheets of polygonal epithelial cells without nuclear groove and intranuclear inclusion. The other component was composed of trabecular clusters of columnar cells containing round to slender nuclei with “salt and pepper” chromatin. Immunocytochemical analysis revealed that synaptophysin was expressed in the latter component. Therefore, a cytodiagnosis of strumal carcinoid was made. Histopathological analyses confirmed the diagnosis of strumal carcinoid. Albeit rare, carcinoid tumor occurs in the ovary, and the recognition of characteristic nuclear features and cellular arrangement leads to correct cytodiagnosis. Presence of struma ovarii component suggests an ovarian origin. Moreover, immunocytochemical analysis for neuroendocrine markers aids its differential diagnosis from granulosa cell tumor and carcinoma arising from struma ovarii.     

Cytokeratin and CD30 expression in dysgerminoma

Dysgerminoma is a malignant germ cell tumor of the ovary that shares morphological, immunophenotypic, and genetic features with its testicular counterpart, seminoma. Recent evidence supports the hypothesis that seminoma can differentiate into non-seminomatous germ cell tumor types. The progression of these tumors can be measured by their acquisition of the potential to express cytokeratin intermediate filaments, a characteristic specific to epithelial differentiation. Although testicular seminomas have been widely investigated, little is known about cytokeratin or E-cadherin expression in dysgerminomas. We investigated 26 formalin-fixed, paraffin-embedded ovarian dysgerminomas with immunohistochemical stains for CAM5.2, AE1/AE3, epithelial membrane antigen, cytokeratin 7, cytokeratin 20, high-molecular-weight keratin, and E-cadherin. In addition, we investigated the CD30 and vimentin immunoreactivity of these tumors. Immunoreactivity for CAM5.2 and for AE1/AE3 was present in more than 10% of neoplastic cells in 5 (19.2%) of 26 cases and in 2 (7.7%) of 26 cases, respectively. Cytokeratin 7 showed only focal positivity and never showed positive staining in greater than 10% of dysgerminoma cells. E-cadherin staining was positive in 2 cases showing weak membranous immunostaining in more than 10% of cells. Vimentin immunoreactivity was observed in only 2 dysgerminomas, both of which had less than 10% of the neoplastic cells staining. Cytokeratin 20, epithelial membrane antigen, high-molecular-weight keratin, and CD30 were consistently negative in all cases. Our study demonstrates that cytokeratin expression in dysgerminomas is not unusual and is consistent with the hypothesis that dysgerminomas have the capacity to differentiate along epithelial lines. Furthermore, the immunohistochemical staining patterns for cytokeratins, E-cadherin, and CD30 in dysgerminomas need to be considered when assessing differential diagnoses in difficult cases of primary ovarian tumors.     

Unilateral malignant struma ovarii in a case of bilateral ovarian teratoma with raised CA-125 level: a rare case with treatment dilemmas

Struma ovarii are specialized form of mature ovarian teratoma comprised predominantly of thyroid tissue (>50%). Most of the struma ovarii are benign; rarely can they undergo malignant transformation. Elevated CA-125 levels with benign struma ovarii have been seen in only 5 cases in literature. The association of malignant struma ovarii and high CA-125 levels with pseudo-Meig syndrome has been reported in only 2 cases in English literature. We describe a case of a 46-year-old multigravida who presented with an abdominal mass and raised CA-125 levels. Radiological investigations revealed bilateral cystic adnexal masses with ossified elements on left side suggesting a teratoma. Intraoperative frozen section and final pathology revealed bilateral teratoma with follicular variant of papillary thyroid carcinoma arising in the left ovary. To the best of our knowledge, this is the first case of malignant struma ovarii in combination with bilateral teratoma. The dilemmas related to preoperative diagnoses with elevated CA-125 levels, mimicking an epithelial ovarian neoplasm; intraoperative frozen section consultation; management and follow-up issues in this rare malignancy are discussed.     

Adenomatoid tumor of the adrenal gland: case report with immunohistochemical study

Adrenal adenomatoid tumor (AT) is a recently recognized disease with marked male predominance. Herein is presented a case of adrenal AT incidentally found in a 30-year-old man and results of immunohistochemical examination of the tumor. The left adrenal gland, weighing 17 g, contained a mass measuring 3 x 2.5 x 2.5 cm in the cortical tissue. Cut surface showed a relatively well-circumscribed firm tumor with a white solid appearance. Histologically, the tumor had the typical appearance of AT described in the genital tract. Immunohistochemically, the tumor cells were positive for calretinin, D2-40, WT1, mesothelial cell antigen, CA125, thrombomodulin, vimentin and cytokeratins (stained by AE1 + AE3, OV-TL 12/30, CAM5.2 and MNF116), and negative for endothelial markers (CD31, CD34 and factor VIII-related antigen) and CD56. CD56-positive adrenocortical cells were diffusely scattered in the tumor, especially in its periphery. Immunohistochemistry of estrogen, progesterone and androgen receptors was negative. These findings confirm mesothelial origin of the tumor and suggest that this tumor has little relation to sex hormone despite male predominance.