Gliomatosis peritonei (GP) indicates the peritoneal implantation of mature neuroglial tissue and is usually accompanied by ovarian mature or immature teratoma. Here, we report a case of ovarian immature teratoma associated with gliomatosis involving the peritoneum, lymph nodes and Douglas' pouch, where gliomatosis coexisted with endometriosis. As far as we know, only seven cases of GP have been reported as coexisting with endometriosis. Eight cases with mature glial tissue in the lymph nodes, i.e., nodal gliomatosis, have been published either in association with GP or in its absence. Metaplasia of pluripotent coelomic stem cells has been suggested to be responsible for the pathogenesis of endometriosis and GP rather than implantation metastases of ovarian teratomatous tumor with varying maturation. This theory is also applied to GP independently of ovarian teratomatous tumors. To the best of our knowledge, nodal gliomatosis coexisting with GP and also involving endometriosis has not yet been reported.
Gliomatosis peritonei; Endometriosis; Immature teratoma; Ovary; Lymph nodes
Here, we report the case of a 43-year-old female who was diagnosed with a cotyledonoid dissecting leiomyoma (CDL) of the uterus. CDL is a recently described and extremely rare variant of a benign leiomyoma that can grossly masquerade as a malignancy. The 13-cm sized tumor was located primarily on the extrauterine surface as an intrauterine continuity, which showed dark red, congested, bulbous protuberances. It was multinodular appearance, encasing the bilateral adnexae and the left iliac vein. Microscopically, the nodules were separated by extensive hydropic degeneration. The nodules were composed of cigar-shaped spindle cells with no mitosis, cellular pleomorphism or coagulation necrosis. They also showed an intravascular luminal growth pattern. CDL with intravascular growth was diagnosed after excluding intravascular leiomyomatosis, disseminated peritoneal leiomyomatosis, and benign metastasizing leiomyoma. The present case is the second reported case of CDL in Korea. Recognition of this rare and bizarre, malignancy-mimicking leiomyoma is crucial to prevent inappropriate treatment.
Cotyledonoid; Leiomyoma; Intravascular; Uterus
The effect of insufficient node sampling in patients with rectal cancer managed by neoadjuvant chemoradiation followed by surgery has not been clearly determined. We evalulated the impact of insufficient sampling or even abscence of lymph nodes in the specimen on survival in patients at high-risk (T3, T4 or node positive) for rectal cancer.
We conducted a single institution, retrospective analysis of all patients who underwent surgical rectal resection following neoadjuvant chemoradiation for treatment of mid to lower rectal cancer between 1997 and 2009. ypNX was defined as the absence of lymph nodes retrieved in the resected specimen.
A total of 132 patients underwent resection for treatment of rectal cancer following neoadjuvant chemoradiation. Ninety four patients (71.2%) were considered as having node-negative disease, including ypNx and ypN0. In 38 patients (28.8%), the primary tumor was associated with regional lymph node metastases (ypNpos). The mean number of retrieved nodes per specimen was 14.2, respectively. The five-year overall survival from initial operation for the ypNx group was 100%, respectively. The estimated five-year overall survival for ypN0 and ypNpos was 84.0% and 60.3%, respectively (P =0.001). No significant differences in overall survival were observed between the ypNx and ypN0 group (P =0.302).
Absence of recovered LN in resected specimens after neoadjuvant chemoradiation was observed in 7.6% of specimens. Absence of LN should not be regarded as a risk factor for poor survival or as a sign of less radical surgery.
Rectal neoplasm; Surgery; Neoadjuvant chemoradiation
Herein, we report a case of micronodular thymoma with lymphoid stroma in a previously healthy 73-year-old male. Thymectomy was performed. The solid and macrocystic masses were encapsulated with focal invasion. The solid portion consisted of nodules of bland-looking spindle or round epithelial cells in lymphoid stroma containing prominent germinal centers. The epithelial cells had moderate amount of cytoplasm and occasional mucin production. The cystic portion was lined with cuboidal epithelium. According to World Health Organization (WHO) classification, the mass was diagnosed as a micronodular thymoma with lymphoid stroma accompanied by a pre-existing multilocular thymic cyst. Micronodular thymoma with lymphoid stroma, a possible variant of type A thymoma, is an extremely rare tumor. This so-called "unusual" variant may imply the schematic weakness of the current WHO classification that cannot cover all morphologic types. Further study is recommended for clarification of this variant and its incorporation into the current classification.
Thymoma; Thymic cyst; Lymphoid tissue; Germinal center
Optical coherence tomography (OCT) is an interferometry-based imaging modality that generates high-resolution cross-sectional images of the retina. Circumpapillary retinal nerve fiber layer (cpRNFL) and optic nerve head assessments are the mainstay of glaucomatous structural measurements in OCT. However, because these measurements are not always available or precise, it would be useful to have another reliable indicator. The macula has been suggested as an alternative scanning location for glaucoma diagnosis. Using time-domain (TD-) OCT, macular measurements have shown to provide good glaucoma diagnostic capabilities. With the adoption of spectral-domain OCT, which allows a higher image resolution than TD-OCT, segmentation of inner macular layers becomes possible. These layers are specifically prone to glaucomatous damage and thickness measurements show a comparable performance to that of glaucomatous cpRNFL measurements. The role of macular measurements for detection of glaucoma progression is still under investigation. More sophisticated measurement and analysis tools that can amplify the advantages of macular measurements are expected. For example, improvement of image quality would allow better visualization, development of various scanning modes would optimize macular measurements, and further refining of the analytical algorithm would provide more accurate segmentation. With these achievements, macular measurement can be an important surrogate for glaucomatous structural assessment.
Sertoli-Leydig tumors tend to relapse early and due to their rarity, limited data are available regarding a role of chemotherapy in the management of Sertoli-Leydig cell tumors. We present a case of recurrent ovarian Sertoli-Leydig cell tumor whose salvage treatment was successful with paclitaxel and carboplatin chemotherapy.
Ovary; Sertoli-Leydig cell tumor; Salvage therapy
In this study, we characterized a putative peroxidase Prx1 of Candida albicans by: 1) demonstrating the thioredoxin-linked peroxidase activity with purified proteins, 2) examining the sensitivity to several oxidants and the accumulation of intracellular reactive oxygen species with a null mutant (prx1Δ), a mutant (C69S) with a point mutation at Cys69, and a revertant, and 3) subcelluar localization. Enzymatic assays showed that Prx1 is a thioredoxin-linked peroxidase which reduces both hydrogen peroxide (H2O2) and tert-butyl hydroperoxide (t-BOOH). Compared with two other strong H2O2 scavenger mutants for TSA1 and CAT1, prx1Δ and C69S were less sensitive to H2O2, menadione and diamide at all concentrations tested, but were more sensitive to low concentration of t-BOOH. Intracellular reactive oxygen species accumulated in prx1Δ and C69S cells treated with t-BOOH but not H2O2. These results suggest that peroxidase activity of Prx1 is specified to t-BOOH in cells. In both biochemical and physiological cases, the evolutionarily conserved Cys69 was found to be essential for the function. Immunocytochemical staining revealed Prx1 is localized in the cytosol of yeast cells, but is translocated to the nucleus during the hyphal transition, though the significances of this observation are unclear. Our data suggest that PRX1 has a thioredoxin peroxidase activity reducing both t-BOOH and H2O2, but its cellular function is specified to t-BOOH.
hydrogen peroxide; peroxidase; PRX1; tert-butyl hydroperoxide; translocalization
Myeloid sarcoma is a rare extramedullary myeloid tumor, which is frequently misdiagnosed when no evidence of leukemia is initially observed. Here, we report on a peculiar case of a 49-year-old man afflicted with multiple masses in the jejunum, the superior mesentery, and the serosa of the transverse colon, without leukemic manifestation. The tumor was composed of undifferentiated small round cells containing eosinophilic cytoplasm, which were negative for myeloperoxidase, nonspecific esterase, lysozyme, terminal deoxynucleotidyl transferase, leukocyte common antigen, CD3, CD4, CD15, CD20, CD30, CD43, CD56, CD68/PG-M1, CD79a, human melanoma black-45, c-kit, and CD34 with positivity only for CD68/KP1, CD99, and vimentin. Under electron microscopy, those cells had abundant membrane-bound cytoplasmic granules that measured 200 to 300 nm in diameter, which were consistent with granulocytic azurophilic granules. The tumor was finally diagnosed as a myeloid sarcoma. The presence of non-leukemic myeloid sarcomas showing immunonegativity for conventional myeloid-leukemic markers necessitated a diagnosis by ultrastructural observation.
Sarcoma, myeloid; Jejunum; CD68/KP1; CD99; Microscopy, electron
Metastatic calcification is rare; it is found during autopsy in patients who underwent hemodialysis. Diffuse calcium precipitation of small and medium-sized cutaneous vessels, known as calciphylaxis, can result in progressive tissue necrosis secondary to vascular calcification. This condition most commonly involves the skin; however, a rare occurrence of visceral calciphylaxis has been reported. Here we report on an autopsy case. Despite a thorough evaluation, and even performing an autopsy, the underlying cause of acute-onset hypercalcemia, resulting in the production of pulmonary calciphylaxis and metastatic renal calcification associated with acute respiratory and renal failure, could not be determined. Metastatic calcification often lacks specific symptoms, and the degree of calcification is a marker of the severity and chronicity of the disease. This unusual autopsy case emphasizes the importance of rapidly progressing visceral calciphylaxis, as well as its early detection.
Calcification; Hypercalcemia; Calciphylaxis
We report two cases of follicular adenoma of the thyroid with extensive extracellular mucin deposition. Fine needle aspiration in Case 1 showed singly discohesive polygonal cells in a granular mucinous background. They contained abundant eosinophilic cytoplasm, nuclear irregularities, and frequent nuclear inclusions with occasional bizarre mitoses. A right lobectomy was done. In Case 2, a 47-year-old Caucasian woman with multinodular goiter had total thyroidectomy and a yellow-tan nodule was found within the right lobe. Both tumors were well-encapsulated masses with thick capsules. Each was characterized by microfollicles without papillae in a mucinous stroma. Tumor cells were positive for thyroglobulin and negative for calcitonin, CEA, galectin-3, HBME-1, and CK19. The extracellular mucin stained with Alcian-blue and colloidal iron but not with mucicarmine and D-PAS. No BRAF gene mutation was detected. Because there were neither capsular nor vascular invasions, both cases were diagnosed as follicular adenomas of the thyroid with extensive extracellular mucin deposition, which as proposed by the WHO classification can be categorized as a mucinous variant of follicular adenoma. Retrospectively, frequent nuclear inclusions and the absence of nuclear grooves in the mucin-containing background of cytologic smears and histologic sections were shared by those of mucin-producing papillary carcinoma. It is unclear whether it belongs to an existing category of thyroid neoplasm with mucin production or whether it is truly a new tumor variant. Furthermore, pathologists should pay attention to avoid misdiagnosis of this variant of follicular neoplasm that shows an overlapping cytology with that of papillary carcinoma.
thyroid; mucin; follicular adenoma; aspiration; fine needle
Pulmonary cystic and cavitary lesions caused by diverse etiologies are commonly encountered in chest imaging. The terms "cyst" and "cavity" are used to describe air-filled regions in the center of a nodule or consolidation of the lung. To date, only radiologic aspects of these lesions have been addressed. The morphologies of pulmonary cystic and cavitary lesions exhibit a broad spectrum, ranging from benign to malignant pulmonary diseases of acquired or congenital origin, including variable infectious diseases. In this review, we summarized the differential diagnosis of pathological entities to provide pathologists and radiologists with an overview of the diseases most commonly associated with pulmonary cystic and cavitary lesions in adults and children. The results showed slightly different patterns in the distribution of the diseases in the two groups. The most common causes of cavitary lesions include malignancy and infection in adults, and congenital malformation in children. Therefore, identification of pathologic entities correlating with the radiologic findings, clinical course, and location of the lesion is important in the evaluation of cystic and cavitary lung lesions in order to avoid unnecessary surgical procedures or delayed treatment.
Lung; Cysts; Cavity; Congenital; Malignant neoplasms
To establish and validate a formula to predict spectral domain (SD)-optical coherence tomography (OCT) retinal nerve fiber layer (RNFL) thickness from time domain (TD)-OCT RNFL measurements and other factors.
SD-OCT and TD-OCT scans were obtained on the same day from healthy participants and patients with glaucoma. Univariate and multivariate linear regression relationships were analyzed to convert average Stratus TD-OCT measurements to average Cirrus SD-OCT measurements. Additional baseline characteristics included age, sex, intraocular pressure, central corneal thickness, spherical equivalent, anterior chamber depth, optic disc area, visual field (VF) mean deviation, and pattern standard deviation. The formula was generated using a training set of 220 patients and then evaluated on a validation dataset of 105 patients.
The training set included 71 healthy participants and 149 patients with glaucoma. The validation set included 27 healthy participants and 78 patients with glaucoma. Univariate analysis determined that TD-OCT RNFL thickness, age, optic disc area, VF mean deviation, and pattern standard deviation were significantly associated with SD-OCT RNFL thickness. Multivariate regression analysis using available variables yielded the following equation: SD-OCT RNFL = 0.746 × TD-OCT RNFL + 17.104 (determination coefficient [R2] = 0.879). In the validation sample, the multiple regression model explained 85.6% of the variance in the SD-OCT RNFL thickness.
The proposed formula based on TD-OCT RNFL thickness may be useful in predicting SD-OCT RNFL thickness. Other factors associated with SD-OCT RNFL thickness, such as age, disc area, and mean deviation, did not contribute to the accuracy of the final equation.
Glaucoma; Retinal nerve fiber layer; Spectral domain optical coherence tomography; Time domain optical coherence tomography
Here we report the first Korean case of a girl who developed noninvasive squamous cell carcinoma of the vulva at the age of 16 years. She was taking tacrolimus, an immunosuppressive agent, after living-related liver transplantation. The vulvar masses were microscopically proved as vulvar intraepithelial neoplasm II and III, even squamous cell carcinoma in situ. Human papillomavirus subtypes (69 and 73) and human papillomavirus types (66, 70, 73, and 43) were detected in the vulvar mass and the cervicovaginal smear, respectively. The outcome of liver transplantation for children has been markedly improved during the last several decades. However, the present case highlights the need to perform periodic genital examinations for the adolescents after liver transplantation. In addition to the high risk and probable high subtypes, uncommonly found human papillomavirus subtypes were extracted from her vulvar cancer. The present case is the first to show the possible relationship between previously unknown and uncommon human papillomavirus subtypes and pediatric post-transplant vulvar squamous cell carcinoma. More attention should be paid to the vulvar and cervical surveillance of pediatric transplant recipients by both medical specialists and general physicians.
Children; Human papillomavirus; Liver transplantation; Squamous cell carcinoma; Vulva
A 62-yr-old man presented with a 5-yr history of intermittent abdominal distention and pain. These symptoms persisted for several months and subsided without treatment. A diagnosis of suspected small bowel lymphoma was made based on plain radiograph and computerized tomogram findings, and he was referred to our institution for further evaluation. Segmental resection of the small intestine was performed and the diagnosis of marginal zone B-cell lymphoma associated with amyloidosis was made. This is the first case of marginal zone B-cell lymphoma of mucosa-associated lymphoid tissue (MALT) in the small intestine associated with amyloidosis in Korea.
Gastrointestinal Tract; Lymphoma; Lymphoma, B-Cell, Marginal Zone; Amyloidosis
The purpose of this study was to investigate the prevalence and characteristics of glaucoma in the population of the rural Korean town, Sangju.
Residents of Sangju aged greater than 50 years old were included in this study. Participants completed an interview examining their medical and ophthalmic history Information was collected on the participants' uncorrected and best corrected vision scores, slit lamp examination results, angle width measurements using the Van Herick technique, gonioscopy if the angle width was less than 1 / 4 angle, intraocular pressure (IOP) assessed with the Goldmann applanation tonometry, optic disc examination results, and a visual field test results using frequency-doubling perimetry in cases in which glaucoma was suspected. Glaucoma was diagnosed according to the criteria described by the International Society for Geographic and Epidemiological Ophthalmology.
1,118 residents aged greater than 50 years were examined initially from a population of 2,984 people. Of these, 671 subjects (60%) participated in this study. The prevalence of glaucoma was determined to be 3.4% (95% confidence interval [CI], 2.1-4.8). Open-angle glaucoma with low IOP was determined to be the most common form with a prevalence rate as high as 2.5% (95% CI, 1.8-3.7). Additionally, primary angle closure glaucoma was determined to have a prevalence rate of 0.3% (95% CI, 0.1-0.9). Open-angle glaucoma with low IOP accounted for 94.4% of the open-angle glaucoma cases.
The prevalence of glaucoma among the population of rural Sangju was 3.4%, and open-angle glaucoma with low IOP was the most common form accounting for 94.4% of the total number of cases.
Epidemiology; Glaucoma; Open angle glaucoma; Prevalence
A 60-year-old woman who had experienced two episodes of amaurosis fugax in her right eye presented with vision loss. Two weeks earlier, at a private clinic, she was diagnosed with impending central retinal vein occlusion (CRVO) of the right eye and received an intravitreal injection of bevacizumab. Two weeks after this injection she was diagnosed with ischemic CRVO. At 11-weeks post-presentation, extremely ischemic features were observed with fluorescein angiographic findings of severe vascular attenuation and extensive retinal capillary obliteration. At 22-weeks post-presentation she was diagnosed with neovascular glaucoma; she experienced no visual improvement over the following several months.
Bevacizumab; Central retinal vein occlusion
A 51-yr-old man presented with an enlarged right testis for two months. The radically resected testis showed a relatively well-circumscribed ovoid mass, nearly replacing the normal architecture with central cystic changes. Microscopically, the mass was composed of ovoid shaped tumor cells of a moderately differentiated squamous cell carcinoma (SCC). The central portion of the mass was filled with well-formed laminated keratinous materials and the remnant cavity lined by dysplastic squamous epithelium, indicated SCC may be derived from an epidermal cyst. SCC is among the most common types of neoplasm afflicting human beings, but it is rare in the testis. To our knowledge, this is the second report of the testicular squamous cell carcinoma occurring in a patient without other primary tumors, and the firstly reported case in Korea.
Carcinoma, Squamous Cell; Testis
Most of thyroid lymphomas are B-lineage, and T-cell lymphomas are rare. Here, we report a case of primary thyroid T-cell lymphoma associated with Hashimoto's thyroiditis. A 48-yr-old woman presented with incidentally found neck mass. Histologically, the resected right lobe of the thyroid was replaced by monomorphic small atypical lymphoid cells with lymphoepithelial lesion-like change, most of which were immunoreactive for CD3, CD8, βF-1, and TIA-1. Peripheral T-cell lymphoma, unspecified, was finally diagnosed after molecular study for TCR-γ gene rearrangement. This is the second case of cytotoxic T-cell lymphoma reported in the thyroid gland so far. Unique association between thyroid follicles and neoplastic lymphocytes may be characteristic feature of this type of T-cell lymphoma.
Lymphoma, T-Cell; Thyroid Gland; T-Lymphocytes, Cytotoxic; Lymphoma, B-Cell, Marginal Zone
To report the experiences at our institute of pars plana vitrectomy (PPV) in combination with intravitreal triamcinolone acetonide (IVTA) injection in patients with retained lens material after cataract surgery.
The medical records of patients who underwent PPV between January 2005 and December 2006 after complicated cataract surgery in which lens material was dropped into the vitreous cavity were reviewed retrospectively.
Five eyes of five patients were treated by vitrectomy and removal of the dislocated lens material and were administered an adjuvant intravitreal injection of 4 mg triamcinolone acetonide at the end of surgery. IVTA administration induced a rapid improvement in vision within six weeks postoperatively. Final visual acuity was 20/32 or better in all patients. Inflammatory cystoid macular edema (CME) that existed preoperatively was promptly improved after IVTA. Intraocular pressure increased transiently in one patient. No procedure-related complications were observed.
PPV with additional IVTA was successfully performed in five eyes with retained lens material and resulted in a marked resolution of vitreous inflammation, an improvement in CME and rapid visual recovery. The authors recommend that IVTA administration should be investigated more thoroughly as an adjunct during the management of retained lens material.
Cataract; Cystoid macular edema; Dropped nucleus; Retained lens material; Triamcinolone acetonide
Clear cell ependymoma was included in the World Health Organization classification of the nervous system in 1993, and all the reported cases, except for two in the spinal cord, were located in the brain, mainly in the supratentorial compartment. Astrocytomas outnumber ependymomas in the spinal cord, and the two entities partly share cytologic findings such as long, bipolar glial processes and oval to round nuclei resembling those seen in pilocytic astrocytoma. Here, we report the first Korean case of intramedullary clear cell ependymoma of the spinal cord, which is the third case situated in the spinal cord in the literature. The crush smear revealed round-to-oval nuclei with occasional nuclear eosinophilic inclusion and rare nuclear grooves. Cytoplasm had fluffy eosinophilic glial processes, and acellular fibrillary zone. On hematoxylin-eosin stain, oval to round tumor cells had large central nuclei with indistinct nucleoli and a moderate amount of clear cytoplasm, i.e. perinuclear halo, mimicking oligodendroglioma. Perivascular pseudorosettes and ependymal clefts were rarely found. In retrospect, perinuclear halo was absent on crush smears. Ultrastructurally, they had extensive surface microvilli and edematous cytoplasm filled with abundant glial filaments and microlumens with or without microvilli. Intercellular long cell junctions of the zipper-like zonula adherens type were found.
Ependymoma, Clear Cell; Spinal Cord; Crush Smear; Microscopy, Electron
Primary cardiac tumors are extremely rare and can originate within the heart or be the result of tumor spread from other sites. We report a female patient with a pulmonary vein tumor extending into the left atrium that had a suspicious primary malignant origin with a sacral metastatic carcinoma. The patient was admitted complaining of pain in her buttock area as a result of a sacral tumor. It was believed that the sacral tumor was a metastasis from the imaging study and clinical manifestation. The primary malignant origin was evaluated. The chest CT showed a left atrium thrombus-like lesion without a pulmonary abnormality. After a transesophageal echocardiogram, the patient was diagnosed with a pulmonary vein tumor extending to the left atrium. The patient was given palliative radiotherapy for the sacral pain. Initially, the clinical impression was a metastatic sacral tumor with a thromboembolism of the left atrium. However, this patient was finally diagnosed with a pulmonary vein tumor with a left atrium extension by a transesophageal echocardiogram.
Pulmonary vein tumor; Left atrium tumor
Neurofibromatosis type 1 (von Recklinghausen's disease, NF-1) is an autosomal-dominant neurocutaneous disorder characterized by abnormal skin pigmentation (café au lait spots and axillary freckling), cutaneous and plexiform neurofibromas, skeletal dysplasias, and Lisch nodules (pigmented iris hamartomas). Gastrointestinal stromal tumors (GISTs) are the most common tumors of mesenchymal origin in the gastrointestinal tract, mesentery, omentum, and retroperitoneum. Here, we report a case of GIST in the ileum of a 76-year-old woman previously diagnosed as NF-1. She was admitted due to sudden onset of abdominal pain. Contrast enhanced CT scan revealed a moderately defined, peripherally enhanced soft tissue mass of about 8.8×7.3cm, originating from the small bowel in the left of the abdomen. Surgical excision was performed and the tumor was found to be composed of tumor cells that were positive for c-kit protein. The patient started imatinib treatment a month later, but stopped medication due to dyspepsia after a few months and eventually progressed after 18 months
Gastrointestinal Stromal Tumors; Neurofibromatosis
Methylation of p16 is an important mechanism in cervical carcinogenesis. However, the relationship between cervical squamous cell carcinoma (SCC) and Epstein-Barr virus (EBV) remains controversial. Here, we explored whether EBV infection and/or p16 gene inactivation would play any role in cervical carcinogenesis. Eighty-two specimens included 41 invasive SCCs, 30 cervical intraepithelial neoplasm (CIN; CIN 1, 11 cases, CIN II, 3 cases, CIN III 16 cases) and 11 nonneoplastic cervices. EBV was detected by polymerase chain reaction (PCR) for EBNA-1 and in situ hybridization for EBER-1. The p16 methylation-status and the expression of p16 protein were studied by methylation-specific PCR and immunohistochemistry, respectively. The materials were divided into four groups: 1) nonneoplastic cervices, 2) CIN I, 3) CIN II-III and 4) invasive SCCs. p16 methylation and p16 immunoexpressions increased in CIN and invasive SCCs than nonneoplastic tissue. p16-methylation and p16-immunoreactivities were higher in the EBV-positive group (p=0.009, p<0.001) than in the EBV-negative group. EBV was detected more frequently in CIN and SCCs than nonneoplastic cervices. In conclusion, a correlation between p16 methylation, p16 immunoreactivity and the detection of EBV strongly suggested that the cooperation of EBV and p16 gene may play a synergic effect on cell cycle deregulation.
Protein p16; Methylation; Herpesvirus 4, Human; Epstein-Barr Virus Infections; Cervix Neoplasms; Carcinogenesis
Angiosarcoma of the thyroid has long been a controversial entity, and it is histologically defined as cleft-like anastosmosing spaces lined by large, atypical cells of endothelial lineage. However, clear-cut separation between the angiosarcoma and anaplastic carcinoma of the thyroid is difficult because they yield nearly the same clinical prognosis and overlapping histologic findings. We report a case of thyroid neoplasm composed of minimally invasive well differentiated follicular carcinoma and angiosarcoma with intervening transitional area. Immunohistochemically, the angiosarcomatous portion showed focal immunoreactivity for endothelial markers such as CD31, CD34, Ulex europaeus 1 lectin, factor VIII-related antigen, and immunonegativity for epithelial markers including pancytokeratin, epithelial membrane antigen and thyroglobulin, whereas the reverse was demonstrated in the minimally invasive follicular carcinomatous portion. The follicular carcinoma portion was positive for thyroid transcription factor-1 (TTF-1). Each component showed ultrastructural findings of epithelial and endothelial differentiation, respectively. The present case was unique in that angiosarcoma of the thyroid was confirmed by immunohistochemistry and electron microscopy, as well as light microscopy, and also coexisted with a minimally invasive well differentiated follicular carcinoma in the same mass. This combination has never been described in the literature. Although restricted to a single case, the present case further supports that angiosarcoma is a true existent entity rather than a variant of anaplastic carcinoma.