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PubMed2003

Corticomedullary mixed tumor of the adrenal gland with concurrent adrenal myelolipoma.

We report the case of a corticomedullary mixed tumor of the adrenal gland in a 55-year-old woman with a left adrenal mass who presented with mild symptoms of Cushing syndrome and an elevated urinary cortisol level. The patient underwent a left adrenalectomy. A well-circumscribed 2.5-cm mass, composed of an admixture of adrenal cortical cells and pheochromocytes, and an incidental 0.7-cm myelolipoma were present in the resected left adrenal gland. The diagnosis of adrenal corticomedullary mixed tumor was confirmed by both immunohistochemistry and electron microscopy. To our knowledge, this is the eighth well-documented report of this rare tumor.

Adrenal Cortex Neoplasms↗

PubMed2001

Corticomedullary mixed tumor of the adrenal gland.

Corticomedullary mixed tumors of the adrenal gland are quite rare, with only five well-documented cases reported in the literature.(1-4) Herein, we report the light microscopic and immunohistochemical features of two cases of this rare tumor. Patient 1 is a 34-year-old woman who presented with hypertension, hair loss, and amenorrhea of 1-year duration. Patient 2 is a 52-year-old woman who presented with flank pain and what appeared to be a renal mass on arteriogram with no history of hypertension, Cushing's syndrome, or other endocrine abnormalities. At surgery, the tumor was noted to arise from the adrenal gland rather than the kidney and adrenalectomy was performed. In both cases, the surgically resected specimens consisted of a well-circumscribed, single adrenal mass surrounded by a rim of uninvolved adrenal cortical tissue. The tumors were composed of adrenal cortical cells intimately admixed with pheochromocytes. Immunohistochemical studies highlighted these two cellular components. The pheochromocytes were strongly reactive with chromogranin and the sustentacular cells with S-100 protein, whereas the adrenal cortical cells reacted specifically with inhibin. Thus, we report two additional cases of mixed corticomedullary tumor of the adrenal gland. Ann Diagn Pathol 5:304-308, 2001. This is a US government work. There are no restrictions on its use.

Adrenal Cortex↗

PubMedDate not supplied

Papillary glioneuronal tumor--a new tumor entity.

Glioneuronal neoplasms of the CNS comprises a heterogeneous group of generally low-grade tumors expressing glial and neuronal cells of varying differentiation. Recently, a new variant of the glioneuronal tumors has been identified. We present a case of a glioneuronal tumor located in the left frontal lobe of a 16-year-old boy who developed seizures 6 months after brain concussion. MR scan demonstrated an irregular, but well circumscribed, mixed cystic and solid tumor with contrast enhancement in the solid part. Histology showed a papillary glioneuronal tumor. The tumor is indolent with no sign of recurrence after gross total resection.

Adolescent↗

PubMed2006

[Analysis of the relationship between pathology and recurrence of primary lacrimal epithelial tumors].

OBJECTIVE: To analyze the relationship between pathology and recurrence of primary lacrimal epithelial tumors. METHODS: 128 cases of primary lacrimal epithelial tumors including benign mixed tumor (74 cases, 57.8%), adenoid cystic carcinoma (22 cases, 17.2%) and malignant mixed tumor (18 cases, 14.1%) were subjected in the study. Pathological features were analyzed and compared with their recurrence. RESULTS: The recurrent rate of benign mixed tumor, adenoid cystic carcinoma and malignant mixed tumor was 23.0%, 18.2% and 27.8%, respectively. The recurrence of benign mixed tumor was statistically related to pathological classification and encapsulates. CONCLUSIONS: Primary lacrimal epithelial tumors show variant types and high recurrent rate. The pathological features were decisive in diagnosis, selection of treatment and the time of follow-up.

Adult↗

PubMed2005

[Histological classification of human gliomas: state of art and controversies].

The histological classification of human gliomas remains in 2005 a challenge. The aim is to define the histological type of glioma (astrocytic, oligodendrocytic or mixed) and the grade in order to classify the patients and give them an accurate treatment. Although the standard remains the WHO classification, this classification suffered from lack of reproducibility among pathologists. In particular this classification does not take into account the intrinsic morphological heterogeneity of infiltrative gliomas and does not discriminate the tumour cells from the residual brain parenchyma. According to the WHO classification, infiltrative gliomas encompass astrocytic gliomas (diffuse astrocytomas grade II, anaplastic astrocytomas grade III and glioblastomas grade IV), oligodendroglial tumours (oligodendrogliomas grade II, anaplastic oligodendrogliomas grade III) and mixed gliomas (oligoastrocytomas grade II and anaplastic oligoastrocytomas grade III). Circumscribed gliomas mainly corresponds to pilocytic astrocytomas (grade I). In contrast, the Sainte Anne classification takes into account the macroscopic informations provided by imaging techniques and the tumour growth patterns. Three distinct tumour growth patterns may be seen in gliomas, type I: tumor tissue only, type II: tumour tissue and isolated tumor cells permeating the brain parenchyma (ITC) and type III: ITCs only and no tumor tissue. According to the Sainte Anne classification, gliomas are divided into astrocytic gliomas (pilocytic astrocytomas, structure type I, glioblastomas structure type II) and oligodendrogliomas and mixed oligoastrocytomas (grade A: lack of contrast enhancement and lack of endothelial hyperplasia, structure type III; and grade B: contrast enhancement or endothelial hyperplasia, structure type II and III). In the future the glioma classification has to be unique and should take into account clinical data, neuroradiological and histological features and results of molecular biology.

Astrocytoma↗

PubMed2002

Lung tumors with mixed histologic pattern. Clinico-pathologic characteristics and prognostic significance.

OBJECTIVE: To analyze and compare clinico-pathologic characteristics and survival between lung tumors with mixed histologic pattern and our population of resected lung tumors with single histology in the same period. METHODS: From January 1993 to December 1999, 1158 patients received resection for lung tumors. Of these, 59 (5.1%) presented a mixed histologic pattern on the surgical specimen. There were 48 men and 11 women (mean age 64 years, range 43-79). Three groups of tumors were identified: adenosquamous carcinoma, combined neuroendocrine + non-neuroendocrine carcinoma (NNEC) and biphasic tumors (epithelial + mesenchymal malignant components) represented by carcinosarcoma and blastoma. The combined neuroendocrine tumors were further divided in small cell lung carcinoma (SCLC) + large cell neuroendocrine carcinoma (LCNEC)/NNEC and other neuroendocrine tumors/NNEC. Clinico-pathologic characteristics, pTNM and survival were analyzed and compared to our population of resected lung tumors with single histology. RESULTS: There were 33 adenosquamous carcinomas, 19 combined SCLC+LCNEC/NNEC, two other neuroendocrine tumors/NNEC and five biphasic tumors (three carcinosarcomas and two blastomas). Among adenosquamous carcinomas, high cell grading (G2 or G3), advanced stage (IIIa or higher) and intratumoral perineural invasion were significantly more evident than in the single histology population. Among combined neuroendocrine/NNEC, high cell grading (G3) and intratumoral vascular invasion were significantly more evident than in the single histology population. Among biphasic tumors, all were at early stages and showed high cell grading (G3). Three-year survival rates were 46% in the single histology group, 28% in the adenosquamous group and 21% in the combined SCLC + LCNEC/NNEC. The difference among the three groups was significant (P = 0.013). Median survival of biphasic tumors was 19 months (range 8-37). CONCLUSIONS: Lung tumors with mixed histologic pattern are rare tumors. Adenosquamous carcinoma and combined SCLC + LCNEC/NNEC present a more aggressive clinico-pathologic behaviour and reduced survival as compared to the single histology population of resected lung tumors.

Adult↗

PubMed1999

Heterotopic endochondrial ossification with mixed tumor formation in C3(1)/Tag transgenic mice is associated with elevated TGF-beta1 and BMP-2 expression.

Transgenic mice which express the simian virus 40 large T-antigen (Tag) under the regulatory control of the hormone responsive rat C3(1) gene develop unusual lesions of heterotopic bone growth associated with mixed tumor formation arising from eccrine sweat glands found only in the foot pads of mice, ischiocavernosus muscle adjacent to bulbourethral glands and occasionally the salivary and mammary glands. These lesions are very similar to mixed tumors arising in several types of human cancers. Based upon electron microscopic examination and immunocytochemical analyses of cellular differentiation markers, the mixed proliferative lesions in this transgenic mouse model begin with the Tag-induced proliferation of epithelial and myoepithelial cells. The proliferation of these two types of cells results in hyperplasia and adenomatous transformation of the epithelial component, whereas the proliferating myoepithelial cells undergo metaplasia to form chondrocytes which deposit extracellular matrix, including collagen fibers. Cartilage develops focally between areas of epithelial proliferation and subsequently ossifies through a process of endochondrial bone formation. The metaplasia of myoepithelial cells to chondrocytes appears to require the inductive interaction of factors produced by the closely associated proliferating epithelial cells, including members of the TGF-beta superfamily. We demonstrate that TGF-beta1 protein accumulates in the extracellular matrix of the lesions, whereas RNA in situ hybridization reveals that BMP-2, another strong inducer of heterotopic bone formation, is overexpressed by the proliferating epithelial cells during the development of ectopic bone. The formation of sarcomatous tumors within the mixed tumors appears to be androgen-dependent and more frequent in mice lacking a normal allele of p53. This process of cartilage and bone induction may mimic epithelial-mesenchymal interactions which occur during embryonic bone formation. These transgenic mice may provide new insights into the processes of ectopic endochondrial bone formation associated with mixed tumor formation and serve as a useful model for human heterotopic bone disease.

Actins↗

PubMed2006

Novel low-grade glioneuronal neoplasm presenting in an octogenarian: case report and review of the literature.

Glioneuronal neoplasms are rare tumors that typically affect patients in the first three decades of life. Since the publication of the World Health Organization (WHO) 2000 classification of tumors, further variants of these tumors have been reported. We present an 83-year-old gentleman who presented with a history of ataxia and weight loss. MRI and CT scan revealed a ring-enhancing bihemispheric lesion in the premotor cortex consistent with a malignant primary brain tumor crossing the corpus collosum. The patient underwent a sterotactic biopsy with drainage of the cystic component. Histopathologic studies revealed a mixed glioneuronal tumor with benign characteristics. A craniotomy was performed and the tumor was resected. Postoperatively, the patient has been followed with serial MRI scans with no evidence of disease recurrence at 27 months. Glioneuronal tumors are extremely uncommon in the octogenarian population, however, it is important to include them in the differential diagnosis of intracerebral masses. They are histopathologically quite heterogeneous, and in this report we present a novel subtype. Radiographically, these lesions can mimic more aggressive primary brain tumors.

Aged↗

PubMed2001

Immunohistochemical evaluation of PCNA and Ki-67 in ameloblastoma.

Thirty-two ameloblastoma tissues were immunohistochemically studied using monoclonal anti-proliferating cell nuclear antigen (PCNA) and anti-Ki-67 antibodies. Positive cells were evaluated and analyzed in relation to the WHO classification, cytological pattern of the outer layer cell, clinical appearance, tumor location, radiographic appearance and patient's age. In regard to the cytological pattern of the outer layer cells, the basal cell type had significantly higher PCNA and Ki-67 (P<0.05) labeling indices than the cuboidal cell type. The solid type had significantly higher PCNA and Ki-67 (P<0.05) labeling indices than the cystic and the mixed type. The labeling index of the younger patient was found to be the lowest, the middle age was in the middle and the older patient was the highest. These results indicated that the proliferating activities of ameloblastomas are quite variable, and the evaluations of Ki-67 and PCNA seem to be good indicators to assess the proliferating activity of each type of ameloblastomas.

Adolescent↗

PubMed1998

Sialoglycoconjugates in primary and recurrent mixed tumors of lacrimal glands.

We examined the activity of Griffonia simplicifolia II agglutinin (GS2) before and after neuraminidase treatment in 2 cases of primary benign mixed tumor (BMT) and a case of recurrent mixed tumor (MT). Histologically, each type of MT is comprised of epithelial and mesenchymal components. GS2, which showed no binding to either structural component in the primary BMT, was specifically bound to the mesenchymal component in the recurrent MT. After neuraminidase treatment, there was a marked increase in GS2-binding levels in the epithelial component of the primary BMT and in both components of the recurrent MT. These results might reveal a prominent sialylation in the recurrent MT.

Adult↗

PubMed2000

Sporadic unilateral vestibular schwannoma with islets of meningioma: case report.

OBJECTIVE AND IMPORTANCE: Vestibular schwannomas with meningioma islets have been rarely reported in the literature; they have been observed only among patients with neurofibromatosis Type II. We present a case of a sporadic mixed tumor in a patient without neurofibromatosis Type II that was not suspected before surgery. CLINICAL PRESENTATION: A 59-year-old female patient presented with clinical signs of progressive loss of hearing. Her family history did not include evidence of neurological diseases. Magnetic resonance imaging scans revealed a typical unilateral vestibular schwannoma. INTERVENTION: The tumor presented with invasion of the surrounding arachnoid membrane, as well as Cranial Nerves VII and VIII. Preservation of the facial nerve with complete removal of the tumor was not possible. Therefore, Cranial Nerve VII reconstruction was performed. CONCLUSION: The concomitant occurrence of schwannomas and meningiomas infiltrating the arachnoid membrane might be related to poor clinical outcomes for patients with neurofibromatosis Type II, with respect to preservation of facial and acoustic nerves. Among sporadic schwannomas, this phenomenon is extremely rare.

Female↗

PubMed2004

Sarcomatoid hepatocellular carcinoma with hepatoblastoma-like features in an adult.

A mixed epithelial and mesenchymal tumor of the liver arising in an adult is rare and is mostly classified as sarcomatoid hepatocellular carcinoma (HCC). In this study, a case of sarcomatoid HCC in an adult with hepatoblastoma (HB)-like features, which produced difficulty in the differential diagnosis between sarcomatoid HCC and mixed HB, is presented. The epithelial component of the tumor composed of poorly differentiated HCC, Edmondson's grade III, and more primitive components, which were embryonal and small cell undifferentiated components of HB-like areas. The small undifferentiated cells surrounded HCC and the embryonal component of HB-like area, and revealed transition partly to areas of rhabdomyosarcoma. A small portion of chondrosarcoma was also noted. Immunohistochemical analysis showed that HCC and the embryonal component of HB-like areas expressed alpha-fetoprotein (AFP) and cytokeratin 8. The small undifferentiated cells were negative for AFP but stained with cytokeratin 8 as well as CD56, which is a marker of primitive cells in many sarcoma and HB. It is not certain whether small undifferentiated cells belong to hepatic progenitor cells or primitive mesenchymal cells. Polymerase chain reaction-single-strand conformation polymorphism analysis for beta-catenin mutation using microdissection revealed no mutation of any components. A review was undertaken of the cases previously reported as adult hepatoblastoma without detailed immunohistochemical study and consider many of them may be sarcomatoid HCC. These primitive and sarcomatoid components would be arising from the dedifferentiation process of HCC.

Aged↗

PubMed2005

Nested stromal epithelial tumor of the liver: six cases of a distinctive pediatric neoplasm with frequent calcifications and association with cushing syndrome.

Rare cases of nonhepatocytic mixed stromal and epithelial tumors of the liver with associated calcification and ossification have been described previously. We report 6 similar cases in children, including 2 cases associated with ectopic ACTH production. The patients were between 2 and 14 years of age at diagnosis. All tumors presented as a solitary liver mass with no extrahepatic involvement. Two adolescent females with palpable abdominal tumors presented with Cushing syndrome that abated after excision of the tumors. The other children had tumors identified incidentally on imaging studies or at laparotomy. All tumors were well circumscribed, ranging in size from 4.0 to 30.0 cm in greatest diameter. Histologically, they shared an organoid arrangement of cellular nests that were comprised of an admixture of both spindled and epithelioid cells. These cellular nests were surrounded by a band of delicate myofibroblasts and set in a dense fibrous stroma that contained slit-like to dilated blood vessels. A variable proliferation of bile ducts extended from the fibrous stroma and focally surrounded the cellular nests. One case showed a sheet-like overgrowth of the nested cells with associated necrosis. The cellular nest cells were immunoreactive for EMA, CD56, neuron specific enolase, pan-cytokeratin (4 of 6 cases), vimentin (5 of 6 cases), and WT-1 amino terminus (4 of 6 cases). Cytokeratin and EMA stained mostly epithelioid nest cells, with vimentin and WT-1 staining predominantly the spindled nest cells. The 3 cases from adolescent females showed immunoreactivity for ACTH in the nested cell population but not in the surrounding stromal cells. Immunohistochemical stains for synaptophysin and chromogranin were negative in all cases. Psammomatous calcifications were present focally in 2 cases and were extensive in 3 cases. Ossification or osteoid formation was present in 4 cases. The 1 patient whose tumor had sheet-like overgrowth of the nested cell population had a local recurrence with multiple hepatic nodules 1 year following the original resection. A 2-year-old patient has been subsequently diagnosed with nephroblastomatosis and Wilms tumor of the kidney. Follow-up information was available in an additional 3 patients with no tumor recurrence or metastatic disease at 2, 3, and 14 years.

Adolescent↗

PubMed1996

Expression in human lung cancer cell lines of genes of prohormone processing and the neuroendocrine phenotype.

Lung tumor cells and cell lines, principally the histologically classified small cell lung cancer, are characterized by the expression of neuroendocrine (NE) features including AADC (aromatic amino acid decarboxylase, previously called DOPA decarboxylase) and the production of many peptide hormones. The general mechanisms by which most aspects of the NE phenotype affect the clinical behavior of lung tumor cells are unknown, but it is well recognized that peptide hormones can have systemic effects (paraneoplastic syndromes) and several have been shown to be autocrine growth factors for cancer cells. In order to determine the relationship between expression of different aspects of the NE phenotype in lung cancer cell lines, we have compared expression of a gene required for biosynthesis of some active peptide hormones (PAM, peptidylglycine alpha-amidating monooxygenase) to the gene for AADC in 32 lung cancer cell lines. Expression of these genes was quantified by both steady state Northern blot analysis and radiochemical enzymatic activity measurements. To ensure a range of expression of NE markers, non-small cell lung cancer (NSCLC) cell lines were chosen to include several which had previously been shown to express NE markers, and several small cell lung cancer (SCLC) cell lines with previous low levels of AADC were included. PAM enzyme activity and Northern blot analysis showed a two to three log variation in levels of expression in both the small cell and non-small cell lines. A smaller range was found for AADC expression. Using the highly sensitive PAM enzyme assays, all cell lines were found to express detectable PAM. PAM activities were secreted into the growth medium of all cell lines. There was no simple correlation apparent between AADC and PAM gene expression in the lung cancer cell lines. However, classic small cell lines demonstrated high levels of expression of both PAM and AADC genes, as did the carcinoid subset of the NSCLC lines. NSCLC lines expressed levels of PAM mRNA and enzyme activities equivalent to those of SCLC but had infrequent expression of AADC (principally only carcinoid NSCLC expressed AADC). These data demonstrate that separate aspects of the NE phenotype can be differentially expressed in lung cancer histological sub-types. Expression of PAM enzymes in all sub-types of lung cancer suggests that peptide prohormone activation may be a common mechanism for autocrine growth stimulation even in non-Ne NSCLC cell lines, or may reflect maintenance in cell lines of a common pathway of lung tumor promotion.

Biomarkers, Tumor↗

PubMed2006

Classification of malignant pediatric renal tumors by gene expression.

BACKGROUND: The most common malignant renal tumors of childhood are Wilms tumor (WT), clear cell sarcoma of the kidney (CCSK), cellular mesoblastic nephroma (CMN), and rhabdoid tumor of the kidney (RTK). Because these tumors present significant diagnostic difficulties, the goal was to define diagnostically useful signatures based on gene expression. PROCEDURES: Gene expression analysis using oligonucleotide arrays was performed on a training set of 47 tumors (10 CCSKs, 9 CMNs, 8 RTKs, and 20 WTs). Classifiers were developed for each tumor type using variations of compound covariate class predictor. The classifiers were applied to an independent test set of 72 tumors (3 CMN, 7 CCSK, 4 RTK, and 58 WT). Central review diagnosis was utilized as the gold standard. Correlation with the institutional diagnosis and qualitative estimation of confidence levels at the time of central review were noted. RESULTS: Within the training set, classifiers resulted in no errors when >10 genes were utilized. Top genes in each classifier were verified using quantitative reverse transcription-polymerase chain reaction (RT-PCR). Applying the classifiers to the test set, 71 of 72 tumors were correctly classified with a confidence level of >99%. The exception was incorrectly classified by the gold standard. In comparison, by histopathology 31% of the non-WT were not accurately classified by the local institution, and 29% were classified with <95% confidence on central review. CONCLUSIONS: Classifiers based on gene expression provide diagnostic confidence and accuracy greater than that of pathologic analysis alone. Tumors that show ambiguous gene expression profiles are those that are also pathologically and molecularly ambiguous and merit further analysis.

Confidence Intervals↗

PubMed2007

Desmoplastic small round cell tumor in childhood: the St. Jude Children's Research Hospital experience.

BACKGROUND: Desmoplastic small round cell tumor (DSRCT) is a rare, primarily intra-abdominal tumor that has a poor outcome with current therapies. PROCEDURE: We retrospectively reviewed patient characteristics, presenting symptoms, tumor pathology, treatment, and outcome of 11 pediatric patients with DSRCT at our institution. RESULTS: The cohort included 1 female and 10 male patients. Median age at diagnosis was 14 years (range 5-21 years). In eight (73%) patients, the primary tumor was abdominal or pelvic, and in one patient each, it was submental, mediastinal, and paratesticular. Nine (82%) patients had metastatic disease. All tumors showed polyphenotypic differentiation by immunohistochemistry. The EWS-WT1 transcript was detected in six of seven tumors tested. One tumor showed rhabdomyoblastic differentiation after therapy. All patients received chemotherapy; eight underwent surgical resection, seven received primary site radiation, and four received myeloablative chemotherapy with stem-cell support. Three (27%) patients are alive 23 months, 8 years, and 10 years from diagnosis. Two died of treatment-related toxicity, six died of disease. None of the patients in whom surgery and initial chemotherapy failed to induce complete remission survived. CONCLUSIONS: DSRCT is an aggressive malignancy that does not respond well to contemporary treatments, and patients who do not enter complete remission after initial chemotherapy and surgery appear to have a particularly dismal outcome. Patients with localized extra-abdominal disease have a better prognosis, most likely due to increased feasibility of resection. Better understanding of molecular and genetic mechanisms of tumorigenesis and treatment-related changes may contribute to development of more effective therapy for DSRCT.

Abdominal Neoplasms↗

PubMed2004

[Malignant gastrointestinal stromal tumor (GIST) of the papilla vateri. A rare tumor entity].

We present the case report of a 68-year-old female patient who had a malignant gastrointestinal stromal tumor of the papilla of Vater. The abdominal CT showed a coin-shaped lesion in the liver (segment VIII). Intraoperative rapid histological examination detected an old parasitic hepatic cyst. To exclude metastasis, we performed duodenopancreatectomy with curative intention. The postoperative course was without complications and neither chemotherapy nor other adjunct treatment was necessary. Because of heterogeneity, the different localization, and in this case the rare localization, surgery of gastrointestinal stromal tumors is difficult. However, we adhered to oncological and therapeutic standards of surgery for papillary carcinoma. This case is discussed based on a review of the literature. However, until now there has been no case report of gastrointestinal stromal tumor of the papilla of Vater in the literature.

Aged↗

PubMed2004

Pulmonary metastasectomy for pediatric solid tumors.

Most pediatric thoracic malignancy is pulmonary disease secondary to solid tumors of childhood. The management of isolated pulmonary metastases in adulthood is well documented. Little has been published to document the long-term outcome of pulmonary metastasectomy in childhood. A retrospective study was undertaken to assess the results of surgery for isolated pulmonary metastases. Twenty children underwent surgery over 12 years (mean follow-up 8 years). Five had Wilms' tumor (mean age 51 months), eight had osteogenic sarcoma (mean age 141 months), three had rhabdomyosarcoma (mean age 92 months), two had hepatoblastoma (mean age 30 months) and two had teratoma (mean age 72 months). Four had bilateral synchronous metastases and thoracotomies, and one had bilateral metachronous metastases and thoracotomies. Nineteen children were discharged well within 10 days of surgery. There was one early complication: a death due to pneumonia. Four children subsequently died postoperatively with cranial metastases (mean 29 months postoperatively). The remaining 16 children remain alive and well. As part of the combined therapy, these results would support an aggressive surgical approach to this disease. Preoperative assessment should include contrast enhanced computed tomogram of the head and chest as well as chest X-ray taken immediately preoperatively to exclude metastases. Bilateral synchronous and metachronous thoracotomy is well tolerated in childhood.

Child↗