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PubMed2001

Synovial sarcoma in children: surgical lessons from a single institution and review of the literature.

BACKGROUND: Synovial sarcomas are malignant high-grade, soft-tissue neoplasms that account for 7% to 8% of all malignant soft-tissue tumors and are the most common nonrhabdomyosarcoma soft-tissue sarcomas in pediatric patients. STUDY DESIGN: A retrospective review of the records of children younger than 17 years with synovial sarcoma treated at the University of Texas MD Anderson Cancer Center from 1966 until 1999 was undertaken. Primary site, tumor size, tumor margins, surgical treatment, adjuvant therapy, local and distant recurrence, and survival were recorded for 42 patients. Overall survival (OS) and progression-free survival (PFS) rates were calculated by the Kaplan-Meier method. The PFS and OS comparisons were performed using the log-rank test. RESULTS: Forty-four patients were identified, but two patients were excluded because of incomplete records. The median followup duration for the 42 patients was 8.8 years (range 0.2 to 22.4 years). The 5-year progression-free survival and overall survival rates were 75.6% and 87.7%, respectively. Eleven patients were dead and four others had progressed but were alive without evidence of disease after further therapy. Intergroup Rhabdomyosarcoma Study (IRS) grouping and tumor invasiveness were found to be significant prognostic indicators (p < 0.01 and p = 0.02, respectively). Patients with initial gross total resection (IRS Groups I and II) and noninvasive tumors (T1) were most likely to have prolonged PFS and OS. Patients with small tumors (<5 cm) (p = 0.09) had better PFS and OS. Adjuvant radiation therapy appeared to be of benefit, and chemotherapy did not seem to impact PFS or OS. Tumors > or = 5 cm are associated with increased risk of local recurrence and distant metastases. CONCLUSIONS: Complete resection with clear, yet not necessarily large, margins remains the treatment of choice for synovial sarcoma in children. Adjuvant radiation therapy should possibly be considered in patients with clear margins (IRS Group I) and in patients with microscopic residual tumor (IRS Group II). Chemotherapy did not seem to impact PFS or OS. Lymph nodes should be evaluated for local regional disease.

Adolescent↗

PubMed1995

Post-radiation primary intranodal leiomyosarcoma.

An unusual case of post-radiation primary intranodal leiomyosarcoma of the cervical lymph node is presented. The patient was a 32-year-old white man who was treated in 1986 with hemiglossectomy and right neck dissection, followed by radiation therapy, for squamous cell carcinoma of the tongue. Six years later, he presented with an enlarged left cervical lymph node which on biopsy showed a high grade spindle cell malignant neoplasm. Immunohistochemistry and electron microscopic study verified the smooth muscle origin of the tumour. Extensive work-up for a second primary lesion was negative. Primary intranodal leiomyosarcoma is extremely rare. We briefly discuss the histological differential diagnosis of spindle cell lesions of lymph nodes, leiomyosarcoma of the vessel wall and radiation-associated sarcoma.

Adult↗

PubMed2004

Immunohistochemical detection of ZAP-70 in 341 cases of non-Hodgkin and Hodgkin lymphoma.

Using immunohistochemical methods, we evaluated zeta-associated protein (ZAP)-70 expression in 341 cases of non-Hodgkin and Hodgkin lymphoma. In B-cell NHL, ZAP-70 was positive in five of six (83%) precursor B-lymphoblastic lymphoma, 11 of 37 (30%) chronic lymphocytic leukemia/small lymphocytic lymphoma (CLL/SLL), five of 39 (13%) mantle cell lymphoma, one of 12 (8%) Burkitt lymphoma, and one of 12 (8%) nodal marginal zone B-cell lymphoma. In 22 cases of CLL/SLL, seven of nine (78%) with unmutated IgVH genes expressed ZAP-70, compared with one of 13 (8%) with mutated IgVH genes (P=0.0015 Fisher's exact test). ZAP-70 expression was not detected in diffuse large B-cell lymphoma (n=26), extranodal marginal zone B-cell lymphoma of mucosa-associated lymphoid tissue (n=24), follicular lymphoma (n=21), plasma cell myeloma/plasmacytoma (n=10), lymphoplasmacytic lymphoma (n=10), or splenic marginal zone lymphoma (n=6). In T/NK-cell NHL, ZAP-70 was positive in all extranodal natural killer (NK) / T-cell lymphoma, nasal-type (n=6) and enteropathy-type T-cell lymphoma (n=4), four of five (80%) subcutaneous panniculitis-like T-cell lymphoma, six of eight (75%) mycosis fungoides, three of five (60%) precursor T-lymphoblastic lymphoma, 10 of 17 (59%) peripheral T-cell lymphoma, two of four (50%) blastic NK-cell lymphoma, one of three (33%) T-cell prolymphocytic leukemia, 13 of 52 (25%) anaplastic large cell lymphoma, and one of six (17%) angioimmunoblastic T-cell lymphoma. Seven of 12 (58%) cutaneous CD30-positive lymphoproliferative disorders were also ZAP-70-positive. In Hodgkin lymphoma, ZAP-70 was negative in neoplastic cells in all cases tested. ZAP-70 staining in B-cell lymphomas and reactive T cells was predominantly nuclear with variable cytoplasmic staining. By contrast, ZAP-70 staining in T/NK-cell lymphomas was heterogeneous, and a shift from predominantly nuclear to predominantly cytoplasmic staining was observed, particularly in those neoplasms with high-grade morphology. In summary, ZAP-70 is expressed by many lymphoma types, correlates with immunoglobulin heavy-chain variable region gene mutational status in CLL/SLL, and can be detected reliably using immunohistochemical methods.

Blotting, Western↗

PubMed2000

Low expression of p27 and low proliferation index do not correlate in hairy cell leukaemia.

The molecular basis accounting for the peculiar clinical and biological features of hairy cell leukaemia (HCL) is currently unknown. Deregulation of cell cycle genes plays a significant role in oncogenesis and there is considerable evidence suggesting that Cdk inhibitors (Ckis) function as tumour suppressors. We and others have recently demonstrated low expression of Cki p27 in very aggressive neoplasms and high-grade lymphomas. To investigate whether HCL cases express normal p27 protein, as in other low-grade lymphomas with a low proliferation index, 58 cases of HCL were characterized using a sensitive biotin-streptavidin-immunoperoxidase technique and specific antibodies against p27. All HCL cases showed either no or very weak reactivity, in contrast to other types of low-grade B-cell lymphoma [22 cases of chronic lymphocytic leukaemia (CLL), 12 cases of gastric marginal B-cell lymphoma (MALT), 16 cases of follicular lymphomas and two cases of splenic marginal zone lymphomas]. To investigate the possible mechanism(s) accounting for the low p27 expression observed in hairy cells, multiple approaches were used. According to these molecular studies, low levels of p2 7 are not as a result of (1) increased ubiquitin-mediated degradation, (2) decreased levels of p27 transcription or (3) p27 somatic mutations and/or allelic loss. These findings suggest that low p27 protein expression in HCL may be achieved through post-transcriptional regulation. Finally, our data demonstrate that p27 expression in HCL does not correlate with either cell cycle progression or proliferation index, suggesting that low levels of p27 in hairy cells may be associated with their unique stage of B-cell differentiation and/or the activation of as yet unknown pathways.

Blotting, Western↗

PubMed2001

Localized amyloidosis and extramedullary plasmacytoma involving the larynx of a child.

Extramedullary plasmacytoma of the larynx and localized laryngeal amyloidosis are 2 uncommon disease entities that are exceedingly rare in children. We report a case of a 12-year-old girl presenting with progressive hoarseness who was subsequently found to have extramedullary plasmacytoma coexisting with localized amyloidosis involving the larynx. Results from the immunohistochemical and molecular studies showed that the tumor cells of the plasmacytoma were monoclonal (kappa-restricted), strongly supporting their neoplastic nature. The biochemical nature of the amyloid deposits was also shown to be of kappa immunoglobulin light chain, suggesting the pathogenetic relationship between the plasmacytoma and amyloid deposition in the larynx of this patient. There was no other evidence of malignancy or amyloidosis elsewhere. On a follow-up period of 4 years, this patient was well and asymptomatic. We believe that this represents the first case in the literature showing the coexistence of extramedullary plasmacytoma and localized amyloidosis of the larynx in children. This case also supports the concept that localized laryngeal amyloidosis may be a manifestation of low-grade B-cell neoplasms.

Amyloidosis↗

PubMed1990

Lasting remissions in hairy-cell leukemia induced by a single infusion of 2-chlorodeoxyadenosine.

2-Chlorodeoxyadenosine is a simple purine nucleoside that has previously been shown to be effective in the treatment of low-grade malignant disorders of lymphoid tissue, including chronic lymphocytic leukemia and non-Hodgkin's lymphoma. Because of these encouraging results, we treated 12 patients with another low-grade B-cell neoplasm, hairy-cell leukemia. The patients received 2-chlorodeoxyadenosine (0.1 mg per kilogram of body weight per day) by continuous infusion for seven days. All the patients responded to treatment. Eleven had complete remissions characterized by the normalization of peripheral blood and bone marrow and disappearance of tumor masses. The longest remission has been 3.8 years. None of the patients have relapsed, and the median duration of remission has been 15.5 months. No serious toxic reactions occurred as a result of 2-chlorodeoxyadenosine therapy. These results suggest that 2-chlorodeoxyadenosine may be the most effective therapy available for hairy-cell leukemia. The administration of 2-chlorodeoxyadenosine resulted in a higher rate of complete remission than is observed with interferon alfa, and it required no maintenance therapy. Its toxicity may be lower than that of deoxycoformycin, and the responses were achieved with single courses of treatment.

2-Chloroadenosine↗

PubMed1989

Intermediate filament expression in astrocytic neoplasms.

Immunohistochemical analysis of 30 paraffin-embedded astrocytic neoplasms was performed to correlate the expression of intermediate filament proteins with histologic subtype. Each tumor was studied with monoclonal antibodies to keratin, vimentin, desmin, 200-kd neurofilament protein, and glial fibrillary acidic protein (GFAP). Immunoreactivity with the anti-keratin monoclonal antibodies AE1 and AE3 was demonstrated in 24 cases (80%) including 4 of 6 (66%) well-differentiated astrocytomas (WDAs), 10 of 12 (83%) anaplastic astrocytomas (ANAs), and 10 of 12 (83%) glioblastomas multiforme (GBMs). These cases were further studied with the monoclonal antikeratin antibodies 34 beta E12 and 34 beta H11. Of the 24 AE1/AE3-positive cases, 14 (58%) reacted with 34 beta E12. None of the cases was reactive with 34 beta H11. Vimentin expression was demonstrated in 24 cases (80%), including 2 of 6 (33%) WDAs, 11 of 12 (92%) ANAs, and 11 of 12 (92%) GBMs. Coexpression of keratin and vimentin was observed in 20 cases (67%), including 2 of 6 WDAs, 9 of 12 (75%) ANAs, and 9 of 12 (75%) GBMs. Immunoreactivity with GFAP antibody was present in all 30 (100%) cases, but none of the tumors was reactive with antibodies to desmin or 200-kd neurofilament protein. These findings demonstrate that expression of both keratin and vimentin intermediate filaments is common in astrocytic neoplasms regardless of histologic grade.

Astrocytoma↗

PubMed2004

Serous carcinoma of the ovary and peritoneum with metastases to the breast and axillary lymph nodes: a potential pitfall.

Metastasis of ovarian or peritoneal serous carcinoma to the breast and/or axillary lymph nodes is a rare event. Nevertheless, its recognition and distinction from mammary carcinoma are of great clinical importance because the treatment and prognosis differ significantly. Eighteen cases of ovarian or peritoneal serous carcinoma metastatic to the breast and/or axillary LNs from a 14-year period (1990-2003) were retrieved from our files. Clinical information was obtained from the patients' charts. The age of the patients ranged from 21 to 67 years (median, 55 years). The primary tumors included 14 ovarian serous carcinomas (11 high grade and 3 low grade; 2 of the low-grade tumors presented as serous tumors of low malignant potential and recurred as low-grade serous carcinoma) and 4 peritoneal serous carcinomas (3 high grade and 1 low grade). Of the ovarian neoplasms, 1 was stage I and 10 were stage III tumors; the breast and/or axillary lymph node metastases were discovered on average 30 months after presentation (range, 7-135 months). Three of the ovarian serous carcinomas were stage IV tumors; in 1 case, there were axillary lymph node metastases at initial presentation; and in 2 cases, breast and/or axillary lymph node metastases developed at 18 and 102 months. Two of the 4 patients with peritoneal serous carcinoma presented with stage IV disease, having synchronous breast and axillary lymph node metastases; the other 2 patients developed them at 11 and 16 months after presentation. Four patients had multiple breast lesions and 8 patients had a single metastasis. In 4 cases, the breast metastases were initially interpreted as infiltrating ductal carcinoma. The remaining 6 patients had axillary lymph node involvement only. The metastases in 17 of the cases had papillary features, with psammoma bodies present in 4. Immunoperoxidase studies for GCDFP-15 and WT-1 were performed in 4 cases; all 4 were positive for WT-1 and negative for GCDFP-15. Follow-up was available for 17 patients, with 7 patients known to be dead from disease (survival range, 2-31 months) after the development of metastatic disease to the breast or axillary lymph nodes. Ten patients were alive with disease at their last follow-up, which ranged from 1 to 30 months after the breast or axillary LN metastasis developed. Metastases to the breast or axillary lymph nodes from ovarian and/or peritoneal serous carcinomas are uncommon. Most of the patients in whom metastatic disease develops have a known history of advanced stage ovarian or peritoneal carcinoma. Breast and/or axillary LN involvement at initial presentation can occur but is rare. Differentiation between metastatic and primary tumors of the breast is of great importance because treatment and prognosis differ significantly. Clinical history, the presence of papillary architecture, and WT-1 expression are useful in establishing the correct diagnosis.

Adult↗

PubMed2003

Minimal focus of adenocarcinoma on prostate biopsy: clinicopathological correlations.

AIMS: To establish the clinicopathological features of minimal volume prostate adenocarcinoma on prostate biopsy. METHODS: Twenty four cases of minimal adenocarcinoma diagnosed on prostate biopsy and treated by radical prostatectomy were reviewed. RESULTS: The major microscopic criteria were nuclear enlargement (22 of 24), infiltrative pattern (19 of 24), prominent nucleoli (19 of 24), intraluminal eosinophilic secretions (15 of 24), and high grade intraepithelial neoplasia associated (11 of 24). Sixteen of 24 cases were assigned a Gleason score 6 on biopsy. When the whole gland was assessed, 22 of these tumours were localised to the prostate (stage pT2), and only two cases were stage pT3. CONCLUSIONS: Minimal focus of adenocarcinoma on prostate biopsy is not an uncommon finding. It is usually an intermediate grade and localised stage neoplasm.

Adenocarcinoma↗

PubMed1987

CT computerised stereotactic biopsy for low density CT lesions presenting with epilepsy.

Thirty five patients presenting with epilepsy alone and non-enhancing low-density lesion on the CT scan underwent a computer-assisted CT-guided stereotactic biopsy with stereotactic angiographic control. There was no mortality or morbidity in this series and the diagnostic yield was 97%. Thirty four patients had low grade intra-axial neoplasms. After an estimation of the pathological extent of the tumour, three patients underwent a computer-assisted stereotactic laser resection and 28 patients had radiotherapy.

Adult↗

PubMedDate not supplied

Extranodal marginal zone B-cell lymphoma of malt type involving the cavernous sinus.

Mucosa-associated lymphoid tissue (MALT) lymphomas are low-grade B-cell neoplasms that occur in a variety of extranodal sites. Only rarely has this entity been discovered as a primary tumor involving the dura of the cavernous sinus. We report the case of a 46 year old woman who presented with a mass involving the cavernous sinus that was originally diagnosed as a meningioma by MRI. However, at surgery the mass was found to be an extranodal marginal zone B-cell lymphoma of the MALT type. The patient underwent partial excision of the lesion followed by radiation. There are only seven other cases of marginal zone B-cell lymphoma involving the intracranial dura reported in the literature. These patients were females who presented with intracranial lesions thought to be consistent with meningioma by preoperative radiographic imaging. MALT lymphomas of the dura, as their counterparts in other organs, appear to have favorable clinical outcomes and excellent long-term prognoses with local therapy alone.

Brain Neoplasms↗

PubMed1999

Microscopic mimicry of lymphomas: diagnostic pitfalls.

A common, critical challenge for the pathologist is the distinction between lymphoma and lymphoma simulators. Ancillary studies are effective in making this distinction and should be used in descending order of their likely reliability according to the particular differential diagnostic consideration at hand. Clinical case information is essential to avoid misinterpretation. Lymphoma simulators can be grouped into several major categories: nonlymphoid neoplasms, usually high-grade, simulating high-grade lymphomas; chronic lymphoid hyperplasias simulating low-grade lymphomas; and acute lymphoid hyperplasias simulating high-grade lymphomas.

Diagnosis, Differential↗

PubMedDate not supplied

[Effect of certain aspects of female sexual behavior on the development of cervical intraepithelial neoplasia].

The paper presents results of investigation of certain parameters of female sexual activity and their effect on the appearance of squamous intraepithelial cervical lesions (SIL). This correlation was investigated in the context of Human Papilloma Virus (HPV) infection, in view of the role attributed to certain types of these viruses in the appearance and development of cervical SIL. Research was done on 1000 patients suffering from low histological grade (LSIL) or high histological grade (HSIL) intraepithelial cervical neoplasms. It transpired that such lesions were most frequent in women in the second, third and fourth decade of life, and that one third of pathological lesions was a function of age. Human Papilloma Virus infection was detected in 65% of patients. The frequency of HPV negative results or presence of non-oncogenic types of viruses (6 and 11) was significantly higher in LSIL type lesions, while the frequency of detection of oncogenous HPV types (16, 18, 31 and 33) was significantly higher in more severe stadiums of the disease. Of the investigated parameters of sexual activity, early onset of such activity and several sexual partners proved to be significant. The number of parturitions and abortions did not appear to be significant risk factors for the onset and development of these diseases.

Adolescent↗

PubMed2001

Large-cell neuroendocrine carcinoma of the uterine cervix--a clinicopathological study of five cases.

OBJECTIVE: The present study describes 5 cases of large-cell neuroendocrine carcinoma (LCNEC) of the uterine cervix, evaluating their clinical features and pathological profiles. METHODS: Clinical data were obtained from the patients' clinical files at the combined gynaecological-oncology unit of Johannesburg Hospital and the University of the Witwatersrand Medical School, Johannesburg, South Africa. A histopathological diagnosis was obtained after biopsy material from all 5 patients was examined microscopically and subjected to immunohistochemical staining with MNF116 (pankeratin) synaptophysin and chromagranin A, all of which are neuroendocrine markers. Two patients received pelvic radiotherapy only. None of the 5 patients in this series received chemotherapy or underwent surgery. RESULTS: All 5 patients were adult females, with an average age of 57.3 years. The majority were multiparous, with the most common presenting complaint being vaginal bleeding. Three of the 5 patients presented with advanced-stage cervical carcinoma, with evidence of metastases in 2 of them. Treatment responses and long-term survival in our series proved to be disappointing as 3 of the 5 patients died in less than 6 months. On histopathological examination, all 5 tumours showed features of a high-grade poorly differentiated malignant neoplasm with ulceration and extensive tumour necrosis including trabecular and organoid growth patterns. All 5 neoplasms also showed strong immunoreactivity for MNF116, while their endocrine nature was confirmed by staining for synaptophysin in all cases. None of the tumours showed positive straining for chromagranin A. CONCLUSIONS: LCNECs are rare tumours and distinct from other neoplasms of the uterine cervix. The results of this study reaffirm the biologically aggressive nature of this uncommon tumour and its very unfavourable prognosis.

Aged↗

PubMed2004

Leiomyosarcoma of the urinary bladder: a case report.

A case of leiomyosarcoma of the urinary bladder occurring in a middle aged lady with immunohistochemical confirmation is presented for its rarity with emphasis on grading of smooth muscle neoplasms of the urinary bladder.

Diagnosis, Differential↗

PubMed2005

[Clear cell carcinoma of salivary gland: a clinicopathologic study of 10 cases].

OBJECTIVE: To analyze the clinicopathologic features of clear cell carcinoma of salivary gland. METHODS: All cases of salivary gland tumors enrolled from January 1985 to May 2004 were reviewed, and ten cases of CCC were identified and analyzed by clinicopathology and immunohistochemistry. RESULTS: Seven tumors were from palate, one was from base of tongue, one in retromolar region, and one in floor of mouth. The mean age of five men and five women was 55 years. Microscopically, the tumors were predominantly composed of trabeculae, cords, nests, solid sheets of clear cells and a few eosinophilic cells, and the two kinds of tumor cells were surrounded by hyalinized stroma. Immunohistochemically, EMA and CK8 were positive in all cases, CK18 in five cases, CKHMW in two cases. CK10/13, S-100, SMA and Calponin were negative in all cases. There was no evidence of recurrence of disease or metastasis in eight followed-up patients. CONCLUSIONS: CCC is a distinctive low grade malignant salivary gland neoplasm, arising mainly in the minor salivary glands, and it may originate from intercalated-duct reserve cells.

Adenocarcinoma, Clear Cell↗

PubMed2004

Solid pseudopapillary tumor of the pancreas in a young Malay woman.

Solid pseudopapillary tumors of the pancreas are very rare, low-grade malignant potentially curable neoplasms. Despite having non-specific symptomatology, they have typical features such as being more common in young women, and classically presenting as large abdominal masses. Accurate diagnosis is important because long-term survival hinges on complete resection of the tumor.

Adolescent↗

PubMed2006

Epithelial myoepitheial carcinoma of minor salivary gland--low grade malignant tumor presenting with nodal metastasis.

Epithelial myoepithelial carcinoma (EMC) is a rare low grade malignant salivary gland neoplasm that most commonly occurs in the parotid gland but can also arise in minor salivary glands. We report a case of primary epithelial myoepithelial carcinoma of minor salivary gland in a 25 year old women who presented with swelling left cheek of one year duration and bilateral submandibular lymphadenopathy. A mass causing erosion of mandible, thyroid cartilage and masseter muscle was identified on CT scan. This was excised and histological examination revealed a mixture of ductal structures consisting of inner dark cells and outer clear cells seen in solid sheets. Immunohistochemical analysis showed the clear cells to be weakly positive for S100 and smooth muscle actin (SMA) and ductal cells to be positive for cytokeratin (CK) and epithelial membrane antigen (EMA). The characteristic morphological and immunohistochemical features aided in the diagnosis of epithelial myoepithelial carcinoma.

Actins↗