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PubMed2006

Alveolar soft-part sarcoma of the retroperitoneum.

A 33-year-old woman was referred to Iida Municipal Hospital because of left back pain. Computed tomography showed a tumor (17 x 11 x 10 cm) in the left retroperitoneal space. T1- and T2-weighted magnetic resonance imaging showed an inhomogeneous mass with marginal blood vessels. The tumor was resected via lumbar oblique incision with the thoraco-abdominal approach. The tumor weighed 1800 g and consisted of nests of 5-100 large, loosely arranged, polygonal cells, surrounded by capillaries, resembling alveoli. The tumor cells were rich in cytoplasm, containing periodic acid-Schiff- and diastase-positive granules and typical crystals. The pathological diagnosis was alveolar soft-part sarcoma (ASPS). Alveolar soft-part sarcoma is a rare soft-tissue tumor that accounts for approximately 0.5-1% of soft-tissue sarcomas. Such tumors originating in the retroperitoneal space are extremely rare. Herein is reported a case of ASPS of the retroperitoneum with radiological and pathological findings.

Adult↗
PubMed1983

Alveolar soft-part sarcoma. A review on its histogenesis and further studies based on electron microscopy, immunohistochemistry, and biochemistry.

To date, the histogenesis of alveolar soft part sarcoma has been considered to be of paraganglioma origin, striated muscle cell origin, or as a malignant granular cell myoblastoma, neural neoplasm, or renin-producing tumor. Further studies for these existing theories were performed based on various methods. The negative formaldehyde-induced fluorescence and the immunohistochemical absence of neuron-specific enolase were against the paraganglioma theory. The immunohistochemical absence of myelin proteins (P2 protein and PO protein) and S-100 protein were against the malignant granular cell myoblastoma and neural neoplasm theory. Furthermore, there was a totally negative immunohistochemical finding for renin in the tumor cells, and the biochemical relationship of the tumor and renin was completely negated. The contradiction in a well-known report that the components of crystals were considered to be Z-band materials such as tropomyosin was referred to based on recent myological data. Concurrently, the absence of tropomyosin was immunohistochemically demonstrated. Hence, the issues on the histogenesis of alveolar soft part sarcoma and the identity of the characteristic crystalloids remain open for discussion.

Adult↗
PubMed1998

Alveolar soft-part sarcoma: further evidence by FISH for the involvement of chromosome band 17q25.

A cytogenetic study of an alveolar soft-part sarcoma, a rare tumor of probably myogenic origin, demonstrated a t(X;17)(p11;q25) as the sole chromosomal abnormality. Dual- and triple-color fluorescence in situ hybridization, performed on metaphase and interphase cells, confirmed the translocation between chromosomes X and 17 and demonstrated that this translocation resulted in loss of 17q25. Involvement of 17q25 has been described in four previously published cases of alveolar soft-part sarcoma, but without further characterization. Compared to our karyotype, it seems that the derivative chromosome 17 observed in the reported cases could also be the result of a t(X;17) with possible loss of the 17q25 band. If so, a 17q25 deletion and/or chromosome rearrangement between Xp and 17q leading either to a gene fusion or gene disruption could play an important role in the pathogenesis of alveolar soft-part sarcoma.

Child↗
PubMed2000

Alveolar soft-part sarcoma of the cheek: report of a case with a review of the literature.

An alveolar soft-part sarcoma (ASPS) occurring in the cheek is described, with a review of the literature. The subject was a 25-year-old woman who presented with a large swelling in the left cheek. The lesion, initially diagnosed as a benign tumor on radiographic and computed tomographic examinations, was surgically excised. Histopathological examination of the mass revealed a nest-like or organoid cellular arrangement as confirmed by three-dimensional computer graphic reconstruction. The cytoplasm of the large polygonal tumor cells contained abundant diastase-resistant, PAS-positive inclusions, ultrastructurally confirmed to be crystalloid materials. The tumor cells were immunoreactive for anti-myoglobin, sarcomeric actin, and neuron-specific enolase antibodies, suggesting a myogenic origin.

Adult↗
PubMed2004

Alveolar soft-part sarcoma presenting with multiple intracranial metastases.

A 28-year-old man presented with history of raised intracranial pressure and one episode of generalized tonic clonic seizures. Computed Tomogram revealed multiple contrast enhancing intracranial lesions. Biopsy of one of the lesions was reported as metastatic alveolar soft part sarcoma. He was advised whole brain radiotherapy.

Adult↗
PubMed1999

Chromosome rearrangement at 17q25 and xp11.2 in alveolar soft-part sarcoma: A case report and review of the literature.

BACKGROUND: Despite the characteristic histopathologic appearance of alveolar soft-part sarcoma (ASPS), its histogenesis remains unclear, and cytogenetic analysis of ASPS is limited to eight cases so far because of the extreme rarity of this disease. METHODS: The authors document a cytogenetic study of a primary case of ASPS in which a modern spectral karyotyping technique was used. RESULTS: A standard cytogenetic analysis of the primary tumor cells with G-banding revealed 46,XY, add(17)(q25) in 23 of 25 metaphases analyzed. This structural rearrangement of chromosome 17, involving band q25, was also present in 5 of 8 ASPS cases in the literature. Moreover, with the spectral karyotyping technique, the additional part of the long arm of chromosome 17 in the current case was found to originate from chromosome X, resulting in a final tumor karyotype of 46, XY, add(17)(q25).ish der(17)t(X;17) (p11.2;q25)(wcpX+). CONCLUSIONS: This case report documents a clonal chromosome abnormality of der(17)t(X;17)(p11.2;q25) in ASPS. The results of the current study indicate that further molecular analyses focused on 17q25 and Xp11.2 are of interest and could help to elucidate the pathogenesis of ASPS.

Child↗
PubMedDate not supplied

Alveolar soft-part sarcoma of the head and neck: clinical and imaging features in five cases.

BACKGROUND AND PURPOSE: Alveolar soft-part sarcoma (ASPS) of the head and neck is an extremely rare malignancy. Although the clinical and imaging features of this tumor have been reported, a periodic review of unusual tumors is useful. The purpose of this study was to describe the clinical and imaging features of ASPS of the head and neck. METHODS: Between January 1990 and May 2004 at our institution, five head and neck ASPS were diagnosed in five patients (two male and three female patients; age range, 4-22 years). Clinical and imaging findings were reviewed retrospectively. Imaging studies consisted of contrast material-enhanced CT (in four patients), MR imaging (in four patients), and digital subtraction angiography (in two patients). RESULTS: The locations of the tumor were tongue in two cases, larynx in one case, buccal space in one case, and paravertebral space in one case. This tumor appeared as a large lobulating-contoured mass with high signal intensity and flow voids on T2-weighted images and showed strong enhancement on contrast-enhanced CT and MR images. Preoperative angiography showed high vascularity. Wide surgical excisions were performed in four cases. Mean follow-up periods were 16 months (range, 6-30 months), and no recurrence was noted except for the laryngeal case. CONCLUSION: ASPS should be included in the differential diagnosis of head and neck tumor when a slow-growing, large mass with high signal intensity and flow voids on T2-weighted images and strong enhancement on contrast-enhanced CT or MR image is seen, particularly in young female patients.

Adolescent↗
PubMed1995

Alveolar soft-part sarcoma of the female genital tract: a report of nine cases and review of the literature.

Nine alveolar soft-part sarcomas of the female genital tract (four previously reported) occurred in patients 14-38 (mean 29) years of age. The most common clinical presentation was abnormal uterine bleeding. Two tumors were located in the vagina, three in the cervix or lower uterine segment, three in the uterine corpus, and one in the broad ligament. The tumors were solid and well circumscribed, ranging in maximum dimension from 1 to 9.8 (average 3.6) cm and had the characteristic histologic features of alveolar soft-part sarcoma. Granules that were diastase resistant and positive by periodic acid-Schiff (PAS) were present in all the tumors, and PAS-positive, diastase-resistant crystals were present in eight of them. Follow-up information ranging from 9 months to 17 years (average 62 months) was available for all the patients. One patient died of metastatic disease 25 months after diagnosis; eight other patients had uneventful follow-up periods ranging from 9 months to 17 years (average 67 months). The differential diagnosis of these tumors is discussed and the literature reviewed.

Adolescent↗
PubMed1990

Alveolar soft-part sarcoma of the tongue. Report of a case.

A case of alveolar soft-part sarcoma (ASPS) of the tongue in a 19-year-old boy is presented. He underwent a hemiglossectomy and received chemotherapy and has been free of disease for 3 years. The origin of intracytoplasmic periodic acid-Shiff(PAS)-positive crystals found in the tumor and the histogenesis of ASPS are briefly discussed.

Adult↗
PubMed2002

Metastatic alveolar soft-part sarcoma of the intracranial skull base: case report.

A patient with an intracranial skull base alveolar soft-part sarcoma, thought on preoperative imaging to be a meningioma, is presented. The mass was removed and identified by pathological evaluation to be an alveolar soft-part sarcoma. Postoperative investigation revealed widespread systemic disease. The histologic and clinical characteristics of this unusual tumor and the implications for prognosis and treatment are discussed.

Case Reports↗
PubMed2005

Oral alveolar soft-part sarcoma: review of literature and case report with immunohistochemistry study for prognostic markers.

OBJECTIVE: To describe a case of alveolar soft-part sarcoma (ASPS) affecting the tongue of a child and to study prognostic imunohistochemical markers for the disease. STUDY DESIGN: Tissue sections were incubated with primary antibodies reactive to neuron-specific enolase (NSE), vimentin, desmin, S-100 protein, cytokeratins AE1-AE3, EMA, neurofilament, synaptophysin, and muscle-specific actin (MSA), and for prognostic markers, including Ki-67, p53, bcl-2, bax, and nm23. RESULTS: Histologically, the tumor showed a proliferation of large polygonal cells with PAS-positive diastase-resistant intracytoplasmatic material, arranged in an alveolar growth pattern. Diffuse positive reaction for neuron specific enolase (NSE), focal reactivity for desmin and S-100 protein, strong positivity for nm23 and bax, but weak reaction for p53 and Ki-67 were found. No bcl-2-positive cells were noted. CONCLUSION: These immunohistochemical findings may reflect the less aggressive behavior of ASPS in oral tissues.

Actins↗
PubMed1989

Orbital nonchromaffin paraganglioma. A case report and review of the literature.

Nonchromaffin paraganglioma (NCP), also called glomus body tumor or chemodectoma, is rarely found in the orbit. The behavior of orbital nonchromaffin paraganglioma may potentially be more aggressive than in other head and neck locations. Diagnosis depends on electron microscopic demonstration of membrane-bound neurosecretory granules. Results of histopathologic study show a well-circumscribed lesion without a true capsule with alveolar or organoid arrangements of epithelioid cells within a reticulin framework with thin-walled blood vessels. Cells are polygonal with round or oval nuclei containing rare mitotic figures and pale-staining cytoplasm. Differential diagnosis includes alveolar soft-part sarcoma, alveolar rhabdomyosarcoma, neuroblastoma, carcinoid, and granular cell tumor. Of 29 previously reported cases of orbital NCP, 16 have been reclassified as alveolar soft-part sarcoma. The authors report a patient with an electron microscopically established orbital NCP, with the history of a contralateral glomus jugulare tumor irradiated 14 years previously.

Adult↗