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1/404. Surgically created fourth-third cranial nerve communication: temporary success in a child with bilateral third nerve hamartomas. Case report.

    Shortly after birth, an otherwise healthy infant developed eye deviation and ptosis due to a hamartomatous lesion of the interpeduncular segment of the right oculomotor nerve. The left nerve became similarly involved when the child was 1.5 years of age. Direct nerve repair was not possible. Instead, the trochlear nerve was divided and its proximal end was attached to the distal end of the third nerve. Elevation of the upper eyelid and partial adduction of the eye developed gradually over the ensuing 3 to 5 months. Both functions were lost after an additional 2 months, presumably as a result of tumor recurrence or neuroma formation. This case report shows that surgically created fourth-third cranial nerve communication is feasible and may merit consideration under similar circumstances.
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2/404. Diagnostic utility of Mullerian inhibiting substance determination in patients with primary and recurrent granulosa cell tumors.

    OBJECTIVES: In this study we evaluated changes in serum Mullerian inhibiting substance (MIS) concentration in a large number of patients with granulosa cell tumors (GCT) to determine whether MIS is elevated at the time of presentation and whether MIS is an index of successful surgical resection and management of recurrences. methods: We retrospectively reviewed MIS levels from 17 subjects prior to tumor resection and studied serial MIS samples from 56 subjects following initial tumor resection. Clinical follow-up information was available for 36 of those with postoperative MIS values. serum MIS was measured by an ELISA. MIS values were compared to a combination of normative values previously established in our laboratory and from more recently obtained samples from older pre- and postmenopausal women, using this assay. RESULTS: serum MIS was elevated pre-operatively in 6 of 8 (75%) subjects with juvenile GCTs and in 7 of 9 (78%) of those with adult GCTs relative to age-matched controls (76% for both types combined). Post-operative clinical correlation was available for 36 patients. There was no clinical recurrence in 21 subjects with normal or undetectable postoperative values, and incompletely resectable tumor or recurrence was identified in 6 of 15 patients with elevated postoperative values. CONCLSUIONS: The results of this study demonstrate that postoperative serum MIS concentrations may be used to evaluate the completeness of tumor removal following initial surgery and that serial MIS determinations may allow the detection of recurrences.
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3/404. Molecular assessment of clonality leads to the identification of a new germ line TP53 mutation associated with malignant cystosarcoma phyllodes and soft tissue sarcoma.

    Cystosarcoma phyllodes (CSP) is a rare breast neoplasm composed of stromal and epithelial elements. It usually runs a benign course but it may metastasize. In a 31-year-old patient with recurring CSP, a mesenchymal tumor in the leg developed. The question arose whether the latter tumor could be a metastasis from the CSP, which would have major treatment consequences. The problem was addressed using molecular methods, i.e., comparison of the pattern of polymorphic repeat markers on chromosome 17p as well as single strand conformation polymorphism analysis and sequencing of exons 5 to 8 of the TP53 gene in both tumor and normal tissue. An identical pattern of loss of heterozygosity in both breast tumors was demonstrated, but a different pattern was shown in the tumor in the leg. This led to the conclusion that the latter tumor had to be a new primary tumor. A mutation in codon 162 of the TP53 gene was found in the tumor tissue as well as in the normal tissue of this patient. This germ line mutation leads to the replacement of isoleucine by asparagine and most likely has functional consequences. In all four examined tumors of this patient, the normal TP53 allele was lost. This is strong evidence that this germ line TP53 mutation causes the genesis of these two rare primary mesenchymal tumors in this young patient. The current study exemplifies the power of molecular diagnostic methods in investigating the specific clinical problem of clonal relation between two separate tumors. The germ line mutation found in codon 162 of the TP53 gene and the association with cystosarcoma phyllodes have not been described previously.
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4/404. Transformation of ameloblastic fibroma to fibrosarcoma.

    The direct transformation of an ameloblastic fibroma into a fibrosarcoma in a 16-year-old Caucasian male is reported. Although no ameloblastic epithelium was found in the recurrent tumor, the odontogenic origin of the fibrosarcoma was evident. The ameloblastic fibrosarcoma and the fibrosarcoma of identical odontogenic origin represent an entity which should be distinguished from conventional fibrosarcoma as these tumors demonstrate different clinical behaviors.
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5/404. Primary cutaneous Ki-1(CD30) positive anaplastic large cell lymphoma in childhood.

    Primary cutaneous Ki-1(CD30) positive anaplastic large cell lymphoma (ALCL) is an unusual tumor in the pediatric population. However, the nodal-based form of the disease compared with other histologic subsets of childhood non-Hodgkin's lymphomas (NHL) more frequently involves skin, soft tissue, and bone. The objective of this article is to determine the histologic and immunologic characteristics of childhood primary cutaneous Ki-1(CD30) positive ALCL and its prognosis. The clinical data, histologic features and immunohistochemical profiles of skin biopsy specimens from 3 children with cutaneous Ki-1(CD30) positive lymphoma were reviewed. A literature search was performed and disclosed information on 5 childhood cases. The 3 patients with primary cutaneous Ki-1(CD30) positive ALCL all presented similarly as rapidly growing masses initially and clinically believed to be infectious/reactive processes. The diagnosis was established on the basis of histopathologic examination and immunohistochemical studies. Histologic sections revealed an extensive infiltrate of tumor cells extending throughout the entire dermis into the subcutaneous fat with frank ulceration in 1 patient. No significant epidermotropism was noted. Tumor cells exhibited striking cellular pleomorphism and a high mitotic rate with numerous atypical mitoses. Inflammatory cells were present in all patients. The tumor cells stained positively for Ki-1 antigen (CD30), epithelial membrane antigen, and for T-cell markers (UCHL-1, CD3). One of 3 cases, however, failed to stain for leukocyte common antigen (LCA). No clinically apparent adenopathy was observed in any of the patients. In all instances the patients developed recurrent disease in the skin at sites separate from the primary location. None of the patients demonstrated any involvement of lymph nodes, bone marrow, or other organ systems. All patients were treated with chemotherapy with good response. Primary cutaneous Ki-1(CD30) positive lymphoma is rare in children and is characterized by recurrences. The prognosis seems to be favorable.
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6/404. Transnasal endoscopic repair of congenital defects of the skull base in children.

    OBJECTIVE: To examine imaging findings and methods of endoscopic treatment of congenital skull base defects in children. DESIGN: Retrospective study and case series. SETTING: Academic tertiary care center. patients: Four patients (aged 12 and 14 months and 8 and 13 years) were included from 1995 to 1997. Three presented with a nasal glioma, which was recurrent in 1 case. The fourth patient presented with bacterial meningitis due to a spontaneous cerebrospinal fluid leak. Computed tomography and magnetic resonance imaging were used to locate the defect of the skull base. INTERVENTION: Transnasal endoscopic resection of the glioma or the meningocele, with immediate repair of the skull base defects using free mucosal flaps and/or pediculized mucosal flaps and/or conchal cartilage together with fibrin glue and nasal packing during a 3-week period. RESULTS: None of the 4 patients has experienced recurrent cerebrospinal fluid leaks or postoperative meningitis. CONCLUSIONS: The transnasal endoscopic repair of congenital meningoceles is a reliable technique in select pediatric patients. Computed tomography and magnetic resonance imaging provide information that can be used to help the surgical procedure.
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7/404. Malignant glial tumor arising from the site of a previous hamartoma/ganglioglioma: coincidence or malignant transformation?

    Gangliogliomas are generally considered benign tumors. Although more commonly found in the brain, spinal cord ganglioglioma is a well established, albeit infrequent, entity. We describe a 2-decade clinical course of a patient initially diagnosed with a thoracolumbar 'glial-neuronal hamartoma' at age 4. Seventeen years after his first operation, local recurrence was noted. Despite subsequent multiple gross total resections and adjuvant therapy, histologic features became increasingly ominous and ultimately proved fatal. This is an unusual report and histologic presentation of a resected spinal cord ganglioglioma recurring as an anaplastic ependymoma/astrocytoma and subsequently a glioblastoma. It is quite likely that the originally resected ganglioglioma was actually part of a primitive neuroectodermal tumor which had undergone extensive maturation.
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8/404. Histologic transformation of polymorphous low-grade adenocarcinoma of salivary gland.

    Polymorphous low-grade adenocarcinoma of salivary gland origin (PLGA) was initially described in 1984 and has since become an established clinicopathologic entity. Owing to the indolent nature of PLGA and its relatively recent description, the full clinicopathologic spectrum of this entity has not been elucidated fully. Transformation to a histologically different-appearing lesion or progression to a higher histologic grade has not been reported. We describe 2 PLGAs arising in the palate and associated with multiple locoregional recurrences that were treated with excision and radiation therapy. This was followed by histologic transformation to a higher grade neoplasm after 17 and 26 years, respectively. The histologic appearance after transformation was characterized by a predominantly solid and cystic growth pattern, nuclear atypia with prominent nucleoli, and foci of necrosis. High-grade transformation of PLGA may occur after a protracted clinical course with multiple recurrences of typical PLGA. The possible role of radiation therapy as an initiator of this transformation merits further study. Tumors with these histologic features should not be included under the rubric of typical PLGA. Segregation of these neoplasms will allow further study of their biologic potential, particularly with regard to possible increased rates of local recurrence and metastasis.
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9/404. A case of incomplete Currarino triad with malignant transformation.

    A case of an incomplete Currarino triad is reported. The baby underwent an emergency laparotomy due to a life-threatening intestinal obstruction caused by severe rectal stenosis. During the posterosagittal anorectoplasty (PSARP), a presacral teratoma was identified and resected. The tumor recurred three times; she initially responded to chemotherapy, but nonetheless died at the age of 4 years. In cases with evidence of anorectal stenosis, a presacral mass should be suspected. PSARP is the best choice of treatment for both the anorectal anomaly and excision of the presacral mass. The presacral region should be followed up closely for recurrence of the tumor.
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10/404. Odontogenic ghost cell carcinoma: report of four new cases and review of the literature.

    Only 12 odontogenic ghost cell carcinomas (OGCC) have been reported in the English language literature to date. This article reports four additional cases of this rare odontogenic tumour and examines them in relation to those previously described. Judging from the number of published cases, the OGCC is more prevalent in Asians than other racial groups, occurs more often in the maxilla than the mandible, and is slightly more common in males than females. Histologically, elements of a benign calcifying odontogenic cyst (COC) can be identified in all the malignant variants, either separated or admixed with the malignant epithelial component. The latter can consist of either small basaloid cells or large epithelial cells. Despite the differing histological presentations, the biological behavior of the tumour is unpredictable, with some cases characterized by relatively indolent growth and others by a locally aggressive and potentially fatal course. The tumour apparently arises most often from malignant transformation of a preexisting benign COC, although it may also develop from other odontogenic tumours.
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