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Pituicytoma: A Rare But Recurring Tumor [Meeting Abstract]

Punsoni, Michael; Tanpitukpongse, Teerath P.; Ortiz, A. Orlando; Holtzman, Robert N.; Drexler, Steven
ISSN: 0002-9173
CID: 3461532

Carbohydrate antigen 19-9 and carcinoembryonic antigen immunostaining in benign multicystic mesothelioma of the peritoneum [Case Report]

Holtzman RN; Heymann AD; Bordone F; Marinoni G; Barillari P; Wahl SJ
A 58-year-old Italian man was incidentally discovered to have an elevated carbohydrate antigen 19-9 (CA-19-9) level of 132 U/mL on routine blood testing. Multisystem imaging studies revealed multiple benign-appearing cysts of the liver and single cysts in the pancreas and kidney parenchyma. Throughout 14 months, fluctuations were observed in the elevated serum CA-19-9 levels from 99 to 450 U/mL. Serum carcinoembryonic antigen (CEA) and other tumor markers were normal. Laparoscopy disclosed multiple cystic lesions on the surface of the liver, on the serosal surface of the ileum, and in the mesentery. Electron microscopy characterized the cells as mesothelial. The pathologic diagnosis was benign multicystic mesothelioma of the peritoneum (BMMP). Aspirated fluid from the liver cyst revealed CA-19-9 levels at 28 500 U/mL, strongly linking the elevated serum CA-19-9 levels with mesothelial cyst secretion. Immunostaining was positive for CA-19-9, CEA, and cancer antigen 125 (CA-125). We believe this is the first documented instance of CA-19-9 and CEA secretion in BMMP
PMID: 11419983
ISSN: 0003-9985
CID: 36460

Acute ventricular hemorrhage in adults with hydrocephalus managed by corpus callosotomy and fenestration of the septum pellucidum. Report of three cases [Case Report]

Holtzman, R N; Brust, J C; Ainyette, I G; Bowers, P P; Tikofsky, R S; Lliguin, H M; Hughes, J E
Three patients with hypertension-induced basal ganglia or thalamic hemorrhage and ventricular rupture underwent corpus callosotomy and fenestration of the septum pellucidum. A patient with a left thalamic hemorrhage underwent surgery on an emergency basis and made a complete physical recovery, although she retained mild psychomotor deficits. Another patient with a large right basal ganglia hemorrhage who also underwent surgery on an emergency basis retained a spastic left hemiparesis without evident psychomotor deficits. The third patient with a left thalamic and basal ganglia hemorrhage, who was initially awake and then lapsed into stupor days later, underwent surgery, but did not recover consciousness. Hydrocephalus was reversed and effectively controlled in all three patients without having to perform a shunt placement procedure.
PMID: 11453378
ISSN: 0022-3085
CID: 5035892

Pituicytoma: a distinctive low-grade glioma of the neurohypophysis [Case Report]

Brat, D J; Scheithauer, B W; Staugaitis, S M; Holtzman, R N; Morgello, S; Burger, P C
Pituicytoma is a rare, poorly characterized tumor of the sella and suprasellar region that is distinct morphologically from other local tumors and is thought to be derived from neurohypophyseal pituicytes. Clinical data, neuroimaging studies, and microsections were reviewed from nine such low-grade gliomas. Immunostains for glial, neuronal, and proliferation markers were performed on all nine tumors and six control neurohypophyses. Three tumors were studied ultrastructurally. Six men and three women, age 30 to 83 years (mean, 48 years), presented with visual symptoms, headache, or hypopituitarism. Magnetic resonance images showed solid, discrete, contrast-enhancing masses, four within the sella and five in the suprasellar space. The tumors consisted of sheets and/or fascicles of plump spindle cells with slightly fibrillar cytoplasm and slightly pleomorphic, oval-to-elongate nuclei with pinpoint nucleoli. Extracellular mucin was prominent in one tumor. Rosenthal fibers, granular bodies, and Herring bodies (granular axonal dilatations characteristic of the normal neurohypophysis) were lacking. Mitoses were rare or absent. MIB-1 labeling indices were low (0.5-2%). Tumor cells were strongly reactive for vimentin and S-100 protein, variably positive for glial fibrillary acidic protein, and nonreactive for synaptophysin and neurofilament protein. Cytoplasm varied in electron density and contained intermediate filaments. Neither meningothelial nor ependymal features were noted. Two tumors recurred at 20 and 26 months after subtotal resection, but none of the six completely resected tumors have done so. Pituicytomas are discrete, largely noninfiltrative low-grade gliomas of the sellar region that occur in adults. Their histologic appearance is distinct from pilocytic and ordinary, infiltrative astrocytomas. The distinction between pituicytoma and normal neurohypophysis is aided by the latter's content of axons, Herring bodies, and perivascular anucleate zones rich in axonal terminations. Although curable by total excision, subtotal resection can be associated with recurrence.
PMID: 10716149
ISSN: 0147-5185
CID: 5035882

Lobular capillary hemangioma of the cauda equina. Case report [Case Report]

Holtzman RN; Brisson PM; Pearl RE; Gruber ML
This 56-year-old woman presented with a 1-year history of low-back pain, sciatica, and paresthesias in the right S-1 dermatome. On examination the patient was shown to have a right-sided Lasegue's sign, normal strength, hypalgesia in the right S-1 dermatome, and a slight diminution of the right Achilles tendon reflex. Magnetic resonance imaging revealed a 2-cm intradural enhancing lesion at the level of the L-4 vertebra. Laminectomy of L3-L5 vertebrae was performed, and intradural exploration disclosed a blueberry-appearing tumor that was surrounded by an intense arachnoiditis and attached to the right S-1 nerve root. A cystic collection of cerebrospinal fluid was seen caudal to the tumor. Complete removal required transection of the adherent nerve root fascicles. Histological analyses indicate that the lesion was a lobular capillary hemangioma, which, to the authors' knowledge, appears to be one of the first recorded examples of such a case
PMID: 10199255
ISSN: 0022-3085
CID: 36035

Immunohistochemical detection of intracranial vasa vasorum: a human autopsy study

Connolly, E S; Huang, J; Goldman, J E; Holtzman, R N
The existence of intracranial vasa vasorum supplying the larger vessels of the circle of Willis has long been debated. Much of this debate results from contradictory findings of microanatomic studies in a variety of nonprimate species. Recently, however, a growing body of evidence seems to suggest that in certain pathological situations, such as human intracranial atherosclerosis, vasa vasorum are present. In an effort to determine whether intracranial vasa vasorum are present in humans without clinical evidence of intracranial vascular disease, we studied the circle of Willis in five autopsy specimens using immunohistochemistry. Antiserum to the endothelial-specific antigen, Factor VIII, revealed staining of 10- to 20-microns vascular channels in the outer media and adventitial layers. Staining was present in the proximal carotid, middle cerebral (M1), and anterior cerebral (A1) arteries but could not be detected in M2 or A2 segments. Hematoxylin and eosin staining was additionally helpful in identifying the nuclear morphology of the endothelial cells lining these channels, as well as the presence of erythrocytes within them. We conclude that in nonpathological settings, endothelial-lined channels exist in the proximal intracranial vessels of humans. These vessels might represent intracranial vasa vasorum, which in turn might play a role in pathological conditions, such as atheroma formation, intracranial dissection, and vasospasm.
PMID: 8692400
ISSN: 0148-396x
CID: 5036012

Nondural-based lumbar clear cell meningioma. Case report [Case Report]

Holtzman, R N; Jormark, S C
This 32-year-old man had noticed right leg pain for 4 years and developed classic right sciatica after heavy lifting, followed by episodes of buckling of both legs 1 month prior to admission. His medical history included congenital left abducens palsy. Examination revealed a right Lasègue's sign and Fajersztajn's sign with mild weakness of the right extensor hallucis longus. Magnetic resonance imaging revealed a 1.5 x 2.0-cm enhancing intradural lesion at the L3-4 level. Following laminectomy of L-3 and L-4 and intradural exposure, the tumor was found to be draped loosely by the roots of the cauda equina and attached to a single root without any adherence to dura. Transection of the adherent fascicles and typical microdissection of arachnoidal filaments permitted its complete removal without violation of the capsule, allowing the preservation of a large fascicle. The patient's recovery was uneventful. Postoperatively, a mild right lateral foot hypalgesia and diminution of the right ankle jerk implicated the S-1 root. Histological and immunohistochemical analyses diagnosed the specimen as a clear cell meningioma.
PMID: 8592230
ISSN: 0022-3085
CID: 5036002

Synovial sarcoma of the suboccipital region of the neck [Case Report]

Lombardi, L J; Cleri, D J; Horten, B C; LaMarca, C A; Holtzman, R N
A synovial sarcoma (SS) is an uncommon malignant soft-tissue tumor, which in spite of its name does not arise from synovial tissue. It is so named because of its histologic similarity to synovium. An SS originates from mesenchyme, not from synoviocytes and usually manifests as a biphasic tumor with both malignant-epithelial and spindle-cell components. Monophasic epithelial and spindle-cell presentations may cause a diagnostic dilemma. Diagnosis should include immunocytochemistry using cytokeratin and/or epithelial membrane antigen; vimentin further helps to eliminate any histologic confusion. These tumors are most commonly found in the extremities. When located near a joint, invasion occurs only by secondary extension. Rarely are SSs found in the neck, especially in the posterior aspect, as reported here.
PMID: 7552151
ISSN: 1078-4519
CID: 5035992

The diagnosis and treatment of cerebral mycotic aneurysms

Brust, J C; Dickinson, P C; Hughes, J E; Holtzman, R N
Seventeen patients were treated for 28 documented cerebral mycotic aneurysms. Initial neurological symptoms were attributable to aneurysm rupture in only 7 patients, and in 3 of them symptoms did not suggest subarachnoid hemorrhage. Six patients presented with embolic infarction and 1 with meningitis; in 3 patients it was uncertain if aneurysm rupture occurred. Four patients had rupture of at least one aneurysm while receiving appropriate antibiotic treatment and another had rupture at the conclusion of therapy. Of 20 aneurysms followed angiographically or with computed tomography during medical treatment, 10 became smaller or disappeared and 10 remained unchanged or enlarged, 1 with fatal rupture. Eight ruptured aneurysms were surgically excised; 2 of the patients with ruptured aneurysms died and 2 had residual aphasia or cognitive impairment. All 4 patients whose only surgery was for an unruptured aneurysm made uneventful recoveries. Recognizing the retrospective and anecdotal nature of our data and the differing views of previous investigators, we recommend: (1) that careful neurological examination, computed tomography, and (unless contraindicated) lumbar puncture be performed on any patient with endocarditis; (2) that those with neurological abnormalities not attributable to systemic toxicity, including pleocytosis in the cerebrospinal fluid or apparent infarction on computed tomographic scans, undergo four-vessel cerebral angiography; (3) that single accessible mycotic aneurysms in medically stable patients be promptly excised, with individualization of multiple or proximal aneurysms; and (4) that repeat angiography be performed at the conclusion of antibiotic therapy in patients requiring long-term anticoagulation.(ABSTRACT TRUNCATED AT 250 WORDS)
PMID: 2327735
ISSN: 0364-5134
CID: 5035902

Bilateral symptomatic intraspinal T12-L1 synovial cysts [Case Report]

Holtzman, R N; Dubin, R; Yang, W C; Rorat, E; Liu, H M; Leeds, N E
A 72-year-old man presented with several months of increasing lumbar pain, sciatica, lower extremity weakness, numbness in his buttocks and posterior thighs, burning sensations in his scrotum, and urinary incontinence. Myelogram-computed tomography scan demonstrated a high grade incomplete block at the T12-L1 level due to bilateral synovial cysts and simultaneously a high grade partial block at L4-L5 due to spinal stenosis. Laminectomy of the T-12 vertebra and partial laminectomy of the L-1 vertebra with excision of both synovial cysts and laminectomies of the L-4 and L-5 vertebrae with foraminotomies resulted in a reversal of the patient's symptomatology.
PMID: 3629450
ISSN: 0090-3019
CID: 5035922