Oligodendroglioma and juvenile pilocytic astrocytoma presenting as synchronous primary brain tumors

Case report with histological and molecular differentiation of the tumors and review of the literature

Restricted access

✓ Multiple metastatic brain tumors and multifocal primary brain tumors of a single histological type are well described in the literature. The concurrent presence of multiple primary brain tumors with different histological characteristics, however, is very rare. The authors describe the first known case in which an oligodendroglioma and a juvenile pilocytic astrocytoma (JPA) presented as synchronous primary brain tumors in the same patient.

This 43-year-old man presented with a 2-month history of progressive headaches, nausea, and vomiting. Magnetic resonance imaging demonstrated an enhancing heterogeneous right medial cerebellar lesion and a larger calcified, nonenhancing, heterogeneous right frontal lesion with surrounding edema and a mass effect. The results of a metastatic workup were unremarkable.

The patient underwent an initial right frontotemporal craniotomy and a subsequent suboccipital craniectomy 2 years later for resection of the posterior fossa lesion. Histological examination revealed the frontal and cerebellar lesions to be an oligodendroglioma and JPA, respectively. A molecular analysis detected a deletion of chromosome 1p36 in the oligodendroglioma, but not in the JPA. After the initial operation, the patient received follow-up care for his oligodendroglioma, but eventually required temozolomide for tumor progression. His condition remains stable both neurologically and according to imaging studies.

The authors describe the first known case in which a low-grade oligodendroglioma and a JPA presented as synchronous primary brain tumors. They review the literature on multiple primary brain tumors with different histological characteristics and discuss potential mechanisms for the development of these lesions.

Article Information

Address reprint requests to: Randy Jensen, M.D., Ph.D., Department of Neurosurgery, University of Utah, 3B409 SOM, 30 North 1900 East, Salt Lake City, Utah 84132–2303. email: randy.jensen@hsc.utah.edu.

© AANS, except where prohibited by US copyright law.

Headings

Figures

  • View in gallery

    Neuroimaging studies revealing a calcified 6-cm nonenhancing heterogeneous right frontal lesion. Left: Contrast-enhanced CT scan demonstrating calcification and no enhancment. Right: Axial T1-weighted MR image obtained after Gd administration.

  • View in gallery

    Magnetic resonance images demonstrating a right medial 2 × 1.5 × 2—cm cerebellar mass containing both a cystic and an enhancing solid component. The lesion is exerting a mild mass effect on the fourth ventricle. Left: Axial T1-weighted image. Right: Axial T1-weighted image obtained after Gd administration.

  • View in gallery

    Photomicrographs of sections from each lesion. Upper: Tissue demonstrating a diffusely infiltrating glial tumor. The tumor cells contain primarily round regular nuclei with mild variations in size and shape. There are areas in which a fine meshwork of thin-walled vessels separate groups of tumor cells. Satellitosis of neurons by the tumor cells and focal calcification are also seen. The cellular architecture is consistent with that of an oligodendrocyte displaying a characteristic “fried egg” appearance. No mitosis, necrosis, or endothelial proliferation is seen. Lower: Section of the tumor removed from the patient's posterior fossa during the second operation. This specimen consists of hypercellular glial tissue with numerous pilocytic processes and Rosenthal fibers. Mild cellular atypia is present and the tissue is clearly different from that of the supratentorial oligodendroglioma (upper). A final diagnosis of JPA was made. H & E, original magnification × 40.

  • View in gallery

    Results of an FISH performed using a 1p36 probe (CEB 108/T7). Left: The majority (84%) of oligodendroglioma nuclei that were counted demonstrated a loss of chromosome 1p36, which is manifested by a single signal per nuclei. Right: Conversely, the majority (71%) of the nuclei of cells from the JPA demonstrate two signals, making it statistically distinct according to the FISH analysis of 1p36 (p < 0.0001, chi-square test).

References

  • 1.

    Arieti S: Multiple meningioma and meningiomas associated with other brain tumors. J Neuropathol Exp Neurol 3:2552701944Arieti S: Multiple meningioma and meningiomas associated with other brain tumors. J Neuropathol Exp Neurol 3:255–270 1944

    • Search Google Scholar
    • Export Citation
  • 2.

    Batzdorf UMalamud N: The problem of multicentric gliomas. J Neurosurg 20:1221361963Batzdorf U Malamud N: The problem of multicentric gliomas. J Neurosurg 20:122–136 1963

    • Search Google Scholar
    • Export Citation
  • 3.

    Bingas BBrunngraber CV: Das gleichzeitige Vorkommen von Meningeom und Glioblastom. Zentralbl Neurochir 24:2712751964Bingas B Brunngraber CV: Das gleichzeitige Vorkommen von Meningeom und Glioblastom. Zentralbl Neurochir 24:271–275 1964

    • Search Google Scholar
    • Export Citation
  • 4.

    Butti GGiordana MTPaoletti Pet al: Multiple primary intracranial tumors of different cell types: association of anaplastic astrocytoma and acoustic neurinoma—with review of the literature. Surg Neurol 18:3363421982Butti G Giordana MT Paoletti P et al: Multiple primary intracranial tumors of different cell types: association of anaplastic astrocytoma and acoustic neurinoma—with review of the literature. Surg Neurol 18:336–342 1982

    • Search Google Scholar
    • Export Citation
  • 5.

    Cohnheim J: Vorlesungen Uber Allgemeine Pathologie. Berlin: A Hirshwald1877Cohnheim J: Vorlesungen Uber Allgemeine Pathologie. Berlin: A Hirshwald 1877

    • Search Google Scholar
    • Export Citation
  • 6.

    Deen HG JrLaws ER Jr: Multiple primary brain tumors of different cell types. Neurosurgery 8:20251981Deen HG Jr Laws ER Jr: Multiple primary brain tumors of different cell types. Neurosurgery 8:20–25 1981

    • Search Google Scholar
    • Export Citation
  • 7.

    Gokalp HZErdogan AEgemen Net al: Multiple intracranial tumors of different cell types. Neurosurgery 27:4634661990Gokalp HZ Erdogan A Egemen N et al: Multiple intracranial tumors of different cell types. Neurosurgery 27:463–466 1990

    • Search Google Scholar
    • Export Citation
  • 8.

    Kasantikul VShuangshoti SPreechayudh Pet al: A combined neurilemmoma and angioma of the parasellar region. Case report. J Neurosurg 67:3073111987Kasantikul V Shuangshoti S Preechayudh P et al: A combined neurilemmoma and angioma of the parasellar region. Case report. J Neurosurg 67:307–311 1987

    • Search Google Scholar
    • Export Citation
  • 9.

    Madonick MJShapiro JHThorac RM: Multiple diverse primary brain tumors. Neurology 11:4304361961Madonick MJ Shapiro JH Thorac RM: Multiple diverse primary brain tumors. Neurology 11:430–436 1961

    • Search Google Scholar
    • Export Citation
  • 10.

    Marra ARamponi GGrimaldi G: Simultaneous occurrence of right supratentorial meningioma and glioblastoma multiforme. Case report. Acta Neurochir 36:83911977Marra A Ramponi G Grimaldi G: Simultaneous occurrence of right supratentorial meningioma and glioblastoma multiforme. Case report. Acta Neurochir 36:83–91 1977

    • Search Google Scholar
    • Export Citation
  • 11.

    Miki KKawamoto KKawamuro Yet al: A rare case of Maffucci's syndrome combined with tuberculum sellae enchondroma, pituitary adenoma and thyroid adenoma. Acta Neurochir 87:79851987Miki K Kawamoto K Kawamuro Y et al: A rare case of Maffucci's syndrome combined with tuberculum sellae enchondroma pituitary adenoma and thyroid adenoma. Acta Neurochir 87:79–85 1987

    • Search Google Scholar
    • Export Citation
  • 12.

    Myerson PG: Multiple tumors of the brain of diverse origin. J Neuropathol Exp Neurol 1:4064151942Myerson PG: Multiple tumors of the brain of diverse origin. J Neuropathol Exp Neurol 1:406–415 1942

    • Search Google Scholar
    • Export Citation
  • 13.

    Pearl GSMirra SSMiles ML: Glioblastoma multiforme occurring 13 years after treatment of a medulloblastoma. Neurosurgery 6:5465511980Pearl GS Mirra SS Miles ML: Glioblastoma multiforme occurring 13 years after treatment of a medulloblastoma. Neurosurgery 6:546–551 1980

    • Search Google Scholar
    • Export Citation
  • 14.

    Raghavan RBalani JPerry Aet al: Pediatric oligodendrogliomas: a study of molecular alterations on 1p and 19q using fluorescence in situ hybridization. J Exp Neuropathol Exp Neurol 62:5305372003Raghavan R Balani J Perry A et al: Pediatric oligodendrogliomas: a study of molecular alterations on 1p and 19q using fluorescence in situ hybridization. J Exp Neuropathol Exp Neurol 62:530–537 2003

    • Search Google Scholar
    • Export Citation
  • 15.

    Robinson RG: A second brain tumour and irradiation. J Neurol Neurosurg Psychiatry 41:100510121978Robinson RG: A second brain tumour and irradiation. J Neurol Neurosurg Psychiatry 41:1005–1012 1978

    • Search Google Scholar
    • Export Citation
  • 16.

    Rubinstein LJ (ed): Tumors of the Central Nervous System. Atlas of Tumor PathologySer 2Fasc 6. Washington, DC: Armed Forces Institute of Pathology1972Rubinstein LJ (ed): Tumors of the Central Nervous System. Atlas of Tumor Pathology Ser 2 Fasc 6. Washington DC: Armed Forces Institute of Pathology 1972

    • Search Google Scholar
    • Export Citation
  • 17.

    Sackett JFStenwig JTSongsirikul P: Meningeal and glial tumors in combination. Neuroradiology 7:1531601974Sackett JF Stenwig JT Songsirikul P: Meningeal and glial tumors in combination. Neuroradiology 7:153–160 1974

    • Search Google Scholar
    • Export Citation
  • 18.

    Schnegg JFde Tribolet N: Neurinoma of the facial nerve associated with a parietal meningioma. Surg Neurol 21:19221984Schnegg JF de Tribolet N: Neurinoma of the facial nerve associated with a parietal meningioma. Surg Neurol 21:19–22 1984

    • Search Google Scholar
    • Export Citation
  • 19.

    Schoenberg BS: Multiple primary neoplasms and the nervous system. Cancer 40 (Suppl 4):196119671977Schoenberg BS: Multiple primary neoplasms and the nervous system. Cancer 40 (Suppl 4):1961–1967 1977

    • Search Google Scholar
    • Export Citation
  • 20.

    Shishkina VLKasumova SIUSnigireva RIAet al: Sochetanie Kraniofaringiomy s adenomoi gipofiza ikhordomi bliumenbakhova skata. Zh Vopr Neiroktir Im N N Burdenko 6:52541981Shishkina VL Kasumova SIU Snigireva RIA et al: Sochetanie Kraniofaringiomy s adenomoi gipofiza ikhordomi bliumenbakhova skata. Zh Vopr Neiroktir Im N N Burdenko 6:52–54 1981

    • Search Google Scholar
    • Export Citation
  • 21.

    Shuangshoti SNetsky MG: Brain tumors of mixed mesenchymal neuroepithelial origin. Case report. J Neurosurg 34:8088131971Shuangshoti S Netsky MG: Brain tumors of mixed mesenchymal neuroepithelial origin. Case report. J Neurosurg 34:808–813 1971

    • Search Google Scholar
    • Export Citation
  • 22.

    Strong AJSymon LMacGregore BJLet al: Coincidental meningioma and glioma: Report of two cases. J Neurosurg 45:4554581976Strong AJ Symon L MacGregore BJL et al: Coincidental meningioma and glioma: Report of two cases. J Neurosurg 45:455–458 1976

    • Search Google Scholar
    • Export Citation
  • 23.

    Swischuk LEBryan RN: Double midline intracranial atypical teratomas: a recognizable neuroendocrinologic syndrome. AJR 122:5175241974Swischuk LE Bryan RN: Double midline intracranial atypical teratomas: a recognizable neuroendocrinologic syndrome. AJR 122:517–524 1974

    • Search Google Scholar
    • Export Citation
  • 24.

    Tanaka JGarcia JHNetsky MGet al: Late appearance of meningioma at the site of partially removed oligodendroglioma. Case report. J Neurosurg 43:80851975Tanaka J Garcia JH Netsky MG et al: Late appearance of meningioma at the site of partially removed oligodendroglioma. Case report. J Neurosurg 43:80–85 1975

    • Search Google Scholar
    • Export Citation
  • 25.

    Waga SHanda H: Radiation-induced meningioma: with review of literature. Surg Neurol 5:2152191976Waga S Handa H: Radiation-induced meningioma: with review of literature. Surg Neurol 5:215–219 1976

    • Search Google Scholar
    • Export Citation
  • 26.

    Willis RA: The pathology of the tumors of children in Cameron RWright GP (eds): Pathological MonographsSeries II. Edinburg: Oliver and Boyd1962 pp 2025Willis RA: The pathology of the tumors of children in Cameron R Wright GP (eds): Pathological Monographs Series II. Edinburg: Oliver and Boyd 1962 pp 20–25

    • Search Google Scholar
    • Export Citation
  • 27.

    Yamada KHatayama TOhta Met al: Coincidental pituitary adenoma and parasellar meningioma: Case report. Neurosurgery 19:2672701986Yamada K Hatayama T Ohta M et al: Coincidental pituitary adenoma and parasellar meningioma: Case report. Neurosurgery 19:267–270 1986

    • Search Google Scholar
    • Export Citation

TrendMD

Metrics

Metrics

All Time Past Year Past 30 Days
Abstract Views 93 93 4
Full Text Views 133 133 0
PDF Downloads 86 86 0
EPUB Downloads 0 0 0

PubMed

Google Scholar