Head ache, bouts of vomitng and blurring of vision .
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Selected MRI image at first presentation, after resection of cranipharyngioma and after onset of metastatic recurrence in right temporal fossa
MRI shows a mass with a cystic component filled with proteinatious fluid in the suprasellar region causing compression of optic chiasma.
This patient underwent right parietal craniotomy for suprasellar craniopharyngioma in 2008. The histology indicated a adamantinous craniopharyngioma.
It was believed to be completely resected surgically and the immediate post operative MRI imaging one year after surgery confirmed complete resection without residual tumor (not shown). No radiotherapy given. He was on hormone replacement therapy with cortisol and thyroxine.
3 years after the surgery, the child again developed visual symptoms and head ache. MRI done after this second symptomatic presentation demonstrated a cystic lesion in the right temporoparietal region with an enhancing wall. There was no recurrence at the site of surgery. Since the symptoms were mild the patient was followed up for 18 months.
Therefore repeat right parietal craniotomy was performed to resect this lesion. The biopsy again confirmed a metastatic seedling of adamantinous craniopharyngioma.
5 years later
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The MRI after 2 years demonstrated further enlargement of this implant metastasis.
Our patient had a recurrence along the surgical route in the right middle cranial fossa. The approximate time period between surgery and recurrence was two years. There were reported 17 cases in the available literature out of which 5 were under the age of 18 years. In the most of the cases metastases have occurred along the surgical route. There were two recurrences in the spinal cord and the rest were at ectopic sites not related to surgical route 10,11. In one case report a patient has presented with two metastases 4: one in the temporal fossa at 3 years and another in the frontal space at 10 years 7.
These patients are usually given hormonal replacement therapy 7. Our patient was treated with thyroxine, cortisol and DDAVP. He underwent repeat surgical resection of the metastasis in March 2013.
Two mechanisms have been postulated for these ectopic recurrences.
- The first is metastasis often occurs along the surgical route, as in our case
- Spreading to distant locations via cerebrospinal fluid has been suggested 3. Therefore regular follow-up is necessary, even after supposedly total resection.
- 1 Mark Bernstein, Mitchel S. Berger-. Neuro-Oncology: The Essentials of Neuro-Oncology: Thieme 18-19
- 2. Sekine S, Shibata T, Kokubu A, et al (December 2002). "Craniopharyngiomas of adamantinomatous type harbor beta-catenin gene mutations". Am. J. Pathol. 161 (6): 1997–2001
- 3. Frangou et al, Metastatic Craniopharyngioma: Case report and literature review Child Nerv Syst (2009) 25: 1143-1147,
- 4. Novegno et al, Ectopic recurrences of craniopharyngioma Novegno et al, child nerv syst (2002) 18:468-473
- 4. Fuentes S, Metellus P, Dufour H, Do L, Grisoli F , Postoperative intracranial seeding of craniopharyngioma. Three case reports and a review of the literature Neuro-Chirurgie 2002, 48(4):345-350
- 5. Ito Mamoru, Jamshidi, Jamshid , Yamanaka Kazunori , Does Craniopharyngioma Metastasize? Case Report and Review of the Literature. Neurosurgery: April 2001 :48 : 4 ; 933-936
- 6. Nomura H, Kurimoto M, Nagai S, Hayashi N, Hirashima Y, Tsukamoto E, Endo S , Multiple intracranial seeding of craniopharyngioma after repeated surgery—case report. (2002) Neurol Med Chir (Tokyo) 42:268–271
- 8. Bikmaz, K ,Guerrero C et al, Ectopic Recurrence of Craniopharyngiomas: Case Report Neurosurgery: February 2009 : 64 :2 ;382-388
- 10. Jacek M. Malik, Remote recurrence of craniopharyngioma in the epidural space Case report , Journal of Neurosurgery November 1992 . 77 : 5 804-807
- 11. Dae Kyu Lee,et al Postoperative spinal seeding of craniopharyngioma Case report Journal of Neurosurgery April 2001 94 : 4 617-620
- 12. Gupta K, Kuhn MJ, Shevlin DW, Wacaser LE Metastatic craniopharyngioma. (1999) AJNR Am J Neuroradiol 20:1059–1060
- 9. Ashvin T. Ragoowansi, Postoperative ectopic craniopharyngioma-Case report Journal of Neurosurgery April 1991, 74 :4 653-655
- 7. Liu, Jeannette M ,Ectopic Recurrence of Craniopharyngioma after an Interhemispheric Transcallosal Approach: Case Report. Neurosurgery: March 2002 : 50 : 639-645