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LETTERS TO EDITOR
Year : 2018  |  Volume : 66  |  Issue : 2  |  Page : 564-566

Distant supratentorial recurrence of craniopharyngioma


1 Department of Neurosurgery, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad, Telangana, India
2 Department of Pathology, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad, Telangana, India

Date of Web Publication15-Mar-2018

Correspondence Address:
Dr. Alugolu Rajesh
Department of Neurosurgery, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad - 500 082, Telangana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.227330

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How to cite this article:
Egoor A, Rajesh A, Uppin MS, Saradhi M V. Distant supratentorial recurrence of craniopharyngioma. Neurol India 2018;66:564-6

How to cite this URL:
Egoor A, Rajesh A, Uppin MS, Saradhi M V. Distant supratentorial recurrence of craniopharyngioma. Neurol India [serial online] 2018 [cited 2019 Aug 22];66:564-6. Available from: http://www.neurologyindia.com/text.asp?2018/66/2/564/227330




Sir,

Recurrences, even following complete excision of craniopharyngiomas, are well known, with most (up to 57%) of them occurring loco- regionally.[1] Ectopic recurrences, cranial and spinal, have been largely reported in the pediatric population.[2],[3] Here, we report a case of an ectopic recurrence of a craniopharyngioma 2 years following its initial diagnosis and treatment.

A 24-year old male patient presented with headache and bitemporal hemianopia along with a suprasellar lesion [Figure 1]. Our working diagnosis was a craniopharyngioma. He was operated by a right supraorbital, keyhole craniotomy, subfrontal corridor, wherein a gross total resection was performed. Histopathologically, it was reported as adamantinomatous craniopharyngioma. The patient's visual symptoms improved but he had persistent diabetes insipidus and hypothyroidism (thyroxine (T) 4: 3.5 nmg/dl; T3: 10 nmg/dl; thyroid-stimulating hormone: 0.2 mIU/dl; and, cortisol: 12 μg/dl) in the postoperative period. He was treated with tab desmopressin 0.1 mg daily and thyroid replacement (thyroxine 100 μg daily). His follow-up magnetic resonance imaging (MRI) of the brain at 6 months [Figure 2], 12 months and 18 months postoperatively, showed no recurrence.
Figure 1: Magnetic resonance imaging of the brain showing a suprasellar lesion distinct from the normal sella, hypointense on T1-weighted sagittal section (a), hyperintense onT2-weighted coronal section (b) and having heterogenous contrast enhancement (c)

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Figure 2: A 12-month follow-up magnetic resonance imaging scan showing the craniotomy defect (red arrow) and no evidence of any residual or recurrence lesion

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At 2 years, he developed a severe left-sided headache, and computed tomography scan of the brain (done at an outside facility) showed a hypodense lesion with peripheral rim of calcification in the left parietal region [Figure 3]. MRI of the brain showed an extra-axial, well-defined ovoid lesion in the left parietal region which was hypointense on T1-weighted, hyperintense on T2-weighted/fluid-attenuated inversion recovery (FLAIR) images with peripheral rim enhancement on contrast [Figure 4].
Figure 3: Computed tomographic scan, axial sections showing a hypodense lesion with peripheral rim of calcification in the left parietal region

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Figure 4: Magnetic resonance imaging of the brain showing an extraaxial, well-defined ovoid lesion in the left parietal region, hypointense on T1-weighted (a) and hyperintense on T2-weighted (b) images, with a peripheral rim enhancement on contrast administration (c)

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A left parietal craniotomy targeted over the lesion revealed a well-defined, extra-axial, cystic lesion with straw-colored fluid, which was excised in toto [Figure 5].
Figure 5: Intraoperative picture showing a cystic, well-defined lesion being removed in-toto

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Histopathological examination showed a cystic lesion lined by stratified squamous epithelium and wet keratin, that on a higher magnification showed nests and trabeculae of cells with peripheral palisading and central stellate reticulum, which was characteristic of an adamantinomatous craniopharyngioma (the same histological type as the primary tumor) [Figure 6]. He recovered well and was under constant follow-up.A follow-up MRI after 24 months showed no evidence of a residual or recurrent lesion [Figure 7].
Figure 6: (a and b) Cystic lesion lined by stratified squamous epithelium and wet keratin. Hematoxylin and eosin ×20. (c and d) Nests and trabeculae of cells with peripheral palisading and central stellate reticulum. Hematoxylin and eosin ×400

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Figure 7: A 6-month follow-up magnetic resonance imaging showing no evidence of residual or recurrent lesion on T1-weighted (a), T2-weighted (b) and contrast (c) axial sections

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Local recurrence of craniopharyngiomas is not a rare occurrence with the reported incidence of being upto 57%.[3],[4] However, an ectopic recurrence is a rare phenomenon with only 45 cases reported till date in the English literature.

There are two defined mechanisms for the ectopic recurrences viz., seeding along the surgical route during the primary resection, the most common route of ectopic spread, or the spillage into the cerebrospinal fluid (CSF), which may allow viable tumor cells to migrate within the subarachnoid and even the intraventricular space.

In our report, it was a left parietal recurrence in a patient who was operated via a right subfrontal approach, which ruled out the possibility of recurrence due to seeding along the surgical route.

On reviewing the literature regarding the mechanism of ectopic spread of a craniopharyngioma, 20 cases were speculated to have spread via a CSF dissemination, 24 cases via the direct seeding of the surgical pathway, and one case was believed to have spread by both the mechanisms. The mean time to recurrence via the CSF dissemination was approximately 7.6 years; whereas, in cases with seeding along the surgical pathway, it was 5.2 years. However, in the present case, the recurrence occurred within 2 years.

This report emphasizes the importance of careful handling of the tumor while resection is taking place, and avoidance of tumor spillage into the CSF, which might lead to recurrences. The importance of a long-term follow-up and a maintaining a high index of suspicion for recurrences in these patients is also highlighted.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Bunin GR, Surawicz TS, Witman PA, Preston-Martin S, Davis F, Bruner JM. The descriptive epidemiology of craniopharyngioma. J Neurosurg 1998;89:547-51.  Back to cited text no. 1
    
2.
Adamson TE, Wiestler OD, Kleihues P, Yasargil MG. Correlation of clinical and pathological features in surgically treated craniopharyngiomas. J Neurosurg1990; 73:12-7.  Back to cited text no. 2
    
3.
Narla S, Govindraj J, Chandrasekar K, Sushama P. Craniopharyngioma with malignant transformation: Review of literature. Neurol India 2017;65:418-20.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Clark SW, Kenning TJ, Evans JJ. Recurrent ectopic craniopharyngioma: A case report and review of the literature. Nagoya J Med Sci 2015;77:297-306.  Back to cited text no. 4
    


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  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]



 

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