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 Table of Contents     
CASE REPORT
Year : 2022  |  Volume : 37  |  Issue : 3  |  Page : 268-270  

An unusual case of glioblastoma multiforme, presenting as skeletal superscan


Department of Nuclear Medicine, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India

Date of Submission30-Dec-2021
Date of Decision02-Mar-2022
Date of Acceptance16-Mar-2022
Date of Web Publication02-Nov-2022

Correspondence Address:
Dr. Girish Kumar Parida
Department of Nuclear Medicine, All India Institute of Medical Sciences, Bhubaneswar - 751 019, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijnm.ijnm_209_21

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   Abstract 


Extracranial metastases of glioblastoma multiforme (GBM) are very rare. The estimated incidence is <2%. We report a case of a 49-year-old woman, who was a known case of GBM in the left temporo-occipital lobe. She was operated and had received radiotherapy and adjuvant chemotherapy for the same. Subsequently, the patient underwent bone scan. On 99 m-Tc methylene diphosphonate (MDP) bone scan, homogenously increased tracer uptake was noted in the axial and appendicular skeletal system, suggesting metastatic skeletal superscan.

Keywords: 99 m-Tc MDP bone scan, bone metastasis, extracranial metastases, glioblastoma multiforme, superscan


How to cite this article:
Bishnoi K, Parida GK, Thavnani R, Patro P S, Agrawal K. An unusual case of glioblastoma multiforme, presenting as skeletal superscan. Indian J Nucl Med 2022;37:268-70

How to cite this URL:
Bishnoi K, Parida GK, Thavnani R, Patro P S, Agrawal K. An unusual case of glioblastoma multiforme, presenting as skeletal superscan. Indian J Nucl Med [serial online] 2022 [cited 2022 Nov 29];37:268-70. Available from: https://www.ijnm.in/text.asp?2022/37/3/268/360275




   Introduction Top


In adults, glioblastoma multiforme (GBM) is the most common malignant primary brain tumor.[1] Grade IV is the highest grade in the WHO classification of brain tumors defined by histopathologic features of endothelial proliferation and necrosis.[2] The incidence of GBM is 3.19 cases/100,000 person years. It has a very poor prognosis with a 5-year survival rate of 4%–5%, and the survival rate at 2 years is only 26%–33%.[3],[4],[5],[6] In the younger age group, specific point mutation of isocitrate dehydrogenase (IDH) 1 or 2 genes are seen to be associated.[7] It has a more favorable outcome. The IDH wild-type is seen in the majority of the GBM.[8],[9] The molecular changes in glioblastoma typically include mutations in genes regulating receptor tyrosine kinase/rat sarcoma/p53/phosphoinositide 3-kinase.[10] Majority of the patients present with a clinical history of 3–6 months. If tumor develops from a low-grade astrocytoma, the symptoms, however, may span over a long period.[11],[12] Occasionally, the symptoms might be mistaken for a stroke.[13] By direct effect, there is necrosis of brain tissue, which gives rise to symptoms depending on the involved region of the brain. About 40%–60% of patients present with focal neural deficit and cognitive impairments. 20%–40% of patients have frontal lobe involvement causing a personality change.[11],[12] Large tumors can lead to gait impairment and incontinence.[13] There is increased intracranial pressure and surrounding edema due to gradual increase in tumor size. This leads to a shift in intracranial contents, resulting in headaches. Headache is a characteristic feature in 30%–50% of GBM patients.[11],[12] Unilaterally localized headache with no specific pain pattern is seen and is often associated with vomiting and papilledema.[13] 20%–40% cases may have seizures depending on the location of tumor cases. It is usually a focal onset episode. This can be simple partial, complex partial, or generalized seizures.[11],[12],[13] Extracranial metastases of GBM are rare with incidence of <2%.[14],[15] The GBM may spread by the hematogenous and lymphatic route.[16] In some cases, direct extension of tumor can be the mode of spread. The rarity of metastatic GBM can be explained by the absence of lymphatics in the central nervous system and the lack of communication between intracranial and extracranial perivascular spaces.[17] Furthermore, the shorter survival time of patients, and the metastatic disease which is still undiagnosed can be the cause.[18] The most commonly affected organ systems extracranially are bones, lymph nodes, and lungs. Although extracranial GBM does not shorten the survival time of patients drastically, the physicians should be aware of its presence. If there are any symptoms referable to these areas of spread, they should be evaluated by the physicians. The approximate median OS for patients with GBM is 12–15 months.[19] Reportedly, cases with local intracranial recurrence have a median OS of 7.5 months.[20] Local continuous growth is the most common form of GBM recurrence which is seen within 2–3 cm from the border of the original lesion.[21],[22],[23] Original tumor location recurrence occurs in more than 90% of the patients with glioma. In 5% of cases, multiple lesions develop after treatment.[24] Rarely, GBM cases may develop new parenchymal lesions which do not exhibit continuous growth patterns, ventricular spread, or dissemination.[25] Uncommon relapse patterns can be seen in midline tumors or those which infiltrate both hemispheres.[26] Subtotal resections are sometimes done in an attempt to preserve neurological function and maintain patient quality of life. This may later lead to residual tumor growth which is seen on imaging scans or manifest as new symptoms. The appearance of residual tumor growth is also defined as tumor recurrence. The term “tumor progression” is considered synonymous with “tumor recurrence” because of the spectrum from which new lesions can develop. The treatment of recurrent GBM is multimodal including surgery, radiotherapy, chemotherapy, and antiangiogenic therapy. Surgical resection of GBM reduces the metastasis risk, and chemotherapy and radiotherapy in metastatic GBM are considered helpful in prolonging survival.[14] Further knowledge in the field of underlying tumor biology, gene therapy, antiangiogenic antagonists, and immunotherapies may help in developing more promising treatment plans.[27]

We report an unusual case in which a patient presented with skeletal superscan and further investigations helped in the diagnosis.


   Case Report Top


A 49-year-old female patient, who was a known case of GBM in the left temporo-occipital lobe for which she had craniotomy. Following which the patient had received adjuvant chemotherapy (temozolomide) and radiotherapy treatment. Subsequently, the patient presented with left-sided body pain. In suspicion for skeletal metastases, the patient was referred for bone scan. Whole-body bone scan was done with IV administration of 20 mCi of 99 m-Tc MDP. Anterior and posterior images [Figure 1]a & [Figure 1]b representing anterior and posterior images, respectively] showed homogenously increased tracer uptake in the axial and appendicular skeleton system with increased bone soft tissue ratio and very faint visualization of the kidneys, suggesting of metastatic superscan. Magnetic resonance imaging spine was also done which showed multiple hyperintense bony lesions in all lumbar vertebrae, bilateral iliac bones and sacrum, favoring skeletal metastases. Subsequently, to look for other visceral metastasis, contrast-enhanced computed tomography (thorax, abdomen, and pelvis) was done, which showed extensive sclerotic lesions in multiple vertebrae, sternum, and bilateral pelvis. Bone marrow biopsy later confirmed the diagnosis and the diagnosis of GBM with diffuse skeletal metastases was made. However, before commencement of any therapy patient succumbed to the disease.
Figure 1: Figure a & b represent anterior and posterior bone scan images respectively, which show homogeneously increased tracer uptake in the axial and appendicular skeleton with increased bone to soft tissue ratio and very faint visualization of the kidneys, suggestive of metastatic super-scan

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   Discussion Top


The natural history of GBM metastases is still not completely defined. The knowledge of clinical factors which promote GBM metastases may explain the mechanisms of tumor cell invasion in the brain. Although extracranial GBM does not decrease the survival time of patients with GBM, the awareness regarding its existence leads to better management of patients. There are very few case reports published, showing skeletal metastases from GBM and only one case report showing presentation as skeletal superscan.[28] In a study done by Manohar et al., the overall incidence of superscan in different types of cancers was 1.3%. It was most commonly seen in patients with prostate cancer followed by breast and lung cancer.[29] The case discussed above is of a patient with GBM who presented with skeletal superscan. A bone scan with diffusely increased skeletal radioisotope uptake relative to soft tissue in association with absent or faint renal activity (“absent kidney sign)” is known as a superscan.[30],[31] Recurrence is also not uncommon and is treated with a multimodal approach including surgery, radiotherapy, chemotherapy, and antiangiogenic therapy. However, further understanding of underlying tumor biology is essential in developing more effective therapies.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Schiff D, Wen P. Cancer Neurology in Clinical Practice. Germany: Humana Press Inc.; 2003.  Back to cited text no. 1
    
2.
Tinchon A, Oberndorfer S, Marosi C, Rudà R, Sax C, Calabek B, et al. Malignant spinal cord compression in cerebral glioblastoma multiforme: A multicenter case series and review of the literature. J Neurooncol 2012;110:221-6.  Back to cited text no. 2
    
3.
Razavi SM, Lee KE, Jin BE, Aujila PS, Gholamin S, Li G. Immune evasion strategies of glioblastoma. Front Surg 2016;3:11.  Back to cited text no. 3
    
4.
Van Meir EG, Hadjipanayis CG, Norden AD, Shu HK, Wen PY, Olson JJ. Exciting new advances in neuro-oncology: The avenue to a cure for malignant glioma. CA Cancer J Clin 2010;60:166-93.  Back to cited text no. 4
    
5.
Carlsson SK, Brothers SP, Wahlestedt C. Emerging treatment strategies for glioblastoma multiforme. EMBO Mol Med 2014;6:1359-70.  Back to cited text no. 5
    
6.
Weathers SP, Gilbert MR. Current challenges in designing GBM trials for immunotherapy. J Neurooncol 2015;123:331-7.  Back to cited text no. 6
    
7.
Louis DN, Perry A, Reifenberger G, von Deimling A, Figarella-Branger D, Cavenee WK, et al. The 2016 World Health Organization classification of tumors of the central nervous system: A summary. Acta Neuropathol 2016;131:803-20.  Back to cited text no. 7
    
8.
Sturm D, Witt H, Hovestadt V, Khuong-Quang DA, Jones DT, Konermann C, et al. Hotspot mutations in H3F3A and IDH1 define distinct epigenetic and biological subgroups of glioblastoma. Cancer Cell 2012;22:425-37.  Back to cited text no. 8
    
9.
Ellingson BM, Lai A, Harris RJ, Selfridge JM, Yong WH, Das K, et al. Probabilistic radiographic atlas of glioblastoma phenotypes. AJNR Am J Neuroradiol 2013;34:533-40.  Back to cited text no. 9
    
10.
Cancer Genome Atlas Research Network. Comprehensive genomic characterization defines human glioblastoma genes and core pathways. Nature 2008;455:1061-8.  Back to cited text no. 10
    
11.
Clarke CR. In: Kumar P, Clark M, editors. Neurological Diseases in Kumar & Clark Clinical Medicine. 6th ed. Edinburgh: Elsevier Saunders; 2005. p. 1244-5.  Back to cited text no. 11
    
12.
Sanli AM, Turkoglu E, Dolgun H, Sekerci Z. Unusual manifestations of primary Glioblastoma Multiforme: A report of three cases. Surg Neurol Int. 2010;1:87.  Back to cited text no. 12
    
13.
Omuro A, DeAngelis LM. Glioblastoma and other malignant gliomas: A clinical review. JAMA 2013;310:1842-50.  Back to cited text no. 13
    
14.
Davis ME. Glioblastoma: Overview of disease and treatment. Clin J Oncol Nurs 2016;20:S2-8.  Back to cited text no. 14
    
15.
Waite KJ, Wharton SB, Old SE, Burnet NG. Systemic metastases of glioblastoma multiforme. Clin Oncol (R Coll Radiol) 1999;11:205-7.  Back to cited text no. 15
    
16.
Zhen L, Yufeng C, Zhenyu S, Lei X. Multiple extracranial metastases from secondary glioblastoma multiforme: A case report and review of the literature. J Neurooncol 2010;97:451-7.  Back to cited text no. 16
    
17.
Piccirilli M, Brunetto GM, Rocchi G, Giangaspero F, Salvati M. Extra central nervous system metastases from cerebral glioblastoma multiforme in elderly patients. Clinico-pathological remarks on our series of seven cases and critical review of the literature. Tumori 2008;94:40-51.  Back to cited text no. 17
    
18.
Kraft M, Lang F, Braunschweig R, Janzer RC. Parotid gland metastasis from glioblastoma multiforme: A case report and review of the literature. Eur Arch Otorhinolaryngol 2008;265:709-11.  Back to cited text no. 18
    
19.
Beauchesne P, Bernier V, Carnin C, Taillandier L, Djabri M, Martin L, et al. Prolonged survival for patients with newly diagnosed, inoperable glioblastoma with 3-times daily ultrafractionated radiation therapy. Neuro Oncol 2010;12:595-602.  Back to cited text no. 19
    
20.
Wong ET, Hess KR, Gleason MJ, Jaeckle KA, Kyritsis AP, Prados MD, et al. Outcomes and prognostic factors in recurrent glioma patients enrolled onto phase II clinical trials. J Clin Oncol 1999;17:2572-8.  Back to cited text no. 20
    
21.
Gaspar LE, Fisher BJ, Macdonald DR, LeBer DV, Halperin EC, Schold SC Jr., et al. Supratentorial malignant glioma: Patterns of recurrence and implications for external beam local treatment. Int J Radiat Oncol Biol Phys 1992;24:55-7.  Back to cited text no. 21
    
22.
Halperin EC, Burger PC, Bullard DE. The fallacy of the localized supratentorial malignant glioma. Int J Radiat Oncol Biol Phys 1988;15:505-9.  Back to cited text no. 22
    
23.
Lee SW, Fraass BA, Marsh LH, Herbort K, Gebarski SS, Martel MK, et al. Patterns of failure following high-dose 3-D conformal radiotherapy for high-grade astrocytomas: A quantitative dosimetric study. Int J Radiat Oncol Biol Phys 1999;43:79-88.  Back to cited text no. 23
    
24.
Choucair AK, Levin VA, Gutin PH, Davis RL, Silver P, Edwards MS, et al. Development of multiple lesions during radiation therapy and chemotherapy in patients with gliomas. J Neurosurg 1986;65:654-8.  Back to cited text no. 24
    
25.
Loeffler JS, Alexander E 3rd, Hochberg FH, Wen PY, Morris JH, Schoene WC, et al. Clinical patterns of failure following stereotactic interstitial irradiation for malignant gliomas. Int J Radiat Oncol Biol Phys 1990;19:1455-62.  Back to cited text no. 25
    
26.
Baumann F, Bjeljac M, Kollias SS, Baumert BG, Brandner S, Rousson V, et al. Combined thalidomide and temozolomide treatment in patients with glioblastoma multiforme. J Neurooncol 2004;67:191-200.  Back to cited text no. 26
    
27.
Hou LC, Veeravagu A, Hsu AR, Tse VC. Recurrent glioblastoma multiforme: A review of natural history and management options. Neurosurg Focus 2006;20:E5.  Back to cited text no. 27
    
28.
Shinya T, Akaki S, Ogata T, Sato S, Kanazawa S. Super scan in a patient with diffuse bone metastases from intracranial glioma. Clin Nucl Med 2007;32:481-3.  Back to cited text no. 28
    
29.
Manohar PR, Rather TA, Khan SH, Malik D. Skeletal metastases presenting as superscan on technetium 99m methylene diphosphonate whole body bone scintigraphy in different type of cancers: A 5-year retro-prospective study. World J Nucl Med 2017;16:39-44.  Back to cited text no. 29
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30.
Brenner AI, Koshy J, Morey J, Lin C, DiPoce J. The bone scan. Semin Nucl Med 2012;42:11-26.  Back to cited text no. 30
    
31.
Love C, Din AS, Tomas MB, Kalapparambath TP, Palestro CJ. Radionuclide bone imaging: An illustrative review. Radiographics 2003;23:341-58.  Back to cited text no. 31
    


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