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CASE REPORT
Year : 2016  |  Volume : 15  |  Issue : 1  |  Page : 50-52

The complimentary role of Methoxy-Isobutyl-Isonitrile and hand-held gamma probe in adamantinoma


1 Department of Nuclear Medicine, Umhlanga Molecular Imaging and Therapy, Centre of Excellence, Umhlanga, South Africa
2 Department of Medical Imaging and Clinical Oncology, Stellenbosch University, and Tygerberg Hospital, South Africa

Date of Web Publication18-Dec-2015

Correspondence Address:
Masha Maharaj
Department of Nuclear Medicine, Umhlanga Molecular Imaging and Therapy, Centre of Excellence, Umhlanga, 4319
South Africa
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DOI: 10.4103/1450-1147.167600

PMID: 26912979

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   Abstract 

Adamantinoma is a rare locally aggressive osteolytic tumor that is found 90% of the time in the diaphysis of the tibia with the remaining lesions found in the fibula and long tubular bones. A case of adamantinoma of the tibia is presented. The added value of nuclear medicine investigations in the workup of this patient is described. A three-phase whole body 99mTc-methylene diphosphonate bone and a whole body 99mTc-methoxy-isobutyl-isonitrile scans were complimentary in the demarcation of viable bone tumor and the assessment of the remainder of the bone and soft tissue to exclude other sites. Intra-operative assistance with a hand-held gamma probe, guided the biopsy of the most metabolically active tumor tissue. Histology revealed a biphasic tumor composed of epithelial and fibrous components, in keeping with an adamantinoma.

Keywords: Adamantinoma, bone scan, gamma probe, methoxy-isobutyl-isonitrile, tumor


How to cite this article:
Maharaj M, Korowlay N, Ellmann. The complimentary role of Methoxy-Isobutyl-Isonitrile and hand-held gamma probe in adamantinoma. World J Nucl Med 2016;15:50-2

How to cite this URL:
Maharaj M, Korowlay N, Ellmann. The complimentary role of Methoxy-Isobutyl-Isonitrile and hand-held gamma probe in adamantinoma. World J Nucl Med [serial online] 2016 [cited 2019 Dec 6];15:50-2. Available from: http://www.wjnm.org/text.asp?2016/15/1/50/167600


   Interesting Image Top


A 28-year-old male patient presented with 3 months history of pain and swelling over the anterior aspect of the left tibia.

Planar X-rays revealed at least one section of confluent lytic areas with an associated pathological fracture, surrounded by cortical thickening and demarcated superiorly by an area of irregular sclerosis [Figure 1]a,[Figure 1]b,[Figure 1]c.
Figure 1:(a) Planar X-ray of left tibia, (b) Magnetic resonance imaging transaxial cut of proximal left tibia, (c) Magnetic resonance imaging left tibia

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Magnetic resonance imaging demonstrated a lobulated lesion involving the midshaft of the left tibia, measuring ± 9 cm in total length. Cartilaginous features in the proximal aspect of the lesion were confirmed by areas of irregular hyperintensity. An irregular hyperintense component in relation to the lytic area and the pathological fracture was observed suggesting extraosseous extension of tumor tissue causing overlying displacement of soft tissue. The radiological features were that of an aggressive Adamantinoma.

The patient was referred to nuclear medicine to further evaluate the extent of the primary lesion and to determine other sites of possible metastases.99m Tc-methylene diphosphonate (99m Tc-MDP) whole body blood pool and 3 h delayed whole body [Figure 2]a bone scan were done. There was a localized area of increased vascularity and inhomogeneous concentration of radiopharmaceutical in the left proximal tibia. Intense localized osteoblastic activity was seen in the superolateral and anterior aspect of the lesion corresponding to the fracture site, a relatively photopenic defect was seen medial and adjacent to this area with a rim of low-grade increased osteoblastic activity. The remainder of the bone scan was normal. A 99m Tc-methoxy-isobutyl-isonitrile (99m Tc-MIBI) whole body scan [Figure 2]b was performed 2 days after the bone scan to determine the extent of the metabolically active component of the tumor. It showed avid uptake corresponding to the photopenic area on the bone scan. The rest of the scan was normal.
Figure 2:(a) Whole body blood pool and whole body delayed images, (b) Whole body bone scan versus whole body methoxy-isobutyl-isonitrile scan

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Directly after the MIBI study the patient was transferred to theater for intraoperative localization, biopsy of the metabolically active tumor tissue and excision of the lesion. This was aided by a hand-held gamma probe (World of Medicine Gamma Finder) [Figure 3]. Multiple fragments of tissue were taken for histology; the largest was 17 × 15 × 5 mm. Counts of more than 20% of background activity were considered as significant. The patient underwent enblock excision of the proximal tibia with preservation of the growth plate. The defect was filled by use of a bone transplant technique and an external fixation was applied.
Figure 3:Largest tissue fragment taken

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Histology revealed a biphasic tumor with epithelial and fibrous components. The epithelial component displayed three different patterns, basaloid [Figure 4]a, fascicular [Figure 4]b, and tubular [Figure 4]c. The fibrous component did not contain features of fibro-osseous dysplasia or osteofibrous dysplasia-like areas. Immunohistochemistry for pancytokeratin [MNF 116] [Figure 4]d revealed a strong expression in the epithelial component and negative in stromal component.
Figure 4:Histology revealed biphasic tumor

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A week post total excision of the tumor a repeat MIBI whole body scan was performed to evaluate for residual active tumor. This scan showed no abnormal uptake [Figure 5]. It was confirmed on histology that the tumor was removed with a clear margin. Postoperatively, the patient did well and long-term follow-up has been planned.
Figure 5: Whole body methoxy-isobutyl-isonitrile scan postsurgery

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   Discussion Top


Adamantinoma is a rare locally aggressive osteolytic tumor, found 90% of the time in the diaphysis of the tibia with remaining lesions found in the fibula and long tubular bones. Adamantinoma typically ranges from 3 to 15 cm in size, and metastases occur in approximately 15-20% of patients. Diagnosis is histological. The tumor is known to have a dominant lytic component. This tumor is insensitive to radiation and chemotherapy.[1],[2]

The accurate assessment of musculoskeletal tumors may be unreliable due to pathological fractures, hemorrhage, calcification, and inflammation on a bone scan. In 1990, Delmon-Moingeon et al., described Sestamibi as an in vivo tumor imaging agent.[3] Better delineation of tumor outlines and cellular activity is an advantage of MIBI scintigraphy, which is helpful in the evaluation of musculoskeletal tumors. No relationship could be found between tumor MIBI uptake and 99m Tc-MDP osteoblastic activity in musculoskeletal tumor's.[4] Although the exact uptake mechanisms of MIBI into the myocardial and tumor cells are not well-understood, it is postulated to be related to blood flow, blood residence time, and the cellular uptake due to passive influx of the lipophilic cation, driven by the plasma and mitochondrial membrane potentials generated in living cells. Elevated potentials are directly related to metabolic state.[5] MIBI is physiologically taken up by the salivary glands, thyroid, heart, liver, spleen, and skeletal muscle. There is physiological hepatobiliary and renal clearance. MIBI has the advantage of being readily available, easy to prepare and cost-effective.


   Conclusion Top


As demonstrated in this case report, molecular imaging techniques can provide complementary information to anatomical imaging techniques in staging, therapeutic monitoring and surveillance as well as additional applications that include the localization of a malignant lesion using a hand held gamma probe. The best test will be defined by local cost, availability, and expertise within a given modality, in addition to patient specific circumstances.

 
   References Top

1.
Pieterse AS, Smith PS, McClure J. Adamantinoma of long bones: Clinical, pathological and ultrastructural features. J Clin Pathol 1982;35:780-6.  Back to cited text no. 1
[PUBMED]    
2.
Jain D, Jain VK, Vasishta RK, Ranjan P, Kumar Y. Adamantinoma: A clinicopathological review and update. Diagn Pathol 2008;3:8.  Back to cited text no. 2
    
3.
Delmon-Moingeon LI, Piwnica-Worms D, Van den Abbeele AD, Holman BL, Davison A, Jones AG. Uptake of the cation hexakis (2-methoxyisobutylisonitrile)-technetium-99m by human carcinoma cell lines in vitro. Cancer Res 1990;50:2198-202.  Back to cited text no. 3
    
4.
Ozcan Z, Burak Z, Erinç R, Dirlik A, Başdemir G, Sabah D, et al. Correlation of 99mTc-sestamibi uptake with blood-pool and osseous phase 99mTc-MDP uptake in malignant bone and soft-tissue tumours. Nucl Med Commun 2001;22:679-83.  Back to cited text no. 4
    
5.
Moretti JL, Hauet N, Caglar M, Rebillard O, Burak Z. To use MIBI or not to use MIBI? That is the question when assessing tumour cells. Eur J Nucl Med Mol Imaging 2005;32:836-42.  Back to cited text no. 5
    


    Figures

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



 

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