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Table of Contents
LETTER TO EDITOR
Year : 2020  |  Volume : 3  |  Issue : 3  |  Page : 612-614

Primary intraosseous leiomyosarcoma of the clavicle secondary to radiation therapy


1 Department of Surgical Oncology, Cancer Institute (WIA), Chennai, Tamil Nadu, India
2 Department of Medical Oncology, Cancer Institute (WIA), Chennai, Tamil Nadu, India

Date of Submission22-Feb-2020
Date of Decision01-Apr-2020
Date of Acceptance02-Apr-2020
Date of Web Publication19-Sep-2020

Correspondence Address:
Arvind Krishnamurthy
Department of Surgical Oncology, Cancer Institute (WIA), 38, Sardar Patel Rd., Adyar, Chennai - 600 036, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/CRST.CRST_54_20

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How to cite this article:
Krishnamurthy A, Das A, Reddy R, Dhanushkodi M. Primary intraosseous leiomyosarcoma of the clavicle secondary to radiation therapy. Cancer Res Stat Treat 2020;3:612-4

How to cite this URL:
Krishnamurthy A, Das A, Reddy R, Dhanushkodi M. Primary intraosseous leiomyosarcoma of the clavicle secondary to radiation therapy. Cancer Res Stat Treat [serial online] 2020 [cited 2020 Oct 26];3:612-4. Available from: https://www.crstonline.com/text.asp?2020/3/3/612/295522



Postirradiation sarcoma is a rare but a potentially lethal complication following radiation therapy.[1] While there have been a few case reports of postirradiation leiomyosarcomas (LMS), a vast majority of them were reports of LMS originating from the soft tissues. To the best of our knowledge, there have been just two earlier reports of primary intraosseous LMS of the bone, originating within the field of radiation, secondary to prior radiation therapy.[2],[3] We report an additional case and briefly review the limited available literature.

A 30-year-old gentleman reported to our cancer center with a hard tumor of 3 months duration, overlying the right collar bone. He had a past history of carcinoma nasal cavity, cT4aN2bM0, 10 years back, and was treated then with concurrent chemoradiation (66 Gray of external-beam radiation therapy to the primary and bilateral neck along with three cycles of three weekly cisplatin 100 mg/m2). Clinical examination revealed a bony hard 8 cm × 7 cm, nontender mass arising from the medial end of the right clavicle [Figure 1]. A positron-emission tomography–computed tomography (CT) scan done as a part of the metastatic workup revealed an isolated tracer uptake (standardized uptake value: 16.25) in the medial half of the right clavicle with an associated soft-tissue component involving the right sternoclavicular joint and the adjacent muscles [Figure 2]a and b]. A core-needle biopsy from the mass lesion was consistent with a diagnosis of a high-grade malignant LMS. A CT angiography revealed the mass to be supplied by the branches of the subclavian vessels; in addition, there was compression but no infiltration of the subclavian vessels [Figure 3]a and [Figure 3]b. A diagnosis of primary intraosseous LMS secondary to radiation therapy was made.
Figure 1: Timeline of treatment

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Figure 2: (a and b) A positron-emission tomography–computed tomography scan revealed an isolated tracer uptake (standardized uptake value: 16.25) in the medial half of the right clavicle with an associated soft-tissue component involving the right sternoclavicular joint and the adjacent muscles

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Figure 3: (a and b) A computed tomography angiography revealed the mass to be supplied by the branches of the subclavian vessels; in addition, there was compression but no infiltration of the subclavian vessels

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The patient was taken up for upfront surgery which entailed a wide excision of the medial end of the right clavicle and the adjoining right first rib, the right sternoclavicular joint, and part of the overlying skin. The resultant defect was reconstructed using a prolene mesh; the adjacent right pectoralis major muscle was transposed over the mesh and a primary closure of the skin was achieved [Figure 4]a, [Figure 4]b, [Figure 4]c, [Figure 4]d. The final histopathology corroborated with the upfront biopsy report of primary intraosseous high-grade LMS, resected with clear margins. The patient was explained about the uncertain role of further chemotherapy and radiotherapy; he wished to remain on regular follow-up.
Figure 4: (a) Intraoperative photograph showing the tumor overlying the medial half of the right clavicle. (b) Intraoperative photograph showing the tumor to be in close proximity to the subclavian vessels. (c) Intraoperative photograph showing the mobilized right pectoralis major muscle over the tumor bed and the subclavian vessels. (d) The resected specimen photograph

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The criteria for postirradiation sarcoma as initially reported by Cahan and Woodard[1] are the occurrence of a second primary tumor within the field of radiation, a relatively long latent period, a histopathological diagnosis of a sarcoma, and an initial absence of a primary tumor either by microscopy or radiology. Primary intraosseous LMS is a rare sarcoma, accounting for just about 0.7% of all the primary malignant bony tumors.[4] Prior reports have suggested that the median time interval from the completion of external-beam radiotherapy to the detection of postirradiation sarcoma was around 13.2 years,[5] (range: 3.4–22.8 years). The postirradiation LMS in our patient appeared after a latent period of 10 years.

The recommended treatment of choice for primary intraosseous LMS, or for that matter any postirradiation sarcoma, is a radical surgical resection so as to attain clear margins.[6] The exact role of adjuvant chemotherapy and radiation therapy is unclear.[5],[7] The clinical behavior of bone LMS is generally aggressive, and most of the published studies on bone LMS have reported poor long-term prognosis. Our report thus highlights the need for clinicians to be aware of the possibility and implications of the unusual long-term complication of radiation-induced sarcomas.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Cahan WG, Woodard HQ. Sarcoma arising in irradiated bone; report of 11 cases. Cancer 1948;1:3-29.  Back to cited text no. 1
    
2.
Aoki T, Ozeki Y, Watanabe M, Tanaka S, Isaki H, Terahata S. Development of primary leiomyosarcoma of the sternum postirradiation: Report of a case. Surg Today 1998;28:1326-8.  Back to cited text no. 2
    
3.
Sedghizadeh PP, Angiero F, Allen CM, Kalmar JR, Rawal Y, Albright EA. Post-irradiation leiomyosarcoma of the maxilla: Report of a case in a patient with prior radiation treatment for retinoblastoma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;97:726-31.  Back to cited text no. 3
    
4.
Siegel RL, Miller KD, Jemal A. Cancer statistics, 2016. CA Cancer J Clin 2016;66:7-30.  Back to cited text no. 4
    
5.
Wiklund TA, Blomqvist CP, Räy J, Elomaa I, Rissanen P, Miettinen M. Postirradiation sarcoma. Cancer 1991;68:524-31.  Back to cited text no. 5
    
6.
Recine F, Bongiovanni A, Casadei R, Pieri F, Riva N, De Vita A, et al. Primary leiomyosarcoma of the bone: A case report and a review of the literature. Medicine (Baltimore) 2017;96:e8545.  Back to cited text no. 6
    
7.
Turkar S. Current treatment landscape and emerging management options for extremity sarcoma. Cancer Res Stat Treat 2018;1:121-38.  Back to cited text no. 7
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    Figures

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



 

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