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CASE REPORT
Year : 2010  |  Volume : 6  |  Issue : 3  |  Page : 336-338

Osteosarcoma of the mandible - second cancer in a case of Hodgkin's lymphoma post-chemotherapy


Department of Radiotherapy, Medical College Hospitals, 88 College Street, Kolkata - 700 073, West Bengal, India

Date of Web Publication29-Nov-2010

Correspondence Address:
Dodul Mondal
Doctor's Chummery Hostel, Room no - 13, 41, Eden Hospital Road, Kolkata - 700073, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.73349

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 > Abstract 

Osteosarcoma as a second cancer occurring in an adolescent male previously treated with only chemotherapy for Hodgkin's lymphoma is rare. A 14 year old male presented with gradual swelling of right jaw and numbness of lip and chin for last three months. He was treated with only chemotherapy for Hodgkin's lymphoma four years back. Incisional biopsy was taken from mandibular swelling, histopathology suggesting osteosarcoma. Right hemimandibulectomy and reconstruction with metallic implant were done along with excision of right parotid lymph node. Chemotherapy was started with cisplatin and doxorubicin regimen. Absence of radiation as a risk factor and very young age of the patient presenting with a second cancer made this case a rarity and hence the case is being reported.

Keywords: Chemotherapy, Hodgkin′s lymphoma, secondary osteosarcoma


How to cite this article:
De S, Ghosh S, Mondal D, Sur PK. Osteosarcoma of the mandible - second cancer in a case of Hodgkin's lymphoma post-chemotherapy. J Can Res Ther 2010;6:336-8

How to cite this URL:
De S, Ghosh S, Mondal D, Sur PK. Osteosarcoma of the mandible - second cancer in a case of Hodgkin's lymphoma post-chemotherapy. J Can Res Ther [serial online] 2010 [cited 2019 Sep 21];6:336-8. Available from: http://www.cancerjournal.net/text.asp?2010/6/3/336/73349


 > Introduction Top


Osteosarcoma of the jaw is a rare entity, comprising approximately 5% to 8% of all osteosarcomas. [1],[2] The most common histologic subtypes seen in the jaw region are chondroblastic and osteoblastic types, typically of low to intermediate grade. However, osteosarcomas of a higher grade are not infrequent in the jaw; they are often seen in cases that have occurred as second cancer due to radiation or other factors. [3] We report a case of osteoblastic osteosarcoma in a patient with history of Hodgkin's lymphoma treated with only chemotherapy four years back.


 > Case Report Top


A 10 year old boy presented with non tender swelling of the right neck gland in 2004. There were no other symptoms (particularly fever, night sweats, pain). Core biopsy of the neck node was done and histopathology showed mixed cellularity Hodgkin's lymphoma [Figure 1]. As a staging work up chest radiograph, complete blood count, ultrasonography of abdomen revealed no abnormality. Serum alkaline phosphatase was mildly elevated. Clinical diagnosis was Hodgkin's lymphoma, stage IA and he was treated with a regimen of six cycles of ABVD which ended on 15 th June 2005. He achieved complete remission after the end of second cycle of chemotherapy. No radiation was given to the involved field considering his response and age and favorable histology.
Figure 1: Histopathology slide showing features of mixed cellularity Hodgkins lymphoma (H and E, 10×)

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After a gap of four years, in June 2008, he presented with a three month history of a gradually increasing painful swelling on the right side of the jaw that was associated with numbness of the right lower lip and chin. There was no evidence of recurrence at previous site of malignancy. Orthopantomography (OPG) done on July 2008 showing a mass in right cheek involving the mandible [Figure 2]. Incisional biopsy was done from the jaw swelling. Histopathological report was suggestive of osteosarcoma (low grade; having 2-4 mitoses per high power field). Right hemimandibulectomy and reconstruction with titanium plate were done along with excision of right parotid lymph node on 18 th July 2008 [Figure 3]. On macroscopic examination, the operative specimen revealed expansion and destruction of bone measuring 10.5 × 6.5 × 6 cm. Cut surface was solid, firm, fleshy, greyish white in color having gritty feeling at places. Histopathology revealed stellate and spindle cells with cellular and nuclear pleomorphism, nuclear hyperchromatism, and 2-4 abnormal mitosis per field of microscope in collagenous stroma. There were scattered trabeculae of immature bone with osteoblastic rimming [Figure 4]. The final diagnosis was that of an osteoblastic osteosarcoma involving the mandible.
Figure 2: Pre-operative orthopantomography (OPG) showing an illdefined lytic lesion involving the right angle of mandible without any sclerosis or matrix mineralization

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Figure 3: Post operative lateral radiograph of the neck revealing the metallic mandibular implant

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Figure 4: Histopathological examination of the operative specimen showing stellate and spindle cells, with collagenous stroma having areas of osteoblastic foci, suggesting a diagnosis of osteosarcoma (H & E, 400×)

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Post operative recovery was good and he was put on chemotherapy (cisplatin and doxorubicin regimen). Till date, patient has received three cycles of chemotherapy and is asymptomatic with no signs or symptom of residual disease.


 > Discussion Top


Treatment of childhood cancers has become increasingly successful, with a current overall cure rate of more than 70%. [4],[5] Second malignant neoplasms, however, are observed in long-term survivors of childhood cancer, with a 15 year cumulative incidence of 3%-5%. [6],[7],[8],[9] Secondary cancers are the second leading cause of death, after recurrence, among five-year survivors of childhood cancer. [10] Radiation therapy for childhood tumors has been associated with the development of secondary sarcomas, [8],[9],[11] but the association between secondary sarcomas and other factors-e.g., chemotherapy exposure, age, or family history of cancer-have not been well studied. Development of secondary sarcoma was associated with previous radiation exposure, higher dose exposures of anthracyclines or alkylators, or a history of other secondary malignancies. The highest risks were observed among survivors of pediatric soft tissue and bone sarcomas, renal tumors, and Hodgkin's lymphoma. [10]

Our case is unique in the sense that patient developed osteosarcoma within four years of treatment of Hodgkin's lymphoma with only chemotherapy at an early age of 14 years. There is no history of being treated with radiation. The only risk factors in this case were previous treatment with anthracycline (doxorubicin-360mg), previous history of Hodgkin's disease. In the absence of radiation treatment, prior chemotherapy could be implicated as a causative factor in the development of osteosarcoma in our case. The patient was given chemotherapy with cisplatin and doxorubicin. Till date, he has received three cycles of chemotherapy and at present has no features of residual disease. Further more reconstruction with metallic implant created contraindication to treatment with radiation. This caused treatment dilemma as radiation could not be given.

 
 > References Top

1.Clark JL, Unni KK, Dahlin DC, Devine KD. Osteosarcoma of the jaw. Cancer 1983;51:2311-6.   Back to cited text no. 1
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2.Bertoni F, Dallera P, Bacchini P, Marchetti C, Campobassi A. The Istituto Rizzoli-Beretta experience with osteosarcoma of the jaw. Cancer 1991;68:1555-63.   Back to cited text no. 2
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3.Arlen M, Higinbotham NL, Huvos AG, Marcove RC, Miller T, Shah IC. Radiation-induced sarcoma of bone. Cancer 1971;28:1087-99.  Back to cited text no. 3
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4.Dreyer Z, Blatt J, Bleyer A. Late effects of childhood cancer and its treatment. In: Pizzo P, Poplack D, editors. Principles and practice of pediatric oncology. 4 th ed. Philadelphia: Lippincott Williams and Wilkins; 2002. p. 1431-62.  Back to cited text no. 4
    
5.Greenlee RT, Murray T, Bolden S, Wingo PA. Cancer statistics, 2000. CA Cancer J Clin 2000;50:7-33.  Back to cited text no. 5
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6.de Vathaire F, Hawkins M, Campbell S, Oberlin O, Raquin MA, Schlienger JY, et al. Second malignant neoplasms after a first cancer in childhood: Temporal pattern of risk according to type of treatment. Br J Cancer 1999;79:1884-93.  Back to cited text no. 6
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7.Olsen JH, Garwicz S, Hertz H, Jonmundsson G, Langmark F, Lanning M, et al. Second malignant neoplasms after cancer in childhood or adolescence: Nordic Society of Paediatric Haematology and Oncology Association of the Nordic Cancer Registries. BMJ 1993;307:1030-6.  Back to cited text no. 7
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8.Hawkins MM, Draper GJ, Kingston JE. Incidence of second primary tumours among childhood cancer survivors. Br J Cancer 1987;56:339-47.  Back to cited text no. 8
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9.Neglia JP, Friedman DL, Yasui Y, Mertens AC, Hammond S, Stovall M, et al. Second malignant neoplasms in five-year survivors of childhood cancer: Childhood cancer survivor study. J Natl Cancer Inst 2001;93:618-29.  Back to cited text no. 9
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10.Mertens AC, Yasui Y, Neglia JP, Potter JD, Nesbit ME Jr, Ruccione K, et al. Late mortality experience in five-year survivors of childhood and adolescent cancer: The Childhood Cancer Survivor Study. J Clin Oncol 2001;19:3163-72.  Back to cited text no. 10
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11.Meadows AT, Baum E, Fossati-Bellani F, Green D, Jenkin RD, Marsden B, et al. Second malignant neoplasms in children: An update from the Late Effects Study Group. J Clin Oncol 1985;3:532-8.  Back to cited text no. 11
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    Figures

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


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