Journal of Cancer Research and Therapeutics Medknow Publications on behalf of the Association of Radiation Oncologists of India (AROI) ISSN: 0973-1482 EISSN: 1998-4138 Vol. 6, Num. 3, 2010, pp. 336-338

Journal of Cancer Research and Therapeutics, Vol. 6, No. 3, July-September, 2010, pp. 336-338

Case Report

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

Sumitava De, Suparna Ghosh, Dodul Mondal, Prabir K Sur

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

Correspondence Address:Dodul Mondal, Doctor's Chummery Hostel, Room no - 13, 41, Eden Hospital Road, Kolkata - 700073, West Bengal, India, dodulmondal@gmail.com

Code Number: cr10079

PMID: 21119269

DOI: 10.4103/0973-1482.73349

Abstract

Keywords: Chemotherapy, Hodgkin′s lymphoma, secondary osteosarcoma

Introduction

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

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.

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.

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

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

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