Case Study

A case report of chondroblastic osteosarcoma following treatment for Hodgkin lymphoma

Tenille A. Abrahams, Farah Rahman, Sharlene Parasnath

Received: 25 July 2024; Accepted: 15 Oct. 2024; Published: 30 Nov. 2024

Copyright: © 2024. The Author(s). Licensee: AOSIS.
This is an Open Access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Patients diagnosed with Hodgkin lymphoma (HL) have improved survival because of the early use of effective therapies and positron emission tomography (PET) – adapted strategies. However, long-term observational studies have shown that HL survivors have an increased risk of secondary cancers and other complications, which include cardiovascular disease, infertility and endocrinopathies. Therefore, long-term follow-up and screening of HL survivors is important.

Contribution: We report a case of a 22-year-old female with chondroblastic osteosarcoma following treatment of HL. We have devised a simple screening algorithm to ensure comprehensive long-term surveillance of HL survivors.

Keywords: Hodgkin lymphoma; osteosarcoma; radiotherapy; mandible mass; secondary malignancy.

Introduction

Osteosarcoma is one of the recognised secondary malignancies following the treatment of Hodgkin lymphoma (HL).¹ Chemotherapy used in the management of HL, especially the alkylating agents, have long been associated with the development of secondary malignancies. The use of involved field radiotherapy (IFRT) confers an additional risk of oncogenesis.² This case highlights the importance of ongoing surveillance and screening of patients for treatment-related side effects, including secondary malignancies.

Case presentation

A 22-year-old female was treated for HL with six cycles of (doxorubicin [alternative name is adriamycin], bleomycin, vinblastine and dacarbazine) ABVD. An interim positron emission tomography (PET) scan confirmed residual disease. The patient continued further treatment at a private institution where she received one session of radiotherapy to the left supraclavicular area, thereafter, obtaining a complete metabolic remission. Unfortunately, no records of the radiotherapy or chemotherapy received in the private sector were available, and the patient was unable to recall the details of her treatment. She re-presented 7 years later with an extensive left-sided mandibular lesion (Figure 1). Clinical examination revealed a hard, large, fungating tumour with buccal and lingual expansion, significantly impeding jaw mobility. Systemic examination revealed that the disease appeared to be localised to the jaw. Positron emission tomography scan showed metabolically active disease in the left facial mass with evidence of left cervical lymph node involvement and no evidence of distant metastases.

FIGURE 1: New left mandibular lesion. (a) Lateral and (b) anterior views of jaw mass. (c) Coronal view of positron emission tomography scan demonstrating pathological uptake (SUVmax 15.98).

Initially, an incision biopsy of the mandibular mass showed features in keeping with a benign fibro-osseous lesion, favouring an ossifying fibroma. Clinically, the mass was noticed to be increasing in size, prompting histological review. In conjunction with radiological findings, the diagnosis was amended to a chondroblastic osteosarcoma. Based on these findings, the patient was treated surgically with resection of the mandible and placement of a reconstruction plate. Following excision of the mass, the patient was referred to oncology for adjuvant chemotherapy using cisplatin and doxorubicin in conjunction with IFRT and is undergoing treatment at the time of writing this report.

This case illustrates the importance of long-term follow-up and screening of HL patients for complications post-treatment.

Discussion

Advances in the management of HL, including the use of PET – adapted strategies and effective chemo-immunotherapy, have improved long-term survival in HL patients. However, long-term observational studies have shown that HL survivors have an increased risk of secondary malignancies and other complications, including cardiovascular disease, infertility and endocrinopathies.³ There is a growing number of HL survivors, highlighting long-term therapy-related complications.⁴

Hodgkin lymphoma is one of the most curable malignancies.⁴ The use of radiotherapy, alkylating chemotherapy agents and immunosuppressive agents confer additional carcinogenic risk. Environmental, lifestyle and genetic factors are potential determinants for the development of secondary malignancies; therefore, prior radiotherapy exposure as a sole risk factor remains disputable.⁵ Immunosuppressed individuals may lack immune surveillance and may also possess germline mutations, further increasing their risk of secondary malignancies.⁶ An individualised treatment approach with aggressive ‘upfront disease control’, early de-escalation of more toxic chemotherapies and use of newer radiotherapy techniques may help decrease the risk of secondary malignancies.⁷ Treatment modalities for these secondary malignancies may sometimes be limited because of patient factors or previous treatment exposure, making early detection of these malignancies extremely important.⁸

The present case highlights the need for a holistic management approach with emphasis on patient education in order to ensure early healthcare seeking behaviour, especially within the first 5 years following complete remission.⁹ According to the European Society for Medical Oncology (ESMO), a detailed history as well as physical examination should be performed at every visit along with full blood count, blood chemistry testing and examination guided additional investigation.¹⁰

We have devised a screening algorithm based on the National Comprehensive Cancer Network (NCCN)¹¹ and Children’s Oncology Group (COG)¹² recommendations to ensure appropriate and timeous screening of HL survivors in resource-constrained settings (Figure 2).^10,11,12 This simple algorithm can be introduced at all levels of healthcare to ensure early recognition of ‘red flag’ symptoms (Figure 2), prompting early referral to the necessary healthcare level. Although secondary malignancies are the focus of this discussion, other treatment-related complications should actively be sought and treated timeously by a multidisciplinary team.¹³

FIGURE 2: Algorithm for long-term screening of patients with Hodgkin lymphoma.

Conclusion

This case highlights the importance of adequate long-term follow-up of HL survivors. The need for early, appropriate and thorough investigation of complications is of paramount importance in reducing further morbidity and mortality. The use of simple screening algorithms, which may be utilised in non-specialist centres, may assist in early detection and prompt referral for management of long-term complications.

Acknowledgements

The authors would like to thank V. Pillay, N. Gama, J. Karunakaram and the Maxillo-facial and Oral surgery Department from the Inkosi Albert Luthuli Central Hospital, Durban, KwaZulu-Natal, South Africa. The authors would also like to acknowledge A. Madaree from Lancet Laboratories, Durban, KwaZulu-Natal and Bungane from the National Health Laboratory Services, Durban, KwaZulu-Natal.

The authors declare that they have no financial or personal relationships that may have inappropriately influenced them in writing this article.

T.A.A., F.R. and S.P. contributed equally to this work.

Ethical clearance to conduct this study was obtained from Biomedical Research Ethics Committee, Management of Inkosi Albert Luthuli Central (reference no. EXM003/2024).

This research received no special grant from any funding agency in the public, commercial or non-profit sectors.

Data sharing is not applicable to this article as no new data were created or analysed during this study.

The views and opinions expressed in this article are those of the authors and are the product of professional research. They do not necessarily reflect the official policy or position of any affiliated institution, funder, agency or that of the publisher. The authors are responsible for this article’s results, findings and content.

References