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A Rare Case of Pleomorphic Sarcoma Arising in Burn Scar
Mohan J Algotar*, Yogesh S Puri**, Ashok Borisa**, Nikhil Agarkhedkar***
Abstract
Burn scar carcinoma is rare entity in its own. More commonly burn scar carcinoma is mainly Squamous cell carcinoma. Rarely following malignancies are reported in burn scar carcinoma in descending order of frequency; Basal cell carcinoma, Melanoma, Osteogenic Sarcoma, fibro sarcoma, adenocarcinoma and Liposarcoma. We present a case of Pleomorphic Sarcoma in a 20 year old male which presents very rarely in chronic burn scar in lower limb.
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Introduction
Squamous cell carcinoma is most common
histologic type of malignancy found and second most common neoplasm is Basal Cell Carcinoma (BCC).1,2 BCC allegedly occurs when the burn is more superficial and hair follicles and sebaceous glands are intact.3 Other neoplasm reported are Melanoma,2 Osteogenic sarcoma4, Fibro sarcoma and adenocarcinoma1, Liposarcoma3. Burn scar carcinoma is seen more commonly in males with M:F ratio as 3:12,5 found predominantly in adults with average age as 58 years with range from 18-84 years.6 It also has been reported that time from primary burn to the time of onset of carcinoma is inversely proportional to the age of patient at the time of burn injury7. The younger the patient at the time of burns the longer the time required for malignant transformation. We here present a case which has not yet reported in recent literature as a Pleomorphic Sarcoma arising in burn scar in very young male patient of 20 years of age with duration of 10 years in between burn trauma and appearance of malignancy.
Case Report
We present a case of 20 year old man who presented to us with non healing ulcer on right foot. Patient was suffering from recurrent ulceration in the same site. He had suffered from burn of both lower legs at the age of 10 years by hot boiling water. He had undergone Skin Grafting. After some time patient again complained of small ulcer which has progressed to occupy medial aspect of left lower leg in the burn scar, after that wound never healed. He presented to us with non healing ulcer bearing high suspicion of malignancy, we had taken edge biopsy of the ulcer, which turned out to be Pleomorphic Sarcoma. After that we had treated the patient with below knee operation, final histopathological diagnosis (Fig. 1) confirmed with earlier report of Pleomorphic Sarcoma with bone margins involved in malignancy.
Hence patient has been reoperated with above knee amputation.
After which patient regularly followed up for one year by which time patient had developed inguinal lymph node metastasis after which patient did not come for follow up. Thus this signifies importance of high degree of suspicion of rare malignancy, long term follow up of patient in these highly aggressive malignancies
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| Fig. 1a : Higher magnification. |
Fig. 1b : Lower magnification |
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Discussion
Pleomorphic Sarcoma is rare type of malignancy and treatment protocol for this malignancy are not established.8 Marjolin’s ulcer are malignancies that arise at site of chronic injury. Squamous cell carcinoma is found in 2% and BCC in 0.3% of all burn scars. Melanoma, adenoma, fibrosarcoma, liposarcoma and osteogenic sarcoma are all reported in Marjolin’s ulcer but rarely.9
The interval between injury and appearance of malignancy is reported to be 25-40 years; although interval as long as 70 years has been reported.1,10,11,13,14 But in our case interval between burns and malignancy is 10 years. Several investigators have observed; younger the patient more time interval for malignant change.9-11,15 But in our case patient is very young that is 20 years old. This may suggest that Pleomorphic sarcoma have arisen denovo from burn scar and not as complication of burn scar trauma. It is said that Marjolin’s ulcer has low metastasis rate. Explanation for the same given by Treves and Pack12 was relatively avascular connective tissue in these lesions serves as “Barrier against metastasis”. But in our case lymph node metastasis has come up within 6 months of above knee amputation. From western literature amputation, probably not superior to wide local excision for burn scar malignancy.16
But in our case there was early bone and soft tissue invasion and patient has to undergo above knee amputation for malignancy at ankle.
Regional lymph node dissection is indicated whenever nodes are palpable.10,16-18 Lymph node dissection in absence of palpable nodes is however controversial. Bostowick et al19 have recommended lymph node dissection for all patients with burn scar malignancy, because they found that these tumours recur locally in an aggressive fashion. Lifeso and Bull20 showed that histological grading can be helpful in deciding which patients will benefit from local nodal dissection. But in our case patient has not followed up properly.
In conclusion we should have a high degree of suspicion for early diagnosis and management of this high grade tumour and we should pursue our patients to have long term follow-up which may improve prognosis of such patients.
Acknowledgement
We would like to thank Dr. B. M. Subnis Head of the Department of Surgery, Grant Medical College and J. J. Group of Hospitals for granting us permission to publish this case report.
References
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2. Lawrance REA. Carcinoma arising in burn scar. Surg Gynecol Obstet 1952;95:579-88.
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5. Sirsat MV, Shrikhande SS. Histochemical studies on squamous cell carcinoma of the skin arising in burn scars with special reference to histogenesis. Indian J Cancer 1967; 3 : 157-69.
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12. Fordyce JA. Malignant disease of scars and ulcers- Marjolin’s ulcer. Surgery: Its Principles and Practise. Keen WW(ed). Philadelphia, WB Saunders Co. 1911; Vol. 2 : 631-2.
13. Ames FC, Hickey RC. Squamous cell carcinoma of the skin of the extremities. Int Adv Surg Oncol 1980; 3 : 179-99.
14. Luce EA. Malignant epithelial tumours of the lower extremity. Part I: basal cell carcinoma. Contemp Ortop 1986; 12 : 31-9.
15. Lawrence EA. Carcinoma arising in the scars of thrmal burns: with special reference to the influence of the ageat burn on the lenghth of induction period. Surg Gynecol Obstet 1952; 95 : 579-88.
16. Barr LH, Menard JW. Marjoli’s ulcer. LSU experience. Cancer 1983; 52 : 173-5.
17. Berkwits L, Yarkony GM, Lewis V. Marjolin’s ulcer complicating pressure ulcer : case report and literature review. Arch phys Med rehabil 1986; 67 : 831-3.
18. Novick M, Gard DA, Hardy SB, et al. Burn Scar carcinoma: a review and analysis of 46 cases. J Truma 1977; 17 : 809-17.
19. Bostowick J III, Pedergrast WJ Jr, Vesconez LO. Marjolin’s ulcer: an immunologically privileged tomor? Plast Reconstr Surg 1976; 57 : 66-9.
20. Lifeso RM, Bull CA. squamous cell carcinoma of the extremities. Cancer 1985; 55 : 2862-7.
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OFF-PUMP CABG MAY BE BETTER THAN CONVENTIONAL CABG
Off-pump coronary artery bypass grafting (CABG) has similar rates of graft patency to conventional bypass surgery using cardiopulmonary bypass. Al-Ruzzeh and colleagues looked at 168 patients who needed primary isolated CABG and randomised them to conventional or off-pump technique. The authors evaluated graft patency after three months and found that in the conventional group angiographic patency was 92.7% and in the off-pump group it was 92.1%. However, patients in the off-pump group needed a shorter stay in hospital and their neurocognitive function was better preserved at six weeks and six months.
BMJ, 2006; 332 : 1365.
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