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CORRESPONDENCE
Year : 2013  |  Volume : 9  |  Issue : 2  |  Page : 311-313

A case report of osteogenic sarcoma with leprosy


1 Department of Medical Oncology, Tata Memorial Hospital, Mumbai, India
2 Department of Microbiology, Tata Memorial Hospital, Mumbai, India

Date of Web Publication13-Jun-2013

Correspondence Address:
Jyoti Bajpai
Department of Medical Oncology, Tata Memorial Hospital, Mumbai
India
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Source of Support: Nuclear Science and Technology Research Institute (NSTRI), Conflict of Interest: None


DOI: 10.4103/0973-1482.113403

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 > Abstract 

This is a rare case report of osteosarcoma with lepromatous leprosy. A 15 year old male patient presented with swelling around the right knee joint. Imaging and biopsy were consistent with osteosarcoma. After his first cycle of adjuvant chemotherapy (ACT), the patient developed fever, erythematous nodules, perichondritis of ear lobe, and thickened nerves. His slit-skin smear examination showed acid-fast bacilli in clumps, and a diagnosis of multibacillary leprosy was made. He was treated with anti-leprosy medications with steroids, and once his condition stabilized, his ACT was continued. On follow-up, his skin lesions completely recovered.

Keywords: Osteosarcoma, leprosy, chemotherapy


How to cite this article:
Bajpai J, Shetty N, Gupta A, Samar A, Kelkar R. A case report of osteogenic sarcoma with leprosy. J Can Res Ther 2013;9:311-3

How to cite this URL:
Bajpai J, Shetty N, Gupta A, Samar A, Kelkar R. A case report of osteogenic sarcoma with leprosy. J Can Res Ther [serial online] 2013 [cited 2019 Nov 17];9:311-3. Available from: http://www.cancerjournal.net/text.asp?2013/9/2/311/113403


 > Introduction Top


This is a case report of osteogenic sarcoma (OGS) with leprosy. In literature, there have been reports of leprosy with malignancies of alimentary tract, lung cancer, [1] squamous cell carcinoma of trophic ulcer, [2] lymphoma, [3] and leukemia. [4] To the best of our knowledge, there has been no case report on the association of osteosarcoma with leprosy.


 > Case Report Top


A 15 year old boy presented to our center with pain and swelling for 2 months duration around the right knee joint. On examination, he had a 19 × 6cm swelling above his right knee joint, which was hard and non-tender. His X-ray showed a lytic and sclerotic lesion in the distal end of right femur with Codman's triangle and new bone formation, and Magnetic Resonance Imaging (MRI) knee [Figure 1] showed altered signal in distal metaphysis and two-third of diaphysis of the right femur, measuring 19 × 5.3 × 5.7 cm in superoinferior, anteroposterior, and transverse dimensions, with a large periosseous soft tissue component. His Computed Tomography (CT) thorax was normal and bone scan showed increased uptake only at the primary site. Biopsy from the lesion was suggestive of high-grade osteosarcoma. The patient was started on neoadjuvant chemotherapy (NACT) with cisplatin (120 mg/m 2 ), adriamycin (75 mg/m 2 ), and ifosfamide (9 g/m 2 ), and after 3 cycles of NACT, he underwent above-knee amputation, the histopathology of which showed scanty residual viable osteosarcoma and 95% necrosis. Patient was then started on adjuvant chemotherapy (ACT).
Figure 1: MRI femur showing altered signal in distal metaphysis andtwo-third of diaphysis of right femur with a large periosseous soft tissue component.

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Patient developed high-grade fever on the 16 th day of ACT. Two days later, he developed a maculopapular rash over his extremities [Figure 2]. His blood investigations showed hemoglobin 10.8 g/dl and total leukocyte count of 15,000/mm 3 with 90% neutrophils. Two days later, he developed multiple erythematous nodules over bilateral upper limbs on the flexor and extensor part up to mid arm and on the dorsum of feet up to mid leg. We had a differential diagnosis of viral exanthem, drug rash, erythema nodosum, or a form of paraneoplastic feature, with this clinical picture. Examination also revealed bilateral thickened and tender ulnar nerves with madarosis. Four days later, the rash progressed to his face and trunk and he also had perichondritis of the ear lobe. In view of erythematous nodules, thickened nerves, madarosis, and perichondritis in an immunocompromised individual, a suspicion of leprosy was made and we proceeded with a slit-skin smear examination. Meanwhile, the patient was started on oral steroids after which his fever, rash, and perichondritis subsided [Figure 3]. His slit-skin smear showed acid-fast bacilli (AFB) [Figure 4] arranged in clumps , and therefore the diagnosis of leprosy was confirmed.
Figure 2: Maculopapular rash in upper limbs

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Figure 3: Resolution of rash after anti-leprosy medication

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Figure 4: Arrow showing acid-fast bacilli

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He was started on dapsone, rifampicin, and clofazimine as treatment for multibacillary leprosy. His steroids were tapered and stopped. Throughout this period, we had to withhold his chemotherapy and his next cycle was started once the patient was better after a delay of 26 days. The patient has received anti-leprosy medications for 1 month so far and he has been asked to continue the same. He has responded clinically by becoming afebrile and his maculopapular rash over the face and trunk has resolved completely. His earlobe perichondritis also subsided completely. The patient is now being continued on anti-leprosy medication along with ACT and is on regular follow-up at our center.


 > Discussion Top


Leprosy is an infectious disease which has been eradicated in some countries, but cases are still being reported from other parts. In the beginning of this decade, 90% of the cases were reported in just 11 countries, led by India and Brazil. [5] OGS is the commonest primary bone tumor. [6] Since both leprosy and cancer in general are immunocompromised states, there has always been a debate regarding whether there is increased incidence of leprosy in cancer patients. On the corollary, studies have also been done to assess whether there has been any increase in the incidence of malignancy in leprosy patients. Here, we report a case of lepromatous leprosy in a cancer patient who is suffering from osteosarcoma and is on ACT.

The association of leprosy with various visceral and lymphoreticular malignancies is known. [7] In a series of 252 autopsies done in leprosy patients, the incidence of malignant tumor was 33.7% (85 cases), among which carcinoma of the alimentary system was the most common. Of the nonepithelial malignancies, malignant lymphoma is the most common association, and these include Hodgkin's lymphoma and mycoses fungoides. [3],[9] Patients with lepromatous leprosy also have depressed cellular immunity; however, a study of 195 autopsied subjects with leprosy did not reveal an increase in the occurrence of cancer. Thirty-three of the 195 subjects with leprosy or 16.9% died of cancer, which was comparable to an age-matched group of individuals. [8]

A rare association of leprosy with peripheral T-cell lymphoma in an 85-year-old male has been reported by Sethuraman et al.[3] In their patient, slit-skin smear showed bacilli and the skin biopsy was suggestive of peripheral T-cell lymphoma. Sudhakar Rao et al. reported a rare case of leprosy in a chronic lymphocytic leukemia (CLL) patient. [4] In both the patients, there was no nerve thickening; however, in our patient, it was present. In lepromatous leprosy, skin lesions are more pronounced, and whether malignancy due to immunosuppression further worsens it is not known. In both the above cases, skin biopsy was done; but in our patient, it was not done because soon after starting anti-leprosy medication and steroids, the patient showed significant improvement.

A strong humoral response, but relatively absent cell-mediated immunity results in lepromatous leprosy with widespread skin lesions and high bacterial loads. As per a study, deaths from malignant neoplasms were higher than expected among patients with lepromatous leprosy for both sexes (49 observed vs. 44.02 expected), whereas they were lower than expected among patients with tuberculoid leprosy (35 observed vs. 36.83 expected); however, the differences were not statistically significant. Mortalities from cancers of the cervix and the esophagus among females with lepromatous leprosy were significantly higher. [8]


 > Conclusion Top


is a rare case of leprosy in osteosarcoma. The occurrence of leprosy in our patient may be coincidental, but to the best of our knowledge, there are no reports of leprosy in osteosarcoma. Through our patient, we also like to highlight therapeutic issues in cancer patients with leprosy. In our patient, chemotherapy was delayed for 26 days as the patient was unstable due to fever and rash. Since osteosarcoma is a potentially curable malignancy, therapeutic delay might affect the prognosis adversely. It needs to be answered whether the drugs used in chemotherapy for osteosarcoma are so immunosuppressive that might have predisposed our patient to mycobacterial infection.

 
 > References Top

1.Purtilo DT, Pangi C. Incidence of cancer in patients with leprosy. Cancer 1975;35:1259-61.  Back to cited text no. 1
    
2.Swamy S, Durai V, Oommen PK, Rao KS. Cauliflower growth in trophic ulcer of leprosy-a 10 year study. Indian J Lepr 1986;58:48-53.  Back to cited text no. 2
    
3.Sethuraman G, Serisha, Srinivas CR, Byravarathinam S, Raman ML, Smiles L, et al. Leprosy with peripheral T-cell lymphoma: a rare association. Int J Lepr Other Mycobact Dis 2001;69:119-22.  Back to cited text no. 3
    
4.Sudhakar R, Jayashree P, Anjad BS, Varna N, Kotbagi HB. Leprosy in chronic lymphocytic leukemia: A rare case report. J Clin Diagn Res 2011;5:1106-8.  Back to cited text no. 4
    
5.Leprosy-Global situation. Wkly Epidemiol Rec 2000;75:226-31.  Back to cited text no. 5
    
6.Mirabello L, Troisi RJ, Savage SA. Osteosarcoma incidence and survival rates from 1973 to 2004: data from the Surveillance, Epidemiology, and End Results Program. Cancer 2009;115:1531-43.  Back to cited text no. 6
    
7.Furuta M, Obara A, Ishida Y, Harada N, Ozaki M. Leprosy and malignancy: autopsy findings of 252 leprosy patients. Int J Lepr Other Mycobact Dis 1990;58:697-703.  Back to cited text no. 7
    
8.Tokudome S, Kono S, Ikeda M, Kuratsune M, Kumamaru S. Cancer and other causes of death among leprosy patients. J Natl Cancer Inst 1981;67:285-9.  Back to cited text no. 8
    
9.Grossman D, Rapini RP, Osborne B, Duvic M. Emergence of leprosy in a patient with mycosis fungoides. J Am Acad Dermatol 1994;30:313-5.  Back to cited text no. 9
    


    Figures

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



 

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