Chondrosarcoma Presenting in a Rheumatoid Arthritic Foot

Brian Richman, DPM,* Gregg Young, DPM, **
Graydon Harker, MD ***

Abstract: The authors present a case of chondrosarcoma involving the left foot of a 71-year-old patient with long-standing rheumatoid arthritis.  Chondrosarcoma is a malignant tumor that rarely occurs in the foot.  The average duration of symptoms is usually 15 months, with pain and swelling being the most common. ( 1 ) In this case, the patient’s tumor was locally excised, but metastatic disease developed concurrently.  The tumor was poorly responsive to chemotherapy and, despite a temporary response to radiation therapy, it eventually let to the patient’s death. 
Key words:
cartilage, sarcoma.
 

            Bone tumors represent less then 1% of all tumors diagnosed in the United States. ( 2 ) It is estimated that 1-2% of these tumors arise in the foot. ( 3 )  Benign neoplasms outnumber malignant neoplasms by approximately four to one. ( 4 ) Chondrosarcoma is a malignant tumor involving both cartilage and bone.  These tumors represent the third most common primary malignant tumors of bone and are rarely located in the foot. ( 5 & 6 )  Over a 12 year period in Japan, only 4 cases of chondrosarcoma involving the foot were noted in a review of 2,658 malignant bone tumors. ( 7 )  Berlin also noted that chondrosarcomas rarely arise on the small bones of the feet and are more frequently seen involving pelvic bones and the femur. ( 8 )

             The incidence of chondrosarcomas is less than 3% in the feet and is not well understood.  Chondrosarcomas are more common in males than females, with a 3:1 ratio.  Chondrosarcomas of the bones of the hands and feet are rare, and the criteria for differentiating these lesions from the relatively common chondromas of the bones of the hands and feet are not well delineated. ( 9  ) Clinical features include dull pain of several years’ duration, local swelling without inflammation, and disability of a contiguous joint.  The tumor typically appears as a gradually enlarging, painful mass.( 10 )  The mortality rate is unknown in chondrosarcomas in the feet because of the rarity of the disease.  The histologic grade, location of the tumor, and adequacy of surgical excision have a significant effect of survival.  Recurrence of the tumor is highly suggestive of possible malignancy.  Prognosis for survival in chondrosarcoma of the foot appears to be better than for proximally located lesions. ( 10 )  Treatment consists of chemotherapy, radiation, surgical excision, and amputation of the affected extremity.  Early diagnosis and proper medical treatment while the tumor is localized is the greatest chance for survival.

            Rheumatoid arthritis is a systemic disease primarily affecting the joints, whit upper and lower extremity manifestations.  Swelling, pain and limitation of motion are everyday problems to the person living with this disease.  It is marked by inflammatory changed in the synovial membranes and articular structures.  Contracture, deviation, and subluxation of the joint in the hands and feet are common occurrences in the rheumatoid patient.  Painful rheumatoid nodules are often present in the upper and lower extremities.

            We present a case of a man who developed a myxoid chondrosarcoma involving the dorsal left foot in the setting of long-standing rheumatoid arthritis.

 

A Case Report

             A 71-year-old man was first seen in consultation by the Medical Oncology Service at the Salt Lake City Veterans Affairs Medical Center in December 1992.  The patient had a past medical history of rheumatoid arthritis dating to 1985, for which he had received multimodality medical therapy, including low-dose weekly methotrexate.  Several upper and lower extremity subcutaneous nodules had been discovered during that time period that were consistent with rheumatoid nodules. Recently, however, a nodule that had been present on the dorsal left foot for at least 1 year rapidly increased in size.  Evidence of skin breakdown in that area led to resection of the nodule, which had the gross histologic appearance of a rheumatoid nodule.  Within 2 weeks of the resection, however, the nodule had regrown to its original size.  At that time solitary erythematous subcutaneous nodules were also noted bilaterally on the anterior thighs.  The foot lesion was re-excised, and both thigh nodules were biopsied.  Histologic examination of the nodules revealed a high-grade tumor with areas containing predominantly myxoid or spindle components adjacent to each other.  Tumor necrosis was noted, as were a number of mitotic figures.  The features of the tumor were most consistent with a high-grade myxoid chondrosarcoma.

             On physical examination the patient was noted to have bilateral 2 cm x 3 cm erythematous anterior thigh masses and a 3-cm ulcer on the dorsal left foot, with central nodularity.  Further staging revealed the presence of bilateral lung nodules.  The patient was started on combination chemotherapy consisting of mesna, doxorubicin, ifosfamide, and dacarbazine, ( 11) and after one cycle her had slight improvement in the thigh nodules.  However, the foot mass grew rapidly despite the chemotherapy.  The thigh and foot lesions were then treated with external beam photon irradiation, which led to the rapid regressing and near-complete resolution of all three lesions for approximately 8 months.  The tumor subsequently recurred in all three areas, as well as in the lung in the form of isolated nodules.  At that time the patient had significant pain in his left foot and had difficulty ambulating.         

             Photographs of the foot lesion at the time of the recurrence are presented in Figures 1-3. The skin over the proximal dorsal left foot was thickened and immobile.  A 6 cm x 6 cm irregular exophytic tumor mass was present, with areas of central ulceration, necrosis, and bleeding.  Multiple smaller subcutaneous nodules surrounded the larger tumor.  A photograph of the right thigh mass is presented in Figure 4.

            Figure 5 demonstrates the changes in the soft tissues of the foot, as well as erosive changed in the superior aspect of the talus and posterior aspect of the calcaneus.  Amputation below the knee was recommended to the patient for pain control and wound care reasons, but the patient refused.  Intravenous and oral morphine were administered for pan control, and the patient expired related to the progressive tumor in March 1994.  

Discussion

 Bone tumors presenting in the feet should be evaluated to determine if the neoplasms are benign or malignant.  The differential diagnosis for benign tumors involving the feet should include osteoid osteoma, osteoblastoma, enchondroma, osteochondroma, giant cell tumor, unicameral bone cyst, chondroblastoma, chondromyxoid fibroma, and aneurysmal bone cyst.  The differential diagnosis for malignant tumors of the feet should include chondrosarcoma, Ewing’s sarcoma, osteogenic sarcoma, Kaposi’s sarcoma, synovial sarcoma, malignant melanoma, and fibrosarcoma.  Taking a careful history and completing a thorough physical examination of the patient will often lead to the use of the appropriate diagnostic procedure.  Biopsy and / or surgical excision followed by histologic examination of the tissue are essential for establishing a diagnosis.  Although most bone tumors in the feet are benign, malignant tumors do occur and can be difficult to manage for both the physician and the patient.

 

            In the patient described in this report, the relationship of the development of the chondrosarcoma to the long-standing rheumatoid arthritis, or the treatment thereof, is unclear.  There is a clinical association between the treatment of patients with autoimmune diseases such as rheumatoid arthritis, systemic lupus erythematosus, polymyaglia rheumatica, or temporal arteritis with long-term immunosuppressive regimens and the development of Kaposi’s sarcoma. ( 12 )  This form of Kaposi’s sarcoma, which is unrelated to HIV-1 infection, usually presents clinically and pathologically like the classic of African (epidemic) form. ( 13 )

 

            The histologic features of the tumor found in this patient were not consistent with the diagnosis of  Kaposi’s sarcoma, but the pattern of the spread of the tumor noted in this case (subcutaneous nodules) is distinctly different from that of typical soft tissue of bone sarcomas, which metastasize predominantly to the lungs.  We are unaware of an association between the development of other types of sarcomas and either rheumatoid arthritis of long-term immunosuppressive therapy, such as the weekly methotrexate and corticosteroids used to treat this patient. 

 

            Chondrosarcomas are malignant tumors that occur rarely in the bones of the feet.  But when evaluation bone tumors, chondrosarcomas should be included in the differential diagnosis.  Early diagnosis and proper medical management are essential to achieve good local tumor control and to reduce the risk of the subsequent development of distant metastases.  Standard surgical approaches for well-localized tumors include wide local excision of amputation, but should be considered prior to the use of extensive local surgical procedures alone.

 

Summary

             The authors presented a rare case study of a chondrosarcoma involving the left foot of a patient with rheumatoid arthritis.  The patient was unresponsive to chemotherapy but responded to radiation therapy for a short time.  He subsequently developed metastases to the lower extremities and left lung, which led to his death.  The prognosis is unfavorable in most such metastatic tumors.  It is very important to establish the diagnosis early and to treat the tumor aggressively while it is still localized.

 

Acknowledgment

             We thank Marva Jensen, MEd, for reviewing the manuscript.

 

References

 

1.   Pachter MR, Alpert M: Chondrosarcoma of the foot skeleton. J Bone Joint Surg 46A:601-607, 1964

2.   Mirra j, Picci P, Gold R: Bone tumors: Clinical, ratiologic, and pathologic correlations. Vol. 1. Lea & Febiger, Philadelphia, 1989

3.   Chase D, Enzinger F: Epitheloid sarcoma: Diagnosis, prognostic indicators, and treatment. Am J Surg Pathol 9:241-263, 1985

4.   Johnston M: Epidemiology of soft-tissue and bone tumors of the foot. Clin Podiatr Med Surg 10:561-607,1993

5.   Dahlin DC: Bone tumors: General aspects and an analysis of 2,276 cases. Charles C Thomas, Springfield, IL, pp.114-128, 1957 

6.   Dahlin DC, Henderson ED: Chondrosarcoma a surgical and pathological problem.  Review of 212 cases. J Bone Joint Surg 38A: 1025-1038,1956

7.   Miki T, Yamamuro T, Oka M. Urushindani H, Itokazu M: Chondrosarcoma developed in the distal phalangeal bone of the third toe. Clin Orthop Relat Res 136:241-243,1978

8.   Berlin S: Tumors and tumorous conditions of the foot. pp.609-645. In McGlamry E.(ed): Comprehensive textbook of foot surgery: Williams & Wilkins, Baltimore,1974

9.   Dahlin DC, Salvador AH: Chondrosarcomas of bones on the hands and feet: A study of 30 cases. Cancer 34:755-760, 1974

10. Wiss DA: Chondrosarcoma of the first metatarsal. J Surg Oncol 23: 110-112, 1983

11. Elias A, Ryan L, Sulkes A, et al: Response to Mesna, Doxorubicin, ifosfamide, and dacarbazine in 108 patients with metastatic or unresectable sarcoma with no prior chemotherapy. J Clin Oncol 7:1208-1216, 1989

12. Larbre JP, Nicolas JF, Collet P, Larbre B, Llorca G: Kaposi’s sarcoma in a patient with rheumatoid arthritis: Possible responsibility of Captopril in the development of lesions. Rheumatology 18:476-477, 1991

13.      Schottstaedt M, Hurd E, Stone M: Kaposi’s sarcoma in rheumatoid arthritis. Am J Jed 82:1012-1026, 1987

© 2001, Dr. Brian Richman, All Rights Reserved