Arch Orthop Trauma Surg (1991) 111 : 58-60
0rth0paed c andTraumaSurgery
Archivesof
@ Springer-Verlag 1991
Current problem cases
Malignant fibrous histiocytoma in the course of chronic osteomyelitis E. Czerwifiski, A. Skolarczyk, and W. Frasik Department of Orthopaedics, Cracow Academy of Medicine, ul. Kopernika 19a, 31-501 Krakdw, Poland
Summary. Malignant fibrous histiocytoma ( M F H ) was f o u n d in the f e m u r of a 28-year-old m a n who had suffered f r o m chronic osteomyelitis. T h e patient had undergone surgery for a valgus knee deformity and the procedure was complicated by chronic osteomyelitis which lasted for 12 years. F o u r m o n t h s after the onset of painful s y m p t o m s related to the occurrence of M F H , gross b o n e destruction with pathologic fracture and pulmonary metastases were present. T h e patient died 4 m o n t h s later. Histopathological and i m m u n o h i s t o c h e m i c a l examinations indicated the diagnosis of M F H .
Malignant fibrous histiocytoma ( M F H ) is a rare b o n e turnout occurring 10 times less frequently than osteogenic sarcoma. It has b e e n f o u n d that in 28% of cases M F H follows irradiation or pre-existing b o n e diseases such as Paget's disease, fibrous dysplasia, and others [3, 6]. So far there has b e e n no report of M F H associated with chronic osteomyelitis. H o w e v e r , planocellular sinus carOffprint requests to: E. Czerwifiski (address above)
c i n o m a has b e e n described as a complication of chronic b o n e infection in 0.25% of cases [7]. In this report we would like to present a case of M F H which arose in one of our patients being treated for chronic osteomyelitis.
Case report In 1973 we operated a 16-year-old boy for valgus deformity of the right knee caused by epiphyseal dysplasia. Supracondylar osteotomy of the femur was performed (Fig. 1). The boy had an uneventful postoperative recovery with primary wound healing. Nine months later a discharging sinus appeared on the posterior aspect of the thigh. After 3 weeks of unsuccessful antibiotic therapy we removed the plate, d6brided the affected bone and installed suction-irrigation drainage. Staphylococcus aureus K+M + and later Pseudomonas aeruginosa were identified in the culture. Gentamycin and cloxacillin were administered according to the sensitivity of the bacteria. Bone biopsy showed the picture typical of osteomyelitis (Fig. 2). The infection went into the chronic stage and we had to operate on the patient several times: 2, 5 and 14 months later. During the next 7 years there were a few flare-ups of the infection, which settled with antibiotic treatment. After that period the patient was asymptomatic for 4 years.
Fig. la-c. Radiographs of the distal femur taken a before operation b just after surgery and c 9 months later
59
Fig. 2. Biopsy specimen taken during d6bridement of chronic osteomyelitis, showing inflammatory infiltrates and connective tissues between bone trabeculae. H & E, magnification × 175
The 12th year from the onset of osteomyelitis, when the patient was 28 years old, pain occurred in the right knee, gradually increasing over 2 months. The knee was aspirated and opaque, aseptic fluid evacuated. The blood cell count was normal and the erythrocyte sedimentation rate 10mm. Slight bone destruction was noted on the radiograph (Fig. 3a), which was interpreted as consecutive recurrence of the osteomyelitis, and antibiotic treatment was prescribed (cephradine). On the radiograph taken 2 months later, gross bone destruction and pathological fracture were seen (Fig. 3b, c). A trocar biopsy specimen was taken from the focus. Histopathological examination revealed malignant tumour tissue composed of spindle cells, suggesting MFH or synovial sarcoma (Fig. 4). Because of the advanced pulmonary metastases detected and the poor general condition of the patient, surgery and chemotherapy were not attempted. The patient died 8 months after the onset of painful symptoms. Specimens taken from the primary focus and metastases presented the same picture as in the previous biopsy. Immunohistochemical examinations were performed to delineate the type of neoplasm. Positive staining for al-antichymotrypsin was found, while testing for S-100 protein and epithelial membrane antigen gave negative results. Thus, the tumour was classified as MFH.
Fig. 3a-c. Radiographs taken 12 years later, showing a osteolytic and b, c pathological fracture with gross bone destruction
Discussion
Fig. 4. Neoplastic infiltrates in histological specimen taken from site of pathological fracture. H & E, magnification × 178
It is k n o w n that M F H m a y follow pre-existing pathological osseous conditions such as P a g e t ' s disease and fibrous dysplasia. Single cases have b e e n r e p o r t e d in association with e n c h o n d r o m a , giant cell t u m o u r , b o n e infarction, supracondylar f e m o r a l fracture and c e m e n t e d total hip r e p l a c e m e n t [1, 3, 4, 8]. W e would like to add chronic osteomyelitis to this series o f cases. T h e clinical findings in the case p r e s e n t e d confirm the features of s e c o n d a r y M F H described by other authors. S e c o n d a r y M F H appears m o r e frequently after the age of 21 years, the radiographic picture is atypical, and the n e o p l a s m exhibits a high degree of malignancy [5]. O u r patient was 28 ye'ars old, and radiographs of the malignant transformation were misleading. Huvos et al. noticed that the classic radiographic picture of "an osteolytic
60 type of geographic bone destruction" was observed only in the primary M F H . The secondary ones had an atypical appearance with no clear sign of malignancy [5]. Clinical deterioration was very rapid in our patient: pulmonary metastases and pathological fracture were present 4 months after the onset of symptoms, and the patient died another 4 months later. Huvos et al. distinguished three groups of histological patterns of MFH: predominantly fibroblastic, histiocytic, and those with malignant giant cells [5]. The case we present can be assigned to the most frequent fibroblastic group. It seems probable that the processes of bone destruction and reparation in chronic osteomyelitis lasting for several years could stimulate the malignant proliferation of fibroblastic cells. The principles of treatment of M F H are similar to those employed in bone sarcomas. T u m o u r malignancy and local condition are the decisive factors when en bloc excision or amputation is being considered. Pre- and postoperative chemotherapy is recommended. In a series of 136 patients with M F H treated according to the above regime, Huvos et al. saw 53% survival at 5 years [2, 4].
References 1. Bader H, Sponher F, Gerflitzky W, Meyer D (1981) Malignes fibr6ses Histiozytom nach suprakondyl~irer Oberschenkelfraktur. Dtsch Med Wochenschr 106: 336-339 2, Bertoni F, De Santis E, Lavs M, Sanchez Fernandez (1978) Malignant fibrous histiocytoma of bone. Ital J Orthop Traumatol 4: 369-377 3. Boland PJ, Huvos AG (1986) Malignant fibrous histiocytoma of bone. Clin Orthop Rel Res 204:130-134 4. Huvos AG, Heiweil M, Bretsky SS (1985) The pathology of malignant fibrous histiocytoma of bone. Am J Surg Pathol 9: 853-871 5. G6recki A, Wysocki M (1981) Fibrohistiocytoma malignum ossis femoris. Chir Narzadow Ruchu Orthop Pol 44: 495-498 6. Huvos AG, Woodard HR, Heilwell M (1986) Postradiation malignant fibrous histiocytoma of bone. A clinipathologic study of 20 patients. Am J Surg Path 10 : 9-18 7. McAnally AK, Dockerty MB (1949) Carcinoma developing in chronic draining cutaneous sinuses and fistulas. Surg Gynecol Obstet 88 : 87-91 8. Tait NP, Hackind PM, Malcolm AJ (1988) Case reports. Malignant fibrous histiocytoma occurring at the site of a previous total hip replacement Received March 13, 1989
0rth0paed c andTraumaSurgery
Archivesof
@ Springer-Verlag 1991
Current problem cases
Malignant fibrous histiocytoma in the course of chronic osteomyelitis E. Czerwifiski, A. Skolarczyk, and W. Frasik Department of Orthopaedics, Cracow Academy of Medicine, ul. Kopernika 19a, 31-501 Krakdw, Poland
Summary. Malignant fibrous histiocytoma ( M F H ) was f o u n d in the f e m u r of a 28-year-old m a n who had suffered f r o m chronic osteomyelitis. T h e patient had undergone surgery for a valgus knee deformity and the procedure was complicated by chronic osteomyelitis which lasted for 12 years. F o u r m o n t h s after the onset of painful s y m p t o m s related to the occurrence of M F H , gross b o n e destruction with pathologic fracture and pulmonary metastases were present. T h e patient died 4 m o n t h s later. Histopathological and i m m u n o h i s t o c h e m i c a l examinations indicated the diagnosis of M F H .
Malignant fibrous histiocytoma ( M F H ) is a rare b o n e turnout occurring 10 times less frequently than osteogenic sarcoma. It has b e e n f o u n d that in 28% of cases M F H follows irradiation or pre-existing b o n e diseases such as Paget's disease, fibrous dysplasia, and others [3, 6]. So far there has b e e n no report of M F H associated with chronic osteomyelitis. H o w e v e r , planocellular sinus carOffprint requests to: E. Czerwifiski (address above)
c i n o m a has b e e n described as a complication of chronic b o n e infection in 0.25% of cases [7]. In this report we would like to present a case of M F H which arose in one of our patients being treated for chronic osteomyelitis.
Case report In 1973 we operated a 16-year-old boy for valgus deformity of the right knee caused by epiphyseal dysplasia. Supracondylar osteotomy of the femur was performed (Fig. 1). The boy had an uneventful postoperative recovery with primary wound healing. Nine months later a discharging sinus appeared on the posterior aspect of the thigh. After 3 weeks of unsuccessful antibiotic therapy we removed the plate, d6brided the affected bone and installed suction-irrigation drainage. Staphylococcus aureus K+M + and later Pseudomonas aeruginosa were identified in the culture. Gentamycin and cloxacillin were administered according to the sensitivity of the bacteria. Bone biopsy showed the picture typical of osteomyelitis (Fig. 2). The infection went into the chronic stage and we had to operate on the patient several times: 2, 5 and 14 months later. During the next 7 years there were a few flare-ups of the infection, which settled with antibiotic treatment. After that period the patient was asymptomatic for 4 years.
Fig. la-c. Radiographs of the distal femur taken a before operation b just after surgery and c 9 months later
59
Fig. 2. Biopsy specimen taken during d6bridement of chronic osteomyelitis, showing inflammatory infiltrates and connective tissues between bone trabeculae. H & E, magnification × 175
The 12th year from the onset of osteomyelitis, when the patient was 28 years old, pain occurred in the right knee, gradually increasing over 2 months. The knee was aspirated and opaque, aseptic fluid evacuated. The blood cell count was normal and the erythrocyte sedimentation rate 10mm. Slight bone destruction was noted on the radiograph (Fig. 3a), which was interpreted as consecutive recurrence of the osteomyelitis, and antibiotic treatment was prescribed (cephradine). On the radiograph taken 2 months later, gross bone destruction and pathological fracture were seen (Fig. 3b, c). A trocar biopsy specimen was taken from the focus. Histopathological examination revealed malignant tumour tissue composed of spindle cells, suggesting MFH or synovial sarcoma (Fig. 4). Because of the advanced pulmonary metastases detected and the poor general condition of the patient, surgery and chemotherapy were not attempted. The patient died 8 months after the onset of painful symptoms. Specimens taken from the primary focus and metastases presented the same picture as in the previous biopsy. Immunohistochemical examinations were performed to delineate the type of neoplasm. Positive staining for al-antichymotrypsin was found, while testing for S-100 protein and epithelial membrane antigen gave negative results. Thus, the tumour was classified as MFH.
Fig. 3a-c. Radiographs taken 12 years later, showing a osteolytic and b, c pathological fracture with gross bone destruction
Discussion
Fig. 4. Neoplastic infiltrates in histological specimen taken from site of pathological fracture. H & E, magnification × 178
It is k n o w n that M F H m a y follow pre-existing pathological osseous conditions such as P a g e t ' s disease and fibrous dysplasia. Single cases have b e e n r e p o r t e d in association with e n c h o n d r o m a , giant cell t u m o u r , b o n e infarction, supracondylar f e m o r a l fracture and c e m e n t e d total hip r e p l a c e m e n t [1, 3, 4, 8]. W e would like to add chronic osteomyelitis to this series o f cases. T h e clinical findings in the case p r e s e n t e d confirm the features of s e c o n d a r y M F H described by other authors. S e c o n d a r y M F H appears m o r e frequently after the age of 21 years, the radiographic picture is atypical, and the n e o p l a s m exhibits a high degree of malignancy [5]. O u r patient was 28 ye'ars old, and radiographs of the malignant transformation were misleading. Huvos et al. noticed that the classic radiographic picture of "an osteolytic
60 type of geographic bone destruction" was observed only in the primary M F H . The secondary ones had an atypical appearance with no clear sign of malignancy [5]. Clinical deterioration was very rapid in our patient: pulmonary metastases and pathological fracture were present 4 months after the onset of symptoms, and the patient died another 4 months later. Huvos et al. distinguished three groups of histological patterns of MFH: predominantly fibroblastic, histiocytic, and those with malignant giant cells [5]. The case we present can be assigned to the most frequent fibroblastic group. It seems probable that the processes of bone destruction and reparation in chronic osteomyelitis lasting for several years could stimulate the malignant proliferation of fibroblastic cells. The principles of treatment of M F H are similar to those employed in bone sarcomas. T u m o u r malignancy and local condition are the decisive factors when en bloc excision or amputation is being considered. Pre- and postoperative chemotherapy is recommended. In a series of 136 patients with M F H treated according to the above regime, Huvos et al. saw 53% survival at 5 years [2, 4].
References 1. Bader H, Sponher F, Gerflitzky W, Meyer D (1981) Malignes fibr6ses Histiozytom nach suprakondyl~irer Oberschenkelfraktur. Dtsch Med Wochenschr 106: 336-339 2, Bertoni F, De Santis E, Lavs M, Sanchez Fernandez (1978) Malignant fibrous histiocytoma of bone. Ital J Orthop Traumatol 4: 369-377 3. Boland PJ, Huvos AG (1986) Malignant fibrous histiocytoma of bone. Clin Orthop Rel Res 204:130-134 4. Huvos AG, Heiweil M, Bretsky SS (1985) The pathology of malignant fibrous histiocytoma of bone. Am J Surg Pathol 9: 853-871 5. G6recki A, Wysocki M (1981) Fibrohistiocytoma malignum ossis femoris. Chir Narzadow Ruchu Orthop Pol 44: 495-498 6. Huvos AG, Woodard HR, Heilwell M (1986) Postradiation malignant fibrous histiocytoma of bone. A clinipathologic study of 20 patients. Am J Surg Path 10 : 9-18 7. McAnally AK, Dockerty MB (1949) Carcinoma developing in chronic draining cutaneous sinuses and fistulas. Surg Gynecol Obstet 88 : 87-91 8. Tait NP, Hackind PM, Malcolm AJ (1988) Case reports. Malignant fibrous histiocytoma occurring at the site of a previous total hip replacement Received March 13, 1989
