So-Called
Benign Fibrous Histiocytoma:
By J.C. Hoeffel,
F. Boman-Ferrand,
F. Tachet, P. Lascombes,
Vandoeuvre-Les-Nancy, 0 This is a report of a case of benign fibrous histiocytoma involving the spine of a 13-year-old boy. Copyright o 1992 by W.6. Saunders Company INDEX WORDS:
Histiocytoma,
benign, fibrous.
B
ENIGN FIBROUS histiocytoma (BFH) of bone is a lesion histologically indistinguishable from a nonossifying fibroma but clinically distinguishable by location, radiological appearance, pain, and tendency to occur at an older age. I-3We report a pediatric case involving the spine.
Report of a Case A. Czorny, and C. Bernard
France and Nancy, France
The spine-located cases of fibrous histiocytoma of bones have rarely been reported so far.‘O They were male, and aged 18, 24, and 28 years. Lesions invoIved spinous process and to a lesser extent the arch of the cervical spine of C2 (2 cases) and C6 (1 case). Lesions were 3 to 4 cm. The symptoms were persistent neck pain, occipital pain or headache, and weakness of grip. On x-ray the cervical spinous process was expanded and slightly trabeculated. There were no recurrences noticed in the three cases reported with involvement of the cervical spinous process.
CASE REPORT A 13-year-old boy suffered from scoliosis with thoracic spine stiffness. Neurological examination showed no abnormality. X-ray of the spine showed flattening of the 12th vertebral body with osteolysis on the left side. On tomograms (Fig l), a lytic expansile lesion involved the left lamina, the left pedicle, and most of the vertebral body on the left side. Computed tomography (CT) (Fig 2) showed a large osteolytic lesion of the 12th vertebral body mainly on the left side involving also the left pedicle and the left lamina (8 cm2 for 21 cm2 of vertebral surface). The vertebra’s posterior wall was involved but not ruptured and the dural sac was not displaced. The borders of the lesion were slightly slerotic. There was no calcification, no ossification, and no associated soft tissue mass on CT images. The densities were low (-20 HU). After injection of contrast medium, they increased up to +90 HU. The disc was normal. Surgical excision was performed. Through an anterior approach, the vertebral body was resected excepting a small part of the left side. A tibia1 graft was inserted. Through a posterior approach, the left lamina was resected as were the left articular pillar, the left transverse process, and the left pedicle. A graft was inserted to reconstruct the posterior arch of D12. The postoperative phase was uneventful. At microscopic examination (Fig 3), the lesion was made of fibrohistiocytic benign-appearing spindle cells with a few giant cells, foam cells, hemosiderin deposits, and foci of dense fibrous tissue. Two years after surgery, the boy is doing well, without any recurrence. DISCUSSION
We have collected 31 cases4-9 that have been described in the literature under a variety of diagnoses and that may correspond to the so-called BFH of the bones.
From the Departments of Radiology and Orthopaedic Surgery, Children’s Hospital and the Department of Pathology, Brabois Hospital, Vandoeuvre-Les-Nancy, France, and the Department of Neurosurgery, St-J&en Hospital, Nancy, France. Address reprint requests to J.C. Hoefel, Children’s Hospital, 54500 Vandoeuvre, France. Copyright o 1992 by W.B. Saunders Company 0022-3468192/2705-0037$03.OOlO 672
Fig 1. (A) Frontal and (B) lateral CT views. Diffuse osteolysis with peripheral sclerosis of most of the vertebral body, which is flattened.
Journal of Pediatric Surgery, Vol27, No 5 (May), 1992: pp 672-674
BENIGN FIBROUS HISTIOCYTOMA
OF SPINE
673
Fig 3. At microspic examination, benign-appearing round and spindle-shaped cells are associated with foam cells and some chronic inflammatory cells. One multinucleated giant cell in the corner of the figure (H&E, original magnification x400).
Fig 2. CT of the spine. Osteolysis of the vertebral body with involvement of the posterior arch. The peripheral sclerosis is in favor of a long duration process.
Roessner et aIn described a case of benign fibrous histiocytoma of the third and fourth cervical vertebra in a 21-year-old man. Destouet et al7 described a similar case of the second vertebra in a 24-year-old man. A few cases of nonossifying fibroma of the spine have been reported in adults,*2-14and one in a child.15 Adolphe et ali2 reported the case of a 52-year-old woman who had a lesion of the left pedicle of the 12th vertebra with radicular damage. Ruelle et alI4 reported the case of a 29-year-old woman with a lesion of the fifth vertebra and medullary compression. Fabris et alI5 reported the case of a child of 13 years who had a nonossifying fibroma of the left transverse process of the first lumbar vertebra. From the radiological point of view, the usual radiological diagnosis are aneurysmal bone cyst, osteoblastoma, or fibrous dysplasia. The other clinicopathological diagnoses to be discussed are giant cell tumor (GCT), nonossifying fibroma (NOF), and low-grade malignant fibrous histiocytoma (MFH).1°J6 From 5% to 10% of GCT are characterized by massive fibrohistiocytic repair tissues including foam cells. The patients are generally between 20 and 40 years old and the radiograph shows a large epiphyseal, often bubbly, lesion that looks identical to that
seen in GCT. On exhaustive histological examination variable sized foci of conventional giant cells tumor can be found. The usual criteria for diagnosis of NOF in a non-long bone site are: size under 5 to 6 cm, a border of sclerosis, a lobulated or loculated appearance, absence of epiphyseal involvement, a patient aged less than 20, histology identical to the NOF of long bones, and absence of recurrence after excision. Low-grade MFHs may be confused with BFH,17 but are recognized by the following histological features: subtle, but definite nuclear pleomorphism including enlarged single to multiple nucleoli, and occasional atypical mitoses. Fibrohistiocytic tissues with foam cells may be associated with a number of entities as a late phase process (eosinophilic granuloma, NOF, chondroblastoma, GCT, grade I osteosarcoma, fibrous dysplasia). To make the diagnosis of BFH one has to exclude first other much more common pathological entities. If that is done, few cases will fit with the diagnosis of BFH.1°J6 This tumor can grow over a long time and may recur.’ BFH could represent the benign counterpart of MFH. ACKNOWLEDGMENT The authors are grateful to J.M. Mirra for reviewing slides of the reported case.
histological
REFERENCES 1. Dahlin DC: Bone 1978, __ pp 116-121
Tumors
(ed 3). Springfield,
IL, Thomas,
2. Feldman F, Lattes R: Primary malignant fibrous histiocytoma (fibrous xanthoma) of bone. Skeletal Radio1 1:145-160, 1977 3. Huvos AG: Bone Tumors. Diagnosis, Treatment, sis. Philadelphia, PA, Saunders, 1979, pp 297-306
and Progno-
4. Bertoni F, Capanna R, Calderoni P, et al: Case report 223. Benign fibrous histiocytoma. Skeletal Radio1 9:215-217,1983 5. Bertoni F, Calderoni P, Racchini P, et al: Benign fibrous histiocytoma of bone. J Bone Joint Surg (Am) 68:1225-1230,1986 6. Clarke BE, Xipell JM, Thomas DP: Benign fibrous histiocytoma of bone. Am JSurg Path01 9:806-815, 1985 7. Destouet JM, Kyriakos M, Gilula LA: Fibrous histiocytoma
674
(fibroxanthoma) of a cervical vertebra. A case report with a review of the literature. Skeletal Radio1 5241-246, 1980 8. Hermann G, Steiner GC, Sherry HH: Case report 465. Benign fibrous histiocytoma. Skeletal Radio1 17:195-197, 1988 9. Remagen W, Nidecker A, Prein J: Case report 3.59. Benign fibrous histiocytoma. Skeletal Radio1 X:251-253,1986 10. Mirra JM: Personal communication, 1989 11. Roessner A, Immenkamp M, Weidner A, et al: Benign fibrous histiocytoma of bone. Light and electron-microscopic observation. J Cancer Res Clin Oncol101:191-202,198l 12. Adolphe J, Raul P, Ferry A, et al: Fibrome non ossifiant d’un pedicule vertebral. Ann MCd Nancy 22:797-801,1983
HOEFFEL ET AL
13. Zarski S, Kozina W, Wagner T: Case of non-ossifying fibroma situated in the cervical spine. Nemo1 Neurochir Pol 14:685-688,198O 14. Ruelle A, Datti R, Ceppa P. Fibroma non ossificante a localizzazione vertebrale. Caso Clinico Riv Neural 58:150-152, 1988 15. Fabris D, Candiotto S, Mammano S, et al: Antalgic scoliosis due to nonosteogenic fibroma of the Ll neural arch: Report of a case. J Pediatr Orthop 6:103-RX,1986 16. Mirra JM: Bone Tumors: Diagnosis and Treatment. Philadelphia, PA, Lippincott, 1987, pp l-3 17. Clark TD, Stelling CB, Fechner RE: Case report 328. Benign fibrous histiocytoma. Skeletal Radio1 14:149-151,1985
Benign Fibrous Histiocytoma:
By J.C. Hoeffel,
F. Boman-Ferrand,
F. Tachet, P. Lascombes,
Vandoeuvre-Les-Nancy, 0 This is a report of a case of benign fibrous histiocytoma involving the spine of a 13-year-old boy. Copyright o 1992 by W.6. Saunders Company INDEX WORDS:
Histiocytoma,
benign, fibrous.
B
ENIGN FIBROUS histiocytoma (BFH) of bone is a lesion histologically indistinguishable from a nonossifying fibroma but clinically distinguishable by location, radiological appearance, pain, and tendency to occur at an older age. I-3We report a pediatric case involving the spine.
Report of a Case A. Czorny, and C. Bernard
France and Nancy, France
The spine-located cases of fibrous histiocytoma of bones have rarely been reported so far.‘O They were male, and aged 18, 24, and 28 years. Lesions invoIved spinous process and to a lesser extent the arch of the cervical spine of C2 (2 cases) and C6 (1 case). Lesions were 3 to 4 cm. The symptoms were persistent neck pain, occipital pain or headache, and weakness of grip. On x-ray the cervical spinous process was expanded and slightly trabeculated. There were no recurrences noticed in the three cases reported with involvement of the cervical spinous process.
CASE REPORT A 13-year-old boy suffered from scoliosis with thoracic spine stiffness. Neurological examination showed no abnormality. X-ray of the spine showed flattening of the 12th vertebral body with osteolysis on the left side. On tomograms (Fig l), a lytic expansile lesion involved the left lamina, the left pedicle, and most of the vertebral body on the left side. Computed tomography (CT) (Fig 2) showed a large osteolytic lesion of the 12th vertebral body mainly on the left side involving also the left pedicle and the left lamina (8 cm2 for 21 cm2 of vertebral surface). The vertebra’s posterior wall was involved but not ruptured and the dural sac was not displaced. The borders of the lesion were slightly slerotic. There was no calcification, no ossification, and no associated soft tissue mass on CT images. The densities were low (-20 HU). After injection of contrast medium, they increased up to +90 HU. The disc was normal. Surgical excision was performed. Through an anterior approach, the vertebral body was resected excepting a small part of the left side. A tibia1 graft was inserted. Through a posterior approach, the left lamina was resected as were the left articular pillar, the left transverse process, and the left pedicle. A graft was inserted to reconstruct the posterior arch of D12. The postoperative phase was uneventful. At microscopic examination (Fig 3), the lesion was made of fibrohistiocytic benign-appearing spindle cells with a few giant cells, foam cells, hemosiderin deposits, and foci of dense fibrous tissue. Two years after surgery, the boy is doing well, without any recurrence. DISCUSSION
We have collected 31 cases4-9 that have been described in the literature under a variety of diagnoses and that may correspond to the so-called BFH of the bones.
From the Departments of Radiology and Orthopaedic Surgery, Children’s Hospital and the Department of Pathology, Brabois Hospital, Vandoeuvre-Les-Nancy, France, and the Department of Neurosurgery, St-J&en Hospital, Nancy, France. Address reprint requests to J.C. Hoefel, Children’s Hospital, 54500 Vandoeuvre, France. Copyright o 1992 by W.B. Saunders Company 0022-3468192/2705-0037$03.OOlO 672
Fig 1. (A) Frontal and (B) lateral CT views. Diffuse osteolysis with peripheral sclerosis of most of the vertebral body, which is flattened.
Journal of Pediatric Surgery, Vol27, No 5 (May), 1992: pp 672-674
BENIGN FIBROUS HISTIOCYTOMA
OF SPINE
673
Fig 3. At microspic examination, benign-appearing round and spindle-shaped cells are associated with foam cells and some chronic inflammatory cells. One multinucleated giant cell in the corner of the figure (H&E, original magnification x400).
Fig 2. CT of the spine. Osteolysis of the vertebral body with involvement of the posterior arch. The peripheral sclerosis is in favor of a long duration process.
Roessner et aIn described a case of benign fibrous histiocytoma of the third and fourth cervical vertebra in a 21-year-old man. Destouet et al7 described a similar case of the second vertebra in a 24-year-old man. A few cases of nonossifying fibroma of the spine have been reported in adults,*2-14and one in a child.15 Adolphe et ali2 reported the case of a 52-year-old woman who had a lesion of the left pedicle of the 12th vertebra with radicular damage. Ruelle et alI4 reported the case of a 29-year-old woman with a lesion of the fifth vertebra and medullary compression. Fabris et alI5 reported the case of a child of 13 years who had a nonossifying fibroma of the left transverse process of the first lumbar vertebra. From the radiological point of view, the usual radiological diagnosis are aneurysmal bone cyst, osteoblastoma, or fibrous dysplasia. The other clinicopathological diagnoses to be discussed are giant cell tumor (GCT), nonossifying fibroma (NOF), and low-grade malignant fibrous histiocytoma (MFH).1°J6 From 5% to 10% of GCT are characterized by massive fibrohistiocytic repair tissues including foam cells. The patients are generally between 20 and 40 years old and the radiograph shows a large epiphyseal, often bubbly, lesion that looks identical to that
seen in GCT. On exhaustive histological examination variable sized foci of conventional giant cells tumor can be found. The usual criteria for diagnosis of NOF in a non-long bone site are: size under 5 to 6 cm, a border of sclerosis, a lobulated or loculated appearance, absence of epiphyseal involvement, a patient aged less than 20, histology identical to the NOF of long bones, and absence of recurrence after excision. Low-grade MFHs may be confused with BFH,17 but are recognized by the following histological features: subtle, but definite nuclear pleomorphism including enlarged single to multiple nucleoli, and occasional atypical mitoses. Fibrohistiocytic tissues with foam cells may be associated with a number of entities as a late phase process (eosinophilic granuloma, NOF, chondroblastoma, GCT, grade I osteosarcoma, fibrous dysplasia). To make the diagnosis of BFH one has to exclude first other much more common pathological entities. If that is done, few cases will fit with the diagnosis of BFH.1°J6 This tumor can grow over a long time and may recur.’ BFH could represent the benign counterpart of MFH. ACKNOWLEDGMENT The authors are grateful to J.M. Mirra for reviewing slides of the reported case.
histological
REFERENCES 1. Dahlin DC: Bone 1978, __ pp 116-121
Tumors
(ed 3). Springfield,
IL, Thomas,
2. Feldman F, Lattes R: Primary malignant fibrous histiocytoma (fibrous xanthoma) of bone. Skeletal Radio1 1:145-160, 1977 3. Huvos AG: Bone Tumors. Diagnosis, Treatment, sis. Philadelphia, PA, Saunders, 1979, pp 297-306
and Progno-
4. Bertoni F, Capanna R, Calderoni P, et al: Case report 223. Benign fibrous histiocytoma. Skeletal Radio1 9:215-217,1983 5. Bertoni F, Calderoni P, Racchini P, et al: Benign fibrous histiocytoma of bone. J Bone Joint Surg (Am) 68:1225-1230,1986 6. Clarke BE, Xipell JM, Thomas DP: Benign fibrous histiocytoma of bone. Am JSurg Path01 9:806-815, 1985 7. Destouet JM, Kyriakos M, Gilula LA: Fibrous histiocytoma
674
(fibroxanthoma) of a cervical vertebra. A case report with a review of the literature. Skeletal Radio1 5241-246, 1980 8. Hermann G, Steiner GC, Sherry HH: Case report 465. Benign fibrous histiocytoma. Skeletal Radio1 17:195-197, 1988 9. Remagen W, Nidecker A, Prein J: Case report 3.59. Benign fibrous histiocytoma. Skeletal Radio1 X:251-253,1986 10. Mirra JM: Personal communication, 1989 11. Roessner A, Immenkamp M, Weidner A, et al: Benign fibrous histiocytoma of bone. Light and electron-microscopic observation. J Cancer Res Clin Oncol101:191-202,198l 12. Adolphe J, Raul P, Ferry A, et al: Fibrome non ossifiant d’un pedicule vertebral. Ann MCd Nancy 22:797-801,1983
HOEFFEL ET AL
13. Zarski S, Kozina W, Wagner T: Case of non-ossifying fibroma situated in the cervical spine. Nemo1 Neurochir Pol 14:685-688,198O 14. Ruelle A, Datti R, Ceppa P. Fibroma non ossificante a localizzazione vertebrale. Caso Clinico Riv Neural 58:150-152, 1988 15. Fabris D, Candiotto S, Mammano S, et al: Antalgic scoliosis due to nonosteogenic fibroma of the Ll neural arch: Report of a case. J Pediatr Orthop 6:103-RX,1986 16. Mirra JM: Bone Tumors: Diagnosis and Treatment. Philadelphia, PA, Lippincott, 1987, pp l-3 17. Clark TD, Stelling CB, Fechner RE: Case report 328. Benign fibrous histiocytoma. Skeletal Radio1 14:149-151,1985
