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1 Department of Radiology, Wilford Hall USAF Medical Center, 759th MDTS/MTRD,
Ste. 1, 2200 Bergquist Dr., Lackland AFB, TX 78236-5300.
2 Department of Orthopedic Surgery, Wilford Hall USAF Medical Center, 759th
MSGS/MCSO, Lackland AFB, TX 78236-5300.
Received March 6, 2002;
accepted after revision November 11, 2002.
Address correspondence to J. Q. Ly.
Abstract
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MATERIALS AND METHODS. We retrospectively reviewed the radiographs and MR images of 35 patients with soft-tissue hemangiomas of the extremities. The pattern and extent of the osseous change were categorized as periosteal, cortical, or medullary. Symptomatology, size, and proximity of the hemangioma to the adjacent bone were compared with the presence or absence of osseous change. Statistical analysis was performed using the Student's t test.
RESULTS. Osseous change was noted on radiographs in 13 (37%) of 35 patients and on MR images in 11 (31%) of 35 patients with a total of 14 patients (40%) showing osseous change on at least one study. Periosteal change was present in eight (23%) of 35 patients; cortical change, in 11 (31%) of 35 patients; and medullary change, in 10 (29%) of 35 patients. Direct contact between the soft-tissue hemangioma and the adjacent bone was seen in 13 of 14 patients with osseous change. In those patients without osseous change, the average distance between the soft-tissue hemangioma and bone was 1.06 cm (range, 0–4 cm). No correlation was found between symptoms and the presence of osseous change.
CONCLUSION. Soft-tissue hemangiomas of the extremities frequently result in adjacent osseous change that can be categorized as either periosteal, cortical, or medullary. Only medullary changes correspond with hemangioma size, whereas all three categories of change correlate with the proximity of the hemangioma to the adjacent bone. The presence of osseous change does not correlate with patient symptomatology.
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Radiography is frequently the initial imaging study performed on patients with a palpable soft-tissue mass of the extremities, and although the findings of radiographs are often normal, they may show an indeterminate soft-tissue mass. Although specific for soft-tissue hemangioma, the presence of phleboliths lacks sensitivity, occurring in only 20–67% of the patients [3, 5, 7, 8]. Sonography and CT offer improved visualization of soft-tissue hemangiomas [3, 5, 6, 9]. MR imaging reveals several imaging features that are characteristic of a soft-tissue hemangioma, and in addition, its superior soft-tissue contrast makes it the modality of choice for showing the relationship between the mass and the adjacent anatomic structures, thus aiding in preembolization and preoperative planning.
Several recent reports in the literature describe regional bone changes that occur adjacent to soft-tissue hemangiomas [1, 3, 8, 10]. The exact mechanism leading to these changes remains unknown. In an attempt to further describe the types of bone abnormalities that can occur and to further define the relationship between soft-tissue hemangiomas and these adjacent osseous changes, we retrospectively studied 35 pathologically proven cases of soft-tissue hemangioma. We categorized the osseous change as periosteal, cortical, or medullary, as shown on radiographs and MR images, and then correlated these changes with both the size of the lesion and with the proximity of the lesion to the adjacent bone. We further correlated the various categories of osseous change with the presence or absence of pain in an attempt to determine if the osseous abnormalities are responsible for the pain that is often associated with soft-tissue hemangiomas of the extremities.
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Osseous change was noted in at least one of the three categories (periosteal, cortical, medullary) on radiographs in 13 (37%) of 35 patients and on MR images in 11 (31%) of 35 patients with a total of 14 (40%) of 35 patients showing osseous change on at least one study. A combination of two categories of osseous change was present in seven (20%) of 35 patients, and all three categories of osseous change were present in four (11%) of 35 patients. Periosteal change (Figs. 1A, 1B) was present in eight (23%) of 35 cases. Of these eight, periosteal change was noted on both MR images and radiographs in four patients (50%), on radiographs alone in three patients (37.5%), and on MR images alone in a single patient (12.5%). Six patients showed nonaggressive periosteal change. One patient showed aggressive-appearing periosteal spiculations involving the anterior aspect of the distal fibula, whereas a final patient showed both irregular (aggressive-appearing) periosteal reaction involving the distal radius and thick solid (nonaggressive-appearing) periosteal reaction involving the distal ulna (Figs. 2A, 2B, 2C). Cortical change (Figs. 3A, 3B, 3C and 4A, 4B, 4C, 4D) was present in 11 (31%) of 35 patients. Of these, cortical change was noted on both MR images and radiographs in seven (64%) of 11 patients, on radiographs alone in two (18%) of 11 patients, and on MR images alone in two (18%) of 11 patients. Findings in three patients showed cortical thickening; six showed cortical erosion; two, cortical tunneling; and three, cortical osteopenia. Medullary change (Figs. 2A, 2B, 2C and 4A, 4B, 4C, 4D) was present in 10 (29%) of 35 patients. Of these 10 patients, medullary change was noted on both MR images and radiographs in four (40%), on radiographs alone in three (30%), and on MR images alone in three (30%). Findings of radiographs of five patients revealed medullary osteopenia, and although diffuse cortical osteopenia overlapping the medullary space can mimic medullary osteopenia, three of these patients also had marrow signal change on MR images, further confirming the presence of medullary change. Findings on radiographs in one patient showed medullary sclerosis, and findings on MR images in seven patients showed signal change in the medullary space.
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A significant difference was seen in the size of the lesion in those patients with medullary change (mean size, 147.0 cm3) compared with those without (mean size, 54.9 cm3; p = 0.029). There was no difference for periosteal or cortical change. Significant differences were seen in the proximity of the lesion to bone for those patients with any type of osseous change (periosteal, 0.0 cm vs 0.9 cm, [p < 0.001]); cortical, 0.09 cm vs 0.9 cm [p = 0.001]; and medullary, 0.1 cm vs 0.9 cm [p = 0.001]). Direct contact between the soft-tissue hemangioma and the adjacent bone was seen in 13 of 14 patients with osseous change. In a single case, a combination of cortical and medullary change was present in the absence of direct contact between the soft tissue and the adjacent bone (the mass was 1 cm from the adjacent bone). In those patients with no osseous change, the average distance between the soft-tissue hemangioma and bone was 1.06 cm (range, 0–4 cm). Mean size (volume) of all soft-tissue hemangiomas was 32 cm3, and the mean proximity to the nearest bone was 0.65 cm. Phleboliths were noted in 17 (49%) of 35 patients and were the only finding on radiographs in nine of 35 patients (26%). A soft-tissue mass on radiographs was present in 14 (40%) of 35 patients.
Eight patients presented with a history of pain, with or without a palpable mass. The remaining 27 patients gave no history of pain but presented with either a palpable mass or with no documented chief complaint. Of the eight patients with a history of pain, four showed osseous change, and four showed no evidence of osseous change. No significant correlation was found between the presence of pain and osseous abnormality.
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Osseous Findings
There have been several reports of reactive changes in osseous structures
adjacent to soft-tissue hemangiomas
[1,
3,
8,
10]. Sung et al.
[8] reported three cases
showing elliptical cortical hyperostosis, six cases showing coarsened
trabeculae, and four types of periosteal reactions—lobulated solid,
continuous solid, undulating, and thin solid. Other reports describe the
presence of periosteal reaction
[1,
3], cortical erosions
[3], and cortical thickening
[1,
3,
10]. The results of our study
show that soft-tissue hemangiomas are frequently associated with adjacent
osseous change, which can be divided into one of three categories: periosteal,
cortical, and medullary.
The periosteal changes observed in our patients were predominantly nonaggressive in appearance and were solid and continuous or undulating (Figs. 1A, 1B). These periosteal findings are helpful because they indicate a nonaggressive lesion such as a soft-tissue hemangioma, especially in the presence of an adjacent soft-tissue mass and phleboliths. Aggressive periosteal reaction was present in two cases. The first occurred in a 24-year old man with a left lower extremity soft-tissue hemangioma with associated short periosteal spiculations located on the anterior surface of the distal fibula. Sometimes called a sunburst periosteal reaction, this pattern is typically associated with an aggressive process such as Ewing's sarcoma, osteosarcoma, or infection [18]. The second case occurred in a 64-year-old man with a forearm hemangioma with an irregular periosteal reaction along the distal diaphysis of the radius (Figs. 2A, 2B, 2C) and a nonaggressive-appearing thick solid periosteal reaction along the distal diaphysis of the ulna (not shown in Figs. 2A, 2B, 2C). These findings support the previous report that aggressive-appearing periostitis can occur in association with an adjacent soft-tissue hemangioma [1].
Four different types of cortical change have been previously reported; these include erosions, thickening, tunneling, and osteopenia [8]. The presence of cortical destruction is suggestive of an aggressive or malignant process rather than of an adjacent benign soft-tissue tumor. Awareness that cortical change can occur adjacent to a soft-tissue hemangioma ensures consideration of this lesion in developing a differential diagnosis. Cortical thickening was present in three of 11 patients with cortical change and usually implies a nonaggressive process (Figs. 2A, 2B, 2C, 3A, 3B, 3C, 4A, 4B, 4C, 4D). Thickened or hyperostotic cortex can also occur in association with other benign entities such as stress fracture, cortical hemangioma, and osteoid osteoma. Tunneling of the cortex (sometimes referred to as a pseudopermeative cortex) is observed radiographically because of the multiple tiny holes in the cortex that, when seen overlying the intramedullary space, can mimic a true intramedullary permeative process (Fig. 4C). The pseudopermeative appearance has been reported to occur in the presence of hemangiomas, osteoporosis, and radiation therapy. The fourth type of cortical response noted in our study is osteopenia, which can occur in both aggressive (when severe, focal, or rapid) and nonaggressive (when diffuse) lesions.
Medullary change was the third and final category of osseous abnormality. On radiographs, osteopenia was the most frequent type of intramedullary change observed. In a single case, however, intramedullary sclerosis was present. To our knowledge, the only other report of intramedullary change seen on radiographs was a series of cases with coarsened trabeculae [8]. Intramedullary signal change was noted on MR imaging (Figs. 1B, 2C, and 4D) adjacent to soft-tissue hemangiomas; interestingly, this was the only osseous change that correlated with the size of the adjacent hemangioma. Those hemangiomas with associated medullary change were on the average three times larger (147 cm3) than those without medullary change (54.9 cm3).
All cases with marrow signal abnormality on MR imaging showed decreased signal intensity on T1-weighted and increased signal intensity on T2-weighted images relative to normal adjacent marrow signal. Although we have no pathologic proof from these cases to definitively state the cause of these signal changes, we hypothesize that in most cases the changes represented either marrow edema or hematopoietic conversion associated with localized hyperemia. In two cases however, the soft-tissue hemangiomas appeared to extend into the adjacent bone, and in these cases, signal abnormalities may have been caused by angiomatous involvement of the bone. In all cases however, both the bulk and the center of the hemangiomas were located in the soft tissues and not in the bone and were thought to have originated in the soft tissues.
It has been reported that patients with hemangiomas of the extremity frequently present with a history of pain [13, 14, 16], and bone involvement might be responsible for these symptoms. However, no correlation was found between the presence of symptoms and the presence of osseous change.
Mechanism of Osseous Change
Several theories may explain the precise mechanism by which osseous change
occurs adjacent to soft-tissue masses, and more than one mechanism is likely
responsible for the full range of changes that occur
[4,
19,
20,
21]. When a mass lies in
direct contact with bone, pressure effects may result in either osseous
resorption or in reactive new bone formation. Resorption may result from
either a direct pressure effect or from hyperemic effects of the adjacent
mass. Smooth erosion of the cortical surface is often seen adjacent to a
soft-tissue mass but provides little information regarding the differential
diagnosis because this type of resorption can result from either a malignant
or a benign process [19].
Cortical resorption in the presence of adjacent sclerosis may imply a slowly
evolving process and is therefore more compatible with a benign process
[19,
20], whereas irregular
cortical resorption or destruction in the presence of medullary involvement or
periostitis is indicative of a more rapidly developing process such as
infection or malignancy. Another theory is that soft-tissue masses may result
in stretching or irritation of the adjacent periosteum, which leads to new
bone formation [14]. In cases
of diffuse hemangiomatosis of an extremity, overgrowth of the osseous
structures of the entire extremity has been observed, and this may result from
an increase in the oxygen saturation of the venous blood caused by extensive
arteriovenous shunting in the hemangioma
[14,
22,
23].
The findings of this study show that the proximity of the hemangioma to the adjacent bone correlates with all three categories of osseous change: periosteal, cortical, and medullary. The fact that in nearly every case with osseous change the hemangioma directly contacts the underlying bone tends to support the theory that direct pressure effects from the adjacent mass lead to the osseous change but may also support the theory that hyperemia can lead to adjacent osseous change. Medullary changes were not only related to the proximity of the adjacent hemangiomas but also correlated with the size of the lesion. Although this speculation was not conclusively proven in this study, the larger lesions would be expected to result in increased localized blood flow and hyperemia relative to the smaller lesions, and this explanation seems to be the most likely responsible for medullary changes seen on either radiographs or MR images.
Although solid, smooth, or undulating cortical thickening was a common finding in this series, there were two cases of aggressive-appearing periosteal reaction. Most cortical and periosteal changes would suggest a benign or slow-growing process. The medullary changes noted in this series have been more commonly associated with malignant processes in previous literature [19]. Although soft-tissue hemangiomas typically result in nonaggressive-appearing osseous abnormalities, more aggressive-appearing changes could also occur.
In summary, soft-tissue hemangiomas of the extremities are frequently associated with adjacent osseous change that can be categorized as periosteal, cortical, or medullary. Although all three categories of change correspond with the proximity of the soft-tissue hemangioma to bone, only the size of the lesion corresponds with the presence of medullary change. Pain, which occasionally occurs in the presence of a soft-tissue hemangioma, does not appear to be related to the presence or absence of osseous change.
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