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Spinal angiolipomas revisited: two case reports.

Approximately 60 cases of spinal angiolipomas have been described in the medical literature. Extradural tumours predominate. Lesions with a bony component, the infiltrating subgroup, were reported in 10 patients. Intradural angiolipomas were found three times. Several excellent review articles are available, but a systematic comparison of the characteristics of the two major varieties, infiltrating and non-infiltrating spinal extradural angiolipomas, has not so far been made. The authors operated on two patients with spinal angiolipomas and found one of them the infiltrating and the other the non-infiltrating type. Then, they proceeded to a review of all publish cases of infiltrating angiolipomas. With a knowledge of recently reported data on the subject the authors compared essential clinical features of both varieties of tumours. They share identical clinical characteristics. Differences found in age, sex or location were not statistically significant. Mode of onset and signs and symptoms present on admission were essentially similar. As could be anticipated, removal was more often complete in non-infiltrating tumours, but outcome was good or fair in more than 85% of cases in both groups. Involvement of bone by spinal angiolipomas does not imply a worsening in the prognosis.

Adult

Angiolipoma of suprasellar region.

An angiolipoma of the suprasellar region occurred in an 8-year-old boy. Review of 5 intracranial angiolipomas, including the current case, revealed that four were in the sellar region of one man and three women. One was in the thalamus of a man. The average age of the patients was 49 years, and average size of the tumors was 2.5 cm across. The angiolipomas associated with the sellar region often created abnormalities of the eye viz exophthalmos, decreased vision, and palsy of the oculomotor nerve. Sudden onset has ensued in a massively hemorrhagic thalamic angiolipoma. The characteristic low density with negative Hounsfield unit values in CT scan and hyperdensity in MRI of the brain suggested adipose tissue component of the angiolipoma. The angiography has depicted the angiomatous component of the latter. The combined characteristic CT scan, MRI, and angiographic features should demonstrate both vascular and adipose tissue elements of the angiolipomas before pathologic examination.

Adult

Case report: gastric angiolipoma with chronic hemorrhage and severe anemia.

Lipomas are benign tumors that are common in other sites but rare in the stomach. They are usually submucosal and, when symptomatic, are most often accompanied by gastrointestinal hemorrhage. Angiolipoma is a not uncommon benign lipomatous neoplasm with a characteristic vascular component that almost exclusively occurs in the subcutaneous tissue. A case of gastrointestinal angiolipoma, which appears to be only the second one reported, is discussed. The patient, a 69-year-old man, presented with signs and symptoms of chronic hemorrhage and severe anemia (hemoglobin 6, hematocrit 19.9). The patient's condition was found to have resulted from ulceration of the gastric mucosa overlying a submucosal angiolipoma, and the anemia resolved after local surgical resection of the tumor. Electron microscopic studies supported the light microscopic diagnosis of angiolipoma. The absence of fibrin thrombi in this and other nonsubcutaneous angiolipomas and the possible significance of the vascularity of these tumors is discussed. The literature regarding gastric lipoma and angiolipoma is reviewed.

Aged

Cellular angiolipoma.

Three examples of cellular angiolipoma are presented. They occurred as part of multiple, occasionally painful, subcutaneous nodules on the extremities and trunk of healthy men. There was no familial tendency. Grossly, the cellular angiolipomas were small lesions, approximately 1 cm. Histologically, dense cellular angiomatous tissue comprised more than 95% of the lesions. Much of this component contained prominent spindle cells. Lesser degrees of involvement in the other subcutaneous nodules illustrated the continuum of histology between cellular angiolipomas and more typical angiolipomas. The differential diagnosis of cellular angiolipomas includes spindle cell lipoma, Kaposi's sarcoma, and other vascular tumors. The most important distinguishing features are encapsulation, intravascular fibrin thrombi, septation, association with other more typical angiolipomas, and occurrence in healthy individuals.

Adult

Spinal angiolipomas: MR features.

PURPOSE: To determine the MR features of spinal angiolipomas and to compare these findings with their histologic appearance. METHODS: The MR examinations of three patients with surgically proved angiolipomas were reviewed for tumor location and extent, signal characteristics, and pattern of contrast enhancement, and were then compared with the histologic findings. RESULTS: Four tumors were found in the three patients, all located in the posterior epidural compartment, averaging about 2.5 vertebral bodies in length. On noncontrast T1-weighted images, all lesions were inhomogeneous and hypointense relative to epidural fat. Inhomogeneous enhancement was seen in three lesions on postcontrast T1-weighted images obtained with fat-saturation techniques. Angiolipomas were least conspicuous on T2-weighted images. A high vascular content correlated with the presence of large hypointense regions on T1-weighted images. CONCLUSION: Spinal angiolipomas are typically hyperintense on noncontrast T-1-weighted images relative to other tumors. Angiolipomas that contain large hypointense foci on noncontrast T1-weighted images can be expected to have a high degree of vascularity.

Adult

Angiolipoma of the parotid: case report and review.

An angiolipoma is a benign adipose tissue tumor that can occur as either a circumscribed or an infiltrating lesion and represents up to 17% of all lipomas. Most angiolipomas occur in teenagers and young adults, and are rarely described in younger children. Angiolipoma generally arises on the extremities or trunk, and it is unusual when found in the head and neck. An angiolipoma of the parotid region was suspected in an infant girl after contrast-enhanced computerized tomographic scan, and was confirmed when the mass was surgically excised. The clinical characteristics, radiological appearance, and histopathology of angiolipoma are discussed.

Female

Infiltrating angiolipoma in the foot.

A case of infiltrating angiolipoma of the right lower extremity is reported. Angiolipomas are benign neoplasms of adipose tissue with a rich vascular component and are classified as either infiltrating or noninfiltrating. Noninfiltrating angiolipomas are seen in young individuals, present as painful, soft, subcutaneous nodules and are treated by enucleation. Infiltrating angiolipoma is a rare neoplasm with only 23 previously reported cases. These lesions are usually unencapsulated or rarely partially encapsulated and tend to infiltrate bony, neural, muscular, and fibrocollagenous tissue. Treatment of infiltrating angiolipomas is aimed at wide excision with radiotherapy indicated for cases of recurrence.

Amputation, Surgical

Subacute paraparesis induced by venous thrombosis of a spinal angiolipoma: a case report.

STUDY DESIGN: A case report of spinal extradural angiolipoma, a rare tumor that can cause spinal cord compression, is presented with a complete review of the literature related to this disorder. OBJECTIVES: To discuss venous thrombosis involving the angiolipoma in the development of subacute paraparesis. SUMMARY OF BACKGROUND DATA: This case shows that venous thrombosis of a spinal angiolipoma can precipitate the subacute onset of paraparesis. METHODS: Medical history, physical findings, and the results of imaging and histopathologic studies were analyzed to elucidate the pathogenesis of the patient's subacute onset of paraparesis. A bilateral T3-T7 laminectomy was performed, and although the tumor was extremely hemorrhagic, it was mobilized easily off the compressed dura to achieve resection. RESULTS: The postoperative course was uneventful. One month after her surgery, the patient's myelopathic symptoms had resolved, and the she was able to return to work. CONCLUSION: Because the prognosis after surgical management of these lesions is favorable, the diagnosis of thrombosis involving a spinal angiolipoma should be considered in the differential diagnosis of subacute spinal cord compression.

Acute Disease

Mast cells in angiolipomas and hemangiomas of human skin: are they important for angiogenesis?

To characterize the potential role of mast cells (MC) in angiogenesis, this study tests the hypothesis that MC may be more abundant in angiolipomas than in classic lipomas. MC counts were compared in 13 subcutaneous angiolipomas and 15 subcutaneous classic lipomas stained with Giemsa. Angiolipomas had ten times as many MC as did classic lipomas (25.34 +/- 2.83 versus 2.41 +/- 0.37 per mm2, mean +/- SE). To clarify whether this difference was primary (angiogenic activity) or secondary to the increased vascularity, MC were counted in 8 longstanding cutaneous capillary hemangiomas versus 13 cutaneous capillary hemangiomas of recent onset (pyogenic granulomas). If MC were mediating primary angiogenesis, one would expect them to be present in greater numbers in early than in late hemangiomas. To the contrary, however, long-standing hemangiomas were found to have significantly more MC than had those of recent onset (52.48 +/- 14.99 versus 6.59 +/- 3.37 per mm2, mean +/- SE). These results suggest that MC may not play an essential, early role in the proliferation of blood vessels in angiolipomas and hemangiomas, but rather may be related to maturation of blood vessels in these tumors.

Angiolipoma

Angiolipoma of thalamus presenting with abrupt onset suggestive of cerebrovascular disease.

A 25-year-old man had angiolipoma of the left thalamus producing sudden severe headache, coma, right hemiparalysis, and aphasia as usually observed in cerebrovascular disease. The patient remained right hemiparalytic and aphasic after removal of the thalamic lesion. A vascular lesion was also detected angiographically in the left lateral ventricular choroid plexus but was not excised. Only 3 intracranial angiolipomas including the current instance were on record. However, it was only in our patient that the lesion suddenly presented with intracerebral hemorrhage. The pathogenesis of the angiolipoma was suggested to be associated with maldifferentiation of mesenchyme and/or its derivatives toward the vascular and adipose tissue within the same mass, resulting in the formation of the angiolipoma.

Adult

Infiltrating spinal angiolipoma causing myelopathy: case report.

We present a case of an infiltrating spinal angiolipoma demonstrating extension into the vertebral body and the spinal epidural space. The infiltration into the epidural space caused myelopathy. About 40 cases of spinal angiolipoma and angiomyolipoma have been reported; however, only a few cases have been the infiltrating type. The radiological findings were similar to those of vertebral hemangioma, but poor enhancement of the angiolipoma on contrast-enhanced computed tomographic scans differentiated between them. The infiltrating epidural tumor was removed, and the clinical symptoms improved remarkably. Total removal of the tumor and stabilization of the involved vertebral body using the anterolateral approach may be desirable when a diagnosis of angiolipoma or angiomyolipoma is confirmed preoperatively.

Aged

Spinal angiolipomas. Report of three cases.

Spinal angiolipomas are distinct, benign lesions composed of mature lipocytes admixed with abnormal blood vessels. Three new cases of spinal angiolipoma are presented and 34 previously reported cases are analyzed. The 37 total cases (23 females and 14 males) ranged in age from 17 to 73 years (mean 43 years; median 45 years). The mean age of the female patients was older than that for the males (45.0 vs. 41.6 years; p < 0.001, Student's t-test) and most were peri- or postmenopausal. Prior to diagnosis, 97% of the patients had weakness of the lower extremities, 94% had sensory dysfunction, 84% had hyperreflexia and spasticity, 51% had sphincter dysfunction, and 41% had back pain lasting from 1 to 180 months (mean 28 months). Five (22%) of the 23 female patients were pregnant and two had exhibited significant weight gain coincident with the onset of symptoms. The angiolipomas were extradural in 35 patients and intramedullary in two; seven of the extradural lesions infiltrated the surrounding bone. The tumors extended from C-6 to L-4 and had a predilection for the midthoracic region (53% of cases). Plain radiographs were abnormal in 11 (39%) of 28 patients and in all patients with bone infiltration. Myelograms were abnormal in 97% of 32 patients and showed a complete block in 63% of patients. Computerized tomography (CT) and magnetic resonance (MR) imaging revealed the fat-density lesions in all cases studied. There was vascular enhancement in three of five cases with contrast-infused CT and in the one case with gadolinium-infused MR imaging. All patients improved following resection of the epidural lesions and internal decompression of the intramedullary lesions. It is concluded that spinal angiolipomas predominantly affect women. They involve the thoracic (especially the midthoracic) region, and produce symptoms and signs of spinal compression and, in some cases, bone erosion and pathological fractures. Their symptomatology can be exacerbated by pregnancy and weight gain, suggesting that vascular engorgement and the presence of obesity influence their evolution. Their preponderance in older, peri-, or postmenopausal women, and their clinical exacerbation in pregnant women support a role for hormonal influence. Magnetic resonance imaging is the investigation of choice for the diagnosis of these lesions. Surgery is universally successful in relieving symptoms.

Adult

[Multiple angiolipomas--analgesics therapy with doxepin].

Angiolipomas are rare benign tumours of the subcutaneous fat; they are sometimes solitary but their occurrence is more frequently multiple. Angiolipomas can be differentiated from lipomas clinically by their pronounced tenderness and histologically by their variable vascularization. The disease occurs mostly in young adults, the sites of predilection being the trunk and proximal extremities. Multiple angiolipomas have to be differentiated from other lipomatoses, especially from adiposis dolorosa (Dercum's disease). The case reported in this paper was characterized by typical clinical and histological findings. The systemic administration of acetylsalicylic acid, diclofenac, ketotifen, ranitidine, tramadol, tilidine combined with naloxone did not provide adequate pain relief. In contrast, the therapeutic efficiency of the antidepressant doxepin, which also displays antihistaminic effects, suggests a possible role of mediators in the development of pain in angiolipomas.

Biopsy

[Spinal extradural angiolipoma: a case report].

This 60-year-old man had been well until four years prior to admission, when he developed slowly progressive weakness of the lower extremities. On admission he was found to have a spastic paraparesis, sensory disturbance below the level of T10 and mild sphincter dysfunction. Conventional myelography and CT myelography demonstrated an epidural mass located from T5 to T8 vertebral level. MRI revealed that the epidural mass was fusiform and markedly enhanced by the contrast medium. Laminectomy was performed and a fibrous tumor red in color was subtotally removed. Histological findings were consistent with those of angiolipoma. Angiolipoma is a rare tumor in the central nervous system and is regarded as a clinical entity different from a spinal lipoma. Occurrence of angiolipoma in the central nervous system has been reported in 40 cases in the world literature. Thirty nine cases of them occurred in the spinal canal, especially in the dorsal epidural space of the thoracic region. The fusiform shape demonstrated in this case might also be characteristic of angiolipoma in this region.

Epidural Neoplasms

Cytogenetic analysis of subcutaneous angiolipoma: further evidence supporting its difference from ordinary pure lipomas: a report of the CHAMP Study Group.

Subcutaneous angiolipomas are benign soft-tissue lesions consisting of two mesenchymal elements (i.e., adipose tissue and blood vessels) and having distinct clinical features. They usually are multiple, with an obvious male predominance, and hereditary occurrence has been described. Twenty subcutaneous angiolipomas from 10 patients with typical clinical and morphologic features were reviewed. All lesions had a normal karyotype. This finding is in striking contrast with ordinary lipomas, spindle-cell and pleomorphic lipomas, lipoblastomas, and hibernomas, most of which have characteristic clonal chromosomal aberrations. The normal karyotype of subcutaneous angiolipoma as well as its distinct clinical and morphologic features suggest a different pathogenesis from pure lipomas.

Adult

The treatment of multiple angiolipomas by liposuction surgery.

BACKGROUND: Recently, liposuction surgery has been widely accepted for body contouring or other noncosmetic applications, including lipoma and angiolipomatosis. OBJECTIVE: We treated a case of multiple angiolipomas at 49 years of age to confirm the utility of liposuction. METHODS: Liposuction under general anesthesia was performed for angiolipomas distributed over the trunk. RESULTS: The patient has been followed for 2 years without recurrence. CONCLUSION: We believe that liposuction is a safe and easy method of removing multiple angiolipomas.

Adipose Tissue