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Spinal extradural angiolipoma: A report of two cases and review of the literature
Surg Neurol 1990;34:173-8
173
Spinal Extradural Angiolipoma: A Report of Two Cases and Review of the Literature J o h n A. A n s o n , M . D . , G e o r g e R. C y b u l s k i , M . D . , a n d M a r c R e y e s , M . D . Departments of Neurosurgery and Pathology, Cook County Hospital, Hektoen Institute, Chicago, Illinois
AnsonJA, CybulskiGR, ReyesM. Spinal extraduralangiolipoma:a report of two cases and reviewof the literature. Surg Neurol 1990;34:173-8. Extradural angiolipomas are rare tumors that can produce spinal cord compression. Two patients with thoracic spinal angiolipoma are presented that were treated with surgical resection and radiation. The histological and clinical features of the 18 previously reported cases of these tumors are discussed. KEY WORDS: Angiolipoma; Spinal cord tumor; Spinal cord compression
Extradural angiolipomas are a rare cause of spinal cord compression, with only 18 cases reported in the literature [ 1-15]. This report describes two cases of thoracic spinal angiolipoma and reviews the previously reported cases.
Case R e p o r t s Case 1
A 58-year-old Hispanic woman was admitted to Cook County Hospital with a history of progressively worsening mid-thoracic back pain for 3 months, numbness of both legs for 2 months, and increasing leg weakness for 2 weeks with inability to walk. She had no history of trauma and no other significant medical history. Examination revealed that cranial nerves and upper extremities were normal. There was no spinal tenderness or pain with straight leg raising, and normal rectal sphincter tone. There was moderate muscle weakness throughout both lower extremities, and decreased sensation to pinprick, light touch, and proprioception below the T5-T6 level. Deep tendon reflexes in the legs were very hyperactive and Babinski signs were present bilaterally.
Address reprint requeststo: George R. Cybulski,M.D., Divisionof Neurosurgery, Northwestern University Medical School, 233 East Erie, Suite 500, Chicago,Illinois60611. Received August28, 1989; accepted March 13, 1990.
© 1990by ElsevierSciencePublishingCo.,Inc.
X-rays of the spine were normal. Myelography by lumbar and cervical routes revealed complete block to contrast flow from T3 to T5 (Figure 1). Postmyelogram computed tomography (CT) scan showed a large extradural mass located mostly posteriorly and on the right side of the spinal canal, with anterolateral displacement of the thecal sac (Figure 2). The patient was taken to the operating room and a T2-T7 bilateral laminectomy was performed. A soft, gray, friable mass was encountered extending from T2 to T6 posterior to the dural sac and wrapping anteriorly around the sac on the right side at the T3-T4 level. Profuse bleeding from the mass was encountered, although the mass dissected easily from the underlying dura mater. The mass was removed from the posterior aspect of the spinal canal where it had completely compressed the cord. A remnant of tissue extending into the right anterolateral gutter at T3-T4 could not be resected and was left in place. The postoperative course was uneventful, and magnetic resonance imaging (MRI) was performed on the sixth postoperative day, showing decompression of the spinal cord with a small amount of residual tumor anterolaterally at T4 (Figure 3). A 3000-rad course of radiation was given starting on the tenth postoperative day. Physical therapy was initiated and the patient was discharged to a rehabilitation setting. On last examination 10 weeks after surgery she was ambulating independently and had normal strength and sensation. She continues to improve with physical therapy. Histological examination revealed red soft tissue showing adipose tissue, with multiple thin-walled vascular channels, consistent with angiolipoma (Figure 4). Case 2
A 65-year-old black woman with a history of Alzheimer's-type dementia, hypertension, and heart disease presented to Cook County Hospital with progressive leg weakness for 2 days, with inability to walk since the preceding day. She had no pain or incontinence. Examination revealed a flaccid paraplegia with areflexia and a dense sensory loss below the T6 level, includ0090-3019/90/$3.50
174
Surg Neurol 1990;34:173-8
Figure 1. (A) Lumbar anteroposterior, (B) cervical anteroposterior, and (C) lumbar lateral myelogram views showing extradural compression of thecal sac from an apparent posterior and right-sided mass.
ing pain, light touch, pinprick, and proprioception. There was no spinal tenderness or deformity. Rectal sphincter tone was decreased. X-rays o f the spine were normal. Myelography showed complete block to contrast flow from T1 to T6. Postmyelogram CT scan showed a posterior extradural mass that continued down to T9, with compression and anterior displacement of the thecal sac (Figure 5). The patient was paraplegic for approximately 24 hours prior to surgery. She underwent T1 to T7 laminectomy, exposing an abundance of extradural clot and red, friable tumor posterior to the dural sac. The predominant mass lesion was extradural clot from T1 to T7. The lesion was grossly completely removed. Postoperatively, there was return o f sensation to pain but no return of motor function. H e r postoperative course was otherwise unremarkable and she was transferred to a rehabilitation facility 2 weeks after surgery. Histological examination o f the tumor was similar to case 1, showing fibroadipose tissue with numerous thin-walled blood vessels, consistent with angiolipoma. Discussion Angiolipomas o f the spinal canal are rare tumors, with only 18 cases reported in the literature (Table 1). Angiolipoma was first described by Berenbruch [3] in 1890,
Anson et al
and has been found to occur slightly more commonly in females, with a male to female ratio o f 1 : 1.4, and usually occurs in middle age, with an age range o f 16 to 67 years, with 65% occurring in the fifth and sixth decades. Fourteen cases were located in the thoracic spine, two in the cervicothoracic spine, two in the thoracolumbar spine, and one in the lumbar spine. Symptoms o f back or neck pain were often o f long duration, with all patients having symptoms for greater than 1 month and 70% for greater than a year. Patients most commonly had longstanding back pain and then developed progressive neurologic symptoms secondary to cord compression in all cases. As with other vascular tumors, presentation or
Figure 2. Postmyelogram CT scan at the T4 levd showing anterolateral displacement of the thecal sac by an extradural soft tissue mass (arrow).
Spinal Extradural Angiolipoma
Surg Neurol 1990;34:173-8
175
A
Figure 3. (A) PostoperatioeMRI showing spinal cord d¢compression and a small amount of residual tumor apl~aring as a high signal antsrolatrral mass at T4 (an,owL (B) Line drawing demonstrating the extent of involvemont of the spine by tumor (T__).
deterioration during pregnancy may occur, and pregnancy appeared to be significant in four of the 11 female cases [1,5,6]. Cull et al [5], in their report of two such cases, hypothesized that blood and fluid volume changes caused increased cord compression as well as possible tumor vascular "steal" or pulsatile effects on the adjacent spinal cord. The histologic appearance of angiolipomas shows a meshwork of adipose cells with numerous thin-walled vascular channels scattered among them. The vascular channels are lined with a single layer of flattened endothelial cells and there are occasional small, well-formed arteries seen. This appearance is similar to the more common angiolipomata of skeletal muscle [7], but differs from the angiomyolipomata often found in the kidney, which have also been reported to occur in the extradural space causing cord compression [14]. The latter tumors have hyperplastic smooth muscle in the vessel
B
walls and often osteoid trabeculae within the adipose tissue. Spinal angiolipoma was initially considered to be a hypervascular variant of spinal lipomas, but clinically an angiolipoma appears to be a distinct entity. Angiolipomas, with the exception of one intrameduUary tumor [13], are extradural, while spinal lipomas frequently are located intraparenchymally. Angiolipomas also do not have the association with congenital spinal dysraphic abnormalities that spinal hpomas do. In addition, angiolipomas are usually only loosely adherent to surrounding tissue, making complete excision often feasible, while lipomas often have marked adhesion and infiltration into surrounding tissues, making complete removal difficult.
A
Figure 4. (A) Tumor section from case 1 showing typical mixture of mature fat cells and numerous narrow vascular channels (hematoxylin and eosin stain, bar = 1 O0 Izm). (B) Tumor section from case 2 also demonstrates the mixture of fat cells admixed with vascular channels (hematoxylin and eosin stain, bar = 100 Izm).
,)) //
~!,~!i~ ¸, ~
A
B
Figure 5. (A) Lino drawing demonstrating the relationship of the tumor (T_) to the spinal cord (f~.). (B) Postmydogram CT scan at the T7 level showing reappearance of intrathocal raetrizamide with compression of the thecal sac by a posterior soft tissue mass (arrowL
T a b l e 1. Clinical and Radiologic Features of Cases of Spinal Angiolipoma Reported in the Literature Case no.
1 2 3 4 5 6 7 8 9
Age
Duration of
Level of
Sex
(yr)
symptoms
lesion
X-ray findings"
M F F F
16 51 20 43
2.5 mo 4.5 yr 16 yr 1 yr
Thoracic T7-T8 Thoracic T6-T10
--~
F M M
30 67 33
3 yr 1 yr 2.5 mo
T6-T8 T8-T9 C6-T5
Taylor et al [15]
F F
51 44
9 yr 2 yr
T3-T7 T11-L1
Gonzalez-Crussi et al [7] Cull et al [5]
20 50 45 46 50 52 42 46 27
3 yr 14 yr 5 yr 2 yr 1 mo 5 mo 1 yr 2 yr 5 mo
L1-L4 T8-T9 T6-T8 T3-T5 T4-T6 T4-T6 T4-T5 T11-L2 C6-T2
58 65
3 mo 2 days
T2-T6 T1-T8
Author
Berenbruch [3] Liebscher [9] Balado and Morea [1] Petit-Dutaillia and Christophe [14] Ehni and Love [6] Bucy and Ritchey [4]
10 11 12 13 14 15 16 17 18
Mikit et al [10] Padovani et al [11] Padovani et al [11] Hanakita and Koyama [8] Bardosi et al [2] Palkovic et al [12]
F F F F M M M M M
19 20
Present study Present study
F F
Abbreviations: F, female; M, male; MR/, magnetic resonance imaging. • Description of block refers to myelogram findings.
Partial block* Partial block Partial block Pedicle erosion T 1 - T 5 (Klippel-Feil syndrome) Partial block T3 Pedicle erosion T11-L1, partial block T10-T11 Lamina erosion L3 Partial block T 8 - T 9 Partial block T8 Complete block T 3 - T 5 Complete block T6 Partial block T6 Partial block T5 Complete block T11-L2 Complete block C6-T2, intramedullary tumor on MRI Complete block T 3 - T 5 Complete block T 1 - T 6
Treatment--outcome Operation~autopsy Autopsy Autopsy Operationwrecovery Operation~recovery Operation~recovery Operation, 2050 rads--recovery Operationmrecovery Operation--recovery Operation, 3018 rads--recovery Operarton--recovery Operarlon--recovery Operatton--recovery Operattonmrecovery Operatlon--partial recovery Operar~on--recovery Operataon--recovery Operauon--recovery Operation, 3016 rads--recovery Operation--minimal improvement
178
Surg Neurol 1990;34:173-8
The histogenesis of angiolipomas is still unclear. One theory, proposed by Ehni and Love [6], suggests they originate from primitive pluripotential mesenchymal cells that can differentiate into lipomatous, angiomatous, or mixed tissue. Others consider angiolipomas to be true hamartomas. One ultrastructural study of a spinal angiolipoma, by Bardosi et al [2], reported finding secretory activity by the adipocytes with lipidlike material in perivascular granules. The nature and function of the material, which is released by exocytosis into the vascular channel, are not known. Angiolipomas appear to be slow growing, as evidenced by the prolonged course of most patients, but may cause sudden deterioration by thrombosis or hemorrhage, as well as vascular enlargement. Treatment is surgical excision, which can often be complete. In the absence of complete surgical removal, adjuvant radiation therapy should be considered to prevent further growth of residual tumor. In two of the three previously reported cases of incomplete tumor removal [4,7,13], local radiation therapy was given postoperatively in doses of 2050-3018 rads [4,7], with no evidence of recurrence at 24 and 37 months, respectively. Lastly, the outcome of most patients is very good after surgical excision, often in spite of severe progressive neurologic deterioration preoperatively. All except one [14] of the 16 patients treated surgically since 1928 have made a good or complete recovery.
We would like to thank Mrs. E. Daniels for her assistance in the preparation of the manuscript, and Dr. F. Homsi for assistance in the preparation of the photomicrographs.
Anson et al
References I. Balado M, Morea R. Hemangioma extramedular producieudo paraplejias durante el embarzo. Arch Argent Neuro11928; 1:345-51. 2. Bardosi A, Schoake I, Friede RL, Roessmann V. Extradural spinal angiolipoma with secretory activity. Virchows Arch 1985;406:253-9. 3. Berenbruch K. Ein Fall yon multiplen Angiolipomen kombiniert mit eniem Angiom des Ruckenmarks. Tubingen, 1890. 4. Bucy PC, Ritchey H. Klippel-Feil's syndrome associated with compression at the spinal cord by an extradural hemangiolipoma. J Neurosurg 1947;4:476-81. 5. Cull DJ, Erdohazi M, Symon L. Extradural hemangiolipoma in the spinal canal. Two cases presenting during pregnancy. Acta Neurochir 1978;45:187-93. 6. Ehni G, Love JG. Intraspinal lipomas: report of cases; review of the literature, and clinical and pathologic study. Arch Neurol Psychiatry 1945;53:1-28. 7. Gonzalez-Crussi F, Enneking WF, Arean VM. Infiltrating angiolipoma. J Bone Joint Surg 1966;48:1111-23. 8. Hanakita J, Koyama T. Hemangiolipoma. No Shinkei Geka 1982;10:313-6. 9. Liebscher C. Angiolipom des Wirbelchanls mit compression des Ruckenmarkes. Prager Medicinische Wochenschrift 1901;26:189-91. 10. Miki T, Oka M, Shima M, Hirofuji E, Tanaka S. Spinal angiolipoma. Acta Neurochir 1981;58:115-9. 11. Padovani R, Tognetti F, Speranza S, Pozzati E. Spinal extrathecal hemangiolipomas: report of two cases and review of the literature. Neurosurgery 1982;11:674-7. 12. Palkovic S, Wassmann H, Bonse R, Kashab M. Angiolipoma of the spinal cord: magnetic resonance imaging and microsurgical management. Surg Neurol 1988;29:243-5. 13. Pearson J, Stellar S, Feigin I. Angiomyolipoma: long-term care following radical approach to malignant-appearing benign intraspinal tumor. J Neurosurg 1970;33:466-70. 14. Petit-Dutaillia D, Christophe J. Compression meduUaire par volumineux angio-lipome extra-dural: ablation chirurgicate (Guerison). Rev Neurol (Paris) 1931;2:284-7. 15. Taylor J, Harries BJ, Schurr PH. Extrathecal haemangiolipomas of the spinal canal. B r J Surg 1951;39:1-7.
173
Spinal Extradural Angiolipoma: A Report of Two Cases and Review of the Literature J o h n A. A n s o n , M . D . , G e o r g e R. C y b u l s k i , M . D . , a n d M a r c R e y e s , M . D . Departments of Neurosurgery and Pathology, Cook County Hospital, Hektoen Institute, Chicago, Illinois
AnsonJA, CybulskiGR, ReyesM. Spinal extraduralangiolipoma:a report of two cases and reviewof the literature. Surg Neurol 1990;34:173-8. Extradural angiolipomas are rare tumors that can produce spinal cord compression. Two patients with thoracic spinal angiolipoma are presented that were treated with surgical resection and radiation. The histological and clinical features of the 18 previously reported cases of these tumors are discussed. KEY WORDS: Angiolipoma; Spinal cord tumor; Spinal cord compression
Extradural angiolipomas are a rare cause of spinal cord compression, with only 18 cases reported in the literature [ 1-15]. This report describes two cases of thoracic spinal angiolipoma and reviews the previously reported cases.
Case R e p o r t s Case 1
A 58-year-old Hispanic woman was admitted to Cook County Hospital with a history of progressively worsening mid-thoracic back pain for 3 months, numbness of both legs for 2 months, and increasing leg weakness for 2 weeks with inability to walk. She had no history of trauma and no other significant medical history. Examination revealed that cranial nerves and upper extremities were normal. There was no spinal tenderness or pain with straight leg raising, and normal rectal sphincter tone. There was moderate muscle weakness throughout both lower extremities, and decreased sensation to pinprick, light touch, and proprioception below the T5-T6 level. Deep tendon reflexes in the legs were very hyperactive and Babinski signs were present bilaterally.
Address reprint requeststo: George R. Cybulski,M.D., Divisionof Neurosurgery, Northwestern University Medical School, 233 East Erie, Suite 500, Chicago,Illinois60611. Received August28, 1989; accepted March 13, 1990.
© 1990by ElsevierSciencePublishingCo.,Inc.
X-rays of the spine were normal. Myelography by lumbar and cervical routes revealed complete block to contrast flow from T3 to T5 (Figure 1). Postmyelogram computed tomography (CT) scan showed a large extradural mass located mostly posteriorly and on the right side of the spinal canal, with anterolateral displacement of the thecal sac (Figure 2). The patient was taken to the operating room and a T2-T7 bilateral laminectomy was performed. A soft, gray, friable mass was encountered extending from T2 to T6 posterior to the dural sac and wrapping anteriorly around the sac on the right side at the T3-T4 level. Profuse bleeding from the mass was encountered, although the mass dissected easily from the underlying dura mater. The mass was removed from the posterior aspect of the spinal canal where it had completely compressed the cord. A remnant of tissue extending into the right anterolateral gutter at T3-T4 could not be resected and was left in place. The postoperative course was uneventful, and magnetic resonance imaging (MRI) was performed on the sixth postoperative day, showing decompression of the spinal cord with a small amount of residual tumor anterolaterally at T4 (Figure 3). A 3000-rad course of radiation was given starting on the tenth postoperative day. Physical therapy was initiated and the patient was discharged to a rehabilitation setting. On last examination 10 weeks after surgery she was ambulating independently and had normal strength and sensation. She continues to improve with physical therapy. Histological examination revealed red soft tissue showing adipose tissue, with multiple thin-walled vascular channels, consistent with angiolipoma (Figure 4). Case 2
A 65-year-old black woman with a history of Alzheimer's-type dementia, hypertension, and heart disease presented to Cook County Hospital with progressive leg weakness for 2 days, with inability to walk since the preceding day. She had no pain or incontinence. Examination revealed a flaccid paraplegia with areflexia and a dense sensory loss below the T6 level, includ0090-3019/90/$3.50
174
Surg Neurol 1990;34:173-8
Figure 1. (A) Lumbar anteroposterior, (B) cervical anteroposterior, and (C) lumbar lateral myelogram views showing extradural compression of thecal sac from an apparent posterior and right-sided mass.
ing pain, light touch, pinprick, and proprioception. There was no spinal tenderness or deformity. Rectal sphincter tone was decreased. X-rays o f the spine were normal. Myelography showed complete block to contrast flow from T1 to T6. Postmyelogram CT scan showed a posterior extradural mass that continued down to T9, with compression and anterior displacement of the thecal sac (Figure 5). The patient was paraplegic for approximately 24 hours prior to surgery. She underwent T1 to T7 laminectomy, exposing an abundance of extradural clot and red, friable tumor posterior to the dural sac. The predominant mass lesion was extradural clot from T1 to T7. The lesion was grossly completely removed. Postoperatively, there was return o f sensation to pain but no return of motor function. H e r postoperative course was otherwise unremarkable and she was transferred to a rehabilitation facility 2 weeks after surgery. Histological examination o f the tumor was similar to case 1, showing fibroadipose tissue with numerous thin-walled blood vessels, consistent with angiolipoma. Discussion Angiolipomas o f the spinal canal are rare tumors, with only 18 cases reported in the literature (Table 1). Angiolipoma was first described by Berenbruch [3] in 1890,
Anson et al
and has been found to occur slightly more commonly in females, with a male to female ratio o f 1 : 1.4, and usually occurs in middle age, with an age range o f 16 to 67 years, with 65% occurring in the fifth and sixth decades. Fourteen cases were located in the thoracic spine, two in the cervicothoracic spine, two in the thoracolumbar spine, and one in the lumbar spine. Symptoms o f back or neck pain were often o f long duration, with all patients having symptoms for greater than 1 month and 70% for greater than a year. Patients most commonly had longstanding back pain and then developed progressive neurologic symptoms secondary to cord compression in all cases. As with other vascular tumors, presentation or
Figure 2. Postmyelogram CT scan at the T4 levd showing anterolateral displacement of the thecal sac by an extradural soft tissue mass (arrow).
Spinal Extradural Angiolipoma
Surg Neurol 1990;34:173-8
175
A
Figure 3. (A) PostoperatioeMRI showing spinal cord d¢compression and a small amount of residual tumor apl~aring as a high signal antsrolatrral mass at T4 (an,owL (B) Line drawing demonstrating the extent of involvemont of the spine by tumor (T__).
deterioration during pregnancy may occur, and pregnancy appeared to be significant in four of the 11 female cases [1,5,6]. Cull et al [5], in their report of two such cases, hypothesized that blood and fluid volume changes caused increased cord compression as well as possible tumor vascular "steal" or pulsatile effects on the adjacent spinal cord. The histologic appearance of angiolipomas shows a meshwork of adipose cells with numerous thin-walled vascular channels scattered among them. The vascular channels are lined with a single layer of flattened endothelial cells and there are occasional small, well-formed arteries seen. This appearance is similar to the more common angiolipomata of skeletal muscle [7], but differs from the angiomyolipomata often found in the kidney, which have also been reported to occur in the extradural space causing cord compression [14]. The latter tumors have hyperplastic smooth muscle in the vessel
B
walls and often osteoid trabeculae within the adipose tissue. Spinal angiolipoma was initially considered to be a hypervascular variant of spinal lipomas, but clinically an angiolipoma appears to be a distinct entity. Angiolipomas, with the exception of one intrameduUary tumor [13], are extradural, while spinal lipomas frequently are located intraparenchymally. Angiolipomas also do not have the association with congenital spinal dysraphic abnormalities that spinal hpomas do. In addition, angiolipomas are usually only loosely adherent to surrounding tissue, making complete excision often feasible, while lipomas often have marked adhesion and infiltration into surrounding tissues, making complete removal difficult.
A
Figure 4. (A) Tumor section from case 1 showing typical mixture of mature fat cells and numerous narrow vascular channels (hematoxylin and eosin stain, bar = 1 O0 Izm). (B) Tumor section from case 2 also demonstrates the mixture of fat cells admixed with vascular channels (hematoxylin and eosin stain, bar = 100 Izm).
,)) //
~!,~!i~ ¸, ~
A
B
Figure 5. (A) Lino drawing demonstrating the relationship of the tumor (T_) to the spinal cord (f~.). (B) Postmydogram CT scan at the T7 level showing reappearance of intrathocal raetrizamide with compression of the thecal sac by a posterior soft tissue mass (arrowL
T a b l e 1. Clinical and Radiologic Features of Cases of Spinal Angiolipoma Reported in the Literature Case no.
1 2 3 4 5 6 7 8 9
Age
Duration of
Level of
Sex
(yr)
symptoms
lesion
X-ray findings"
M F F F
16 51 20 43
2.5 mo 4.5 yr 16 yr 1 yr
Thoracic T7-T8 Thoracic T6-T10
--~
F M M
30 67 33
3 yr 1 yr 2.5 mo
T6-T8 T8-T9 C6-T5
Taylor et al [15]
F F
51 44
9 yr 2 yr
T3-T7 T11-L1
Gonzalez-Crussi et al [7] Cull et al [5]
20 50 45 46 50 52 42 46 27
3 yr 14 yr 5 yr 2 yr 1 mo 5 mo 1 yr 2 yr 5 mo
L1-L4 T8-T9 T6-T8 T3-T5 T4-T6 T4-T6 T4-T5 T11-L2 C6-T2
58 65
3 mo 2 days
T2-T6 T1-T8
Author
Berenbruch [3] Liebscher [9] Balado and Morea [1] Petit-Dutaillia and Christophe [14] Ehni and Love [6] Bucy and Ritchey [4]
10 11 12 13 14 15 16 17 18
Mikit et al [10] Padovani et al [11] Padovani et al [11] Hanakita and Koyama [8] Bardosi et al [2] Palkovic et al [12]
F F F F M M M M M
19 20
Present study Present study
F F
Abbreviations: F, female; M, male; MR/, magnetic resonance imaging. • Description of block refers to myelogram findings.
Partial block* Partial block Partial block Pedicle erosion T 1 - T 5 (Klippel-Feil syndrome) Partial block T3 Pedicle erosion T11-L1, partial block T10-T11 Lamina erosion L3 Partial block T 8 - T 9 Partial block T8 Complete block T 3 - T 5 Complete block T6 Partial block T6 Partial block T5 Complete block T11-L2 Complete block C6-T2, intramedullary tumor on MRI Complete block T 3 - T 5 Complete block T 1 - T 6
Treatment--outcome Operation~autopsy Autopsy Autopsy Operationwrecovery Operation~recovery Operation~recovery Operation, 2050 rads--recovery Operationmrecovery Operation--recovery Operation, 3018 rads--recovery Operarton--recovery Operarlon--recovery Operatton--recovery Operattonmrecovery Operatlon--partial recovery Operar~on--recovery Operataon--recovery Operauon--recovery Operation, 3016 rads--recovery Operation--minimal improvement
178
Surg Neurol 1990;34:173-8
The histogenesis of angiolipomas is still unclear. One theory, proposed by Ehni and Love [6], suggests they originate from primitive pluripotential mesenchymal cells that can differentiate into lipomatous, angiomatous, or mixed tissue. Others consider angiolipomas to be true hamartomas. One ultrastructural study of a spinal angiolipoma, by Bardosi et al [2], reported finding secretory activity by the adipocytes with lipidlike material in perivascular granules. The nature and function of the material, which is released by exocytosis into the vascular channel, are not known. Angiolipomas appear to be slow growing, as evidenced by the prolonged course of most patients, but may cause sudden deterioration by thrombosis or hemorrhage, as well as vascular enlargement. Treatment is surgical excision, which can often be complete. In the absence of complete surgical removal, adjuvant radiation therapy should be considered to prevent further growth of residual tumor. In two of the three previously reported cases of incomplete tumor removal [4,7,13], local radiation therapy was given postoperatively in doses of 2050-3018 rads [4,7], with no evidence of recurrence at 24 and 37 months, respectively. Lastly, the outcome of most patients is very good after surgical excision, often in spite of severe progressive neurologic deterioration preoperatively. All except one [14] of the 16 patients treated surgically since 1928 have made a good or complete recovery.
We would like to thank Mrs. E. Daniels for her assistance in the preparation of the manuscript, and Dr. F. Homsi for assistance in the preparation of the photomicrographs.
Anson et al
References I. Balado M, Morea R. Hemangioma extramedular producieudo paraplejias durante el embarzo. Arch Argent Neuro11928; 1:345-51. 2. Bardosi A, Schoake I, Friede RL, Roessmann V. Extradural spinal angiolipoma with secretory activity. Virchows Arch 1985;406:253-9. 3. Berenbruch K. Ein Fall yon multiplen Angiolipomen kombiniert mit eniem Angiom des Ruckenmarks. Tubingen, 1890. 4. Bucy PC, Ritchey H. Klippel-Feil's syndrome associated with compression at the spinal cord by an extradural hemangiolipoma. J Neurosurg 1947;4:476-81. 5. Cull DJ, Erdohazi M, Symon L. Extradural hemangiolipoma in the spinal canal. Two cases presenting during pregnancy. Acta Neurochir 1978;45:187-93. 6. Ehni G, Love JG. Intraspinal lipomas: report of cases; review of the literature, and clinical and pathologic study. Arch Neurol Psychiatry 1945;53:1-28. 7. Gonzalez-Crussi F, Enneking WF, Arean VM. Infiltrating angiolipoma. J Bone Joint Surg 1966;48:1111-23. 8. Hanakita J, Koyama T. Hemangiolipoma. No Shinkei Geka 1982;10:313-6. 9. Liebscher C. Angiolipom des Wirbelchanls mit compression des Ruckenmarkes. Prager Medicinische Wochenschrift 1901;26:189-91. 10. Miki T, Oka M, Shima M, Hirofuji E, Tanaka S. Spinal angiolipoma. Acta Neurochir 1981;58:115-9. 11. Padovani R, Tognetti F, Speranza S, Pozzati E. Spinal extrathecal hemangiolipomas: report of two cases and review of the literature. Neurosurgery 1982;11:674-7. 12. Palkovic S, Wassmann H, Bonse R, Kashab M. Angiolipoma of the spinal cord: magnetic resonance imaging and microsurgical management. Surg Neurol 1988;29:243-5. 13. Pearson J, Stellar S, Feigin I. Angiomyolipoma: long-term care following radical approach to malignant-appearing benign intraspinal tumor. J Neurosurg 1970;33:466-70. 14. Petit-Dutaillia D, Christophe J. Compression meduUaire par volumineux angio-lipome extra-dural: ablation chirurgicate (Guerison). Rev Neurol (Paris) 1931;2:284-7. 15. Taylor J, Harries BJ, Schurr PH. Extrathecal haemangiolipomas of the spinal canal. B r J Surg 1951;39:1-7.