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Microscopic section of the hemangioma demonstrates bone lamella and closely opposed benign endothelial-lined blood vessels along with erythrocytes in the lumen of the vessels (hematoxylin and eosin, ×10)
Source publication
In this paper, we report a case of vertebral hemangioma during pregnancy in a 21-year-old woman presenting with paraparesis of rapid onset. An emergency MRI scan of the dorsal spine showed a lesion of the ninth thoracic vertebra with extradural extension and marked spinal cord compression. A cesarean section was done, and this was followed by emerg...
Citations
... The vast majority of these lesions are asymptomatic. [11] The term "aggressive hemangioma" refers to a vertebral hemangioma with extraosseous extension. [2] The affected vertebra is usually located between T3 and T8. ...
Rationale:
Compressive myelopathy and compression fracture of aggressive vertebral hemangioma after parturition is a rare condition. Vertebral body compression fracture and high serum progesterone lead to extraosseous hemangioma enlargment cause narrowing the spinal canal which contribute to compressive myelopathy relate to pregnancy.
Patient concerns:
We report a case of compressive myelopathy and compression fracture of aggressive vertebral hemangioma after parturition in a 35-year-old woman. The patient complained unable to walk and experienced intense pain in the back.
Diagnosis:
Based on the clinical features and imaging studies, the patient underwent a T4-T6 laminectomy. Histopathology consistent with vertebral hemangioma.
Interventions:
The patient underwent laminectomy for decompression. After subperiosteal dissection of the paraspinal muscles and exposure of the laminae, there was no involvement of the lamina by the tumor. The epidural tumor was removed through the spaces lateral to the thecal sac. Vertebroplasty was performed through T5 pedicles bilaterally and 7 ml of polymethylmethacrylate (PMMA) cement was injected. T4-T6 pedicle screw fixation was performed for segmental fixation and fusion.
Outcomes:
Six months after resection of the tumor the patient remained asymptomatic. She reported no low back pain and had returned to her normal daily activities, with no radiographic evidence of recurrence on MRI. Physical examination revealed that superficial and deep sensation was restored to normal levels in the lower extremities.
Lessons:
The occurrence of compressive myelopathy of pregnancy related vertebral hemangiomas is quite unusual. It can lead to serious neurologic deficits if not treated immediately. So, prompt diagnosis is important in planning optimal therapy and preventing morbidity for patients.
... [15] There are a few case reports in the literature that discuss the treatment of aggressive VHs during pregnancy; however, most are sporadic case reports, and the treatment remains controversial. [8][9][10][11][12][14][15][16][17][18][19][20][21][22][23][24][25][26][27][28][29][30][31][32] Our hospital has treated 95 patients with aggressive VHs in the past 15 years, including 3 with progressive neurological deficits that developed during their pregnancies. Here, we report our experience of aggressive VHs during pregnancy and the results of a literature review of articles published in the past 30 years. ...
... Epidural hemangiomas without vertebral body involvement, hemangiomas without symptoms, and cases with incomplete data were excluded (Table 1). [8][9][10][11][12][14][15][16][17][18][19][20][21][22][23][24][25][26][27][28][29][30][31][32] A total of 32 cases (including the present cases) were identified in literature published in the past 30 years. The average age at diagnosis was 27.1 years (range, [19][20][21][22][23][24][25][26][27][28][29][30][31][32][33][34][35]. ...
... [8][9][10][11][12][14][15][16][17][18][19][20][21][22][23][24][25][26][27][28][29][30][31][32] A total of 32 cases (including the present cases) were identified in literature published in the past 30 years. The average age at diagnosis was 27.1 years (range, [19][20][21][22][23][24][25][26][27][28][29][30][31][32][33][34][35]. The symptoms started in the first trimester of gestation in one (3.1%) ...
Rationale:
Vertebral hemangiomas (VHs), one of the most common benign tumors of the spine, can be aggressive, which is a rare condition and causes neurological deficits. Pregnancy is related to the worsening of aggressive VHs. The diagnosis and treatment of aggressive VHs remain challenging, especially for pregnant cases.
Patient concerns:
We report 3 cases of aggressive VH in women who developed progressive neurological deficits during pregnancy among 95 patients treated for aggressive VH in our hospital in the past 15 years.
Diagnoses and interventions:
All 3 patients experienced progressive deterioration of neurological function and pain at 13, 28, and 41 weeks' gestation. On radiological examination, VHs were the suspected radiological diagnoses in 2 patients; 1 patient was preoperatively misdiagnosed with a spinal metastatic tumor. All 3 patients underwent decompression surgery with intraoperative vertebroplasty and/or postoperative radiotherapy. The pathological diagnosis after surgery was all hemangiomas.
Outcomes:
In all 3 patients, there were no tumor recurrences, and neurological functions remained normal at the last follow-up of 75, 38, and 15 months after the treatment, respectively.
Lessons:
Pregnancy might lead to the onset of aggressive VHs. The diagnosis and treatment of VHs during pregnancy remain controversial due to concern for both maternal and fetal safety. Timely surgery could preserve neurological function. Decompression surgery by laminectomy followed by adjuvant therapies require less skill and have a shorter surgery time, and can be considered more appropriate for aggressive VHs with pregnancy.
... The third trimester, in particular, represents a high-risk period where previously asymptomatic vertebral hemangiomas may cause a variety of neurological symptoms such as radicular pain, sensory and motor deficits, even incontinence. 3,4 The implicated mechanisms include pregnancy-related hemodynamic, mechanical, and hormonal changes which result in vascular distension and growth of these vessel-rich tumours. 1 Herein, we describe an unusual case of vertebral fracture due to a hemangioma presenting as postpartum back pain following epidural analgesia. The possible mechanisms involved in this complication along with the differential diagnosis and management of such cases are discussed. ...
... In females, radicular pain and neurological deficits associated with vertebral hemangiomas may develop during pregnancy. 4,5 Compression of the inferior vena cava by the gravid uterus and elevation of intra-abdominal pressure enhance the blood flow through the vertebral venous plexus. This mechanism, along with a 30-50% increase in circulating blood volume, may gradually lead to expansion of an existing vertebral hemangioma. 1 Another implicated factor is possibly maternal progesterone which affects venous distensibility; moreover, estrogens may contribute to the enlargement of vertebral hemangiomas via their growth effects on endothelium. ...
( Can J Anesth . (2015) 62:901–906)
Vertebral hemangiomas are benign vascular tumors that tend to develop within the vertebra and occur in roughly 10% of the population. These tumors do not typically grow quickly and do not cause any symptoms as long as they remain within the vertebra. However, if these tumors grow beyond the spine’s bony structures, they can produce symptoms related to nerve root compression or compression of the spinal cord. Similarly aggressive development of vertebral hemangiomas can become apparent in pregnant patients, especially during the third trimester when previously undiagnosed tumors may begin to cause a number of neurological symptoms, including radicular pain, sensory and motor deficits, and incontinence. This article details an atypical case in which vertebral hemangiomas resulted in a vertebral fracture that presented at first as postpartum back pain following epidural analgesia.
The patient in question was a 32-year-old North African pregnant woman who was at 40+4 weeks gestation. She had previously experienced 3 uncomplicated deliveries without epidural analgesia. At 162 cm in height and weighing 96 kg, she had gained roughly 27 kg during this pregnancy, which was significantly more than what she had gained with her other pregnancies. The patient did not have a history of back pain or trauma and did not suffer from any neurological or coagulation disorders. She had never received general or neuraxial anesthesia before this pregnancy.
... She was alerted of the risk of worsening of neurological conditions and irreversible paraplegia. Guthkelch [21] 34 T6 1 month Death Askenasy and Behmoaram [20] 34 T10 15 days Complete Fields and Jones [19] 28 T10 3 months Complete Newman [18] 32 L3 8 months Complete Newman [18] 36 T4 1 month Complete Newman [18] 32 T4-5 3 months Death Nelson [17] 28 T2-4 1 month Partial Esparza et al. [16] 24 T5-7 2 months Complete Faria et al. [15] 32 T4 6 months Complete Lavi et al. [14] 28 T4-6 1 month Partial Schwartz et al. [13] 30 T5 1 month Complete Liu and Yang [12] 20 T4 1 month Complete Redekop and Del Maestro [11] 32 T12 4 months Partial Tekkök et al. [2] Po T5 40 days Complete Castel et al. [5] 28 T8 few days Partial Chi et al. [4] 24 C7 25 days Partial Inamasu et al. [6] 33 L2 10 days Complete Yüksel et al. [9] 28 T9 2 months Complete Vijay et al. [7] 26 T11 8 days Complete Kiroglu et al. [8] 36 T4 few days Complete Schwartz et al. [3] Po T11 2 days Complete Shinozaki et al. [10] 28 T2 few days Complete Blecher et al. [27] 37 The decision making process also involved a gynecologist, a radiotherapist, and an interventional radiologist. The final decision was to keep the patient lying in bed under corticoid treatment until the fetus maturity detected by ultrasound imaging and functional exams could allow performing a caesarian operation. ...
Type and timing of treatment for symptomatic hemangiomas in pregnant females are challenging due to fetus survival and conflicts in neurological recovery. In this article, we report a 40-year-old female patient at pregnancy week 23 with a complicated hemangioma at T1 level. Physical examination revealed an incomplete spastic paraplegia. Patient did not accept any surgery due to child's death risk. Patient was started corticoid treatment and no more weight bearing was allowed. At the 28th week of pregnancy, the patient underwent cesarean section immediately followed by selective arterial embolization, decompression, fixation, and radiotherapy. At two-year follow-up, the patient was pain free, without any signs of local recurrence and with complete neurological recovery. A multidisciplinary approach is mandatory to save the life of the fetus without damaging the spinal cord functions of the mother.
... The third trimester, in particular, represents a high-risk period where previously asymptomatic vertebral hemangiomas may cause a variety of neurological symptoms such as radicular pain, sensory and motor deficits, even incontinence. 3,4 The implicated mechanisms include pregnancy-related hemodynamic, mechanical, and hormonal changes which result in vascular distension and growth of these vessel-rich tumours. 1 Herein, we describe an unusual case of vertebral fracture due to a hemangioma presenting as postpartum back pain following epidural analgesia. The possible mechanisms involved in this complication along with the differential diagnosis and management of such cases are discussed. ...
... In females, radicular pain and neurological deficits associated with vertebral hemangiomas may develop during pregnancy. 4,5 Compression of the inferior vena cava by the gravid uterus and elevation of intra-abdominal pressure enhance the blood flow through the vertebral venous plexus. This mechanism, along with a 30-50% increase in circulating blood volume, may gradually lead to expansion of an existing vertebral hemangioma. 1 Another implicated factor is possibly maternal progesterone which affects venous distensibility; moreover, estrogens may contribute to the enlargement of vertebral hemangiomas via their growth effects on endothelium. ...
Vertebral hemangiomas are benign vascular tumours of the bony spine which are usually asymptomatic. Pregnancy-related anatomical and hormonal changes may lead to expansion of hemangiomas and development of neurological symptoms. We present an unusual case of vertebral fracture due to an undiagnosed hemangioma presenting as postpartum back pain following epidural analgesia.
A multiparous female with an unremarkable history developed intense lumbar pain after vaginal delivery under epidural analgesia. The pain was attributed to tissue trauma associated with the epidural technique. The patient had no clinical improvement with analgesics, and her symptoms deteriorated over the following days. A magnetic resonance imaging scan revealed an acute fracture of the second lumbar vertebra (L2) with epidural extension and mild compression of the dural sac, suggesting hemangioma as the underlying cause. The patient underwent successful spinal surgery with pedicle screw fixation to stabilize the fracture.
Vertebral fractures secondary to acute expansion of a vertebral hemangioma rarely occur during vaginal delivery. In such cases, the labour epidural technique and analgesia may challenge the physician in making the diagnosis. Postpartum severe back pain should be thoroughly investigated even in the absence of neurological deficits, and osseous spinal pathology should be considered in the differential diagnosis.
... They cause pain that can be related to an osseous expansion or a pathological fracture and neurological deficits that are caused by compression of the neural elements [3]. In pregnancy, advanced hemangiomas may be seen and these require treatment [4]. Superior mesenteric artery (SMA) syndrome, first defined by Rokitansky [5] in 1842, occurs as a result of compression of the third section of the duodenum between the aorta and the SMA. ...
In pregnancy, advanced vertebral hemangiomas may be seen, and these require treatment. The case reported here is of a 35-year-old female in the 32nd week of pregnancy who was admitted to the orthopaedics clinic with a history of backache and difficulty walking. A burst fracture of L1 associated with a vertebral hemangioma was identified with an L3 compression fracture secondary to osteoporosis. The local kyphosis angle between T12 and L2 was 27°. Kyphotic deformity was corrected and postoperatively, the measured T12-L2 local kyphotic angle was 9°. Twelve hours postoperatively, oral nutrition was allowed, but she developed nausea and vomiting and twenty-four hours postoperatively, an electrolyte imbalance developed. Postoperatively, the patient was diagnosed with superior mesenteric artery syndrome. To the best of our knowledge, this is the first reported case of superior mesenteric artery syndrome, which occurred following the correction of a kyphotic deformity that had developed secondary to an advanced hemangioma in pregnancy.
... 13 Since that first publication, 27 new cases have been described (Table 1). 1,[3][4][5][6][7][8][9][10][11][13][14][15][16][18][19][20][21][22][23][24][25][26][27][28][29] The diagnosis was usually made during the third trimester (22 of 27 cases). The mean maternal age at the time of diagnosis was 30 years. ...
... Of the 27 cases reported in the literature (Table 1), 12 were described at gestational ages corresponding to severe prematurity (less than 32 weeks of gestation). [6][7][8][9]11,[19][20][21]24,26,28,29 Only half of these cases were treated antepartum. 8,[19][20][21]28 Only 1 of the 8 cases of prematurity (between 32 and 36 weeks of gestation) 1,3,10,13-15,19,23 was treated antepartum. ...
... The postpartum surgical management performed by most authors is laminectomy (12 of 19 cases), 1,[3][4][5]7,9,10,14,22,26,27,29 without a complementary procedure in half of the cases. Neurological recovery was observed in all but 1 case in this group; 26 recovery was only partial after laminectomy and the patient was subsequently treated by embolization and corpectomy. ...
Symptomatic vertebral hemangiomas during pregnancy are rare, as only 27 cases have been reported in the literature since 1948. However, symptomatic vertebral hemangiomas can be responsible for spinal cord compression, in which case they constitute a medical emergency, which raises management difficulties in the context of pregnancy. Pregnancy is a known factor responsible for deterioration of these vascular tumors. In this paper, the authors report 2 clinical cases of symptomatic vertebral hemangiomas during pregnancy, including 1 case of spontaneous fracture that has never been previously reported in the literature. The authors then present a brief review of the literature to discuss emergency management of this condition. The first case was a 28-year-old woman at 35 weeks of gestation, who presented with paraparesis. Spinal cord MRI demonstrated a vertebral hemangioma invading the body and posterior arch of T-3 with posterior epidural extension. Laminectomy and vertebroplasty were performed after cesarean section, allowing neurological recovery. The second case involved a 35-year-old woman who presented with spontaneous fracture of T-7 at 36 weeks of gestation, revealing a vertebral hemangioma with no neurological deficit, but it was responsible for pain and local instability. Treatment consisted of postpartum posterior interbody fusion. With a clinical and radiological follow-up of 2 years, no complications and no modification of the hemangiomas were observed. A review of the literature reveals discordant management of these rare cases, which is why the treatment course must be decided by a multidisciplinary team as a function of fetal gestational age and maternal neurological features.
... In another case of vertebral hemangioma during pregnancy in a 21-year-old woman presenting with paraparesis of rapid onset, cesarean section was performed, and this was followed by emergency laminectomy with improvement of symptoms and neurologic deficit [4]. ...
... 10,28 A factor that appears to increase the risk of developing neurological symptoms in previously quiescent hemangiomas is pregnancy. 4,16,30,34,35,37 It has been hypothesized that the increase in intraabdominal pressure caused by the growing fetus augments blood flow to the vertebral venous plexus and that increased estrogen levels may enhance endothelial growth in hemangiomas. 10 Several features help to explain the large size of our patient's hemangioma, its aggressive growth, and the acute onset of neurological deficits. ...
Vertebral hemangiomas are common entities that rarely present with neurological deficits. The authors report the unusual case of a large L-3 vertebral hemangioma with epidural extension in a 27-year-old woman who presented with hip flexor and quadriceps weakness, foot drop, and leg pain. The characteristics of the mass on magnetic resonance imaging suggested an aggressive, hypervascular lesion. The patient underwent embolization of the lesion followed by direct intralesional injection of ethanol. Significant resolution of clinical symptoms was observed immediately after the procedure and at her follow-up visits. Follow-up imaging studies obtained 9 months after the procedure also documented a considerable reduction in the size of the hemangioma with minimal loss of vertebral height and a mild kyphosis at the affected level. On repeated imaging studies obtained 21 months postoperatively, the size of the hemangioma and the degree of vertebral body compression were stable. As demonstrated in this case, patients with vertebral hemangiomas can present with acute nerve root compression and signs and symptoms similar to those of disc herniation. Vertebral hemangiomas can be treated effectively with interventional techniques such as embolization and ethanol injection.
... T-5 paraplegia PP laminectomy complete Abi-Fadel et al., 1997 28 34 T-9 paraplegia PP laminectomy, radiotherapy complete Castel et al., 1999 27 31 T-8 paraplegia PP laminectomy complete Schwartz et al., 2000 29 41 T-11 paraplegia PP corpectomy & fusion, radiotherapy complete Chi et al., 2005 26 29 C-7 paraplegia AP corpectomy & fusion complete Inamasu et al., 2006 20 34 L-2 cauda equina syndrome PP laminectomy, vertebroplasty, posterior fusion complete Yuksel et al., 2007 21 31 T-9 paraparesis PP laminectomy complete Vijay et al., 2008 ...
Fifty-nine cases of vertebral hemangioma were seen at the Mayo Clinic between 1980 and 1990. Vertebral hemangiomas were discovered incidentally in 35 patients, while pain was the presenting complaint in 13 patients. Five patients presented directly with progressive neurological deficit requiring surgery, and six patients had surgery elsewhere for spinal cord compression and were referred for follow-up evaluation. To better define the natural history of these lesions, a historical review of these patients was conducted; progression of an asymptomatic or painful lesion to neurological symptoms was found in only two cases (mean follow-up period 7.4 years, range 1 to 35 years). New-onset back pain followed by subacute progression (mean time to progression 4.4 months, range 0.25 to 12 months) of a thoracic myelopathy was the most common presentation for patients with neurological deficit. Initially, all 11 patients with spinal cord compression underwent decompressive surgery with full neurological recovery. Recurrent neurological symptoms were observed in three of six patients following subtotal tumor resection and postoperative administration of 1000 cGy or less radiation therapy (mean follow-up period 8.7 years, range 1 to 17 years). No recurrences were noted in four patients who had subtotal excision plus radiotherapy between 2600 and 4500 cGy. One other patient had gross total tumor removal without radiotherapy and has not had a recurrence. Based on these patients and a review of the literature, the authors recommend annual neurological and radiological examinations for patients with hemangiomas associated with pain, especially young females with thoracic lesions in whom spinal cord compression is most likely to develop. Radiation therapy or embolization is an effective therapeutic alternative for patients with severe medically refractory pain. Regular follow-up monitoring for patients with asymptomatic lesions is unnecessary unless pain develops at the appropriate spinal level. It is concluded that management of patients with a progressive neurological deficit should include preoperative angiography and embolization, decompressive surgery with the approach determined by the degree of vertebral involvement and site of spinal cord compression, and postoperative radiation therapy in patients following subtotal tumor removal. Operative management and complications are discussed.
