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Benign metastasizing leiomyoma of the cervical spine 31 years after uterine leiomyoma resection
Journal of Clinical Neuroscience xxx (2015) xxx–xxx
Contents lists available at ScienceDirect
Journal of Clinical Neuroscience journal homepage: www.elsevier.com/locate/jocn
Case Report
Benign metastasizing leiomyoma of the cervical spine 31 years after uterine leiomyoma resection Aldo F. Berti b, Alejandro Santillan a,⇑, Luis A. Velasquez a a b
Department of Neurology, University of Texas Health Science Center at San Antonio, 8300 Floyd Curl Drive MSC 7883, San Antonio, TX 78229, USA Miami Neurosurgical Center, South Miami, FL, USA
a r t i c l e
i n f o
Article history: Received 8 February 2015 Accepted 14 February 2015 Available online xxxx Keywords: Cervical spine Leiomyoma Metastasis
a b s t r a c t We report a 74-year-old woman presenting with a leiomyoma of the cervical spine 31 years after uterine leiomyoma resection. Benign metastasizing leiomyoma to the cervical spine is very rare. To the best of our knowledge, this is the fourth reported patient with a leiomyoma metastasizing to the cervical spine and that with the longest latency period for this type of tumor, 31 years. The pathological features were typical of leiomyoma. Ó 2015 Elsevier Ltd. All rights reserved.
1. Introduction Leiomyomas are benign tumors composed of smooth muscle and vascular collagenous tissue, mainly occurring in the uterus. Benign metastasizing leiomyoma (BML) is an extrauterine smooth muscle tumor that occurs in patients with a current or prior history of uterine leiomyoma. Leiomyomas of the cervical spine are extremely rare, with only three reported patients with cervical leiomyoma to date [2–4]. We report a 74-year-old woman with metastasizing leiomyoma of the cervical spine and a history of uterine leiomyoma resection 31 years ago. A literature review of all other reported patients was also performed. 2. Case report A 74-year-old right handed Hispanic woman with a history of hysterectomy in 1982 presented with a history of moderate to severe neck pain radiating to the right shoulder and lateral aspect of the arm, associated with tingling sensation of the right upper extremity. At physical examination, there was no weakness of the upper and lower extremities, but there was brachioradialis hyperreflexia and sensory loss in the right upper arm and lateral fingers. An MRI showed a C6 vertebral body metastasis with flattening of the spinal cord ventrally due to the epidural component within the canal (Fig. 1). An anterior approach was performed using intraoperative somatosensory-evoked potentials for ⇑ Corresponding author. Tel.: +1 7037982246. E-mail address: [email protected] (A. Santillan).
resection of the prevertebral mass, C6 corpectomy, epidural mass decompression and resection of the mass from the right C5–6 foramen. Histologic examination of the surgical specimen revealed an intraosseous leiomyoma with no atypia, increased cellularity or mitotic activity. The neoplastic cells immunoexpressed muscle specific actin, smooth muscle actin and desmin and were negative for CD34, C-kit (CD117) and neurofilament (Fig. 2, 3). The postoperative course was uneventful and she has not had tumor recurrence during her 1 year of follow-up.
3. Discussion We present a very rare patient with a BML of the cervical spine, and a hysterectomy performed 31 years prior. BML was first described by Steiner in 1939 [1]. Leiomyomas are benign tumors of well-differentiated smooth muscle tissue and vascular collagenous tissue. In most patients, metastasis is to the pelvic cavity, retroperitoneum, heart, lung and lymph nodes. To the best of our knowledge, this is only the fourth reported patient in the literature with a BML of the cervical spine [2–4]. Symptoms may vary from neck pain [4], as seen in our patient, to progressive spastic myeloradiculopathy [2]. It is important to note that leiomyomas without histological malignant features rarely metastasize. Treatment options include hormonal manipulation [3] and surgery for symptomatic lesions causing secondary neurological deficits [2–4]. BML should be considered in the differential diagnosis of mass lesions of the cervical spine in patients who have a history of uterine leiomyoma.
http://dx.doi.org/10.1016/j.jocn.2015.02.031 0967-5868/Ó 2015 Elsevier Ltd. All rights reserved.
Please cite this article in press as: Berti AF et al. Benign metastasizing leiomyoma of the cervical spine 31 years after uterine leiomyoma resection. J Clin Neurosci (2015), http://dx.doi.org/10.1016/j.jocn.2015.02.031
2
Case Report / Journal of Clinical Neuroscience xxx (2015) xxx–xxx
Fig. 3. Benign metastasizing leiomyoma. Smooth muscle actin immuno staining at 40 magnification confirms the alpha-smooth muscle isoform of actin, consistent with the myometrium origin. (This figure is available in colour at www.sciencedirect.com.)
Conflicts of Interest/Disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication. References
Fig. 1. T2-weighted sagittal cervical spine MRI of a 74-year-old woman. At C6 there is a vertebral metastasis with a posterior convex margin and epidural disease contributing to stenosis at this level.
[1] Steiner PE. Metastasizing fibroleiomyoma of the uterus: report of a case and review of the literature. Am J Pathol 1939;15:89–110. [2] Joseph V, Chacko G, Raghuram L, et al. Benign metastasizing leiomyoma causing spinal cord compression. Surg Neurol 2003;60:575–7 [discussion 577-8]. [3] Hekster RE, Lambooy N, van Hall EV, et al. Hormone-dependent spinal leiomyoma. Surg Neurol 1994;41:330–3. [4] Gatti JM, Morvan G, Henin D, et al. Leiomyomatosis metastasizing to the spine. A case report. J Bone Joint Surg Am 1983;65:1163–5.
Fig. 2. Benign metastasizing leiomyoma. Spindle cells with uniform nuclei and with no signs of atypia are seen on examination of the surgical specimen (hematoxylin and eosin, 40 magnification). (This figure is available in colour at www.sciencedirect.com.)
Please cite this article in press as: Berti AF et al. Benign metastasizing leiomyoma of the cervical spine 31 years after uterine leiomyoma resection. J Clin Neurosci (2015), http://dx.doi.org/10.1016/j.jocn.2015.02.031
Contents lists available at ScienceDirect
Journal of Clinical Neuroscience journal homepage: www.elsevier.com/locate/jocn
Case Report
Benign metastasizing leiomyoma of the cervical spine 31 years after uterine leiomyoma resection Aldo F. Berti b, Alejandro Santillan a,⇑, Luis A. Velasquez a a b
Department of Neurology, University of Texas Health Science Center at San Antonio, 8300 Floyd Curl Drive MSC 7883, San Antonio, TX 78229, USA Miami Neurosurgical Center, South Miami, FL, USA
a r t i c l e
i n f o
Article history: Received 8 February 2015 Accepted 14 February 2015 Available online xxxx Keywords: Cervical spine Leiomyoma Metastasis
a b s t r a c t We report a 74-year-old woman presenting with a leiomyoma of the cervical spine 31 years after uterine leiomyoma resection. Benign metastasizing leiomyoma to the cervical spine is very rare. To the best of our knowledge, this is the fourth reported patient with a leiomyoma metastasizing to the cervical spine and that with the longest latency period for this type of tumor, 31 years. The pathological features were typical of leiomyoma. Ó 2015 Elsevier Ltd. All rights reserved.
1. Introduction Leiomyomas are benign tumors composed of smooth muscle and vascular collagenous tissue, mainly occurring in the uterus. Benign metastasizing leiomyoma (BML) is an extrauterine smooth muscle tumor that occurs in patients with a current or prior history of uterine leiomyoma. Leiomyomas of the cervical spine are extremely rare, with only three reported patients with cervical leiomyoma to date [2–4]. We report a 74-year-old woman with metastasizing leiomyoma of the cervical spine and a history of uterine leiomyoma resection 31 years ago. A literature review of all other reported patients was also performed. 2. Case report A 74-year-old right handed Hispanic woman with a history of hysterectomy in 1982 presented with a history of moderate to severe neck pain radiating to the right shoulder and lateral aspect of the arm, associated with tingling sensation of the right upper extremity. At physical examination, there was no weakness of the upper and lower extremities, but there was brachioradialis hyperreflexia and sensory loss in the right upper arm and lateral fingers. An MRI showed a C6 vertebral body metastasis with flattening of the spinal cord ventrally due to the epidural component within the canal (Fig. 1). An anterior approach was performed using intraoperative somatosensory-evoked potentials for ⇑ Corresponding author. Tel.: +1 7037982246. E-mail address: [email protected] (A. Santillan).
resection of the prevertebral mass, C6 corpectomy, epidural mass decompression and resection of the mass from the right C5–6 foramen. Histologic examination of the surgical specimen revealed an intraosseous leiomyoma with no atypia, increased cellularity or mitotic activity. The neoplastic cells immunoexpressed muscle specific actin, smooth muscle actin and desmin and were negative for CD34, C-kit (CD117) and neurofilament (Fig. 2, 3). The postoperative course was uneventful and she has not had tumor recurrence during her 1 year of follow-up.
3. Discussion We present a very rare patient with a BML of the cervical spine, and a hysterectomy performed 31 years prior. BML was first described by Steiner in 1939 [1]. Leiomyomas are benign tumors of well-differentiated smooth muscle tissue and vascular collagenous tissue. In most patients, metastasis is to the pelvic cavity, retroperitoneum, heart, lung and lymph nodes. To the best of our knowledge, this is only the fourth reported patient in the literature with a BML of the cervical spine [2–4]. Symptoms may vary from neck pain [4], as seen in our patient, to progressive spastic myeloradiculopathy [2]. It is important to note that leiomyomas without histological malignant features rarely metastasize. Treatment options include hormonal manipulation [3] and surgery for symptomatic lesions causing secondary neurological deficits [2–4]. BML should be considered in the differential diagnosis of mass lesions of the cervical spine in patients who have a history of uterine leiomyoma.
http://dx.doi.org/10.1016/j.jocn.2015.02.031 0967-5868/Ó 2015 Elsevier Ltd. All rights reserved.
Please cite this article in press as: Berti AF et al. Benign metastasizing leiomyoma of the cervical spine 31 years after uterine leiomyoma resection. J Clin Neurosci (2015), http://dx.doi.org/10.1016/j.jocn.2015.02.031
2
Case Report / Journal of Clinical Neuroscience xxx (2015) xxx–xxx
Fig. 3. Benign metastasizing leiomyoma. Smooth muscle actin immuno staining at 40 magnification confirms the alpha-smooth muscle isoform of actin, consistent with the myometrium origin. (This figure is available in colour at www.sciencedirect.com.)
Conflicts of Interest/Disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication. References
Fig. 1. T2-weighted sagittal cervical spine MRI of a 74-year-old woman. At C6 there is a vertebral metastasis with a posterior convex margin and epidural disease contributing to stenosis at this level.
[1] Steiner PE. Metastasizing fibroleiomyoma of the uterus: report of a case and review of the literature. Am J Pathol 1939;15:89–110. [2] Joseph V, Chacko G, Raghuram L, et al. Benign metastasizing leiomyoma causing spinal cord compression. Surg Neurol 2003;60:575–7 [discussion 577-8]. [3] Hekster RE, Lambooy N, van Hall EV, et al. Hormone-dependent spinal leiomyoma. Surg Neurol 1994;41:330–3. [4] Gatti JM, Morvan G, Henin D, et al. Leiomyomatosis metastasizing to the spine. A case report. J Bone Joint Surg Am 1983;65:1163–5.
Fig. 2. Benign metastasizing leiomyoma. Spindle cells with uniform nuclei and with no signs of atypia are seen on examination of the surgical specimen (hematoxylin and eosin, 40 magnification). (This figure is available in colour at www.sciencedirect.com.)
Please cite this article in press as: Berti AF et al. Benign metastasizing leiomyoma of the cervical spine 31 years after uterine leiomyoma resection. J Clin Neurosci (2015), http://dx.doi.org/10.1016/j.jocn.2015.02.031