British Journal of Neurosurgery
ISSN: 0268-8697 (Print) 1360-046X (Online) Journal homepage: http://www.tandfonline.com/loi/ibjn20
Intradural tumoral calcinosis of the foramen magnum region: A case report Hao Wang, Yuanli Zhao & Jun Yang To cite this article: Hao Wang, Yuanli Zhao & Jun Yang (2016): Intradural tumoral calcinosis of the foramen magnum region: A case report, British Journal of Neurosurgery, DOI: 10.1080/02688697.2016.1238036 To link to this article: http://dx.doi.org/10.1080/02688697.2016.1238036
Published online: 11 Oct 2016.
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Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=ibjn20 Download by: [Cornell University Library]
Date: 18 October 2016, At: 14:13
BRITISH JOURNAL OF NEUROSURGERY, 2016 http://dx.doi.org/10.1080/02688697.2016.1238036
SHORT REPORT
Intradural tumoral calcinosis of the foramen magnum region: A case report Hao Wang, Yuanli Zhao and Jun Yang Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
ABSTRACT
ARTICLE HISTORY
Tumoral calcinosis is a rare disorder characterized by tumor-like calcified masses commonly occurring in the periarticular regions of the extremities. Conversely, intradural tumoral calcinosis of the spine is extremely rare, with only two previously reported cases. To our knowledge, this is the first intradural case located in the foramen magnum region.
Received 30 December 2014 Accepted 4 September 2016
Background and importance
Intervention 1
Tumoral calcinosis was first named in 1943 and is most commonly observed around the hips, shoulders, elbows, knees, and fingers. Tumoral calcinosis involving the spine is very rare and extradural in most cases.2 Denise et al.2 reported the most large cases of tumoral calcinosis of the spine: 21 cases are all extradural. As for tumoral calcinosis in the intradural region, only two cases have been reported in the literature to date. In one case, the lesion was located in the filum terminale3, and in the lumbar meninges in the other case4. Here, we report a new intradural case, which was found in the foramen magnum region.
Clinical presentation A 22-year-old woman had experienced right neck and shoulder pain for 7 years, which had worsened in the last 3 years. Upon further questioning, the patient told us that her neck suffered from a shock by a high-speed football 9 years ago, and she received short-wave therapy and recovered quickly. Seven years ago, she experienced gradual onset of slight, persistent right neck and shoulder pain, but paid no attention to it. Four years ago, she underwent cervical spine plain radiography, which showed no abnormality (Figure 1(A)). However, 3 years ago, her symptoms worsened. Consequently, she underwent computed tomography (CT) and magnetic resonance imaging (MRI) at her local hospital. The CT scan (Figure 1(B)) revealed a high-density mass located in the right foramen magnum region, and the MR image (Figure 1(C,D)) revealed that the mass was hypointense and hypointense with heterogeneous enhancement on T2 and T1-weighted images, respectively. Due to the perceived risks of surgery, the patient initially refused to undergo surgery. In August 2012, her family finally convinced her undergo surgery. Preoperative MRI performed in our hospital showed that the mass had grown slightly since the last follow-up (Figure 1(E,F)). Physical examination showed limited neck rotation and positive right Hoffman’s sign.
CONTACT Jun Yang
[email protected]
ß 2016 The Neurosurgical Foundation
KEYWORDS Tumoural calcinosis; spine; foramen magnum tumors; neurosurgery
The preoperative diagnosis was schwannoma or meningioma with calcification, which are the most common intradural extramedullary tumors. The suboccipital midline approach was chosen. Intraoperatively, we opened the atlas and foramen magnum and found the lesion located in the right foramen magnum region. The mass, which measured 15 mm in maximal diameter, calcified with ragged margins, was adherent to the lateral medulla and medial right vertebral artery. The lesion was very hard, which necessitated the use of a laser knife and a bone rongeur for excision. The base originated from the dura of the foramen magnum region. We totally removed the mass and burned the base carefully. Postoperative pathological findings indicated tumoral calcinosis (Figure 1(G)). Hematoxylin and eosin stained sections showed some mature irregular host bones and a new bone formation region, accompanied with calcification and reactive histiocytosis. Based on morphologic findings, we concluded that the present case was an unusual case of tumoral calcinosis of the spine. The patient had normal serum calcium and phosphate levels, normal thyrocalcitonin and parathyroid hormone levels, as well as normal renal function. The abdominal ultrasonography findings were all normal. The patient developed meningitis after the operation and recovered through vancomycin treatment. Postoperative CT revealed some small high-density masses left (Figure 1(H)), which were not visible on MR images. On the lumbar spine MR images, we found an asymptomatic arachnoid cyst in the sacral canal. The patient was discharged with only rigidity of the right upper limb and neck, and we suggested long-term follow-up. Six months after the operation, we contacted the patient through e-mail. She reported feeling very well with reduced rigidity. The patient underwent MRI (Figure 1(I)) at her local hospital, which showed no recurrence of the lesion. We asked the patient to visit our hospital for a thorough examination, including a cervical spine CT, 2-year postsurgery.
Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing 100050, China
2
H. WANG ET AL.
Figure 1. (A) Plain radiography of the cervical spine in August 2008. No abnormality is seen. (B) Three-dimensional computed tomography reconstruction image of the mass in June 2009. The mass is seen in the right foramen magnum region with a high density. (C) Sagittal T2-weighted image of the mass in June 2009. A welldefined hypointense mass in the right foramen magnum region is noted. (D) Coronal post-contrast T1-weighted image of the mass in June 2009. The enhancement is obvious and irregular. (E) Sagittal T2-weighted image of the mass in July 2012. The mass had grown slightly since the previous examination. (F) Sagittal postcontrast T1-weighted image of the mass in July 2012. (G) Pathological examination (original magnification, 100) shows some mature irregular host bones and a new bone formation region, accompanied with flocculent calcification and reactive histiocytosis. (H) Postoperative computed tomography scan. Small, high-density residual masses are noted (arrow). (I) Coronal post-contrast T1-weighted image in December 2012. No recurrence is seen.
Discussion Tumoral calcinosis is frequently associated with hereditary disorders of calcium metabolism or renal dialysis but can also occur in degenerated tissues in the absence of systemic disorders.1,2 To our knowledge, our report is the first to document intradural tumoral calcinosis of the foramen magnum region; therefore, the mechanism behind its development is unknown. In our case, we speculate that there may be an association between the patient’s previous neck injury and the development of tumoral calcinosis. Accordingly, some authors1 had suggested that local hemorrhage might lead to deposition of calcium salts, ultimately resulting in post-traumatic tumoral calcinosis. The natural history of idiopathic tumoral calcinosis is not well understood. In all previously reported intradural tumoral calcinosis cases, the patients underwent surgery soon after the lesions were found. However, the patient in our case chose conservative treatment for 3 years, which allowed observation of the change in the lesion over time. Misdiagnose and poor experience of intradural tumoral calcinosis, we chose operation as the first choice at that time. However, the long follow-up result confirm the benign course of this disease. We admit that conservative treatment could be a better way when the lesion had not changed
significantly and the patient had neck pain rather than neurological deficit. As a rare disease of the intradural region, tumoral calcinosis should not be considered the first diagnosis of similar lesions, and a differential diagnosis for neurinoma and meningioma among others should be considered. However, on identification of lesions with calcification or lesions located at high-risk areas, appropriate preoperative preparation such as preparing suitable instruments, electrophysiological monitoring, and blood products, should be performed. And if intradural tumoral calcinosis was suspected, therapeutic schedule should be comprehensive analyzed.
Conclusion Intradural tumoral calcinosis of the spine is a benign disease that is difficult to diagnose. The treatment experience is poor. If it caused progressive neurological symptoms, the operation should be performed. CT and MR images should be analyzed carefully before surgery, long-term follow-up is needed if residual tumor is found postoperatively. Conservative treatment could be considered first if a asymptomatic intradural tumoral calcinosis was highly suspected.
BRITISH JOURNAL OF NEUROSURGERY
Disclosure statement The authors report no conflicts of interest. The authors alone are responsible for the content and writing of this article.
3
References 1. 2.
Funding
3.
This work was supported by National Natural Science Foundation of China [No. 81070978].
4.
Hutt N, Baghla DP, Gulati V, Pastides PS, Beverly MC, Bashir WA. Acral post-traumatic tumoral calcinosis in pregnancy: a case report. J Med Case Rep 2011;5:89. Durant DM, Riley LH, III, Burger PC, McCarthy EF. Tumoral calcinosis of the spine: a study of 21 cases. Spine 2001;26:1673–9. Sharma M, Sinha R, Hussey K, Fouyas IP. Tumoral calcinosis of the filum terminale. Neurosurgery 2005;57:E596. discussion E596 Watanabe A, Isoe S, Kaneko M, Nukui H. Tumoral calcinosis of the lumbar meninges: case report. Neurosurgery 2000;47:230–2.
ISSN: 0268-8697 (Print) 1360-046X (Online) Journal homepage: http://www.tandfonline.com/loi/ibjn20
Intradural tumoral calcinosis of the foramen magnum region: A case report Hao Wang, Yuanli Zhao & Jun Yang To cite this article: Hao Wang, Yuanli Zhao & Jun Yang (2016): Intradural tumoral calcinosis of the foramen magnum region: A case report, British Journal of Neurosurgery, DOI: 10.1080/02688697.2016.1238036 To link to this article: http://dx.doi.org/10.1080/02688697.2016.1238036
Published online: 11 Oct 2016.
Submit your article to this journal
Article views: 6
View related articles
View Crossmark data
Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=ibjn20 Download by: [Cornell University Library]
Date: 18 October 2016, At: 14:13
BRITISH JOURNAL OF NEUROSURGERY, 2016 http://dx.doi.org/10.1080/02688697.2016.1238036
SHORT REPORT
Intradural tumoral calcinosis of the foramen magnum region: A case report Hao Wang, Yuanli Zhao and Jun Yang Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing, China
ABSTRACT
ARTICLE HISTORY
Tumoral calcinosis is a rare disorder characterized by tumor-like calcified masses commonly occurring in the periarticular regions of the extremities. Conversely, intradural tumoral calcinosis of the spine is extremely rare, with only two previously reported cases. To our knowledge, this is the first intradural case located in the foramen magnum region.
Received 30 December 2014 Accepted 4 September 2016
Background and importance
Intervention 1
Tumoral calcinosis was first named in 1943 and is most commonly observed around the hips, shoulders, elbows, knees, and fingers. Tumoral calcinosis involving the spine is very rare and extradural in most cases.2 Denise et al.2 reported the most large cases of tumoral calcinosis of the spine: 21 cases are all extradural. As for tumoral calcinosis in the intradural region, only two cases have been reported in the literature to date. In one case, the lesion was located in the filum terminale3, and in the lumbar meninges in the other case4. Here, we report a new intradural case, which was found in the foramen magnum region.
Clinical presentation A 22-year-old woman had experienced right neck and shoulder pain for 7 years, which had worsened in the last 3 years. Upon further questioning, the patient told us that her neck suffered from a shock by a high-speed football 9 years ago, and she received short-wave therapy and recovered quickly. Seven years ago, she experienced gradual onset of slight, persistent right neck and shoulder pain, but paid no attention to it. Four years ago, she underwent cervical spine plain radiography, which showed no abnormality (Figure 1(A)). However, 3 years ago, her symptoms worsened. Consequently, she underwent computed tomography (CT) and magnetic resonance imaging (MRI) at her local hospital. The CT scan (Figure 1(B)) revealed a high-density mass located in the right foramen magnum region, and the MR image (Figure 1(C,D)) revealed that the mass was hypointense and hypointense with heterogeneous enhancement on T2 and T1-weighted images, respectively. Due to the perceived risks of surgery, the patient initially refused to undergo surgery. In August 2012, her family finally convinced her undergo surgery. Preoperative MRI performed in our hospital showed that the mass had grown slightly since the last follow-up (Figure 1(E,F)). Physical examination showed limited neck rotation and positive right Hoffman’s sign.
CONTACT Jun Yang
[email protected]
ß 2016 The Neurosurgical Foundation
KEYWORDS Tumoural calcinosis; spine; foramen magnum tumors; neurosurgery
The preoperative diagnosis was schwannoma or meningioma with calcification, which are the most common intradural extramedullary tumors. The suboccipital midline approach was chosen. Intraoperatively, we opened the atlas and foramen magnum and found the lesion located in the right foramen magnum region. The mass, which measured 15 mm in maximal diameter, calcified with ragged margins, was adherent to the lateral medulla and medial right vertebral artery. The lesion was very hard, which necessitated the use of a laser knife and a bone rongeur for excision. The base originated from the dura of the foramen magnum region. We totally removed the mass and burned the base carefully. Postoperative pathological findings indicated tumoral calcinosis (Figure 1(G)). Hematoxylin and eosin stained sections showed some mature irregular host bones and a new bone formation region, accompanied with calcification and reactive histiocytosis. Based on morphologic findings, we concluded that the present case was an unusual case of tumoral calcinosis of the spine. The patient had normal serum calcium and phosphate levels, normal thyrocalcitonin and parathyroid hormone levels, as well as normal renal function. The abdominal ultrasonography findings were all normal. The patient developed meningitis after the operation and recovered through vancomycin treatment. Postoperative CT revealed some small high-density masses left (Figure 1(H)), which were not visible on MR images. On the lumbar spine MR images, we found an asymptomatic arachnoid cyst in the sacral canal. The patient was discharged with only rigidity of the right upper limb and neck, and we suggested long-term follow-up. Six months after the operation, we contacted the patient through e-mail. She reported feeling very well with reduced rigidity. The patient underwent MRI (Figure 1(I)) at her local hospital, which showed no recurrence of the lesion. We asked the patient to visit our hospital for a thorough examination, including a cervical spine CT, 2-year postsurgery.
Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing 100050, China
2
H. WANG ET AL.
Figure 1. (A) Plain radiography of the cervical spine in August 2008. No abnormality is seen. (B) Three-dimensional computed tomography reconstruction image of the mass in June 2009. The mass is seen in the right foramen magnum region with a high density. (C) Sagittal T2-weighted image of the mass in June 2009. A welldefined hypointense mass in the right foramen magnum region is noted. (D) Coronal post-contrast T1-weighted image of the mass in June 2009. The enhancement is obvious and irregular. (E) Sagittal T2-weighted image of the mass in July 2012. The mass had grown slightly since the previous examination. (F) Sagittal postcontrast T1-weighted image of the mass in July 2012. (G) Pathological examination (original magnification, 100) shows some mature irregular host bones and a new bone formation region, accompanied with flocculent calcification and reactive histiocytosis. (H) Postoperative computed tomography scan. Small, high-density residual masses are noted (arrow). (I) Coronal post-contrast T1-weighted image in December 2012. No recurrence is seen.
Discussion Tumoral calcinosis is frequently associated with hereditary disorders of calcium metabolism or renal dialysis but can also occur in degenerated tissues in the absence of systemic disorders.1,2 To our knowledge, our report is the first to document intradural tumoral calcinosis of the foramen magnum region; therefore, the mechanism behind its development is unknown. In our case, we speculate that there may be an association between the patient’s previous neck injury and the development of tumoral calcinosis. Accordingly, some authors1 had suggested that local hemorrhage might lead to deposition of calcium salts, ultimately resulting in post-traumatic tumoral calcinosis. The natural history of idiopathic tumoral calcinosis is not well understood. In all previously reported intradural tumoral calcinosis cases, the patients underwent surgery soon after the lesions were found. However, the patient in our case chose conservative treatment for 3 years, which allowed observation of the change in the lesion over time. Misdiagnose and poor experience of intradural tumoral calcinosis, we chose operation as the first choice at that time. However, the long follow-up result confirm the benign course of this disease. We admit that conservative treatment could be a better way when the lesion had not changed
significantly and the patient had neck pain rather than neurological deficit. As a rare disease of the intradural region, tumoral calcinosis should not be considered the first diagnosis of similar lesions, and a differential diagnosis for neurinoma and meningioma among others should be considered. However, on identification of lesions with calcification or lesions located at high-risk areas, appropriate preoperative preparation such as preparing suitable instruments, electrophysiological monitoring, and blood products, should be performed. And if intradural tumoral calcinosis was suspected, therapeutic schedule should be comprehensive analyzed.
Conclusion Intradural tumoral calcinosis of the spine is a benign disease that is difficult to diagnose. The treatment experience is poor. If it caused progressive neurological symptoms, the operation should be performed. CT and MR images should be analyzed carefully before surgery, long-term follow-up is needed if residual tumor is found postoperatively. Conservative treatment could be considered first if a asymptomatic intradural tumoral calcinosis was highly suspected.
BRITISH JOURNAL OF NEUROSURGERY
Disclosure statement The authors report no conflicts of interest. The authors alone are responsible for the content and writing of this article.
3
References 1. 2.
Funding
3.
This work was supported by National Natural Science Foundation of China [No. 81070978].
4.
Hutt N, Baghla DP, Gulati V, Pastides PS, Beverly MC, Bashir WA. Acral post-traumatic tumoral calcinosis in pregnancy: a case report. J Med Case Rep 2011;5:89. Durant DM, Riley LH, III, Burger PC, McCarthy EF. Tumoral calcinosis of the spine: a study of 21 cases. Spine 2001;26:1673–9. Sharma M, Sinha R, Hussey K, Fouyas IP. Tumoral calcinosis of the filum terminale. Neurosurgery 2005;57:E596. discussion E596 Watanabe A, Isoe S, Kaneko M, Nukui H. Tumoral calcinosis of the lumbar meninges: case report. Neurosurgery 2000;47:230–2.
