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Suprasellar tuberculoma associated with unilateral moyamoya phenomenon: Case report
carotid artery along with moyamoya phenomenon.[1,2] However, this phenomenon has not been described with suprasellar tuberculomas. We report one such case. An 8‑year‑old boy presented with features of raised intracranial pressure and worsening of vision for the last 3 months. There was no other relevant history. On examination, the vision was 6/12 in the right eye and 6/24 in the left eye, with bitemporal hemianopia. Magnetic resonance imaging (MRI) showed a T1 isointense and T2 hypointense tumor with small cysts, extending to the floor of third ventricle, suggestive of a suprasellar tumor, probably a craniopharyngioma.
Sir, Suprasellar tuberculomas are rare and commonly present with visual disturbances or features of hypopituitarism. Some suprasellar tumors have been known to be associated with unilateral or bilateral occlusion of internal
a
b
c
d
e
f
g
h
k
l
i
j
Figure 1: (a) CT Head contrast showing a calcified mass in the suprasellar region; (b-f) MRI, T1WI, T2W1, Coronal T2WI, T1WI contrast axial, and coronal, respectively showing a T1 hypo, T2 hypointense lesion with peripheral enhancement; (g and h) MR angiogram images showing complete occlusion of Left ICA; (i-l) DSA, Left ICA AP, and lateral projections showing complete occlusion of left ICA beyond ophthalmic artery with moyamoya collaterals; (c and d) Left ECA AP and lateral projections showing excellent revascularization of from ECA collaterals
Neurology India | Jul-Aug 2014 | Vol 62 | Issue 4
447
[Downloaded free from http://www.neurologyindia.com on Thursday, September 25, 2014, IP: 202.177.173.189] || Click here to download free Android application fo this journal Letters to Editor
In addition, there was complete non‑visualization of left internal carotid artery (ICA), with small flow voids in the region of ICA bifurcation. Digital subtraction angiography (DSA) showed left ICA injection revealed complete occlusion of ICA distal to posterior communicating artery, and formation of some moyamoya collaterals. Bilateral anterior cerebral arteries (ACAs) were filling from the right A2. On ECA injection, the entire cerebral circulation was supplied by multiple dural collaterals, mainly from the middle meningeal artery. There was no stenosis or occlusion on right ICA injection [Figure 1a‑l]. Because the dural collaterals were supplying the brain on the left side, the patient was operated from the right side. A right pterional craniotomy was done, and the tumor was partially decompressed. Partial decompression of the lesion was done, and lamina terminalis was opened for hydrocephalus. Postoperatively, his vision improved to 6/6 in both eyes, and his visual fields normalized. Histopathology report showed an aggregate of lymphocytes, histiocytes, and plasma cells forming an ill‑formed epithelioid cell granuloma with Langhans giant cells. AFB was negative. A provisional diagnosis of tuberculoma was made [Figure 2]. Considering the histopathology report, he was started on anti‑tubercular treatment in weight‑appropriate doses; at 3‑month follow up, he was asymptomatic. Suprasellar tuberculomas are very rare lesions. Sharma et al. described a large series of sellar tuberculomas, which accounted for 1.5% of all sellar pathologies managed in their institution. [3] Other authors have described reports of suprasellar tuberculomas presenting with visual deficits, hypopituitarism, or a
mass associated with tubercular infection elsewhere in the body.[4] Moyamoya syndrome is rarely associated with pediatric suprasellar tumors. It is most common after radiation therapy of the suprasellar tumors.[5] The association of moyamoya syndrome with suprasellar tumors in absence of treatment is very rare. Tsuji et al. presented a rare association of craniopharyngioma with moyamoya syndrome.[6] There are other reports of moyamoya syndrome associated with craniopharyngiomas, and in one case, a growth hormone‑secreting pituitary adenoma and germinoma. This is probably the first case of moyamoya syndrome associated with a suprasellar tuberculoma. There could have been an associated sub‑clinical meningitic syndrome with the tuberculoma, which led to the development of ICA occlusion secondary to arteritis, or it may be an unusual association of 2 diseases. The ICA occlusion and moyamoya syndrome was probably very long‑standing because there was complete revascularization of the entire hemisphere from collaterals from external carotid artery branches, particularly MMA. In the present patient, there was excellent natural revascularization, and hence, no need for treatment of the moyamoya syndrome. In conclusion, the moyamoya syndrome associated with suprasellar tumors is very rare, and this is the first report of its association with suprasellar tuberculoma. It is important to diagnose the vascular occlusion and the extent of revascularization preoperatively to avoid any postoperative ischemia.
Nupur Pruthi, Shrithi Karanth1, M. N. Nagarjun, Paritosh Pandey Departments of Neurosurgery, and 1Neuropathology, National Institute of Mental Health and Neurosciences, Bangalore, Karnataka, India. E‑mail: [email protected]
References 1.
Figure 2: An aggregate of lymphocytes, histiocytes, and plasma cells forming an ill-formed epithelioid cell granuloma (×20, H and E). Inset highlights the Langhans giant cell (×40)
448
Arita K, Uozumi T, Oki S, Kuwabara S, Ohba S, Nakahara T, et al. Moyamoya disease associated with pituitary adenoma‑report of two cases. Neurol Med Chir (Tokyo) 1992;32:753‑7. 2. Lau YL, Milligan DW. Atypical presentation of craniopharyngioma associated with Moyamoya disease. J R Soc Med 1986;79:236‑7. 3. Sharma MC, Arora R, Mahapatra AK, Sarat‑Chandra P, Gaikwad SB, Sarkar C. Intrasellar tuberculoma‑an enigmatic pituitary infection: A series of 18 cases. Clin Neurol Neurosurg 2000;102:72‑7. 4. Nayil K, Singh S, Makhdoomi R, Ramzan A, Wani A. Sellar‑suprasellar tuberculomas in children: 2 cases and literature review. Pediatr Neurol 2011;44:463‑6. 5. Lee HS, Seol HJ, Kong DS, Shin HJ. Moyamoya syndrome precipitated by cranial irradiation for craniopharyngioma in children. J Korean Neurosurg Soc 2011;50:535‑7. 6. Tsuji N, Kuriyama T, Iwamoto M, Shizuki K. Moyamoya disease associated with craniopharyngioma. Surg Neurol 1984;21:588‑92.
Neurology India | Jul-Aug 2014 | Vol 62 | Issue 4
[Downloaded free from http://www.neurologyindia.com on Thursday, September 25, 2014, IP: 202.177.173.189] || Click here to download free Android application fo this journal Letters to Editor
Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.141264
Received: 10-07-2014 Review completed: 23-07-2014 Accepted: 10-08-2014
Pipeline stents for partially thrombosed posterior circulation aneurysms: A word of caution! Sir, A 59‑years‑old lady presented with complaints of recent headaches and double vision. Examination revealed right partial third nerve palsy. Magnetic resonance imaging (MRI) of head showed a globular hemorrhagic lesion (14 × 16 × 18 mm) in the interpeduncular region on the right side pressing on the right crus. Cerebral angiogram showed a large partially thrombosed right P1‑P2 segment posterior cerebral artery (PCA) aneurysm [Figure 1]. The high location of the lesion, reaching up to the third ventricle, made direct clipping difficult. Other surgical options were considered, such as distal PCA bypass and proximal occlusion, and endovascular coiling-both considered high risk in this situation. Therefore, we opted to use a Pipeline Embolization Device (PED) to occlude the aneurysm with flow diversion.
Figure 1: MRI showing the real size of thrombosed PCA aneurysm while cerebral angiogram showing only partial filling of the lumen while the aneurysm can be seen arising from P1-P2 segment of PCA
Neurology India | Jul-Aug 2014 | Vol 62 | Issue 4
Patient underwent successful deployment of two pipeline stents co‑axially to cover the entire length of the neck and no major branches covered [Figure 2], and discharged subsequently without any complications. Her complaints of double vision improved for period of time, but recurrent worsening of the double vision lead to two admissions in the next 3 weeks. MRI showed increasing size of the aneurysm along with further thrombosis causing increased vasogenic edema around the aneurysm and mass effect [Figure 3]. This was interpreted as a reaction to clotting in the aneurysm sac, and she was started on steroids, improving on both occasions. Four weeks after treatment she presented with sudden onset left hemiparesis. Computed tomography angiography (CTA) showed no clot in the right PCA, however it showed new hemorrhage into the aneurysm and extension into the posterior third ventricle [Figure 4]. A small filling of the aneurysm neck could be seen through the stent. Patient was admitted into the critical care unit (CCU) and options were being discussed with the family in terms of; (a) Deploying a new stent within the area of leak or (b) sacrificing the right PCA/ surgical clipping with debulking and aneurysmorraphy. Unfortunately within 30 minutes of the CTA patient blew both pupils and CT [Figure 5] showed massive brainstem hemorrhage and intraventricular hemorrhage (IVH). External ventricular drains were inserted as a last resort but patient succumbed the very next day. Pipeline embolization devices have stormed the endovascular arena in last few years [1] and chiefly their use has been limited to the anterior circulation. With increasing experience, unexpected complications have been reported, being the most common delayed parenchymal hemorrhage and rupture of apparently well‑treated large or giant aneurysms. Kuzmik et al.,[2] described two paraophthalmic aneurysms treated with SILK stents™ where one of aneurysms occluded completely while the other one ruptured. Hampton et al., have described this process of mural destabilization in two different anterior circulation aneurysms[3] hypothesizing that this phenomenon is responsible for continuous growth of treated aneurysms, which may become symptomatic in the early or late period after treatment. The possible explanations of continuous growth of thrombosed aneurysms are parasitization of aneurysm by vasa vasorum,[4] with the formation of a continuous cycle of hemorrhage and thrombosis of vasa vasorum, leading to increase in the size even when the aneurysm is completely excluded from the circulation and no flow is seen within the sac. Clinico‑pathologic studies suggest that the deployment of PEDs in a partially thrombosed aneurysm can increase the risk of rupture in weak points in the vessel wall already covered by thrombus. Lack of wall nutrition due to overlying thrombus might be the underlying reason.[5] 449
Copyright of Neurology India is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
Suprasellar tuberculoma associated with unilateral moyamoya phenomenon: Case report
carotid artery along with moyamoya phenomenon.[1,2] However, this phenomenon has not been described with suprasellar tuberculomas. We report one such case. An 8‑year‑old boy presented with features of raised intracranial pressure and worsening of vision for the last 3 months. There was no other relevant history. On examination, the vision was 6/12 in the right eye and 6/24 in the left eye, with bitemporal hemianopia. Magnetic resonance imaging (MRI) showed a T1 isointense and T2 hypointense tumor with small cysts, extending to the floor of third ventricle, suggestive of a suprasellar tumor, probably a craniopharyngioma.
Sir, Suprasellar tuberculomas are rare and commonly present with visual disturbances or features of hypopituitarism. Some suprasellar tumors have been known to be associated with unilateral or bilateral occlusion of internal
a
b
c
d
e
f
g
h
k
l
i
j
Figure 1: (a) CT Head contrast showing a calcified mass in the suprasellar region; (b-f) MRI, T1WI, T2W1, Coronal T2WI, T1WI contrast axial, and coronal, respectively showing a T1 hypo, T2 hypointense lesion with peripheral enhancement; (g and h) MR angiogram images showing complete occlusion of Left ICA; (i-l) DSA, Left ICA AP, and lateral projections showing complete occlusion of left ICA beyond ophthalmic artery with moyamoya collaterals; (c and d) Left ECA AP and lateral projections showing excellent revascularization of from ECA collaterals
Neurology India | Jul-Aug 2014 | Vol 62 | Issue 4
447
[Downloaded free from http://www.neurologyindia.com on Thursday, September 25, 2014, IP: 202.177.173.189] || Click here to download free Android application fo this journal Letters to Editor
In addition, there was complete non‑visualization of left internal carotid artery (ICA), with small flow voids in the region of ICA bifurcation. Digital subtraction angiography (DSA) showed left ICA injection revealed complete occlusion of ICA distal to posterior communicating artery, and formation of some moyamoya collaterals. Bilateral anterior cerebral arteries (ACAs) were filling from the right A2. On ECA injection, the entire cerebral circulation was supplied by multiple dural collaterals, mainly from the middle meningeal artery. There was no stenosis or occlusion on right ICA injection [Figure 1a‑l]. Because the dural collaterals were supplying the brain on the left side, the patient was operated from the right side. A right pterional craniotomy was done, and the tumor was partially decompressed. Partial decompression of the lesion was done, and lamina terminalis was opened for hydrocephalus. Postoperatively, his vision improved to 6/6 in both eyes, and his visual fields normalized. Histopathology report showed an aggregate of lymphocytes, histiocytes, and plasma cells forming an ill‑formed epithelioid cell granuloma with Langhans giant cells. AFB was negative. A provisional diagnosis of tuberculoma was made [Figure 2]. Considering the histopathology report, he was started on anti‑tubercular treatment in weight‑appropriate doses; at 3‑month follow up, he was asymptomatic. Suprasellar tuberculomas are very rare lesions. Sharma et al. described a large series of sellar tuberculomas, which accounted for 1.5% of all sellar pathologies managed in their institution. [3] Other authors have described reports of suprasellar tuberculomas presenting with visual deficits, hypopituitarism, or a
mass associated with tubercular infection elsewhere in the body.[4] Moyamoya syndrome is rarely associated with pediatric suprasellar tumors. It is most common after radiation therapy of the suprasellar tumors.[5] The association of moyamoya syndrome with suprasellar tumors in absence of treatment is very rare. Tsuji et al. presented a rare association of craniopharyngioma with moyamoya syndrome.[6] There are other reports of moyamoya syndrome associated with craniopharyngiomas, and in one case, a growth hormone‑secreting pituitary adenoma and germinoma. This is probably the first case of moyamoya syndrome associated with a suprasellar tuberculoma. There could have been an associated sub‑clinical meningitic syndrome with the tuberculoma, which led to the development of ICA occlusion secondary to arteritis, or it may be an unusual association of 2 diseases. The ICA occlusion and moyamoya syndrome was probably very long‑standing because there was complete revascularization of the entire hemisphere from collaterals from external carotid artery branches, particularly MMA. In the present patient, there was excellent natural revascularization, and hence, no need for treatment of the moyamoya syndrome. In conclusion, the moyamoya syndrome associated with suprasellar tumors is very rare, and this is the first report of its association with suprasellar tuberculoma. It is important to diagnose the vascular occlusion and the extent of revascularization preoperatively to avoid any postoperative ischemia.
Nupur Pruthi, Shrithi Karanth1, M. N. Nagarjun, Paritosh Pandey Departments of Neurosurgery, and 1Neuropathology, National Institute of Mental Health and Neurosciences, Bangalore, Karnataka, India. E‑mail: [email protected]
References 1.
Figure 2: An aggregate of lymphocytes, histiocytes, and plasma cells forming an ill-formed epithelioid cell granuloma (×20, H and E). Inset highlights the Langhans giant cell (×40)
448
Arita K, Uozumi T, Oki S, Kuwabara S, Ohba S, Nakahara T, et al. Moyamoya disease associated with pituitary adenoma‑report of two cases. Neurol Med Chir (Tokyo) 1992;32:753‑7. 2. Lau YL, Milligan DW. Atypical presentation of craniopharyngioma associated with Moyamoya disease. J R Soc Med 1986;79:236‑7. 3. Sharma MC, Arora R, Mahapatra AK, Sarat‑Chandra P, Gaikwad SB, Sarkar C. Intrasellar tuberculoma‑an enigmatic pituitary infection: A series of 18 cases. Clin Neurol Neurosurg 2000;102:72‑7. 4. Nayil K, Singh S, Makhdoomi R, Ramzan A, Wani A. Sellar‑suprasellar tuberculomas in children: 2 cases and literature review. Pediatr Neurol 2011;44:463‑6. 5. Lee HS, Seol HJ, Kong DS, Shin HJ. Moyamoya syndrome precipitated by cranial irradiation for craniopharyngioma in children. J Korean Neurosurg Soc 2011;50:535‑7. 6. Tsuji N, Kuriyama T, Iwamoto M, Shizuki K. Moyamoya disease associated with craniopharyngioma. Surg Neurol 1984;21:588‑92.
Neurology India | Jul-Aug 2014 | Vol 62 | Issue 4
[Downloaded free from http://www.neurologyindia.com on Thursday, September 25, 2014, IP: 202.177.173.189] || Click here to download free Android application fo this journal Letters to Editor
Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.141264
Received: 10-07-2014 Review completed: 23-07-2014 Accepted: 10-08-2014
Pipeline stents for partially thrombosed posterior circulation aneurysms: A word of caution! Sir, A 59‑years‑old lady presented with complaints of recent headaches and double vision. Examination revealed right partial third nerve palsy. Magnetic resonance imaging (MRI) of head showed a globular hemorrhagic lesion (14 × 16 × 18 mm) in the interpeduncular region on the right side pressing on the right crus. Cerebral angiogram showed a large partially thrombosed right P1‑P2 segment posterior cerebral artery (PCA) aneurysm [Figure 1]. The high location of the lesion, reaching up to the third ventricle, made direct clipping difficult. Other surgical options were considered, such as distal PCA bypass and proximal occlusion, and endovascular coiling-both considered high risk in this situation. Therefore, we opted to use a Pipeline Embolization Device (PED) to occlude the aneurysm with flow diversion.
Figure 1: MRI showing the real size of thrombosed PCA aneurysm while cerebral angiogram showing only partial filling of the lumen while the aneurysm can be seen arising from P1-P2 segment of PCA
Neurology India | Jul-Aug 2014 | Vol 62 | Issue 4
Patient underwent successful deployment of two pipeline stents co‑axially to cover the entire length of the neck and no major branches covered [Figure 2], and discharged subsequently without any complications. Her complaints of double vision improved for period of time, but recurrent worsening of the double vision lead to two admissions in the next 3 weeks. MRI showed increasing size of the aneurysm along with further thrombosis causing increased vasogenic edema around the aneurysm and mass effect [Figure 3]. This was interpreted as a reaction to clotting in the aneurysm sac, and she was started on steroids, improving on both occasions. Four weeks after treatment she presented with sudden onset left hemiparesis. Computed tomography angiography (CTA) showed no clot in the right PCA, however it showed new hemorrhage into the aneurysm and extension into the posterior third ventricle [Figure 4]. A small filling of the aneurysm neck could be seen through the stent. Patient was admitted into the critical care unit (CCU) and options were being discussed with the family in terms of; (a) Deploying a new stent within the area of leak or (b) sacrificing the right PCA/ surgical clipping with debulking and aneurysmorraphy. Unfortunately within 30 minutes of the CTA patient blew both pupils and CT [Figure 5] showed massive brainstem hemorrhage and intraventricular hemorrhage (IVH). External ventricular drains were inserted as a last resort but patient succumbed the very next day. Pipeline embolization devices have stormed the endovascular arena in last few years [1] and chiefly their use has been limited to the anterior circulation. With increasing experience, unexpected complications have been reported, being the most common delayed parenchymal hemorrhage and rupture of apparently well‑treated large or giant aneurysms. Kuzmik et al.,[2] described two paraophthalmic aneurysms treated with SILK stents™ where one of aneurysms occluded completely while the other one ruptured. Hampton et al., have described this process of mural destabilization in two different anterior circulation aneurysms[3] hypothesizing that this phenomenon is responsible for continuous growth of treated aneurysms, which may become symptomatic in the early or late period after treatment. The possible explanations of continuous growth of thrombosed aneurysms are parasitization of aneurysm by vasa vasorum,[4] with the formation of a continuous cycle of hemorrhage and thrombosis of vasa vasorum, leading to increase in the size even when the aneurysm is completely excluded from the circulation and no flow is seen within the sac. Clinico‑pathologic studies suggest that the deployment of PEDs in a partially thrombosed aneurysm can increase the risk of rupture in weak points in the vessel wall already covered by thrombus. Lack of wall nutrition due to overlying thrombus might be the underlying reason.[5] 449
Copyright of Neurology India is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
