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Bilateral medial medullary infarction with distal stenosis of hypoplastic vertebral artery

Lu, Jie-Ping1,2; Wu, Yan1; Xiao, Fang3; Li, Huai-Yu1; Tang, Qi-Qiang1

Section Editor(s): Guo, Li-Shao

doi: 10.1097/CM9.0000000000000171
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1Department of Neurology, The First Affiliated Hospital of USTC, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei, Anhui 230001, China

2Hefei National Laboratory for Physical Sciences at the Microscale, School of Life Sciences, University of Science and Technology of China, Hefei, Anhui 230026, China

3Medical Imaging Center, The First Affiliated Hospital of USTC, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei, Anhui 230001, China.

Correspondence to: Dr. Jie-Ping Lu, Department of Neurology, The First Affiliated Hospital of USTC, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei, Anhui 230001, China E-Mail: Lujp1984@163.com

How to cite this article: Lu JP, Wu Y, Xiao F, Li HY, Tang QQ. Bilateral medial medullary infarction with distal stenosis of hypoplastic vertebral artery. Chin Med J 2019;00:00–00. doi: 10.1097/CM9.0000000000000171

Received 4 December, 2018

This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0

To the Editor: Bilateral medial medullary infarction (MMI) is a rare stroke subtype.[1] It usually leads to quadriplegia, sensory disturbance, hypoglossal palsy, bulbar paralysis, etc.[2] We encountered a patient with rapidly progressive tetraparesis and diffusion-weighted imaging (DWI) exhibited a “heart appearance” sign in the bilateral ventral medulla. Computed tomography angiography (CTA) demonstrated that the left vertebral artery (VA) was hypoplastic and there was an atherosclerotic stenosis in the V4 segment.

Two days prior to admission, a 54-year-old man suddenly suffered from dizziness in the morning, followed by somnolence in the afternoon. During the night, he developed mild left-sided weakness and slurred speech at 11 PM. On the following day, the patient worsened into a rapidly progressive tetraparesis. He had a history of hypertension and diabetes. Neurologic examination revealed dysarthria, flaccid quadriplegia, bilateral Babinski signs without nystagmus, tongue paralysis or sensory disturbance. Pupils and eye movements were normal. The power in his four limbs was 0 of 5. Brain magnetic resonance images obtained 1 day after symptom onset showed a slight hyperintensity in the bilateral anteromedial medulla on axial T2-weighted imaging. DWI exhibited a hyperintense “heart appearance” signal in the corresponding area [Figure 1A]. CTA showed the left VA was wholly slenderer than the contralateral side in the extracranial portion and there was an atherosclerotic stenosis in the V4 segment [Figure 1B]. The patient was treated with aspirin and atorvastatin. On the second day of admission, he received tracheotomy due to dyspnea. The power in his four limbs was still 0 of 5 after 2 weeks in hospital and then the patient was transferred to the rehabilitation hospital. He had a modified Rankin Scale score of four at 3 months after symptom onset.

Figure 1

Figure 1

MMI accounts for less than 1% of ischemic strokes and bilateral MMI is even rarer.[1] The “heart appearance” sign in the ventral medulla on axial DWI is a characteristic imaging finding of bilateral MMI.[3] The common clinical manifestations of bilateral MMI include flaccid quadriplegia, loss of deep sensation, dysarthria, nystagmus, hypoglossal palsy, respiratory failure, etc.[2] This patient is a rare case of pure motor quadriplegia and bulbar dysfunction, and different symptom combinations of bilateral MMI usually depend on the location and extent of the lesion.

A recent systematic review showed that the most common mechanisms of bilateral MMI include large-artery atherosclerosis and small penetrating artery disease.[2] In this case, CTA showed atherosclerotic stenosis at distal portion of the left VA. The underlying mechanisms of bilateral MMI with unilateral VA stenosis are as follows: (1) anatomic variability of perforator branches that supply the bilateral medulla, (2) the extension of thrombus in the vertebrobasilar junction to the contralateral side and (3) bilateral anterior spinal artery originating from the same pathological VA.[2,4]

It is worth noting that the responsible vessel of VA was hypoplastic in this case. Vertebral artery hypoplasia (VAH) is a common congenital anatomical variation and it may contribute to posterior circulation strokes.[5] The ischemic lesions with VAH mainly present in posterior inferior cerebellar artery territory, posterior cerebral artery territory, and pons, while the presentation in bilateral medial medulla is rare.[5]

In summary, the patient with bilateral MMI may present pure motor quadriplegia and clinicians should be aware that the distal stenosis of unilateral hypoplastic VA may be one of the vascular pathologies of bilateral MMI.

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Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initial will not be published and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.

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Conflicts of interest

None.

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References

1. Akimoto T, Ogawa K, Morita A, Suzuki Y, Kamei S. Clinical study of 27 patients with medial medullary infarction. J Stroke Cerebrovasc Dis 2017; 26:2223–2231. doi: 10.1016/j.jstrokecerebrovasdis.2017.05.004.
2. Pongmoragot J, Parthasarathy S, Selchen D, Saposnik G. Bilateral medial medullary infarction: a systematic review. J Stroke Cerebrovasc Dis 2013; 22:775–780. doi: 10.1016/j.jstrokecerebrovasdis.2012.03.010.
3. Jalal MJ, Menon MK. Bilateral medial medullary infarction with a “heart appearance” sign. Neurol India 2016; 64:130doi: 10.4103/0028-3886.178059.
4. Kim JS, Han YS. Medial medullary infarction: clinical, imaging, and outcome study in 86 consecutive patients. Stroke 2009; 40:3221–3225. doi: 10.1161/STROKEAHA.109.559864.
5. Perren F, Poglia D, Landis T, Sztajzel R. Vertebral artery hypoplasia: a predisposing factor for posterior circulation stroke? Neurology 2007; 68:65–67. doi: 10.1212/01.wnl.0000250258.76706.
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