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Moyamoya syndrome with antiphospholipid antibodies: a case report and literature review Z Wang, Z Fu, J Wang, H Cui, Z Zhang and B Zhang Lupus published online 17 June 2014 DOI: 10.1177/0961203314540761 The online version of this article can be found at: http://lup.sagepub.com/content/early/2014/06/17/0961203314540761
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Lupus (2014) 0,
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CASE REPORT
Moyamoya syndrome with antiphospholipid antibodies: a case report and literature review Z Wang1, Z Fu1, J Wang1, H Cui2, Z Zhang3 and B Zhang1 1
Department of Neurology, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, China; 2Department of Integrated ICU, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, China; and 3Department of Obstetrics and Gynaecology, Peking University First Hospital, Beijing, China
Moyamoya syndrome is a term applied to typical moyamoya vasculopathy with well-recognized associated conditions. Several systematic studies on the entities of moyamoya syndrome with autoimmune disease have been reported. We report a case of moyamoya syndrome with antiphospholipid antibodies and provide a review of relevant cases from the literature, describing the clinical characteristics and treatments. Lupus (2014) 0, 1–3. Key words: Moyamoya syndrome; antiphospholipid autoantibodies; antiphospholipid syndrome
Introduction Moyamoya disease (MMD) is an uncommon cerebrovascular disease of unknown aetiology that is characterized by a progressive narrowing of the anterior circulation vasculature of the brain with the formation of a network of collateral vessels.1 Moyamoya syndrome (MMS) is a term applied to typical moyamoya vasculopathy with wellrecognized associated conditions.2 Several systematic studies on the entities of MMS with autoimmune disease have been reported, including systemic lupus erythematosus and autoimmune thyroid disease.3,4 However, there have been no systematic studies on MMS with antiphospholipid antibodies (aPL). We report a case of MMS with aPL and provide a review of relevant cases from the literature, describing the clinical characteristics and treatments. Methods We report the case of a 43-year-old woman who suffered from MMS with aPL. We searched the Correspondence to: Boai Zhang, Department of Neurology, The First Affiliated Hospital of Zhengzhou University, No.1 Jianshe East Road, Erqi District, Zhengzhou, Henan Province, 450052, China. Email: [email protected] Zhen Wang and Zhenqiang Fu contributed equally to this report. Received 25 December 2013; accepted 29 May 2013
English-language literature using the PubMed database from 1957 to December 2013. The following keywords were used: moyamoya, antiphospholipid, anticardiolipin antibodies, anti-beta2-glycoprotein I antibodies, and lupus anticoagulant. All potential references that were cited in these articles were also reviewed.
Results Case report A 43-year-old woman was admitted with sudden onset of right-sided hemiparesis during menstruation. Seven years prior, she had experienced transient right-sided weakness; she also had a history of adenomyosis without treatment for three years. She lacked a history of smoking, migraine headaches, livedo reticularis, spontaneous abortion, or deep vein thrombosis. A neurological examination revealed a power of 3/5 in the right limbs. A brain computed tomography (CT) scan revealed multiple low-density lesions in the bilateral centrum semiovale and basal ganglia without haemorrhage. CT angiography showed an occlusion of the bilateral middle cerebral arteries with rich basal collateral vessels (Supplemental Figure 1, available online with this article). Her echocardiogram and thoracoabdominal CT scans were normal; however, a pelvic ultrasound revealed uterine adenomyosis.
! The Author(s), 2014. Reprints and permissions: http://www.sagepub.co.uk/journalsPermissions.nav
Downloaded from lup.sagepub.com at UNIVERSITE LAVAL on June 19, 2014
10.1177/0961203314540761
XML Template (2014) [14.6.2014–10:36am] //blrnas3/cenpro/ApplicationFiles/Journals/SAGE/3B2/LUPJ/Vol00000/140129/APPFile/SG-LUPJ140129.3d
(LUP)
[1–3] [PREPRINTER stage]
Moyamoya syndrome with antiphospholipid antibodies Z Wang et al.
2
The results of her routine laboratory blood tests were normal. Coagulation studies were also normal. Lupus anticoagulant (LA) was negative. Immunoglobulin (Ig)G, IgA and IgM anticardiolipin antibodies (aCL) levels were 34 U GPL/ml (normal
http://lup.sagepub.com/
Moyamoya syndrome with antiphospholipid antibodies: a case report and literature review Z Wang, Z Fu, J Wang, H Cui, Z Zhang and B Zhang Lupus published online 17 June 2014 DOI: 10.1177/0961203314540761 The online version of this article can be found at: http://lup.sagepub.com/content/early/2014/06/17/0961203314540761
Published by: http://www.sagepublications.com
Additional services and information for Lupus can be found at: Email Alerts: http://lup.sagepub.com/cgi/alerts Subscriptions: http://lup.sagepub.com/subscriptions Reprints: http://www.sagepub.com/journalsReprints.nav Permissions: http://www.sagepub.com/journalsPermissions.nav
>> OnlineFirst Version of Record - Jun 17, 2014 What is This?
Downloaded from lup.sagepub.com at UNIVERSITE LAVAL on June 19, 2014
XML Template (2014) [14.6.2014–10:36am] //blrnas3/cenpro/ApplicationFiles/Journals/SAGE/3B2/LUPJ/Vol00000/140129/APPFile/SG-LUPJ140129.3d
(LUP)
[1–3] [PREPRINTER stage]
Lupus (2014) 0,
1–3
http://lup.sagepub.com
CASE REPORT
Moyamoya syndrome with antiphospholipid antibodies: a case report and literature review Z Wang1, Z Fu1, J Wang1, H Cui2, Z Zhang3 and B Zhang1 1
Department of Neurology, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, China; 2Department of Integrated ICU, The First Affiliated Hospital of Zhengzhou University, Zhengzhou, China; and 3Department of Obstetrics and Gynaecology, Peking University First Hospital, Beijing, China
Moyamoya syndrome is a term applied to typical moyamoya vasculopathy with well-recognized associated conditions. Several systematic studies on the entities of moyamoya syndrome with autoimmune disease have been reported. We report a case of moyamoya syndrome with antiphospholipid antibodies and provide a review of relevant cases from the literature, describing the clinical characteristics and treatments. Lupus (2014) 0, 1–3. Key words: Moyamoya syndrome; antiphospholipid autoantibodies; antiphospholipid syndrome
Introduction Moyamoya disease (MMD) is an uncommon cerebrovascular disease of unknown aetiology that is characterized by a progressive narrowing of the anterior circulation vasculature of the brain with the formation of a network of collateral vessels.1 Moyamoya syndrome (MMS) is a term applied to typical moyamoya vasculopathy with wellrecognized associated conditions.2 Several systematic studies on the entities of MMS with autoimmune disease have been reported, including systemic lupus erythematosus and autoimmune thyroid disease.3,4 However, there have been no systematic studies on MMS with antiphospholipid antibodies (aPL). We report a case of MMS with aPL and provide a review of relevant cases from the literature, describing the clinical characteristics and treatments. Methods We report the case of a 43-year-old woman who suffered from MMS with aPL. We searched the Correspondence to: Boai Zhang, Department of Neurology, The First Affiliated Hospital of Zhengzhou University, No.1 Jianshe East Road, Erqi District, Zhengzhou, Henan Province, 450052, China. Email: [email protected] Zhen Wang and Zhenqiang Fu contributed equally to this report. Received 25 December 2013; accepted 29 May 2013
English-language literature using the PubMed database from 1957 to December 2013. The following keywords were used: moyamoya, antiphospholipid, anticardiolipin antibodies, anti-beta2-glycoprotein I antibodies, and lupus anticoagulant. All potential references that were cited in these articles were also reviewed.
Results Case report A 43-year-old woman was admitted with sudden onset of right-sided hemiparesis during menstruation. Seven years prior, she had experienced transient right-sided weakness; she also had a history of adenomyosis without treatment for three years. She lacked a history of smoking, migraine headaches, livedo reticularis, spontaneous abortion, or deep vein thrombosis. A neurological examination revealed a power of 3/5 in the right limbs. A brain computed tomography (CT) scan revealed multiple low-density lesions in the bilateral centrum semiovale and basal ganglia without haemorrhage. CT angiography showed an occlusion of the bilateral middle cerebral arteries with rich basal collateral vessels (Supplemental Figure 1, available online with this article). Her echocardiogram and thoracoabdominal CT scans were normal; however, a pelvic ultrasound revealed uterine adenomyosis.
! The Author(s), 2014. Reprints and permissions: http://www.sagepub.co.uk/journalsPermissions.nav
Downloaded from lup.sagepub.com at UNIVERSITE LAVAL on June 19, 2014
10.1177/0961203314540761
XML Template (2014) [14.6.2014–10:36am] //blrnas3/cenpro/ApplicationFiles/Journals/SAGE/3B2/LUPJ/Vol00000/140129/APPFile/SG-LUPJ140129.3d
(LUP)
[1–3] [PREPRINTER stage]
Moyamoya syndrome with antiphospholipid antibodies Z Wang et al.
2
The results of her routine laboratory blood tests were normal. Coagulation studies were also normal. Lupus anticoagulant (LA) was negative. Immunoglobulin (Ig)G, IgA and IgM anticardiolipin antibodies (aCL) levels were 34 U GPL/ml (normal
