Turner syndrome with spinal hemorrhage due to vascular malformation
Identifieur interne : 007D96 ( Ncbi/Merge ); précédent : 007D95; suivant : 007D97Turner syndrome with spinal hemorrhage due to vascular malformation
Auteurs : Min Kyung Yu [Corée du Sud] ; Mo Kyung Jung [Corée du Sud] ; Ki Eun Kim [Corée du Sud] ; Ah Reum Kwon [Corée du Sud] ; Hyun Wook Chae [Corée du Sud] ; Duk Hee Kim [Corée du Sud] ; Ho-Seong Kim [Corée du Sud]Source :
- Annals of Pediatric Endocrinology & Metabolism [ 2287-1012 ] ; 2015.
Abstract
Turner syndrome (TS) is a relatively common chromosomal disorder and is associated with a range of comorbidities involving the cardiovascular system. Vascular abnormalities, in particular, are a common finding in cases of TS. However, dissection involving the vertebral arteries is rare. Here, we report the case of a 9-year-old girl with TS who had been treated with growth hormone replacement therapy for the past 3 years. She presented with weakness of both lower legs, and was ultimately diagnosed with spinal hemorrhage due to vascular malformation. We treated her with intravenous high dose dexamethasone (0.6 mg/kg) and she could walk without assistance after 6 days of treatment. In conclusion, when a patient with TS shows sudden weakness of the lower limbs, we should consider the possibility of spinal vessel rupture and try to take spine magnetic resonance imaging as soon as possible. We suggest a direction how to make a proper diagnosis and management of sudden vertebral artery hemorrhage in patients with TS.
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DOI: 10.6065/apem.2015.20.4.235
PubMed: 26817012
PubMed Central: 4722165
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wicri:level="3"><nlm:aff id="A1">Department of Pediatrics, Severance Children's Hospital, Endocrine Research Institute, Yonsei University College of Medicine, Seoul, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Department of Pediatrics, Severance Children's Hospital, Endocrine Research Institute, Yonsei University College of Medicine, Seoul</wicri:regionArea><placeName><settlement type="city">Séoul</settlement><region type="capital">Région capitale de Séoul</region></placeName></affiliation></author><author><name sortKey="Jung, Mo Kyung" sort="Jung, Mo Kyung" uniqKey="Jung M" first="Mo Kyung" last="Jung">Mo Kyung Jung</name><affiliation wicri:level="3"><nlm:aff id="A1">Department of Pediatrics, Severance Children's Hospital, Endocrine Research Institute, Yonsei University College of Medicine, Seoul, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Department of Pediatrics, Severance 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Reum" uniqKey="Kwon A" first="Ah Reum" last="Kwon">Ah Reum Kwon</name><affiliation wicri:level="3"><nlm:aff id="A1">Department of Pediatrics, Severance Children's Hospital, Endocrine Research Institute, Yonsei University College of Medicine, Seoul, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Department of Pediatrics, Severance Children's Hospital, Endocrine Research Institute, Yonsei University College of Medicine, Seoul</wicri:regionArea><placeName><settlement type="city">Séoul</settlement><region type="capital">Région capitale de Séoul</region></placeName></affiliation></author><author><name sortKey="Chae, Hyun Wook" sort="Chae, Hyun Wook" uniqKey="Chae H" first="Hyun Wook" last="Chae">Hyun Wook Chae</name><affiliation wicri:level="3"><nlm:aff id="A1">Department of Pediatrics, Severance Children's Hospital, Endocrine Research Institute, Yonsei University College of Medicine, Seoul, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Department of Pediatrics, Severance Children's Hospital, Endocrine Research Institute, Yonsei University College of Medicine, Seoul</wicri:regionArea><placeName><settlement type="city">Séoul</settlement><region type="capital">Région capitale de Séoul</region></placeName></affiliation></author><author><name sortKey="Kim, Duk Hee" sort="Kim, Duk Hee" uniqKey="Kim D" first="Duk Hee" last="Kim">Duk Hee Kim</name><affiliation wicri:level="3"><nlm:aff id="A2">Department of Pediatrics, Sohwa Children's Hospital, Seoul, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Department of Pediatrics, Sohwa Children's Hospital, Seoul</wicri:regionArea><placeName><settlement type="city">Séoul</settlement><region type="capital">Région capitale de Séoul</region></placeName></affiliation></author><author><name sortKey="Kim, Ho Seong" sort="Kim, Ho Seong" uniqKey="Kim H" first="Ho-Seong" last="Kim">Ho-Seong Kim</name><affiliation wicri:level="3"><nlm:aff id="A1">Department of Pediatrics, Severance Children's Hospital, Endocrine Research Institute, Yonsei University College of Medicine, Seoul, Korea.</nlm:aff><country xml:lang="fr" wicri:curation="lc">Corée du Sud</country><wicri:regionArea>Department of Pediatrics, Severance Children's Hospital, Endocrine Research Institute, Yonsei University College of Medicine, Seoul</wicri:regionArea><placeName><settlement type="city">Séoul</settlement><region type="capital">Région capitale de Séoul</region></placeName></affiliation></author></analytic><series><title level="j">Annals of Pediatric Endocrinology & Metabolism</title><idno type="ISSN">2287-1012</idno><idno type="eISSN">2287-1292</idno><imprint><date when="2015">2015</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Turner syndrome (TS) is a relatively common chromosomal disorder and is associated with a range of comorbidities involving the cardiovascular system. Vascular abnormalities, in particular, are a common finding in cases of TS. However, dissection involving the vertebral arteries is rare. Here, we report the case of a 9-year-old girl with TS who had been treated with growth hormone replacement therapy for the past 3 years. She presented with weakness of both lower legs, and was ultimately diagnosed with spinal hemorrhage due to vascular malformation. We treated her with intravenous high dose dexamethasone (0.6 mg/kg) and she could walk without assistance after 6 days of treatment. In conclusion, when a patient with TS shows sudden weakness of the lower limbs, we should consider the possibility of spinal vessel rupture and try to take spine magnetic resonance imaging as soon as possible. We suggest a direction how to make a proper diagnosis and management of sudden vertebral artery hemorrhage in patients with TS.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Stochholm, K" uniqKey="Stochholm K">K Stochholm</name></author><author><name sortKey="Juul, S" uniqKey="Juul S">S Juul</name></author><author><name sortKey="Juel, K" uniqKey="Juel K">K Juel</name></author><author><name sortKey="Naeraa, Rw" uniqKey="Naeraa R">RW Naeraa</name></author><author><name sortKey="Gravholt, Ch" uniqKey="Gravholt C">CH Gravholt</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Rosen, Km" uniqKey="Rosen K">KM Rosen</name></author><author><name sortKey="Sirota, Dk" uniqKey="Sirota D">DK Sirota</name></author><author><name sortKey="Marinoff, Sc" uniqKey="Marinoff S">SC Marinoff</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Gravholt, Ch" uniqKey="Gravholt C">CH Gravholt</name></author><author><name sortKey="Juul, S" uniqKey="Juul S">S Juul</name></author><author><name sortKey="Naeraa, Rw" uniqKey="Naeraa R">RW Naeraa</name></author><author><name sortKey="Hansen, J" uniqKey="Hansen J">J Hansen</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Buheitel, G" uniqKey="Buheitel G">G Buheitel</name></author><author><name sortKey="Singer, H" uniqKey="Singer H">H Singer</name></author><author><name sortKey="Hofbeck, M" uniqKey="Hofbeck M">M Hofbeck</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Sybert, Vp" uniqKey="Sybert V">VP Sybert</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Muscat, P" uniqKey="Muscat P">P Muscat</name></author><author><name sortKey="Lidov, M" uniqKey="Lidov M">M Lidov</name></author><author><name sortKey="Nahar, T" uniqKey="Nahar T">T Nahar</name></author><author><name sortKey="Tuhrim, S" uniqKey="Tuhrim S">S Tuhrim</name></author><author><name sortKey="Weinberger, J" uniqKey="Weinberger J">J Weinberger</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Chung, J" uniqKey="Chung J">J Chung</name></author><author><name sortKey="Park, Is" uniqKey="Park I">IS Park</name></author><author><name sortKey="Hwang, Sh" uniqKey="Hwang S">SH Hwang</name></author><author><name sortKey="Han, Jw" uniqKey="Han J">JW Han</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Bondy, Ca" uniqKey="Bondy C">CA Bondy</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Bolar, K" uniqKey="Bolar K">K Bolar</name></author><author><name sortKey="Hoffman, Ar" uniqKey="Hoffman A">AR Hoffman</name></author><author><name sortKey="Maneatis, T" uniqKey="Maneatis T">T Maneatis</name></author><author><name sortKey="Lippe, B" uniqKey="Lippe B">B Lippe</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Park, Hk" uniqKey="Park H">HK Park</name></author><author><name sortKey="Lee, Hs" uniqKey="Lee H">HS Lee</name></author><author><name sortKey="Ko, Jh" uniqKey="Ko J">JH Ko</name></author><author><name sortKey="Hwang, It" uniqKey="Hwang I">IT Hwang</name></author><author><name sortKey="Hwang, Js" uniqKey="Hwang J">JS Hwang</name></author></analytic></biblStruct></listBibl></div1></back></TEI><pmc article-type="case-report"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">Ann Pediatr Endocrinol Metab</journal-id><journal-id journal-id-type="iso-abbrev">Ann Pediatr Endocrinol Metab</journal-id><journal-id journal-id-type="publisher-id">APEM</journal-id><journal-title-group><journal-title>Annals of Pediatric Endocrinology & Metabolism</journal-title></journal-title-group><issn pub-type="ppub">2287-1012</issn><issn pub-type="epub">2287-1292</issn><publisher><publisher-name>The Korean Society of Pediatric Endocrinology</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">26817012</article-id><article-id pub-id-type="pmc">4722165</article-id><article-id pub-id-type="doi">10.6065/apem.2015.20.4.235</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Turner syndrome with spinal hemorrhage due to vascular malformation</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Yu</surname><given-names>Min Kyung</given-names></name><degrees>MD</degrees><xref ref-type="aff" rid="A1">1</xref></contrib><contrib contrib-type="author"><name><surname>Jung</surname><given-names>Mo Kyung</given-names></name><degrees>MD</degrees><xref ref-type="aff" rid="A1">1</xref></contrib><contrib contrib-type="author"><name><surname>Kim</surname><given-names>Ki Eun</given-names></name><degrees>MD</degrees><xref ref-type="aff" rid="A1">1</xref></contrib><contrib contrib-type="author"><name><surname>Kwon</surname><given-names>Ah Reum</given-names></name><degrees>MD</degrees><xref ref-type="aff" rid="A1">1</xref></contrib><contrib contrib-type="author" corresp="yes"><name><surname>Chae</surname><given-names>Hyun Wook</given-names></name><degrees>MD</degrees><xref ref-type="aff" rid="A1">1</xref></contrib><contrib contrib-type="author"><name><surname>Kim</surname><given-names>Duk Hee</given-names></name><degrees>MD</degrees><xref ref-type="aff" rid="A2">2</xref></contrib><contrib contrib-type="author"><name><surname>Kim</surname><given-names>Ho-Seong</given-names></name><degrees>MD</degrees><xref ref-type="aff" rid="A1">1</xref></contrib></contrib-group><aff id="A1"><label>1</label>Department of Pediatrics, Severance Children's Hospital, Endocrine Research Institute, Yonsei University College of Medicine, Seoul, Korea.</aff><aff id="A2"><label>2</label>Department of Pediatrics, Sohwa Children's Hospital, Seoul, Korea.</aff><author-notes><corresp>Address for correspondence: Hyun Wook Chae, MD. Department of Pediatrics, Severance Children's Hospital, Endocrine Research Institute, Yonsei University College of Medicine, 50-1 Yonsei-ro, Seodaemun-gu, Seoul 03722, Korea. Tel: +82-2-2019-3355, Fax: +82-2-393-9118, <email>hopechae@yuhs.ac</email></corresp></author-notes><pub-date pub-type="ppub"><month>12</month><year>2015</year></pub-date><pub-date pub-type="epub"><day>31</day><month>12</month><year>2015</year></pub-date><volume>20</volume><issue>4</issue><fpage>235</fpage><lpage>237</lpage><history><date date-type="received"><day>09</day><month>9</month><year>2015</year></date><date date-type="rev-recd"><day>08</day><month>10</month><year>2015</year></date><date date-type="accepted"><day>15</day><month>10</month><year>2015</year></date></history><permissions><copyright-statement>© 2015 Annals of Pediatric Endocrinology & Metabolism</copyright-statement><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc/3.0/"><license-p>This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (<ext-link ext-link-type="uri" xlink:href="http://creativecommons.org/licenses/by-nc/3.0/">http://creativecommons.org/licenses/by-nc/3.0/</ext-link>) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p></license></permissions><abstract><p>Turner syndrome (TS) is a relatively common chromosomal disorder and is associated with a range of comorbidities involving the cardiovascular system. Vascular abnormalities, in particular, are a common finding in cases of TS. However, dissection involving the vertebral arteries is rare. Here, we report the case of a 9-year-old girl with TS who had been treated with growth hormone replacement therapy for the past 3 years. She presented with weakness of both lower legs, and was ultimately diagnosed with spinal hemorrhage due to vascular malformation. We treated her with intravenous high dose dexamethasone (0.6 mg/kg) and she could walk without assistance after 6 days of treatment. In conclusion, when a patient with TS shows sudden weakness of the lower limbs, we should consider the possibility of spinal vessel rupture and try to take spine magnetic resonance imaging as soon as possible. We suggest a direction how to make a proper diagnosis and management of sudden vertebral artery hemorrhage in patients with TS.</p></abstract><kwd-group><kwd>Turner syndrome</kwd><kwd>Vascular malformations</kwd><kwd>Vertebral artery dissection</kwd></kwd-group></article-meta></front><floats-group><fig id="F1" orientation="portrait" position="float"><label>Fig. 1</label><caption><title>T2-weighted magnetic resonance image taken on hospital day 2, shows acute intradural extramedullary spinal hemorrhage (white arrow) at T 11-12.</title></caption><graphic xlink:href="apem-20-235-g001"></graphic></fig><fig id="F2" orientation="portrait" position="float"><label>Fig. 2</label><caption><title>Spinal angiography taken on hospital day 2, shows no remarkable evidence of spinal artery aneurysm or tumor staining (white arrow).</title></caption><graphic xlink:href="apem-20-235-g002"></graphic></fig><fig id="F3" orientation="portrait" position="float"><label>Fig. 3</label><caption><title>Follow-up T2-weighted magnetic resonance image was taken 7 days after discharge, showing the resolving process of the subacute hemorrhage and the still existing suspicious cavernous malformation (white arrows) at T 11-12.</title></caption><graphic xlink:href="apem-20-235-g003"></graphic></fig></floats-group></pmc><affiliations><list><country><li>Corée du Sud</li></country><region><li>Région capitale de Séoul</li></region><settlement><li>Séoul</li></settlement></list><tree><country name="Corée du Sud"><region name="Région capitale de Séoul"><name sortKey="Yu, Min Kyung" sort="Yu, Min Kyung" uniqKey="Yu M" first="Min Kyung" last="Yu">Min Kyung Yu</name></region><name sortKey="Chae, Hyun Wook" sort="Chae, Hyun Wook" uniqKey="Chae H" first="Hyun Wook" last="Chae">Hyun Wook Chae</name><name sortKey="Jung, Mo Kyung" sort="Jung, Mo Kyung" uniqKey="Jung M" first="Mo Kyung" last="Jung">Mo Kyung Jung</name><name sortKey="Kim, Duk Hee" sort="Kim, Duk Hee" uniqKey="Kim D" first="Duk Hee" last="Kim">Duk Hee Kim</name><name sortKey="Kim, Ho Seong" sort="Kim, Ho Seong" uniqKey="Kim H" first="Ho-Seong" last="Kim">Ho-Seong Kim</name><name sortKey="Kim, Ki Eun" sort="Kim, Ki Eun" uniqKey="Kim K" first="Ki Eun" last="Kim">Ki Eun Kim</name><name sortKey="Kwon, Ah Reum" sort="Kwon, Ah Reum" uniqKey="Kwon A" first="Ah Reum" last="Kwon">Ah Reum Kwon</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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