A rare presentation of Klippel–Trenaunay syndrome with bilateral lower limbs
Identifieur interne : 000835 ( Main/Exploration ); précédent : 000834; suivant : 000836A rare presentation of Klippel–Trenaunay syndrome with bilateral lower limbs
Auteurs : Akira Baba [Japon] ; Shinji Yamazoe [Japon] ; Yumi Okuyama [Japon] ; Kanichiro Shimizu [Japon] ; Yuko Kobashi [Japon] ; Yosuke Nozawa [Japon] ; Yohei Munetomo [Japon] ; Takuji Mogami [Japon]Source :
- Journal of Surgical Case Reports [ 2042-8812 ] ; 2017.
Abstract
Klippel–Trenaunay syndrome (KTS) is a vascular lymphatic malformation underlying with bony and soft tissue hypertrophy. It is a rare condition presenting in 1 out of 10 000 people. The growth disturbance due to KTS is more commonly unilateral (85%) than bilateral (12.5%), and most rarely crossed-bilateral (2.5%). A man in his 40s presented to our hospital with a complaint of lower limb discomfort. Radiograph, ultrasonography, computed tomography venography, magnetic resonance (and venography) showed various radiological findings characteristic for KTS. Because the patient was symptomatic, he underwent stripping of bilateral great saphenous vein and varicectomy of bilateral legs. The surgical procedures were undertaken successfully, and there has been no recurrent symptom for about 2 years and a half. In this study, we report a very rare case of bilateral KTS diagnosed by radiological and clinical manifestations with some literature review.
Url:
DOI: 10.1093/jscr/rjx024
PubMed: 28458832
PubMed Central: 5400491
Affiliations:
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strict</wicri:regionArea></affiliation></author><author><name sortKey="Okuyama, Yumi" sort="Okuyama, Yumi" uniqKey="Okuyama Y" first="Yumi" last="Okuyama">Yumi Okuyama</name><affiliation wicri:level="1"><nlm:aff id="rjx024af1"><addr-line>Department of Radiology, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author><author><name sortKey="Shimizu, Kanichiro" sort="Shimizu, Kanichiro" uniqKey="Shimizu K" first="Kanichiro" last="Shimizu">Kanichiro Shimizu</name><affiliation wicri:level="1"><nlm:aff id="rjx024af2"><addr-line>Department of Radiology</addr-line>,<institution>The Jikei University Kashiwa Hospital</institution>,<addr-line>Kashiwa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author><author><name sortKey="Kobashi, Yuko" sort="Kobashi, Yuko" uniqKey="Kobashi Y" first="Yuko" last="Kobashi">Yuko Kobashi</name><affiliation wicri:level="1"><nlm:aff id="rjx024af1"><addr-line>Department of Radiology, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author><author><name sortKey="Nozawa, Yosuke" sort="Nozawa, Yosuke" uniqKey="Nozawa Y" first="Yosuke" last="Nozawa">Yosuke Nozawa</name><affiliation wicri:level="1"><nlm:aff id="rjx024af1"><addr-line>Department of Radiology, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author><author><name sortKey="Munetomo, Yohei" sort="Munetomo, Yohei" uniqKey="Munetomo Y" first="Yohei" last="Munetomo">Yohei Munetomo</name><affiliation wicri:level="1"><nlm:aff id="rjx024af1"><addr-line>Department of Radiology, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author><author><name sortKey="Mogami, Takuji" sort="Mogami, Takuji" uniqKey="Mogami T" first="Takuji" last="Mogami">Takuji Mogami</name><affiliation wicri:level="1"><nlm:aff id="rjx024af1"><addr-line>Department of Radiology, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">28458832</idno><idno 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xml:lang="en" level="a" type="main">A rare presentation of Klippel–Trenaunay syndrome with bilateral lower limbs</title><author><name sortKey="Baba, Akira" sort="Baba, Akira" uniqKey="Baba A" first="Akira" last="Baba">Akira Baba</name><affiliation wicri:level="1"><nlm:aff id="rjx024af1"><addr-line>Department of Radiology, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author><author><name sortKey="Yamazoe, Shinji" sort="Yamazoe, Shinji" uniqKey="Yamazoe S" first="Shinji" last="Yamazoe">Shinji Yamazoe</name><affiliation wicri:level="1"><nlm:aff id="rjx024af1"><addr-line>Department of Radiology, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author><author><name sortKey="Okuyama, Yumi" sort="Okuyama, Yumi" uniqKey="Okuyama Y" first="Yumi" last="Okuyama">Yumi Okuyama</name><affiliation wicri:level="1"><nlm:aff id="rjx024af1"><addr-line>Department of Radiology, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author><author><name sortKey="Shimizu, Kanichiro" sort="Shimizu, Kanichiro" uniqKey="Shimizu K" first="Kanichiro" last="Shimizu">Kanichiro Shimizu</name><affiliation wicri:level="1"><nlm:aff id="rjx024af2"><addr-line>Department of Radiology</addr-line>,<institution>The Jikei University Kashiwa Hospital</institution>,<addr-line>Kashiwa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author><author><name sortKey="Kobashi, Yuko" sort="Kobashi, Yuko" uniqKey="Kobashi Y" first="Yuko" last="Kobashi">Yuko Kobashi</name><affiliation wicri:level="1"><nlm:aff id="rjx024af1"><addr-line>Department of Radiology, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author><author><name sortKey="Nozawa, Yosuke" sort="Nozawa, Yosuke" uniqKey="Nozawa Y" first="Yosuke" last="Nozawa">Yosuke Nozawa</name><affiliation wicri:level="1"><nlm:aff id="rjx024af1"><addr-line>Department of Radiology, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author><author><name sortKey="Munetomo, Yohei" sort="Munetomo, Yohei" uniqKey="Munetomo Y" first="Yohei" last="Munetomo">Yohei Munetomo</name><affiliation wicri:level="1"><nlm:aff id="rjx024af1"><addr-line>Department of Radiology, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author><author><name sortKey="Mogami, Takuji" sort="Mogami, Takuji" uniqKey="Mogami T" first="Takuji" last="Mogami">Takuji Mogami</name><affiliation wicri:level="1"><nlm:aff id="rjx024af1"><addr-line>Department of Radiology, Tokyo Dental College Ichikawa General Hospital, Ichikawa, Chiba</addr-line>,<country>Japan</country></nlm:aff><country xml:lang="fr">Japon</country><wicri:regionArea># see nlm:aff country strict</wicri:regionArea></affiliation></author></analytic><series><title level="j">Journal of Surgical Case Reports</title><idno type="eISSN">2042-8812</idno><imprint><date when="2017">2017</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><title>Abstract</title><p>Klippel–Trenaunay syndrome (KTS) is a vascular lymphatic malformation underlying with bony and soft tissue hypertrophy. It is a rare condition presenting in 1 out of 10 000 people. The growth disturbance due to KTS is more commonly unilateral (85%) than bilateral (12.5%), and most rarely crossed-bilateral (2.5%). A man in his 40s presented to our hospital with a complaint of lower limb discomfort. Radiograph, ultrasonography, computed tomography venography, magnetic resonance (and venography) showed various radiological findings characteristic for KTS. Because the patient was symptomatic, he underwent stripping of bilateral great saphenous vein and varicectomy of bilateral legs. The surgical procedures were undertaken successfully, and there has been no recurrent symptom for about 2 years and a half. In this study, we report a very rare case of bilateral KTS diagnosed by radiological and clinical manifestations with some literature review.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Mulliken, Jb" uniqKey="Mulliken J">JB Mulliken</name></author><author><name sortKey="Glowacki, J" uniqKey="Glowacki J">J Glowacki</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Redondo, P" uniqKey="Redondo P">P Redondo</name></author><author><name sortKey="Aguado, L" uniqKey="Aguado L">L Aguado</name></author><author><name sortKey="Martinez Cuesta, A" uniqKey="Martinez Cuesta A">A Martinez-Cuesta</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kim, Yw" uniqKey="Kim Y">YW Kim</name></author><author><name sortKey="Lee, Bb" uniqKey="Lee B">BB Lee</name></author><author><name sortKey="Cho, Jh" uniqKey="Cho J">JH Cho</name></author><author><name sortKey="Do, Ys" uniqKey="Do Y">YS Do</name></author><author><name sortKey="Kim, Di" uniqKey="Kim D">DI Kim</name></author><author><name sortKey="Kim, Es" uniqKey="Kim E">ES Kim</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Uihlein, Lc" uniqKey="Uihlein L">LC Uihlein</name></author><author><name sortKey="Liang, Mg" uniqKey="Liang M">MG Liang</name></author><author><name sortKey="Fishman, Sj" uniqKey="Fishman S">SJ Fishman</name></author><author><name sortKey="Alomari, Ai" uniqKey="Alomari A">AI Alomari</name></author><author><name sortKey="Mulliken, Jb" uniqKey="Mulliken J">JB Mulliken</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Dasgupta, R" uniqKey="Dasgupta R">R Dasgupta</name></author><author><name sortKey="Fishman, Sj" uniqKey="Fishman S">SJ Fishman</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Mavili, E" uniqKey="Mavili E">E Mavili</name></author><author><name sortKey="Ozturk, M" uniqKey="Ozturk M">M Ozturk</name></author><author><name sortKey="Akcali, Y" uniqKey="Akcali Y">Y Akcali</name></author><author><name sortKey="Donmez, H" uniqKey="Donmez H">H Donmez</name></author><author><name sortKey="Yikilmaz, A" uniqKey="Yikilmaz A">A Yikilmaz</name></author><author><name sortKey="Tokmak, Tt" uniqKey="Tokmak T">TT Tokmak</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Zea, Mi" uniqKey="Zea M">MI Zea</name></author><author><name sortKey="Hanif, M" uniqKey="Hanif M">M Hanif</name></author><author><name sortKey="Habib, M" uniqKey="Habib M">M Habib</name></author><author><name sortKey="Ansari, A" uniqKey="Ansari A">A Ansari</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Gianlupi, A" uniqKey="Gianlupi A">A Gianlupi</name></author><author><name sortKey="Harper, Rw" uniqKey="Harper R">RW Harper</name></author><author><name sortKey="Dwyre, Dm" uniqKey="Dwyre D">DM Dwyre</name></author><author><name sortKey="Marelich, Gp" uniqKey="Marelich G">GP Marelich</name></author></analytic></biblStruct></listBibl></div1></back></TEI><affiliations><list><country><li>Japon</li></country></list><tree><country name="Japon"><noRegion><name sortKey="Baba, Akira" sort="Baba, Akira" uniqKey="Baba A" first="Akira" last="Baba">Akira Baba</name></noRegion><name sortKey="Kobashi, Yuko" sort="Kobashi, Yuko" uniqKey="Kobashi Y" first="Yuko" last="Kobashi">Yuko Kobashi</name><name sortKey="Mogami, Takuji" sort="Mogami, Takuji" uniqKey="Mogami T" first="Takuji" last="Mogami">Takuji Mogami</name><name sortKey="Munetomo, Yohei" sort="Munetomo, Yohei" uniqKey="Munetomo Y" first="Yohei" last="Munetomo">Yohei Munetomo</name><name sortKey="Nozawa, Yosuke" sort="Nozawa, Yosuke" uniqKey="Nozawa Y" first="Yosuke" last="Nozawa">Yosuke Nozawa</name><name sortKey="Okuyama, Yumi" sort="Okuyama, Yumi" uniqKey="Okuyama Y" first="Yumi" last="Okuyama">Yumi Okuyama</name><name sortKey="Shimizu, Kanichiro" sort="Shimizu, Kanichiro" uniqKey="Shimizu K" first="Kanichiro" last="Shimizu">Kanichiro Shimizu</name><name sortKey="Yamazoe, Shinji" sort="Yamazoe, Shinji" uniqKey="Yamazoe S" first="Shinji" last="Yamazoe">Shinji Yamazoe</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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