Kasabach-Merritt Phenomenon: Classic Presentation and Management Options
Identifieur interne : 004958 ( Pmc/Curation ); précédent : 004957; suivant : 004959Kasabach-Merritt Phenomenon: Classic Presentation and Management Options
Auteurs : Priya Mahajan ; Judith Margolin ; Ionela IacobasSource :
- Clinical Medicine Insights: Blood Disorders [ 1179-545X ] ; 2017.
Abstract
Kasabach-Merritt phenomenon (KMP) is a rare consumptive coagulopathy associated with specific vascular tumors, kaposiform hemangioendothelioma, and tufted angioma. Kasabach-Merritt phenomenon, characterized by profound thrombocytopenia, hypofibrinogenemia, elevated fibrin split products, and rapid tumor growth, can be life-threatening. Severe symptomatic anemia may also be present. With prompt diagnosis and management, KMP can resolve and vascular tumors have been shown to regress. This review highlights the clinical presentation, histopathology, management, and treatment of KMP associated with kaposiform hemangioendothelioma, and less frequently tufted angioma. A classic clinical case is described to illustrate the presentation and our management of a patient with KMP.
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DOI: 10.1177/1179545X17699849
PubMed: 28579853
PubMed Central: 5428202
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Kasabach-Merritt Phenomenon: Classic Presentation and Management Options</title><author><name sortKey="Mahajan, Priya" sort="Mahajan, Priya" uniqKey="Mahajan P" first="Priya" last="Mahajan">Priya Mahajan</name></author><author><name sortKey="Margolin, Judith" sort="Margolin, Judith" uniqKey="Margolin J" first="Judith" last="Margolin">Judith Margolin</name></author><author><name sortKey="Iacobas, Ionela" sort="Iacobas, Ionela" uniqKey="Iacobas I" first="Ionela" last="Iacobas">Ionela Iacobas</name></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">28579853</idno><idno type="pmc">5428202</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5428202</idno><idno type="RBID">PMC:5428202</idno><idno type="doi">10.1177/1179545X17699849</idno><date when="2017">2017</date><idno type="wicri:Area/Pmc/Corpus">004959</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">004959</idno><idno type="wicri:Area/Pmc/Curation">004958</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Curation">004958</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Kasabach-Merritt Phenomenon: Classic Presentation and Management Options</title><author><name sortKey="Mahajan, Priya" sort="Mahajan, Priya" uniqKey="Mahajan P" first="Priya" last="Mahajan">Priya Mahajan</name></author><author><name sortKey="Margolin, Judith" sort="Margolin, Judith" uniqKey="Margolin J" first="Judith" last="Margolin">Judith Margolin</name></author><author><name sortKey="Iacobas, Ionela" sort="Iacobas, Ionela" uniqKey="Iacobas I" first="Ionela" last="Iacobas">Ionela Iacobas</name></author></analytic><series><title level="j">Clinical Medicine Insights: Blood Disorders</title><idno type="eISSN">1179-545X</idno><imprint><date when="2017">2017</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Kasabach-Merritt phenomenon (KMP) is a rare consumptive coagulopathy associated with specific vascular tumors, kaposiform hemangioendothelioma, and tufted angioma. Kasabach-Merritt phenomenon, characterized by profound thrombocytopenia, hypofibrinogenemia, elevated fibrin split products, and rapid tumor growth, can be life-threatening. Severe symptomatic anemia may also be present. With prompt diagnosis and management, KMP can resolve and vascular tumors have been shown to regress. This review highlights the clinical presentation, histopathology, management, and treatment of KMP associated with kaposiform hemangioendothelioma, and less frequently tufted angioma. A classic clinical case is described to illustrate the presentation and our management of a patient with KMP.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Kasabach, Hh" uniqKey="Kasabach H">HH Kasabach</name></author><author><name sortKey="Merritt, Kk" uniqKey="Merritt K">KK Merritt</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Rodriguez, V" uniqKey="Rodriguez V">V Rodriguez</name></author><author><name sortKey="Lee, A" uniqKey="Lee A">A Lee</name></author><author><name sortKey="Witman, Pm" uniqKey="Witman P">PM Witman</name></author><author><name sortKey="Anderson, Pa" uniqKey="Anderson P">PA Anderson</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Sarkar, M" uniqKey="Sarkar M">M Sarkar</name></author><author><name sortKey="Mulliken, Jb" uniqKey="Mulliken J">JB Mulliken</name></author><author><name sortKey="Kozakewich, Hp" uniqKey="Kozakewich H">HP 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<front><journal-meta><journal-id journal-id-type="nlm-ta">Clin Med Insights Blood Disord</journal-id><journal-id journal-id-type="iso-abbrev">Clin Med Insights Blood Disord</journal-id><journal-id journal-id-type="publisher-id">Clinical Medicine Insights: Blood Disorders</journal-id><journal-title-group><journal-title>Clinical Medicine Insights: Blood Disorders</journal-title></journal-title-group><issn pub-type="epub">1179-545X</issn><publisher><publisher-name>SAGE Publications</publisher-name><publisher-loc>Sage UK: London, England</publisher-loc></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">28579853</article-id><article-id pub-id-type="pmc">5428202</article-id><article-id pub-id-type="doi">10.1177/1179545X17699849</article-id><article-id pub-id-type="publisher-id">10.1177_1179545x17699849</article-id><article-id pub-id-type="publisher-manuscript">BDX-0038926</article-id><article-categories><subj-group subj-group-type="heading"><subject>Review</subject></subj-group></article-categories><title-group><article-title>Kasabach-Merritt Phenomenon: Classic Presentation and Management Options</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Mahajan</surname><given-names>Priya</given-names></name></contrib><contrib contrib-type="author"><name><surname>Margolin</surname><given-names>Judith</given-names></name></contrib><contrib contrib-type="author"><name><surname>Iacobas</surname><given-names>Ionela</given-names></name><xref ref-type="corresp" rid="c1-10.1177_1179545x17699849"></xref></contrib><aff id="af1-10.1177_1179545x17699849">Department of Pediatrics, Baylor College of Medicine, Vascular Anomalies Center at Texas Children’s Cancer and Hematology Centers, Texas Children’s Hospital, Houston, TX, USA.</aff></contrib-group><author-notes><corresp id="c1-10.1177_1179545x17699849">CORRESPONDING AUTHOR: Ionela Iacobas, Pediatric Hematology-Oncology, Medical Director, TCH Vascular Anomalies Center, Texas Children’s Hospital, Baylor College of Medicine, 1102 Bates Street suite 1025.06 Houston, TX 77030, USA. Email: <email>ixiacoba@txch.org</email></corresp></author-notes><pub-date pub-type="collection"><year>2017</year></pub-date><pub-date pub-type="epub"><day>16</day><month>3</month><year>2017</year></pub-date><volume>10</volume><elocation-id>1179545X17699849</elocation-id><history><date date-type="received"><day>21</day><month>10</month><year>2016</year></date><date date-type="accepted"><day>16</day><month>2</month><year>2017</year></date></history><permissions><copyright-statement>© The Author(s) 2017</copyright-statement><copyright-year>2017</copyright-year><copyright-holder content-type="sage">SAGE Publications Ltd unless otherwise noted. Manuscript content on this site is licensed under Creative Commons Licenses</copyright-holder><license license-type="open-access"><license-p>This article is distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 License (<ext-link ext-link-type="uri" xlink:href="http://www.creativecommons.org/licenses/by-nc/4.0/">http://www.creativecommons.org/licenses/by-nc/4.0/</ext-link>) which permits non-commercial use, reproduction and distribution of the work without further permission provided the original work is attributed as specified on the SAGE and Open Access page(<ext-link ext-link-type="uri" xlink:href="https://us.sagepub.com/en-us/nam/open-access-at-sage">https://us.sagepub.com/en-us/nam/open-access-at-sage</ext-link>).</license-p></license></permissions><abstract><p>Kasabach-Merritt phenomenon (KMP) is a rare consumptive coagulopathy associated with specific vascular tumors, kaposiform hemangioendothelioma, and tufted angioma. Kasabach-Merritt phenomenon, characterized by profound thrombocytopenia, hypofibrinogenemia, elevated fibrin split products, and rapid tumor growth, can be life-threatening. Severe symptomatic anemia may also be present. With prompt diagnosis and management, KMP can resolve and vascular tumors have been shown to regress. This review highlights the clinical presentation, histopathology, management, and treatment of KMP associated with kaposiform hemangioendothelioma, and less frequently tufted angioma. A classic clinical case is described to illustrate the presentation and our management of a patient with KMP.</p></abstract><kwd-group><kwd>Kasabach-Merritt phenomenon (KMP)</kwd><kwd>kaposiform hemangioendothelioma (KHE)</kwd><kwd>thrombocytopenia</kwd><kwd>coagulopathy</kwd><kwd>sirolimus</kwd></kwd-group></article-meta></front><floats-group><fig id="f1-10.1177_1179545x17699849" position="float"><label>Figure 1</label><caption><p>Classic clinical case of Kaposiform hemangioendothelioma with Kasabach-Merritt phenomenon (KMP) diagnosed in a 5-week-old patient. Diagnosis, response to blood product transfusion, and resolution of KMP with sirolimus + steroids therapy. (A) Platelet counts (normal: 150–450 × 103/µL), (B) hemoglobin (normal: 9.5–13.5 g/dL), (C) fibrinogen (normal: 220–440 mg/dL), and (D) <sc>d</sc>-dimer (normal: <0.4 µg/mL).</p></caption><graphic xlink:href="10.1177_1179545X17699849-fig1"></graphic></fig><fig id="f2-10.1177_1179545x17699849" position="float"><label>Figure 2</label><caption><p>Classic clinical case of Kaposiform hemangioendothelioma with Kasabach-Merritt phenomenon (KMP) diagnosed in a 5-week-old patient. Extension of cutaneous involvement. Photos presented with mother’s permission. (A) At the peak of activity of KMP (day 4)—patient with purpura, edema, irregular margins, and extensive size. (B) After 3 weeks of treatment, the cutaneous component improved significantly. (C) After 2 months of therapy with sirolimus and steroids, complete resolution of the cutaneous component was achieved.</p></caption><graphic xlink:href="10.1177_1179545X17699849-fig2"></graphic></fig><table-wrap id="t1-10.1177_1179545x17699849" position="float"><label>Table 1</label><caption><p>Recommended pharmacologic therapies for KHE/TA with KMP.</p></caption><graphic xlink:href="10.1177_1179545X17699849-table1"></graphic></table-wrap></floats-group></pmc></record>Pour manipuler ce document sous Unix (Dilib)
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