Atypical retiform hemangioendothelioma arising in a patient with Milroy disease: a case report and review of the literature.
Identifieur interne : 000583 ( PubMed/Corpus ); précédent : 000582; suivant : 000584Atypical retiform hemangioendothelioma arising in a patient with Milroy disease: a case report and review of the literature.
Auteurs : Aileen Grace P. Arriola ; Laura A. Taylor ; Eseosa Asemota ; Markus D. Boos ; David E. Elder ; Kristy L. Weber ; Robert G. Micheletti ; Paul J. ZhangSource :
- Journal of cutaneous pathology [ 1600-0560 ] ; 2017.
English descriptors
- KwdEn :
- MESH :
- complications : Lymphedema.
- pathology : Hemangioendothelioma, Skin Neoplasms, Vascular Neoplasms.
- Female, Humans, Middle Aged.
Abstract
Retiform hemangioendothelioma (RH) is a rare vascular neoplasm with a high rate of local recurrence and low metastatic potential. We describe an unusual case of RH in a 45-year-old patient with Milroy disease, with a prominent solid component diffusely involving a chronic lymphedematous leg. This case is consistent with the postulated relationship between lymphedema and vascular neoplasms developing as a result of local immune dysfunction, and highlights the need to closely monitor patients with Milroy disease for pathologic changes. Our case highlights a unique example of RH with atypical features. There are several noteworthy unusual clinical and histologic findings including diffuse involvement of an entire limb, solid component with cytologic atypia, D2-40 expression, and first-time-reported association with Milroy disease. Given the atypical histologic presentation of cytologic atypia, solid areas and atypical immunohistochemical profile with D2-40 positivity, this case could cause diagnostic difficulty, especially in the setting of such a broad clinical differential.
DOI: 10.1111/cup.12844
PubMed: 27730656
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Atypical retiform hemangioendothelioma arising in a patient with Milroy disease: a case report and review of the literature.</title><author><name sortKey="Arriola, Aileen Grace P" sort="Arriola, Aileen Grace P" uniqKey="Arriola A" first="Aileen Grace P" last="Arriola">Aileen Grace P. Arriola</name><affiliation><nlm:affiliation>Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Taylor, Laura A" sort="Taylor, Laura A" uniqKey="Taylor L" first="Laura A" last="Taylor">Laura A. Taylor</name><affiliation><nlm:affiliation>Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Asemota, Eseosa" sort="Asemota, Eseosa" uniqKey="Asemota E" first="Eseosa" last="Asemota">Eseosa Asemota</name><affiliation><nlm:affiliation>Clinical observer and research fellow, Department of Dermatology, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Boos, Markus D" sort="Boos, Markus D" uniqKey="Boos M" first="Markus D" last="Boos">Markus D. Boos</name><affiliation><nlm:affiliation>Department of Pediatrics (Dermatology), University of Washington, Seattle, WA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Elder, David E" sort="Elder, David E" uniqKey="Elder D" first="David E" last="Elder">David E. Elder</name><affiliation><nlm:affiliation>Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Weber, Kristy L" sort="Weber, Kristy L" uniqKey="Weber K" first="Kristy L" last="Weber">Kristy L. Weber</name><affiliation><nlm:affiliation>Department of Orthopaedic Surgery, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Micheletti, Robert G" sort="Micheletti, Robert G" uniqKey="Micheletti R" first="Robert G" last="Micheletti">Robert G. Micheletti</name><affiliation><nlm:affiliation>Department of Dermatology, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Zhang, Paul J" sort="Zhang, Paul J" uniqKey="Zhang P" first="Paul J" last="Zhang">Paul J. Zhang</name><affiliation><nlm:affiliation>Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2017">2017</date><idno type="RBID">pubmed:27730656</idno><idno type="pmid">27730656</idno><idno type="doi">10.1111/cup.12844</idno><idno type="wicri:Area/PubMed/Corpus">000583</idno><idno type="wicri:explorRef" wicri:stream="PubMed" wicri:step="Corpus" wicri:corpus="PubMed">000583</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en">Atypical retiform hemangioendothelioma arising in a patient with Milroy disease: a case report and review of the literature.</title><author><name sortKey="Arriola, Aileen Grace P" sort="Arriola, Aileen Grace P" uniqKey="Arriola A" first="Aileen Grace P" last="Arriola">Aileen Grace P. Arriola</name><affiliation><nlm:affiliation>Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Taylor, Laura A" sort="Taylor, Laura A" uniqKey="Taylor L" first="Laura A" last="Taylor">Laura A. Taylor</name><affiliation><nlm:affiliation>Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Asemota, Eseosa" sort="Asemota, Eseosa" uniqKey="Asemota E" first="Eseosa" last="Asemota">Eseosa Asemota</name><affiliation><nlm:affiliation>Clinical observer and research fellow, Department of Dermatology, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Boos, Markus D" sort="Boos, Markus D" uniqKey="Boos M" first="Markus D" last="Boos">Markus D. Boos</name><affiliation><nlm:affiliation>Department of Pediatrics (Dermatology), University of Washington, Seattle, WA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Elder, David E" sort="Elder, David E" uniqKey="Elder D" first="David E" last="Elder">David E. Elder</name><affiliation><nlm:affiliation>Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Weber, Kristy L" sort="Weber, Kristy L" uniqKey="Weber K" first="Kristy L" last="Weber">Kristy L. Weber</name><affiliation><nlm:affiliation>Department of Orthopaedic Surgery, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Micheletti, Robert G" sort="Micheletti, Robert G" uniqKey="Micheletti R" first="Robert G" last="Micheletti">Robert G. Micheletti</name><affiliation><nlm:affiliation>Department of Dermatology, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author><author><name sortKey="Zhang, Paul J" sort="Zhang, Paul J" uniqKey="Zhang P" first="Paul J" last="Zhang">Paul J. Zhang</name><affiliation><nlm:affiliation>Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</nlm:affiliation></affiliation></author></analytic><series><title level="j">Journal of cutaneous pathology</title><idno type="eISSN">1600-0560</idno><imprint><date when="2017" type="published">2017</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Female</term><term>Hemangioendothelioma (pathology)</term><term>Humans</term><term>Lymphedema (complications)</term><term>Middle Aged</term><term>Skin Neoplasms (pathology)</term><term>Vascular Neoplasms (pathology)</term></keywords><keywords scheme="MESH" qualifier="complications" xml:lang="en"><term>Lymphedema</term></keywords><keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>Hemangioendothelioma</term><term>Skin Neoplasms</term><term>Vascular Neoplasms</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Female</term><term>Humans</term><term>Middle Aged</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Retiform hemangioendothelioma (RH) is a rare vascular neoplasm with a high rate of local recurrence and low metastatic potential. We describe an unusual case of RH in a 45-year-old patient with Milroy disease, with a prominent solid component diffusely involving a chronic lymphedematous leg. This case is consistent with the postulated relationship between lymphedema and vascular neoplasms developing as a result of local immune dysfunction, and highlights the need to closely monitor patients with Milroy disease for pathologic changes. Our case highlights a unique example of RH with atypical features. There are several noteworthy unusual clinical and histologic findings including diffuse involvement of an entire limb, solid component with cytologic atypia, D2-40 expression, and first-time-reported association with Milroy disease. Given the atypical histologic presentation of cytologic atypia, solid areas and atypical immunohistochemical profile with D2-40 positivity, this case could cause diagnostic difficulty, especially in the setting of such a broad clinical differential.</div></front></TEI><pubmed><MedlineCitation Status="MEDLINE" Owner="NLM"><PMID Version="1">27730656</PMID><DateCreated><Year>2016</Year><Month>10</Month><Day>12</Day></DateCreated><DateCompleted><Year>2017</Year><Month>08</Month><Day>15</Day></DateCompleted><DateRevised><Year>2017</Year><Month>08</Month><Day>17</Day></DateRevised><Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1600-0560</ISSN><JournalIssue CitedMedium="Internet"><Volume>44</Volume><Issue>1</Issue><PubDate><Year>2017</Year><Month>Jan</Month></PubDate></JournalIssue><Title>Journal of cutaneous pathology</Title><ISOAbbreviation>J. Cutan. Pathol.</ISOAbbreviation></Journal><ArticleTitle>Atypical retiform hemangioendothelioma arising in a patient with Milroy disease: a case report and review of the literature.</ArticleTitle><Pagination><MedlinePgn>98-103</MedlinePgn></Pagination><ELocationID EIdType="doi" ValidYN="Y">10.1111/cup.12844</ELocationID><Abstract><AbstractText>Retiform hemangioendothelioma (RH) is a rare vascular neoplasm with a high rate of local recurrence and low metastatic potential. We describe an unusual case of RH in a 45-year-old patient with Milroy disease, with a prominent solid component diffusely involving a chronic lymphedematous leg. This case is consistent with the postulated relationship between lymphedema and vascular neoplasms developing as a result of local immune dysfunction, and highlights the need to closely monitor patients with Milroy disease for pathologic changes. Our case highlights a unique example of RH with atypical features. There are several noteworthy unusual clinical and histologic findings including diffuse involvement of an entire limb, solid component with cytologic atypia, D2-40 expression, and first-time-reported association with Milroy disease. Given the atypical histologic presentation of cytologic atypia, solid areas and atypical immunohistochemical profile with D2-40 positivity, this case could cause diagnostic difficulty, especially in the setting of such a broad clinical differential.</AbstractText><CopyrightInformation>© 2016 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.</CopyrightInformation></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Arriola</LastName><ForeName>Aileen Grace P</ForeName><Initials>AG</Initials><AffiliationInfo><Affiliation>Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Taylor</LastName><ForeName>Laura A</ForeName><Initials>LA</Initials><AffiliationInfo><Affiliation>Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Asemota</LastName><ForeName>Eseosa</ForeName><Initials>E</Initials><AffiliationInfo><Affiliation>Clinical observer and research fellow, Department of Dermatology, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Boos</LastName><ForeName>Markus D</ForeName><Initials>MD</Initials><AffiliationInfo><Affiliation>Department of Pediatrics (Dermatology), University of Washington, Seattle, WA, USA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Elder</LastName><ForeName>David E</ForeName><Initials>DE</Initials><AffiliationInfo><Affiliation>Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Weber</LastName><ForeName>Kristy L</ForeName><Initials>KL</Initials><AffiliationInfo><Affiliation>Department of Orthopaedic Surgery, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Micheletti</LastName><ForeName>Robert G</ForeName><Initials>RG</Initials><AffiliationInfo><Affiliation>Department of Dermatology, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Zhang</LastName><ForeName>Paul J</ForeName><Initials>PJ</Initials><AffiliationInfo><Affiliation>Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.</Affiliation></AffiliationInfo></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType UI="D002363">Case Reports</PublicationType><PublicationType UI="D016428">Journal Article</PublicationType><PublicationType UI="D016454">Review</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2016</Year><Month>11</Month><Day>16</Day></ArticleDate></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>J Cutan Pathol</MedlineTA><NlmUniqueID>0425124</NlmUniqueID><ISSNLinking>0303-6987</ISSNLinking></MedlineJournalInfo><CitationSubset>IM</CitationSubset><MeshHeadingList><MeshHeading><DescriptorName UI="D005260" MajorTopicYN="N">Female</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D006390" MajorTopicYN="N">Hemangioendothelioma</DescriptorName><QualifierName UI="Q000473" MajorTopicYN="Y">pathology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D006801" MajorTopicYN="N">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D008209" MajorTopicYN="N">Lymphedema</DescriptorName><QualifierName UI="Q000150" MajorTopicYN="Y">complications</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D008875" MajorTopicYN="N">Middle Aged</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D012878" MajorTopicYN="N">Skin Neoplasms</DescriptorName><QualifierName UI="Q000473" MajorTopicYN="Y">pathology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D019043" MajorTopicYN="N">Vascular Neoplasms</DescriptorName><QualifierName UI="Q000473" MajorTopicYN="Y">pathology</QualifierName></MeshHeading></MeshHeadingList><KeywordList Owner="NOTNLM"><Keyword MajorTopicYN="N">Milroy disease</Keyword><Keyword MajorTopicYN="N">chronic lymphedema</Keyword><Keyword MajorTopicYN="N">retiform hemangioendothelioma</Keyword><Keyword MajorTopicYN="N">vascular neoplasm</Keyword></KeywordList></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="received"><Year>2015</Year><Month>12</Month><Day>15</Day></PubMedPubDate><PubMedPubDate PubStatus="revised"><Year>2016</Year><Month>06</Month><Day>15</Day></PubMedPubDate><PubMedPubDate PubStatus="accepted"><Year>2016</Year><Month>10</Month><Day>06</Day></PubMedPubDate><PubMedPubDate PubStatus="pubmed"><Year>2016</Year><Month>10</Month><Day>13</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="medline"><Year>2017</Year><Month>8</Month><Day>16</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate 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