Combined Lymphedema and Capillary Malformation of the Lower Extremity
Identifieur interne : 001281 ( Main/Merge ); précédent : 001280; suivant : 001282Combined Lymphedema and Capillary Malformation of the Lower Extremity
Auteurs : Reid A. Maclellan ; Gulraiz Chaudry ; Arin K. GreeneSource :
- Plastic and Reconstructive Surgery Global Open [ 2169-7574 ] ; 2016.
Abstract
Primary lymphedema and capillary malformation are independent vascular malformations that can cause overgrowth of the lower extremity. We report a series of patients who had both types of malformations affecting the same leg. The condition is unique but may be confused with other types of vascular malformation overgrowth conditions (eg, Klippel–Trenaunay and Parkes Weber).
Our Vascular Anomalies Center and Lymphedema Program databases were searched for patients with both capillary malformation and lymphedema. Diagnosis of lymphedema–capillary malformation was made by history, physical examination, and imaging studies. Because lymphedema–capillary malformation has phenotypical overlap with other conditions, only patients who had imaging confirming their diagnosis were included in the analysis. Clinical and radiological features, morbidity, and treatment were recorded.
Eight patients (4 females and 4 males) had confirmed lymphedema–capillary malformation. Referring diagnosis was Klippel–Trenaunay syndrome (n = 4), diffuse capillary malformation with overgrowth (n = 3), or lymphatic malformation (n = 1). The condition was unilateral (n = 6) or bilateral (n = 2). Morbidity included infection (n = 6), difficulty fitting clothes (n = 6), bleeding or leaking vesicles (n = 5), leg length discrepancy (n = 4), and difficulty ambulating (n = 3). All patients were managed with compression regimens. Operative management was liposuction (n = 3), treatment of phlebectatic veins (n = 3), staged skin/subcutaneous excision (n = 1), and/or epiphysiodesis (n = 1).
Lymphedema and capillary malformation can occur together in the same extremity. Both conditions independently cause limb overgrowth primarily because of subcutaneous adipose deposition. Compression garments and suction-assisted lipectomy can improve the condition. Lymphedema–capillary malformation should not be confused with other vascular malformation overgrowth diseases that have different morbidities and treatments.
Url:
DOI: 10.1097/GOX.0000000000000487
PubMed: 27014547
PubMed Central: 4778889
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Combined Lymphedema and Capillary Malformation of the Lower Extremity</title><author><name sortKey="Maclellan, Reid A" sort="Maclellan, Reid A" uniqKey="Maclellan R" first="Reid A." last="Maclellan">Reid A. Maclellan</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Chaudry, Gulraiz" sort="Chaudry, Gulraiz" uniqKey="Chaudry G" first="Gulraiz" last="Chaudry">Gulraiz Chaudry</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Greene, Arin K" sort="Greene, Arin K" uniqKey="Greene A" first="Arin K." last="Greene">Arin K. Greene</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">27014547</idno><idno type="pmc">4778889</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4778889</idno><idno type="RBID">PMC:4778889</idno><idno type="doi">10.1097/GOX.0000000000000487</idno><date when="2016">2016</date><idno type="wicri:Area/Pmc/Corpus">000847</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">000847</idno><idno type="wicri:Area/Pmc/Curation">000847</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Curation">000847</idno><idno type="wicri:Area/Pmc/Checkpoint">000B09</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Checkpoint">000B09</idno><idno type="wicri:source">PubMed</idno><idno type="wicri:Area/PubMed/Corpus">000914</idno><idno type="wicri:explorRef" wicri:stream="PubMed" wicri:step="Corpus" wicri:corpus="PubMed">000914</idno><idno type="wicri:Area/PubMed/Curation">000914</idno><idno type="wicri:explorRef" wicri:stream="PubMed" wicri:step="Curation">000914</idno><idno type="wicri:Area/PubMed/Checkpoint">000914</idno><idno type="wicri:explorRef" wicri:stream="Checkpoint" wicri:step="PubMed">000914</idno><idno type="wicri:Area/Ncbi/Merge">008091</idno><idno type="wicri:Area/Ncbi/Curation">008091</idno><idno type="wicri:Area/Ncbi/Checkpoint">008091</idno><idno type="wicri:Area/Main/Merge">001281</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Combined Lymphedema and Capillary Malformation of the Lower Extremity</title><author><name sortKey="Maclellan, Reid A" sort="Maclellan, Reid A" uniqKey="Maclellan R" first="Reid A." last="Maclellan">Reid A. Maclellan</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Chaudry, Gulraiz" sort="Chaudry, Gulraiz" uniqKey="Chaudry G" first="Gulraiz" last="Chaudry">Gulraiz Chaudry</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Greene, Arin K" sort="Greene, Arin K" uniqKey="Greene A" first="Arin K." last="Greene">Arin K. Greene</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author></analytic><series><title level="j">Plastic and Reconstructive Surgery Global Open</title><idno type="eISSN">2169-7574</idno><imprint><date when="2016">2016</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><title>Background:</title><p>Primary lymphedema and capillary malformation are independent vascular malformations that can cause overgrowth of the lower extremity. We report a series of patients who had both types of malformations affecting the same leg. The condition is unique but may be confused with other types of vascular malformation overgrowth conditions (eg, Klippel–Trenaunay and Parkes Weber).</p><sec><title>Methods:</title><p>Our Vascular Anomalies Center and Lymphedema Program databases were searched for patients with both capillary malformation and lymphedema. Diagnosis of lymphedema–capillary malformation was made by history, physical examination, and imaging studies. Because lymphedema–capillary malformation has phenotypical overlap with other conditions, only patients who had imaging confirming their diagnosis were included in the analysis. Clinical and radiological features, morbidity, and treatment were recorded.</p></sec><sec><title>Results:</title><p>Eight patients (4 females and 4 males) had confirmed lymphedema–capillary malformation. Referring diagnosis was Klippel–Trenaunay syndrome (n = 4), diffuse capillary malformation with overgrowth (n = 3), or lymphatic malformation (n = 1). The condition was unilateral (n = 6) or bilateral (n = 2). Morbidity included infection (n = 6), difficulty fitting clothes (n = 6), bleeding or leaking vesicles (n = 5), leg length discrepancy (n = 4), and difficulty ambulating (n = 3). All patients were managed with compression regimens. Operative management was liposuction (n = 3), treatment of phlebectatic veins (n = 3), staged skin/subcutaneous excision (n = 1), and/or epiphysiodesis (n = 1).</p></sec><sec><title>Conclusions:</title><p>Lymphedema and capillary malformation can occur together in the same extremity. Both conditions independently cause limb overgrowth primarily because of subcutaneous adipose deposition. Compression garments and suction-assisted lipectomy can improve the condition. Lymphedema–capillary malformation should not be confused with other vascular malformation overgrowth diseases that have different morbidities and treatments.</p></sec></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Greene, Ak" uniqKey="Greene A">AK Greene</name></author><author><name sortKey="Liu, As" uniqKey="Liu A">AS Liu</name></author><author><name sortKey="Mulliken, Jb" uniqKey="Mulliken J">JB Mulliken</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Hassanein, Ah" uniqKey="Hassanein A">AH Hassanein</name></author><author><name sortKey="Mulliken, Jb" uniqKey="Mulliken J">JB Mulliken</name></author><author><name sortKey="Fishman, Sj" uniqKey="Fishman S">SJ Fishman</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Schook, Cc" uniqKey="Schook C">CC Schook</name></author><author><name sortKey="Mulliken, Jb" uniqKey="Mulliken J">JB 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