Elephantiasic pretibial myxedema: insight into and a hypothesis regarding the pathogenesis of the extrathyroidal manifestations of Graves' disease.
Identifieur interne : 004471 ( PubMed/Checkpoint ); précédent : 004470; suivant : 004472Elephantiasic pretibial myxedema: insight into and a hypothesis regarding the pathogenesis of the extrathyroidal manifestations of Graves' disease.
Auteurs : B. Rapoport [États-Unis] ; R. Alsabeh ; D. Aftergood ; S M MclachlanSource :
- Thyroid : official journal of the American Thyroid Association [ 1050-7256 ] ; 2000.
Descripteurs français
- KwdFr :
- MESH :
- analyse : Antigènes CD, Récepteur TSH.
- immunologie : Anticorps monoclonaux, Récepteur TSH.
- métabolisme : Glycosaminoglycanes.
- étiologie : Dermatoses de la jambe, Myxoedème, Éléphantiasis.
- Adulte d'âge moyen, Femelle, Humains, Maladie de Basedow.
English descriptors
- KwdEn :
- Antibodies, Monoclonal (immunology), Antigens, CD (analysis), Elephantiasis (etiology), Female, Glycosaminoglycans (metabolism), Graves Disease (complications), Humans, Leg Dermatoses (etiology), Middle Aged, Myxedema (etiology), Receptors, Thyrotropin (analysis), Receptors, Thyrotropin (immunology).
- MESH :
- chemical , analysis : Antigens, CD, Receptors, Thyrotropin.
- chemical , immunology : Antibodies, Monoclonal, Receptors, Thyrotropin.
- complications : Graves Disease.
- etiology : Elephantiasis, Leg Dermatoses, Myxedema.
- chemical , metabolism : Glycosaminoglycans.
- Female, Humans, Middle Aged.
Abstract
The basis for the extrathyroidal manifestations of Graves' ophthalmopathy (GO) and dermopathy are not well understood. We describe immunohistochemical studies on the skin of a patient with an extreme, elephantiasic form of Graves' dermopathy that developed after periods of prolonged standing with dependent edema. Excision of part of the lesion with subsequent skin grafting from a normal donor site resulted in recurrence of the disease at the original site as well as in development of disease at the donor site. A murine monoclonal antibody reacted with the thyrotropin receptor (TSHR) or a cross-reacting protein in fibroblast-like cells in the patient's upper dermis and, surprisingly, with dermal cells from unaffected individuals. The patient's dermis containing lymphoid follicles comprising B cells and CD3+, CD4+ T cells, with few CD8+ T cells. CD21+ cells (most likely follicular dendritic cells) were also present in the dermis. Based on past and present observations, we raise an unifying hypothesis to explain the diverse extrathyroidal manifestations of Graves' disease and their apparent lack of association with TSHR autoantibodies. As opposed to the present concept that these phenomena relate to site-specific properties on preadipocytes or fibroblasts, we suggest that clinically evidence GO and dermopathy are primarily caused by local factors (particularly in the orbit) superimposed on a systemic, low-grade connective tissue inflammation.
DOI: 10.1089/10507250050137761
PubMed: 11014313
Affiliations:
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Aftergood</name></author><author><name sortKey="Mclachlan, S M" sort="Mclachlan, S M" uniqKey="Mclachlan S" first="S M" last="Mclachlan">S M Mclachlan</name></author></analytic><series><title level="j">Thyroid : official journal of the American Thyroid Association</title><idno type="ISSN">1050-7256</idno><imprint><date when="2000" type="published">2000</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Antibodies, Monoclonal (immunology)</term><term>Antigens, CD (analysis)</term><term>Elephantiasis (etiology)</term><term>Female</term><term>Glycosaminoglycans (metabolism)</term><term>Graves Disease (complications)</term><term>Humans</term><term>Leg Dermatoses (etiology)</term><term>Middle Aged</term><term>Myxedema (etiology)</term><term>Receptors, Thyrotropin (analysis)</term><term>Receptors, Thyrotropin (immunology)</term></keywords><keywords scheme="KwdFr" xml:lang="fr"><term>Adulte d'âge moyen</term><term>Anticorps monoclonaux (immunologie)</term><term>Antigènes CD (analyse)</term><term>Dermatoses de la jambe (étiologie)</term><term>Femelle</term><term>Glycosaminoglycanes (métabolisme)</term><term>Humains</term><term>Maladie de Basedow ()</term><term>Myxoedème (étiologie)</term><term>Récepteur TSH (analyse)</term><term>Récepteur TSH (immunologie)</term><term>Éléphantiasis (étiologie)</term></keywords><keywords scheme="MESH" type="chemical" qualifier="analysis" xml:lang="en"><term>Antigens, CD</term><term>Receptors, Thyrotropin</term></keywords><keywords scheme="MESH" type="chemical" qualifier="immunology" xml:lang="en"><term>Antibodies, Monoclonal</term><term>Receptors, Thyrotropin</term></keywords><keywords scheme="MESH" qualifier="analyse" xml:lang="fr"><term>Antigènes CD</term><term>Récepteur TSH</term></keywords><keywords scheme="MESH" qualifier="complications" xml:lang="en"><term>Graves Disease</term></keywords><keywords scheme="MESH" qualifier="etiology" xml:lang="en"><term>Elephantiasis</term><term>Leg Dermatoses</term><term>Myxedema</term></keywords><keywords scheme="MESH" qualifier="immunologie" xml:lang="fr"><term>Anticorps monoclonaux</term><term>Récepteur TSH</term></keywords><keywords scheme="MESH" type="chemical" qualifier="metabolism" xml:lang="en"><term>Glycosaminoglycans</term></keywords><keywords scheme="MESH" qualifier="métabolisme" xml:lang="fr"><term>Glycosaminoglycanes</term></keywords><keywords scheme="MESH" qualifier="étiologie" xml:lang="fr"><term>Dermatoses de la jambe</term><term>Myxoedème</term><term>Éléphantiasis</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Female</term><term>Humans</term><term>Middle Aged</term></keywords><keywords scheme="MESH" xml:lang="fr"><term>Adulte d'âge moyen</term><term>Femelle</term><term>Humains</term><term>Maladie de Basedow</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">The basis for the extrathyroidal manifestations of Graves' ophthalmopathy (GO) and dermopathy are not well understood. We describe immunohistochemical studies on the skin of a patient with an extreme, elephantiasic form of Graves' dermopathy that developed after periods of prolonged standing with dependent edema. Excision of part of the lesion with subsequent skin grafting from a normal donor site resulted in recurrence of the disease at the original site as well as in development of disease at the donor site. A murine monoclonal antibody reacted with the thyrotropin receptor (TSHR) or a cross-reacting protein in fibroblast-like cells in the patient's upper dermis and, surprisingly, with dermal cells from unaffected individuals. The patient's dermis containing lymphoid follicles comprising B cells and CD3+, CD4+ T cells, with few CD8+ T cells. CD21+ cells (most likely follicular dendritic cells) were also present in the dermis. Based on past and present observations, we raise an unifying hypothesis to explain the diverse extrathyroidal manifestations of Graves' disease and their apparent lack of association with TSHR autoantibodies. As opposed to the present concept that these phenomena relate to site-specific properties on preadipocytes or fibroblasts, we suggest that clinically evidence GO and dermopathy are primarily caused by local factors (particularly in the orbit) superimposed on a systemic, low-grade connective tissue inflammation.</div></front></TEI><pubmed><MedlineCitation Status="MEDLINE" Owner="NLM"><PMID Version="1">11014313</PMID><DateCreated><Year>2001</Year><Month>01</Month><Day>11</Day></DateCreated><DateCompleted><Year>2001</Year><Month>01</Month><Day>11</Day></DateCompleted><DateRevised><Year>2007</Year><Month>11</Month><Day>14</Day></DateRevised><Article PubModel="Print"><Journal><ISSN IssnType="Print">1050-7256</ISSN><JournalIssue CitedMedium="Print"><Volume>10</Volume><Issue>8</Issue><PubDate><Year>2000</Year><Month>Aug</Month></PubDate></JournalIssue><Title>Thyroid : official journal of the American Thyroid Association</Title><ISOAbbreviation>Thyroid</ISOAbbreviation></Journal><ArticleTitle>Elephantiasic pretibial myxedema: insight into and a hypothesis regarding the pathogenesis of the extrathyroidal manifestations of Graves' disease.</ArticleTitle><Pagination><MedlinePgn>685-92</MedlinePgn></Pagination><Abstract><AbstractText>The basis for the extrathyroidal manifestations of Graves' ophthalmopathy (GO) and dermopathy are not well understood. We describe immunohistochemical studies on the skin of a patient with an extreme, elephantiasic form of Graves' dermopathy that developed after periods of prolonged standing with dependent edema. Excision of part of the lesion with subsequent skin grafting from a normal donor site resulted in recurrence of the disease at the original site as well as in development of disease at the donor site. A murine monoclonal antibody reacted with the thyrotropin receptor (TSHR) or a cross-reacting protein in fibroblast-like cells in the patient's upper dermis and, surprisingly, with dermal cells from unaffected individuals. The patient's dermis containing lymphoid follicles comprising B cells and CD3+, CD4+ T cells, with few CD8+ T cells. CD21+ cells (most likely follicular dendritic cells) were also present in the dermis. Based on past and present observations, we raise an unifying hypothesis to explain the diverse extrathyroidal manifestations of Graves' disease and their apparent lack of association with TSHR autoantibodies. As opposed to the present concept that these phenomena relate to site-specific properties on preadipocytes or fibroblasts, we suggest that clinically evidence GO and dermopathy are primarily caused by local factors (particularly in the orbit) superimposed on a systemic, low-grade connective tissue inflammation.</AbstractText></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Rapoport</LastName><ForeName>B</ForeName><Initials>B</Initials><AffiliationInfo><Affiliation>Autoimmune Disease Unit, Cedars-Sinai Research Institute and School of Medicine, University of California, Los Angeles, USA. rapoporb@cshs.org</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Alsabeh</LastName><ForeName>R</ForeName><Initials>R</Initials></Author><Author ValidYN="Y"><LastName>Aftergood</LastName><ForeName>D</ForeName><Initials>D</Initials></Author><Author ValidYN="Y"><LastName>McLachlan</LastName><ForeName>S M</ForeName><Initials>SM</Initials></Author></AuthorList><Language>eng</Language><GrantList CompleteYN="Y"><Grant><GrantID>DK19289</GrantID><Acronym>DK</Acronym><Agency>NIDDK NIH HHS</Agency><Country>United States</Country></Grant></GrantList><PublicationTypeList><PublicationType UI="D002363">Case Reports</PublicationType><PublicationType UI="D016428">Journal Article</PublicationType><PublicationType UI="D013487">Research Support, U.S. Gov't, P.H.S.</PublicationType></PublicationTypeList></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>Thyroid</MedlineTA><NlmUniqueID>9104317</NlmUniqueID><ISSNLinking>1050-7256</ISSNLinking></MedlineJournalInfo><ChemicalList><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D000911">Antibodies, Monoclonal</NameOfSubstance></Chemical><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D015703">Antigens, CD</NameOfSubstance></Chemical><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance 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IdType="doi">10.1089/10507250050137761</ArticleId></ArticleIdList></PubmedData></pubmed><affiliations><list><country><li>États-Unis</li></country><region><li>Californie</li></region><settlement><li>Los Angeles</li></settlement></list><tree><noCountry><name sortKey="Aftergood, D" sort="Aftergood, D" uniqKey="Aftergood D" first="D" last="Aftergood">D. Aftergood</name><name sortKey="Alsabeh, R" sort="Alsabeh, R" uniqKey="Alsabeh R" first="R" last="Alsabeh">R. Alsabeh</name><name sortKey="Mclachlan, S M" sort="Mclachlan, S M" uniqKey="Mclachlan S" first="S M" last="Mclachlan">S M Mclachlan</name></noCountry><country name="États-Unis"><region name="Californie"><name sortKey="Rapoport, B" sort="Rapoport, B" uniqKey="Rapoport B" first="B" last="Rapoport">B. Rapoport</name></region></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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