Severe but reversible pulmonary hypertension in scleromyxedema and multiple myeloma: a case report
Identifieur interne : 000053 ( Main/Exploration ); précédent : 000052; suivant : 000054Severe but reversible pulmonary hypertension in scleromyxedema and multiple myeloma: a case report
Auteurs : Mazen Kreidy [États-Unis] ; Ali Al-Hilli [États-Unis] ; Ralph Yachoui [États-Unis] ; Jeffrey Resnick [États-Unis]Source :
- BMC Pulmonary Medicine [ 1471-2466 ] ; 2020.
Abstract
Scleromyxedema is a progressive, systemic connective tissue disorder characterized by fibro-mucous skin lesions and increased serum monoclonal immunoglobulin levels. Pulmonary involvement occurs in a subset of patients, though the overall prevalence of pulmonary lesions in scleromyxedema is unknown. Since pulmonary hypertension presumably occurs in these patients due to disease progression and development of additional conditions, treatment of the underlying plasma cell dyscrasia and connective tissue disorder may improve pulmonary hypertension symptoms.
An elderly patient with scleromyxedema developed pulmonary hypertension refractory to vasodilator and diuretic therapy and subsequently multiple myeloma that responded to a combination therapy of bortezomib, cyclophosphamide, and dexamethasone treatment.
Treatment of the underlying disease(s) that contributed to pulmonary hypertension development with anti-neoplastic agents like bortezomib may improve cardiopulmonary symptoms secondary to reducing abnormal blood cell counts and paraprotein levels.
Url:
DOI: 10.1186/s12890-019-1020-6
PubMed: 31918690
PubMed Central: 6953266
Affiliations:
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Le document en format XML
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<front><div type="abstract" xml:lang="en"><sec><title>Background</title>
<p id="Par1">Scleromyxedema is a progressive, systemic connective tissue disorder characterized by fibro-mucous skin lesions and increased serum monoclonal immunoglobulin levels. Pulmonary involvement occurs in a subset of patients, though the overall prevalence of pulmonary lesions in scleromyxedema is unknown. Since pulmonary hypertension presumably occurs in these patients due to disease progression and development of additional conditions, treatment of the underlying plasma cell dyscrasia and connective tissue disorder may improve pulmonary hypertension symptoms.</p>
</sec>
<sec><title>Case presentation</title>
<p id="Par2">An elderly patient with scleromyxedema developed pulmonary hypertension refractory to vasodilator and diuretic therapy and subsequently multiple myeloma that responded to a combination therapy of bortezomib, cyclophosphamide, and dexamethasone treatment.</p>
</sec>
<sec><title>Conclusions</title>
<p id="Par3">Treatment of the underlying disease(s) that contributed to pulmonary hypertension development with anti-neoplastic agents like bortezomib may improve cardiopulmonary symptoms secondary to reducing abnormal blood cell counts and paraprotein levels.</p>
</sec>
</div>
</front>
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<affiliations><list><country><li>États-Unis</li>
</country>
<region><li>Californie</li>
<li>Delaware</li>
<li>Wisconsin</li>
</region>
</list>
<tree><country name="États-Unis"><region name="Wisconsin"><name sortKey="Kreidy, Mazen" sort="Kreidy, Mazen" uniqKey="Kreidy M" first="Mazen" last="Kreidy">Mazen Kreidy</name>
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<name sortKey="Al Hilli, Ali" sort="Al Hilli, Ali" uniqKey="Al Hilli A" first="Ali" last="Al-Hilli">Ali Al-Hilli</name>
<name sortKey="Kreidy, Mazen" sort="Kreidy, Mazen" uniqKey="Kreidy M" first="Mazen" last="Kreidy">Mazen Kreidy</name>
<name sortKey="Resnick, Jeffrey" sort="Resnick, Jeffrey" uniqKey="Resnick J" first="Jeffrey" last="Resnick">Jeffrey Resnick</name>
<name sortKey="Yachoui, Ralph" sort="Yachoui, Ralph" uniqKey="Yachoui R" first="Ralph" last="Yachoui">Ralph Yachoui</name>
</country>
</tree>
</affiliations>
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