Chronic alveolar haemorrhage in a paediatric patient: a diagnostic and treatment challenge.
Identifieur interne : 000E96 ( Main/Exploration ); précédent : 000E95; suivant : 000E97Chronic alveolar haemorrhage in a paediatric patient: a diagnostic and treatment challenge.
Auteurs : Ana Freitas [Portugal] ; Virgílio Senra [Portugal] ; Ant Nio Marinho [Portugal] ; Margarida Guedes [Portugal]Source :
- BMJ case reports [ 1757-790X ] ; 2015.
Descripteurs français
- KwdFr :
- Adolescent, Anticorps anticytoplasme des polynucléaires neutrophiles (analyse), Azathioprine (usage thérapeutique), Biopsie, Cyclophosphamide (usage thérapeutique), Femelle, Hormones corticosurrénaliennes (usage thérapeutique), Humains, Hydroxychloroquine (usage thérapeutique), Hémoptysie (anatomopathologie), Hémoptysie (étiologie), Hémorragie (diagnostic), Hémorragie (traitement médicamenteux), Hémorragie (étiologie), Hémosidérose (anatomopathologie), Hémosidérose (traitement médicamenteux), Maladies pulmonaires (anatomopathologie), Maladies pulmonaires (traitement médicamenteux), Myeloperoxidase (métabolisme), Rituximab (administration et posologie), Rituximab (usage thérapeutique), Vascularite (diagnostic), Vascularite (traitement médicamenteux), Vascularite (étiologie).
- MESH :
- administration et posologie : Rituximab.
- analyse : Anticorps anticytoplasme des polynucléaires neutrophiles.
- anatomopathologie : Hémoptysie, Hémosidérose, Maladies pulmonaires.
- diagnostic : Hémorragie, Vascularite.
- métabolisme : Myeloperoxidase.
- traitement médicamenteux : Hémorragie, Hémosidérose, Maladies pulmonaires, Vascularite.
- usage thérapeutique : Azathioprine, Cyclophosphamide, Hormones corticosurrénaliennes, Hydroxychloroquine, Rituximab.
- étiologie : Hémoptysie, Hémorragie, Vascularite.
- Adolescent, Biopsie, Femelle, Humains.
English descriptors
- KwdEn :
- Adolescent, Adrenal Cortex Hormones (therapeutic use), Antibodies, Antineutrophil Cytoplasmic (analysis), Azathioprine (therapeutic use), Biopsy, Cyclophosphamide (therapeutic use), Female, Hemoptysis (etiology), Hemoptysis (pathology), Hemorrhage (diagnosis), Hemorrhage (drug therapy), Hemorrhage (etiology), Hemosiderosis (drug therapy), Hemosiderosis (pathology), Humans, Hydroxychloroquine (therapeutic use), Lung Diseases (drug therapy), Lung Diseases (pathology), Peroxidase (metabolism), Rituximab (administration & dosage), Rituximab (therapeutic use), Vasculitis (diagnosis), Vasculitis (drug therapy), Vasculitis (etiology).
- MESH :
- chemical , administration & dosage : Rituximab.
- chemical , analysis : Antibodies, Antineutrophil Cytoplasmic.
- chemical , metabolism : Peroxidase.
- chemical , therapeutic use : Adrenal Cortex Hormones, Azathioprine, Cyclophosphamide, Hydroxychloroquine, Rituximab.
- diagnosis : Hemorrhage, Vasculitis.
- drug therapy : Hemorrhage, Hemosiderosis, Lung Diseases, Vasculitis.
- etiology : Hemoptysis, Hemorrhage, Vasculitis.
- pathology : Hemoptysis, Hemosiderosis, Lung Diseases.
- Adolescent, Biopsy, Female, Humans.
Abstract
Pulmonary haemosiderosis is characterised by chronic alveolar haemorrhage, which can lead to serious cardiorespiratory complications. Although considered idiopathic in most patients, there are many possible aetiologies. We present a case of an 18-year-old woman with pulmonary haemosiderosis since 4 years of age, with an inconclusive initial study, who was treated with systemic corticosteroids and hydroxychloroquine until the age of 12 years, and azathioprine since then. Multiple exacerbations led to interstitial lung disease with restrictive functional pattern. Unilateral cochlear deafness was diagnosed at the age of 12 years and occasional polyarthralgias were recorded. When she was 16 years of age the study revealed an atypical myeloperoxidase-antineutrophil cytoplasmic antibody (MPO-ANCA) pattern. Cyclophosphamide and rituximab were administered with resolution of respiratory insufficiency and functional disability, without new episodes of alveolar haemorrhage. This case of chronic pulmonary haemorrhage was revealed to be an ANCA vasculitis, the diagnosis of which was possible only after 12 years of symptoms, with clinical and functional improvement with the association of cyclophosphamide and rituximab.
DOI: 10.1136/bcr-2014-206856
PubMed: 25899510
Affiliations:
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Diseases (drug therapy)</term><term>Lung Diseases (pathology)</term><term>Peroxidase (metabolism)</term><term>Rituximab (administration & dosage)</term><term>Rituximab (therapeutic use)</term><term>Vasculitis (diagnosis)</term><term>Vasculitis (drug therapy)</term><term>Vasculitis (etiology)</term></keywords><keywords scheme="KwdFr" xml:lang="fr"><term>Adolescent</term><term>Anticorps anticytoplasme des polynucléaires neutrophiles (analyse)</term><term>Azathioprine (usage thérapeutique)</term><term>Biopsie</term><term>Cyclophosphamide (usage thérapeutique)</term><term>Femelle</term><term>Hormones corticosurrénaliennes (usage thérapeutique)</term><term>Humains</term><term>Hydroxychloroquine (usage thérapeutique)</term><term>Hémoptysie (anatomopathologie)</term><term>Hémoptysie (étiologie)</term><term>Hémorragie (diagnostic)</term><term>Hémorragie (traitement médicamenteux)</term><term>Hémorragie (étiologie)</term><term>Hémosidérose (anatomopathologie)</term><term>Hémosidérose 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xml:lang="en"><term>Hemoptysis</term><term>Hemorrhage</term><term>Vasculitis</term></keywords><keywords scheme="MESH" qualifier="métabolisme" xml:lang="fr"><term>Myeloperoxidase</term></keywords><keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>Hemoptysis</term><term>Hemosiderosis</term><term>Lung Diseases</term></keywords><keywords scheme="MESH" qualifier="traitement médicamenteux" xml:lang="fr"><term>Hémorragie</term><term>Hémosidérose</term><term>Maladies pulmonaires</term><term>Vascularite</term></keywords><keywords scheme="MESH" qualifier="usage thérapeutique" xml:lang="fr"><term>Azathioprine</term><term>Cyclophosphamide</term><term>Hormones corticosurrénaliennes</term><term>Hydroxychloroquine</term><term>Rituximab</term></keywords><keywords scheme="MESH" qualifier="étiologie" xml:lang="fr"><term>Hémoptysie</term><term>Hémorragie</term><term>Vascularite</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adolescent</term><term>Biopsy</term><term>Female</term><term>Humans</term></keywords><keywords scheme="MESH" xml:lang="fr"><term>Adolescent</term><term>Biopsie</term><term>Femelle</term><term>Humains</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Pulmonary haemosiderosis is characterised by chronic alveolar haemorrhage, which can lead to serious cardiorespiratory complications. Although considered idiopathic in most patients, there are many possible aetiologies. We present a case of an 18-year-old woman with pulmonary haemosiderosis since 4 years of age, with an inconclusive initial study, who was treated with systemic corticosteroids and hydroxychloroquine until the age of 12 years, and azathioprine since then. Multiple exacerbations led to interstitial lung disease with restrictive functional pattern. Unilateral cochlear deafness was diagnosed at the age of 12 years and occasional polyarthralgias were recorded. When she was 16 years of age the study revealed an atypical myeloperoxidase-antineutrophil cytoplasmic antibody (MPO-ANCA) pattern. Cyclophosphamide and rituximab were administered with resolution of respiratory insufficiency and functional disability, without new episodes of alveolar haemorrhage. This case of chronic pulmonary haemorrhage was revealed to be an ANCA vasculitis, the diagnosis of which was possible only after 12 years of symptoms, with clinical and functional improvement with the association of cyclophosphamide and rituximab.</div></front></TEI><affiliations><list><country><li>Portugal</li></country></list><tree><country name="Portugal"><noRegion><name sortKey="Freitas, Ana" sort="Freitas, Ana" uniqKey="Freitas A" first="Ana" last="Freitas">Ana Freitas</name></noRegion><name sortKey="Guedes, Margarida" sort="Guedes, Margarida" uniqKey="Guedes M" first="Margarida" last="Guedes">Margarida Guedes</name><name sortKey="Marinho, Ant Nio" sort="Marinho, Ant Nio" uniqKey="Marinho A" first="Ant Nio" last="Marinho">Ant Nio Marinho</name><name sortKey="Senra, Virgilio" sort="Senra, Virgilio" uniqKey="Senra V" first="Virgílio" last="Senra">Virgílio Senra</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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