Chronic alveolar haemorrhage in a paediatric patient: a diagnostic and treatment challenge.
Identifieur interne : 000258 ( PubMed/Curation ); précédent : 000257; suivant : 000259Chronic 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
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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><pubmed><MedlineCitation Status="MEDLINE" Owner="NLM"><PMID Version="1">25899510</PMID><DateCompleted><Year>2016</Year><Month>01</Month><Day>05</Day></DateCompleted><DateRevised><Year>2019</Year><Month>02</Month><Day>01</Day></DateRevised><Article PubModel="Electronic"><Journal><ISSN IssnType="Electronic">1757-790X</ISSN><JournalIssue CitedMedium="Internet"><Volume>2015</Volume><PubDate><Year>2015</Year><Month>Apr</Month><Day>21</Day></PubDate></JournalIssue><Title>BMJ case reports</Title><ISOAbbreviation>BMJ Case Rep</ISOAbbreviation></Journal><ArticleTitle>Chronic alveolar haemorrhage in a paediatric patient: a diagnostic and treatment challenge.</ArticleTitle><ELocationID EIdType="doi" ValidYN="Y">10.1136/bcr-2014-206856</ELocationID><ELocationID EIdType="pii" ValidYN="Y">bcr2014206856</ELocationID><Abstract><AbstractText>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.</AbstractText><CopyrightInformation>2015 BMJ Publishing Group Ltd.</CopyrightInformation></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Freitas</LastName><ForeName>Ana</ForeName><Initials>A</Initials><AffiliationInfo><Affiliation>Department of Pediatrics, Centro Hospitalar do Porto, Porto, Portugal.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Senra</LastName><ForeName>Virgílio</ForeName><Initials>V</Initials><AffiliationInfo><Affiliation>Department of Pediatrics, Centro Hospitalar do Porto, Porto, Portugal.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Marinho</LastName><ForeName>António</ForeName><Initials>A</Initials><AffiliationInfo><Affiliation>Department of Medicine, Centro Hospitalar do Porto, Porto, Portugal.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Guedes</LastName><ForeName>Margarida</ForeName><Initials>M</Initials><AffiliationInfo><Affiliation>Department of Pediatrics, Centro Hospitalar do Porto, Porto, Portugal.</Affiliation></AffiliationInfo></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType UI="D002363">Case Reports</PublicationType><PublicationType UI="D016428">Journal Article</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2015</Year><Month>04</Month><Day>21</Day></ArticleDate></Article><MedlineJournalInfo><Country>England</Country><MedlineTA>BMJ Case Rep</MedlineTA><NlmUniqueID>101526291</NlmUniqueID><ISSNLinking>1757-790X</ISSNLinking></MedlineJournalInfo><ChemicalList><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D000305">Adrenal Cortex Hormones</NameOfSubstance></Chemical><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D019268">Antibodies, Antineutrophil Cytoplasmic</NameOfSubstance></Chemical><Chemical><RegistryNumber>4F4X42SYQ6</RegistryNumber><NameOfSubstance UI="D000069283">Rituximab</NameOfSubstance></Chemical><Chemical><RegistryNumber>4QWG6N8QKH</RegistryNumber><NameOfSubstance UI="D006886">Hydroxychloroquine</NameOfSubstance></Chemical><Chemical><RegistryNumber>8N3DW7272P</RegistryNumber><NameOfSubstance UI="D003520">Cyclophosphamide</NameOfSubstance></Chemical><Chemical><RegistryNumber>EC 1.11.1.7</RegistryNumber><NameOfSubstance UI="D009195">Peroxidase</NameOfSubstance></Chemical><Chemical><RegistryNumber>MRK240IY2L</RegistryNumber><NameOfSubstance UI="D001379">Azathioprine</NameOfSubstance></Chemical></ChemicalList><SupplMeshList><SupplMeshName Type="Disease" UI="C536281">Idiopathic pulmonary hemosiderosis</SupplMeshName></SupplMeshList><CitationSubset>IM</CitationSubset><MeshHeadingList><MeshHeading><DescriptorName UI="D000293" MajorTopicYN="N">Adolescent</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D000305" MajorTopicYN="N">Adrenal Cortex Hormones</DescriptorName><QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D019268" MajorTopicYN="N">Antibodies, Antineutrophil Cytoplasmic</DescriptorName><QualifierName UI="Q000032" MajorTopicYN="N">analysis</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D001379" MajorTopicYN="N">Azathioprine</DescriptorName><QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D001706" MajorTopicYN="N">Biopsy</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D003520" MajorTopicYN="N">Cyclophosphamide</DescriptorName><QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D005260" MajorTopicYN="N">Female</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D006469" MajorTopicYN="N">Hemoptysis</DescriptorName><QualifierName UI="Q000209" MajorTopicYN="N">etiology</QualifierName><QualifierName UI="Q000473" MajorTopicYN="N">pathology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D006470" MajorTopicYN="N">Hemorrhage</DescriptorName><QualifierName UI="Q000175" MajorTopicYN="N">diagnosis</QualifierName><QualifierName UI="Q000188" MajorTopicYN="N">drug therapy</QualifierName><QualifierName UI="Q000209" MajorTopicYN="Y">etiology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D006486" MajorTopicYN="N">Hemosiderosis</DescriptorName><QualifierName UI="Q000188" MajorTopicYN="N">drug therapy</QualifierName><QualifierName UI="Q000473" MajorTopicYN="Y">pathology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D006801" MajorTopicYN="N">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName UI="D006886" MajorTopicYN="N">Hydroxychloroquine</DescriptorName><QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D008171" MajorTopicYN="N">Lung Diseases</DescriptorName><QualifierName UI="Q000188" MajorTopicYN="N">drug therapy</QualifierName><QualifierName UI="Q000473" MajorTopicYN="Y">pathology</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D009195" MajorTopicYN="N">Peroxidase</DescriptorName><QualifierName UI="Q000378" MajorTopicYN="N">metabolism</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D000069283" MajorTopicYN="N">Rituximab</DescriptorName><QualifierName UI="Q000008" MajorTopicYN="N">administration & dosage</QualifierName><QualifierName UI="Q000627" MajorTopicYN="N">therapeutic use</QualifierName></MeshHeading><MeshHeading><DescriptorName UI="D014657" 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