ChloroquineV1, PascalFrancis, Curation, bibRecord, 000006

Non-X-linked hyperimmunoglobulin M syndrome with chronic interstitial pneumonitis

Identifieur interne : 000006 ( PascalFrancis/Curation ); précédent : 000005; suivant : 000007

Non-X-linked hyperimmunoglobulin M syndrome with chronic interstitial pneumonitis

Auteurs : A. Quezada [Chili] ; X. Norambuena [Chili] ; R. Gonzalez [Chili] ; S.-M. Ramos [Chili] ; A. Chavez [Chili] ; S. Gonzalez [Chili]

Source :

Journal of investigational allergology & clinical immunology [ 1018-9068 ] ; 2000.

RBID : Pascal:01-0119408

Descripteurs français

Pascal (Inist)
- Immunoglobulinémie, IgM, Immunoglobulinémie monoclonale, Enfant, Femelle, Etude cas, Pneumopathie interstitielle, Complication, Forme clinique, Exploration immunologique, Traitement, Chloroquine, Chimiothérapie, Immunothérapie, Immunoglobuline, Ligand CD40, Hyperimmunoglobulinémie M syndrome.
Wicri :
- topic : Enfant.

English descriptors

KwdEn :
- CD40 ligand, Case study, Chemotherapy, Child, Chloroquine, Clinical form, Complication, Female, Hyperimmunoglobulinemia M syndrome, IgM, Immunoglobulinemia, Immunoglobulins, Immunological investigation, Immunotherapy, Interstitial pneumonitis, Monoclonal immunoglobulinemia, Treatment.

Abstract

Hyperimmunoglobulin M (IgM) syndrome is a rare primary immunodeficiency disorder, and the non-X-linked form of this syndrome is even more infrequent. We report the clinical case of a 6-year-old girl. Her disease began at the age of 1 year when she experienced bronchial obstruction. When she was 3 years old she developed severe recurrent respiratory infections of unusual clinical course. Serum IgM was elevated and the other serum immunoglobulins were absent. Cellular immune response was impaired, with severe depression of delayed hypersensitive cutaneous response and of proliferative response to mitogens. The CD40 ligand expression decreased. Chest CT scan showed areas of lung condensation, bronchial dilatation and signs suggesting interstitial pneumonitis. The latter was confirmed by a biopsy showing a high number of Langerhans' cells and an early-stage fibrosis. She was treated with antibiotics, inhaled bronchodilators and corticosteroids, intravenous immunoglobulin, chloroquine and prednisone. Despite the substitution therapy, her clinical course was slow, with respiratory infections and oxygen dependance. The follow-up thoracoscopic biopsy performed after 18 months of immunosuppressant therapy showed a progression of fibrosis and a decrease in the inflammatory infiltrate.

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A11	`02`	`1`		`@1 NORAMBUENA (X.)`
A11	`03`	`1`		`@1 GONZALEZ (R.)`
A11	`04`	`1`		`@1 RAMOS (S.-M.)`
A11	`05`	`1`		`@1 CHAVEZ (A.)`
A11	`06`	`1`		`@1 GONZALEZ (S.)`
A14	`01`			`@1 Department of Pediatrics, School of Medicine, University of Chile @3 CHL @Z 1 aut. @Z 4 aut.`
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C01	`01`		`ENG`	@0 Hyperimmunoglobulin M (IgM) syndrome is a rare primary immunodeficiency disorder, and the non-X-linked form of this syndrome is even more infrequent. We report the clinical case of a 6-year-old girl. Her disease began at the age of 1 year when she experienced bronchial obstruction. When she was 3 years old she developed severe recurrent respiratory infections of unusual clinical course. Serum IgM was elevated and the other serum immunoglobulins were absent. Cellular immune response was impaired, with severe depression of delayed hypersensitive cutaneous response and of proliferative response to mitogens. The CD40 ligand expression decreased. Chest CT scan showed areas of lung condensation, bronchial dilatation and signs suggesting interstitial pneumonitis. The latter was confirmed by a biopsy showing a high number of Langerhans' cells and an early-stage fibrosis. She was treated with antibiotics, inhaled bronchodilators and corticosteroids, intravenous immunoglobulin, chloroquine and prednisone. Despite the substitution therapy, her clinical course was slow, with respiratory infections and oxygen dependance. The follow-up thoracoscopic biopsy performed after 18 months of immunosuppressant therapy showed a progression of fibrosis and a decrease in the inflammatory infiltrate.
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C03	`08`	`X`	`FRE`	`@0 Complication @5 08`
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C03	`12`	`X`	`FRE`	`@0 Chloroquine @2 NK @2 FR @5 13`
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C03	`12`	`X`	`SPA`	`@0 Cloroquina @2 NK @2 FR @5 13`
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C03	`15`	`X`	`FRE`	`@0 Immunoglobuline @5 19`
C03	`15`	`X`	`ENG`	`@0 Immunoglobulins @5 19`
C03	`15`	`X`	`SPA`	`@0 Inmunoglobulina @5 19`
C03	`16`	`X`	`FRE`	`@0 Ligand CD40 @4 CD @5 96`
C03	`16`	`X`	`ENG`	`@0 CD40 ligand @4 CD @5 96`
C03	`17`	`X`	`FRE`	`@0 Hyperimmunoglobulinémie M syndrome @4 CD @5 97`
C03	`17`	`X`	`ENG`	`@0 Hyperimmunoglobulinemia M syndrome @4 CD @5 97`
C07	`01`	`X`	`FRE`	`@0 Homme`
C07	`01`	`X`	`ENG`	`@0 Human`
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C07	`03`	`X`	`SPA`	`@0 Inmunodeficiencia @5 38`
C07	`04`	`X`	`FRE`	`@0 Appareil respiratoire pathologie @5 53`
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C07	`04`	`X`	`SPA`	`@0 Aparato respiratorio patología @5 53`
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C07	`05`	`X`	`ENG`	`@0 Lung disease @5 54`
C07	`05`	`X`	`SPA`	`@0 Pulmón patología @5 54`
N21				`@1 078`

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Pascal:01-0119408

Le document en format XML

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<front><div type="abstract" xml:lang="en">Hyperimmunoglobulin M (IgM) syndrome is a rare primary immunodeficiency disorder, and the non-X-linked form of this syndrome is even more infrequent. We report the clinical case of a 6-year-old girl. Her disease began at the age of 1 year when she experienced bronchial obstruction. When she was 3 years old she developed severe recurrent respiratory infections of unusual clinical course. Serum IgM was elevated and the other serum immunoglobulins were absent. Cellular immune response was impaired, with severe depression of delayed hypersensitive cutaneous response and of proliferative response to mitogens. The CD40 ligand expression decreased. Chest CT scan showed areas of lung condensation, bronchial dilatation and signs suggesting interstitial pneumonitis. The latter was confirmed by a biopsy showing a high number of Langerhans' cells and an early-stage fibrosis. She was treated with antibiotics, inhaled bronchodilators and corticosteroids, intravenous immunoglobulin, chloroquine and prednisone. Despite the substitution therapy, her clinical course was slow, with respiratory infections and oxygen dependance. The follow-up thoracoscopic biopsy performed after 18 months of immunosuppressant therapy showed a progression of fibrosis and a decrease in the inflammatory infiltrate.</div>
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<s5>02</s5>
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<fC03 i1="02" i2="X" l="ENG"><s0>IgM</s0>
<s5>02</s5>
</fC03>
<fC03 i1="02" i2="X" l="SPA"><s0>IgM</s0>
<s5>02</s5>
</fC03>
<fC03 i1="03" i2="X" l="FRE"><s0>Immunoglobulinémie monoclonale</s0>
<s5>03</s5>
</fC03>
<fC03 i1="03" i2="X" l="ENG"><s0>Monoclonal immunoglobulinemia</s0>
<s5>03</s5>
</fC03>
<fC03 i1="03" i2="X" l="SPA"><s0>Inmunoglobulinemia monoclonal</s0>
<s5>03</s5>
</fC03>
<fC03 i1="04" i2="X" l="FRE"><s0>Enfant</s0>
<s5>04</s5>
</fC03>
<fC03 i1="04" i2="X" l="ENG"><s0>Child</s0>
<s5>04</s5>
</fC03>
<fC03 i1="04" i2="X" l="SPA"><s0>Niño</s0>
<s5>04</s5>
</fC03>
<fC03 i1="05" i2="X" l="FRE"><s0>Femelle</s0>
<s5>05</s5>
</fC03>
<fC03 i1="05" i2="X" l="ENG"><s0>Female</s0>
<s5>05</s5>
</fC03>
<fC03 i1="05" i2="X" l="SPA"><s0>Hembra</s0>
<s5>05</s5>
</fC03>
<fC03 i1="06" i2="X" l="FRE"><s0>Etude cas</s0>
<s5>06</s5>
</fC03>
<fC03 i1="06" i2="X" l="ENG"><s0>Case study</s0>
<s5>06</s5>
</fC03>
<fC03 i1="06" i2="X" l="SPA"><s0>Estudio caso</s0>
<s5>06</s5>
</fC03>
<fC03 i1="07" i2="X" l="FRE"><s0>Pneumopathie interstitielle</s0>
<s5>07</s5>
</fC03>
<fC03 i1="07" i2="X" l="ENG"><s0>Interstitial pneumonitis</s0>
<s5>07</s5>
</fC03>
<fC03 i1="07" i2="X" l="SPA"><s0>Neumopatía intersticial</s0>
<s5>07</s5>
</fC03>
<fC03 i1="08" i2="X" l="FRE"><s0>Complication</s0>
<s5>08</s5>
</fC03>
<fC03 i1="08" i2="X" l="ENG"><s0>Complication</s0>
<s5>08</s5>
</fC03>
<fC03 i1="08" i2="X" l="SPA"><s0>Complicación</s0>
<s5>08</s5>
</fC03>
<fC03 i1="09" i2="X" l="FRE"><s0>Forme clinique</s0>
<s5>10</s5>
</fC03>
<fC03 i1="09" i2="X" l="ENG"><s0>Clinical form</s0>
<s5>10</s5>
</fC03>
<fC03 i1="09" i2="X" l="SPA"><s0>Forma clínica</s0>
<s5>10</s5>
</fC03>
<fC03 i1="10" i2="X" l="FRE"><s0>Exploration immunologique</s0>
<s5>11</s5>
</fC03>
<fC03 i1="10" i2="X" l="ENG"><s0>Immunological investigation</s0>
<s5>11</s5>
</fC03>
<fC03 i1="10" i2="X" l="SPA"><s0>Análisis inmunológico</s0>
<s5>11</s5>
</fC03>
<fC03 i1="11" i2="X" l="FRE"><s0>Traitement</s0>
<s5>12</s5>
</fC03>
<fC03 i1="11" i2="X" l="ENG"><s0>Treatment</s0>
<s5>12</s5>
</fC03>
<fC03 i1="11" i2="X" l="SPA"><s0>Tratamiento</s0>
<s5>12</s5>
</fC03>
<fC03 i1="12" i2="X" l="FRE"><s0>Chloroquine</s0>
<s2>NK</s2>
<s2>FR</s2>
<s5>13</s5>
</fC03>
<fC03 i1="12" i2="X" l="ENG"><s0>Chloroquine</s0>
<s2>NK</s2>
<s2>FR</s2>
<s5>13</s5>
</fC03>
<fC03 i1="12" i2="X" l="SPA"><s0>Cloroquina</s0>
<s2>NK</s2>
<s2>FR</s2>
<s5>13</s5>
</fC03>
<fC03 i1="13" i2="X" l="FRE"><s0>Chimiothérapie</s0>
<s5>17</s5>
</fC03>
<fC03 i1="13" i2="X" l="ENG"><s0>Chemotherapy</s0>
<s5>17</s5>
</fC03>
<fC03 i1="13" i2="X" l="SPA"><s0>Quimioterapia</s0>
<s5>17</s5>
</fC03>
<fC03 i1="14" i2="X" l="FRE"><s0>Immunothérapie</s0>
<s5>18</s5>
</fC03>
<fC03 i1="14" i2="X" l="ENG"><s0>Immunotherapy</s0>
<s5>18</s5>
</fC03>
<fC03 i1="14" i2="X" l="SPA"><s0>Inmunoterapia</s0>
<s5>18</s5>
</fC03>
<fC03 i1="15" i2="X" l="FRE"><s0>Immunoglobuline</s0>
<s5>19</s5>
</fC03>
<fC03 i1="15" i2="X" l="ENG"><s0>Immunoglobulins</s0>
<s5>19</s5>
</fC03>
<fC03 i1="15" i2="X" l="SPA"><s0>Inmunoglobulina</s0>
<s5>19</s5>
</fC03>
<fC03 i1="16" i2="X" l="FRE"><s0>Ligand CD40</s0>
<s4>CD</s4>
<s5>96</s5>
</fC03>
<fC03 i1="16" i2="X" l="ENG"><s0>CD40 ligand</s0>
<s4>CD</s4>
<s5>96</s5>
</fC03>
<fC03 i1="17" i2="X" l="FRE"><s0>Hyperimmunoglobulinémie M syndrome</s0>
<s4>CD</s4>
<s5>97</s5>
</fC03>
<fC03 i1="17" i2="X" l="ENG"><s0>Hyperimmunoglobulinemia M syndrome</s0>
<s4>CD</s4>
<s5>97</s5>
</fC03>
<fC07 i1="01" i2="X" l="FRE"><s0>Homme</s0>
</fC07>
<fC07 i1="01" i2="X" l="ENG"><s0>Human</s0>
</fC07>
<fC07 i1="01" i2="X" l="SPA"><s0>Hombre</s0>
</fC07>
<fC07 i1="02" i2="X" l="FRE"><s0>Immunopathologie</s0>
<s5>37</s5>
</fC07>
<fC07 i1="02" i2="X" l="ENG"><s0>Immunopathology</s0>
<s5>37</s5>
</fC07>
<fC07 i1="02" i2="X" l="SPA"><s0>Inmunopatología</s0>
<s5>37</s5>
</fC07>
<fC07 i1="03" i2="X" l="FRE"><s0>Immunodéficit</s0>
<s5>38</s5>
</fC07>
<fC07 i1="03" i2="X" l="ENG"><s0>Immune deficiency</s0>
<s5>38</s5>
</fC07>
<fC07 i1="03" i2="X" l="SPA"><s0>Inmunodeficiencia</s0>
<s5>38</s5>
</fC07>
<fC07 i1="04" i2="X" l="FRE"><s0>Appareil respiratoire pathologie</s0>
<s5>53</s5>
</fC07>
<fC07 i1="04" i2="X" l="ENG"><s0>Respiratory disease</s0>
<s5>53</s5>
</fC07>
<fC07 i1="04" i2="X" l="SPA"><s0>Aparato respiratorio patología</s0>
<s5>53</s5>
</fC07>
<fC07 i1="05" i2="X" l="FRE"><s0>Poumon pathologie</s0>
<s5>54</s5>
</fC07>
<fC07 i1="05" i2="X" l="ENG"><s0>Lung disease</s0>
<s5>54</s5>
</fC07>
<fC07 i1="05" i2="X" l="SPA"><s0>Pulmón patología</s0>
<s5>54</s5>
</fC07>
<fN21><s1>078</s1>
</fN21>
</pA>
</standard>
</inist>
</record>

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   |texte=   Non-X-linked hyperimmunoglobulin M syndrome with chronic interstitial pneumonitis
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Serveur d'exploration Chloroquine

Non-X-linked hyperimmunoglobulin M syndrome with chronic interstitial pneumonitis

Non-X-linked hyperimmunoglobulin M syndrome with chronic interstitial pneumonitis

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Abstract

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