Pulmonary cryptococcosis in childhood systemic lupus erythematosus and Sjögren syndrome overlap: a rare opportunistic infection.
Identifieur interne : 001111 ( Main/Exploration ); précédent : 001110; suivant : 001112Pulmonary cryptococcosis in childhood systemic lupus erythematosus and Sjögren syndrome overlap: a rare opportunistic infection.
Auteurs : V L S. Marques [Brésil] ; R C Gomes ; G R Viola ; M M Maia ; G S Durigon ; N E Aikawa ; C. Artur SilvaSource :
- Lupus [ 1477-0962 ] ; 2013.
Descripteurs français
- KwdFr :
- Adolescent, Antifongiques (usage thérapeutique), Biopsie, Cryptococcose (diagnostic), Cryptococcose (immunologie), Cryptococcose (microbiologie), Cryptococcose (traitement médicamenteux), Facteurs de l'âge, Femelle, Humains, Immunosuppresseurs (usage thérapeutique), Infections opportunistes (diagnostic), Infections opportunistes (immunologie), Infections opportunistes (microbiologie), Infections opportunistes (traitement médicamenteux), Lavage bronchoalvéolaire, Lupus érythémateux disséminé (), Lupus érythémateux disséminé (diagnostic), Lupus érythémateux disséminé (immunologie), Lupus érythémateux disséminé (traitement médicamenteux), Mycoses pulmonaires (diagnostic), Mycoses pulmonaires (immunologie), Mycoses pulmonaires (microbiologie), Mycoses pulmonaires (traitement médicamenteux), Résultat thérapeutique, Syndrome de Gougerot-Sjögren (), Syndrome de Gougerot-Sjögren (diagnostic), Syndrome de Gougerot-Sjögren (immunologie), Syndrome de Gougerot-Sjögren (traitement médicamenteux), Tomodensitométrie.
- MESH :
- diagnostic : Cryptococcose, Infections opportunistes, Lupus érythémateux disséminé, Mycoses pulmonaires, Syndrome de Gougerot-Sjögren.
- immunologie : Cryptococcose, Infections opportunistes, Lupus érythémateux disséminé, Mycoses pulmonaires, Syndrome de Gougerot-Sjögren.
- microbiologie : Cryptococcose, Infections opportunistes, Mycoses pulmonaires.
- traitement médicamenteux : Cryptococcose, Infections opportunistes, Lupus érythémateux disséminé, Mycoses pulmonaires, Syndrome de Gougerot-Sjögren.
- usage thérapeutique : Antifongiques, Immunosuppresseurs.
- Adolescent, Biopsie, Facteurs de l'âge, Femelle, Humains, Lavage bronchoalvéolaire, Lupus érythémateux disséminé, Résultat thérapeutique, Syndrome de Gougerot-Sjögren, Tomodensitométrie.
English descriptors
- KwdEn :
- Adolescent, Age Factors, Antifungal Agents (therapeutic use), Biopsy, Bronchoalveolar Lavage, Cryptococcosis (diagnosis), Cryptococcosis (drug therapy), Cryptococcosis (immunology), Cryptococcosis (microbiology), Female, Humans, Immunosuppressive Agents (therapeutic use), Lung Diseases, Fungal (diagnosis), Lung Diseases, Fungal (drug therapy), Lung Diseases, Fungal (immunology), Lung Diseases, Fungal (microbiology), Lupus Erythematosus, Systemic (complications), Lupus Erythematosus, Systemic (diagnosis), Lupus Erythematosus, Systemic (drug therapy), Lupus Erythematosus, Systemic (immunology), Opportunistic Infections (diagnosis), Opportunistic Infections (drug therapy), Opportunistic Infections (immunology), Opportunistic Infections (microbiology), Sjogren's Syndrome (complications), Sjogren's Syndrome (diagnosis), Sjogren's Syndrome (drug therapy), Sjogren's Syndrome (immunology), Tomography, X-Ray Computed, Treatment Outcome.
- MESH :
- chemical , therapeutic use : Antifungal Agents, Immunosuppressive Agents.
- complications : Lupus Erythematosus, Systemic, Sjogren's Syndrome.
- diagnosis : Cryptococcosis, Lung Diseases, Fungal, Lupus Erythematosus, Systemic, Opportunistic Infections, Sjogren's Syndrome.
- drug therapy : Cryptococcosis, Lung Diseases, Fungal, Lupus Erythematosus, Systemic, Opportunistic Infections, Sjogren's Syndrome.
- immunology : Cryptococcosis, Lung Diseases, Fungal, Lupus Erythematosus, Systemic, Opportunistic Infections, Sjogren's Syndrome.
- microbiology : Cryptococcosis, Lung Diseases, Fungal, Opportunistic Infections.
- Adolescent, Age Factors, Biopsy, Bronchoalveolar Lavage, Female, Humans, Tomography, X-Ray Computed, Treatment Outcome.
Abstract
Meningitis is the main manifestation of cryptococcosis in adult systemic lupus erythematosus (SLE) patients, and other organs and systems, such as the lungs, are rarely affected in this fungal infection. To our knowledge, no case of pulmonary cryptococcosis has been described in the pediatric lupus population. Therefore, we report herein one patient with childhood SLE (C-SLE) and Sjögren's syndrome overlap that presented encapsulated Cryptococcus yeast cells in lung tissue. A 14-year-old girl was diagnosed with C-SLE. At the age of 16 years and 5 months, she presented with fever, cough and dyspnea, without headache, vomiting, and also without signs of meningeal irritation or other clinical manifestations. She was being treated with mycophenolate mofetil, hydroxychloroquine and prednisone. Chest radiography and chest computer tomography showed a single nodule in the left posterior apex and three nodular lesions in the left hemithorax respectively. Bronchoalveolar lavage and transbronchial biopsy were normal and without isolation of bacteria or fungi. Voriconazole was empirically introduced for 21 days. Fifteen days after the first biopsy, she underwent open thoracotomy with surgical left lung biopsy and was diagnosed with pulmonary cryptococcosis. Voriconazole was replaced with oral fluconazole and this antifungal therapy was maintained with improvement of clinical manifestations and without marked alteration of radiological images. In conclusion, we report the first case of pulmonary cryptococcosis in Sjögren's and C-SLE patient with a satisfactory clinical response to antifungal therapy. Fungal infections should be excluded in the presence of lung nodules and etiological identification is required for proper treatment.
DOI: 10.1177/0961203313502859
PubMed: 23985965
Affiliations:
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médicamenteux" xml:lang="fr"><term>Cryptococcose</term><term>Infections opportunistes</term><term>Lupus érythémateux disséminé</term><term>Mycoses pulmonaires</term><term>Syndrome de Gougerot-Sjögren</term></keywords><keywords scheme="MESH" qualifier="usage thérapeutique" xml:lang="fr"><term>Antifongiques</term><term>Immunosuppresseurs</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adolescent</term><term>Age Factors</term><term>Biopsy</term><term>Bronchoalveolar Lavage</term><term>Female</term><term>Humans</term><term>Tomography, X-Ray Computed</term><term>Treatment Outcome</term></keywords><keywords scheme="MESH" xml:lang="fr"><term>Adolescent</term><term>Biopsie</term><term>Facteurs de l'âge</term><term>Femelle</term><term>Humains</term><term>Lavage bronchoalvéolaire</term><term>Lupus érythémateux disséminé</term><term>Résultat thérapeutique</term><term>Syndrome de Gougerot-Sjögren</term><term>Tomodensitométrie</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Meningitis is the main manifestation of cryptococcosis in adult systemic lupus erythematosus (SLE) patients, and other organs and systems, such as the lungs, are rarely affected in this fungal infection. To our knowledge, no case of pulmonary cryptococcosis has been described in the pediatric lupus population. Therefore, we report herein one patient with childhood SLE (C-SLE) and Sjögren's syndrome overlap that presented encapsulated Cryptococcus yeast cells in lung tissue. A 14-year-old girl was diagnosed with C-SLE. At the age of 16 years and 5 months, she presented with fever, cough and dyspnea, without headache, vomiting, and also without signs of meningeal irritation or other clinical manifestations. She was being treated with mycophenolate mofetil, hydroxychloroquine and prednisone. Chest radiography and chest computer tomography showed a single nodule in the left posterior apex and three nodular lesions in the left hemithorax respectively. Bronchoalveolar lavage and transbronchial biopsy were normal and without isolation of bacteria or fungi. Voriconazole was empirically introduced for 21 days. Fifteen days after the first biopsy, she underwent open thoracotomy with surgical left lung biopsy and was diagnosed with pulmonary cryptococcosis. Voriconazole was replaced with oral fluconazole and this antifungal therapy was maintained with improvement of clinical manifestations and without marked alteration of radiological images. In conclusion, we report the first case of pulmonary cryptococcosis in Sjögren's and C-SLE patient with a satisfactory clinical response to antifungal therapy. Fungal infections should be excluded in the presence of lung nodules and etiological identification is required for proper treatment. </div></front></TEI><affiliations><list><country><li>Brésil</li></country></list><tree><noCountry><name sortKey="Aikawa, N E" sort="Aikawa, N E" uniqKey="Aikawa N" first="N E" last="Aikawa">N E Aikawa</name><name sortKey="Artur Silva, C" sort="Artur Silva, C" uniqKey="Artur Silva C" first="C" last="Artur Silva">C. Artur Silva</name><name sortKey="Durigon, G S" sort="Durigon, G S" uniqKey="Durigon G" first="G S" last="Durigon">G S Durigon</name><name sortKey="Gomes, R C" sort="Gomes, R C" uniqKey="Gomes R" first="R C" last="Gomes">R C Gomes</name><name sortKey="Maia, M M" sort="Maia, M M" uniqKey="Maia M" first="M M" last="Maia">M M Maia</name><name sortKey="Viola, G R" sort="Viola, G R" uniqKey="Viola G" first="G R" last="Viola">G R Viola</name></noCountry><country name="Brésil"><noRegion><name sortKey="Marques, V L S" sort="Marques, V L S" uniqKey="Marques V" first="V L S" last="Marques">V L S. Marques</name></noRegion></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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