Lymphocytic interstitial pneumonia as a manifestation of SLE and secondary Sjogren's syndrome
Identifieur interne : 001132 ( Main/Exploration ); précédent : 001131; suivant : 001133Lymphocytic interstitial pneumonia as a manifestation of SLE and secondary Sjogren's syndrome
Auteurs : Daniel Garcia [États-Unis] ; Lary Young [États-Unis]Source :
- BMJ Case Reports [ 1757-790X ] ; 2013-08-02.
English descriptors
- Teeft :
- Antinuclear antibody, Autoimmune, Biopsy, Bronchoalveolar lavage, Case report, Chest pain, Emphysematous disease, Further permission, Inducible ischaemia, Interstitial, Interstitial lung disease, Interstitial pneumonia, Laboratory work, Lung biopsy, Lymphocytic, Lymphoma, Many cases, Negative results, Physical examination, Plasma cells, Pneumonia, Primary syndrome, Pulmonary embolism, Pulmonary manifestations, Pulse therapy, Respir crit care, Secondary syndrome, Surgical lung biopsy, Syndrome, Systemic lupus erythematosus, Unusual association, Vital signs.
Abstract
A 47-year-old woman with systemic lupus erythematosus (SLE) diagnosed at age of 35years was admitted for dyspnoea, substernal chest pain, dry mucosas and difficulty in swallowing. Physical examination revealed vesicular breath sounds bilaterally. Laboratory work showed antinuclear antibody (ANA) (speckled pattern, 1:40), positive anti-Sjogren’s syndrome antigen (SSA) and antisingle side band (SSB) and negative double-strand DNA (dsDNA), with normal C3,C4,C50. A high-resolution chest CT scan demonstrated multiple bronchial cysts and diffuse interstitial infiltrates. Surgical lung biopsy revealed emphysematous changes and mild lymphocytic infiltrate around the bronchioles compatible with lymphocytic interstitial pneumonia diagnosis. This case illustrates a patient with primary SLE overlapped by initial manifestations of secondary Sjogren’s syndrome (SS) presenting with associated autoimmune interstitial lung disease. Antibody markers, high-resolution chest CT scan and surgical lung biopsy were essential in evaluating this patient, confirming the interstitial lymphocytic infiltration of the lung. Primary SS (pSS) is the most commonly associated disease to lung interstitial pneumonia (LIP) (25%). High-resolution chest CT scan demonstrates areas of ground-glass attenuation, suggestive of interstitial disease. Surgical lung biopsy shows pathologic increase of mature lymphocyte cells and histiocytes. Most of the cases have a benign presentation and shortly relapse. Superimposed infection, pulmonary fibrosis and lymphoma develop in less than 20% of cases. Corticosteroids are the primary therapy. While pSS is commonly associated with interstitial lung involvement, secondary Sjogren's syndrome (sSS) is only rare. It has been described the initial sSS presentation by Sica symptoms development only, and our case is the first report of LIP presentation as initial manifestation of sSS. Our patient remained stable after corticosteroids and hydoxychloroquine therapy and no progression of disease after 6 months follow-up.
Url:
DOI: 10.1136/bcr-2013-009598
Affiliations:
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Physical examination revealed vesicular breath sounds bilaterally. Laboratory work showed antinuclear antibody (ANA) (speckled pattern, 1:40), positive anti-Sjogren’s syndrome antigen (SSA) and antisingle side band (SSB) and negative double-strand DNA (dsDNA), with normal C3,C4,C50. A high-resolution chest CT scan demonstrated multiple bronchial cysts and diffuse interstitial infiltrates. Surgical lung biopsy revealed emphysematous changes and mild lymphocytic infiltrate around the bronchioles compatible with lymphocytic interstitial pneumonia diagnosis. This case illustrates a patient with primary SLE overlapped by initial manifestations of secondary Sjogren’s syndrome (SS) presenting with associated autoimmune interstitial lung disease. Antibody markers, high-resolution chest CT scan and surgical lung biopsy were essential in evaluating this patient, confirming the interstitial lymphocytic infiltration of the lung. Primary SS (pSS) is the most commonly associated disease to lung interstitial pneumonia (LIP) (25%). High-resolution chest CT scan demonstrates areas of ground-glass attenuation, suggestive of interstitial disease. Surgical lung biopsy shows pathologic increase of mature lymphocyte cells and histiocytes. Most of the cases have a benign presentation and shortly relapse. Superimposed infection, pulmonary fibrosis and lymphoma develop in less than 20% of cases. Corticosteroids are the primary therapy. While pSS is commonly associated with interstitial lung involvement, secondary Sjogren's syndrome (sSS) is only rare. It has been described the initial sSS presentation by Sica symptoms development only, and our case is the first report of LIP presentation as initial manifestation of sSS. Our patient remained stable after corticosteroids and hydoxychloroquine therapy and no progression of disease after 6 months follow-up.</div></front></TEI><affiliations><list><country><li>États-Unis</li></country><region><li>Floride</li></region></list><tree><country name="États-Unis"><region name="Floride"><name sortKey="Garcia, Daniel" sort="Garcia, Daniel" uniqKey="Garcia D" first="Daniel" last="Garcia">Daniel Garcia</name></region><name sortKey="Young, Lary" sort="Young, Lary" uniqKey="Young L" first="Lary" last="Young">Lary Young</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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