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Multi nodular parenchymal pulmonary amyloidosis in primary Sjogren's syndrome: A case report

Fatma Sema Oymak, Ozlem Canoz, Fatih Kurnaz, Mustafa Calis, Afra Yildirim, Nuri Tutar, Asiye Kanbay, Hakan Buyukoglan, Inci Gülmez, Ramazan Demir
European Respiratory Journal 2012 40: P3618; DOI:
Fatma Sema Oymak
1Department of Chest Diseases, Erciyes University Medical School, Kayseri, Turkey
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Ozlem Canoz
2Department of Pathology, Erciyes University Medical School, Kayseri, Turkey
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Fatih Kurnaz
3Department of Internal Diseases, Hematology Unit, Erciyes University Medical School, Kayseri, Turkey
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Mustafa Calis
4Department of Physical Therapy and Rehabilitation, Erciyes University Medical School, Kayseri, Turkey
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Afra Yildirim
5Department of Radiology, Erciyes University Medical School, Kayseri, Turkey
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Nuri Tutar
1Department of Chest Diseases, Erciyes University Medical School, Kayseri, Turkey
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Asiye Kanbay
1Department of Chest Diseases, Erciyes University Medical School, Kayseri, Turkey
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Hakan Buyukoglan
1Department of Chest Diseases, Erciyes University Medical School, Kayseri, Turkey
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Inci Gülmez
1Department of Chest Diseases, Erciyes University Medical School, Kayseri, Turkey
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Ramazan Demir
1Department of Chest Diseases, Erciyes University Medical School, Kayseri, Turkey
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Abstract

Amyloidosis is a rare cause of pulmonary infiltrates in primary Sjögren's syndrome(SS), affecting 0–2% of symptomatic patients. Diffuse multi nodular amyloidosis(DMNPA) is the most common radiological pattern, occurring alone or in association with cysts of varying size. Amyloid can be easily overlooked or mistaken for other entities with similar staining qualities. We describe the case of a 61-year-old woman with DMNPA (AL (λ)) related to SS, in the presence of primary systemic amyloidosis(PSA). She had suffering from cough, hemoptysis and dyspnoea of two -month duration. She had been diagnosed with SS 10 years previously. She had macroglossia and multiple skin nodules. Chest excursions were reduced with crackles in both lungs.Her chest radiograph and CT showed multiple nodules and cysts in both lung fields. Calcification in the nodules and lymphadenopathy were apparent in the mediastinal windows. Spirometry indicated moderate restriction, and BAL fluid was negative for acid-fast bacilli (AFB), fungi. Bronchoscopy with transbronchial lung biopsy revealed hyaline eosinophilic material associated with a giant cell granulomatous reaction, a patchy lymphocytic infiltrate. The congo red stain was positive in this hyaline material and showed apple green birefringence on polarising microscopy. Electrophoresis of serum proteins revealed hypergammaglobulinaemia. Serum immunofixation showed a monoclonal IgGλ. In the bone marrow (BM) plasma cells were increased to 10-15%. PSA (AL) associated with SS was diagnosed. Amyloid deposits were found not only in the lung but also in skin and BM biopsy specimens. Staining for amyloidosis should be performed in patient with SS and pulmonary infiltration.

  • Orphan disease
  • Imaging
  • Interstitial lung disease (connective tissue disease)
  • © 2012 ERS
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Multi nodular parenchymal pulmonary amyloidosis in primary Sjogren's syndrome: A case report
Fatma Sema Oymak, Ozlem Canoz, Fatih Kurnaz, Mustafa Calis, Afra Yildirim, Nuri Tutar, Asiye Kanbay, Hakan Buyukoglan, Inci Gülmez, Ramazan Demir
European Respiratory Journal Sep 2012, 40 (Suppl 56) P3618;

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Multi nodular parenchymal pulmonary amyloidosis in primary Sjogren's syndrome: A case report
Fatma Sema Oymak, Ozlem Canoz, Fatih Kurnaz, Mustafa Calis, Afra Yildirim, Nuri Tutar, Asiye Kanbay, Hakan Buyukoglan, Inci Gülmez, Ramazan Demir
European Respiratory Journal Sep 2012, 40 (Suppl 56) P3618;
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