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Pneumonia was considered, but there was no obvious improvement after using azithromycin and piperacilli/tazobactam for ten days

Pneumonia was considered, but there was no obvious improvement after using azithromycin and piperacilli/tazobactam for ten days. lung disease (IgG4-RLD), a condition characterized by IgG4-positive lymphoplasmacytic cells infiltration in lung Xantocillin and elevated serum IgG4 concentration in most patients, is a recently emerging entity [1]. Reports indicated IgG4-RLD has multiple forms of lung lesions, which is more than previously thought [2-9], but the full spectrum of clinicopathologic feature has not been well described, it appears to be rather nonspecific, so, it is easy to be misdiagnosed as pneumonia with pulmonary consolidations. In addition, the disease always occurs in adults, male predominance [3]. To date, no description of IgG4-RLD in puerperium has been published to our knowledge. Herein, we describe a case of IgG4-RLD in puerperium, who manifested inflammatory conditions and was misdiagnosed to pneumonia. Case report A 24-year-old woman was administrated for management of delivery at 38 weeks of gestation at six weeks ago (gravida 1, para 1), who gave birth by spontaneous delivery and had uneventful antenatal follow-up period. The female newborn had a birth of 3,203 grams and Apgar scores of 8. No obvious deficits were noted during delivery. Three weeks following the delivery, she was admitted to our hospital because of dry cough, coexisting with fever and exertional dyspnea. She had a history of allergic rhinitis. Her body temperature was 38 degree centigrade, and there were no any signs in chest physical examination. Laboratory findings showed erythrocyte sedimentation rate (ESR) was 105 mm/1 H (normal range, 0-20 mm/1 H) and C reaction protein (CRP) was 28.74 mg/L (normal range, 10 mg/L). The total count and classification of white blood cells were 11.4710^9/L (normal range, 4.0-10.010^9/L) and 70.1%. Serum IgG (1860 mg/dl, normal range, Xantocillin 700-1600 mg/dl) and IgM (290 mg/dl, normal range, 50-270 mg/dl) were mildly increased. Other laboratory tests including IgA, IgE, interleukin-6 (IL-6), carcino-embryonic antigen (CEA), carbohydrate antigen-125 (CA-125), CA-153, CA-199, Antinuclear antibodies (ANA), rheumatoid factors (RF), anti-neutrophil cytoplasmic antibody (ANCA) were normal. A chest X-ray and computed tomography (CT) revealed multiple pulmonary consolidations in bilateral lungs (Figure 1A, ?,1B).1B). Pneumonia was considered, but there was no obvious improvement after using azithromycin and piperacilli/tazobactam for ten days. CT scan images found that there were no obvious changes compared with the previous one, then a bronchoscopy and transbronchial lung biopsy (TBLB) were performed and the histopathology of TBLB samples showed fibrosis and infiltration of plasma cells and lymphocytes in alveolar septa. Bronchoalveolar lavage fluid (BALF) revealed macrophage 46%, lymphocytes 35%, neutrophils 8%, eosinophils 11%, CD4/CD8 ratio 0.234. The bacterial examination was negative. It cant be ruled out the possibility of fungal infection, treatment strategy was changed to use moxifloxacin and voriconazole, but there was still no obvious improvement. To obtain a definitive diagnosis and an appropriate treatment, VATS lung biopsy was performed and the final pathological slice found dense lymphoplasmacytic infiltration, fibrosis in a storiform pattern, IgG4 immuno-staining revealed predominant lgG4 positive plasma cells, the percentage of IgG4 positive versus IgG positive plasma cells (IgG4+/IgG+) was 60% (Figure 2A-C). To further investigate the available TBLB-samples, IgG4 immuno-staining was performed and unexpected found that the percentage of IgG4+/IgG+ was 51%. To determine the involvement of other organs, a total-body CT scan examination showed no other abnormality, including the pancreas, Xantocillin salivary gland, bile duct. A diagnosis of isolated IgG4-RLD was made considering the elevation of serum IgG4 concentration (378 mg/dl) and no other organ involvement. Open in a separate window Figure 1 Multiple pulmonary consolidations revealed by Chest CT of the patient with IgG4-RLD prior to treatment (May 22, 2013). Open in a separate window Figure 2 Pathological section of the nodules. A. Dense lymphoplasmacytes infiltrated in the alveolar septa and bronchovascular bundle (H&E staining; magnification 10). B. Fibrosis was found in a storiform pattern (H&E staining; magnification 10). C. IgG4 immuno-staining revealed predominant lgG4+ plasma cells, the percentage of Mouse monoclonal to CRTC3 IgG4+/IgG+ was 60% (magnification, 10). The patient.