PMID- 21038791 OWN - NLM STAT- MEDLINE DCOM- 20110209 LR - 20161125 IS - 0125-877X (Print) IS - 0125-877X (Linking) VI - 28 IP - 2-3 DP - 2010 Jun-Sep TI - A combination of intravenous immunoglobulin and pulse methylprednisolone extended survival in pulmonary alveolar proteinosis with chronic interstitial pneumonitis: a case report. PG - 200-5 AB - Pulmonary alveolar proteinosis (PAP) is characterized by intra-alveolar accumulation of lipoproteinaceous material. The severe chronic pulmonary disease and susceptibility to pulmonary infection is a prominent feature of the disease. We reported a case of postnatal-onset PAP and chronic interstitial pneumonitis in a girl with chronic respiratory distress since she was 5 months of age. A lung biopsy confirmed the diagnosis. The therapeutic bronchoalveolar lavages, a short trial of granulocyte colony-stimulation factor (G-CSF) and a combination of low dose methylprednisolone and hydroxychloroquine were used at different times without noting satisfactory improvement. Intravenous immunoglobulin (IVIG) and pulse methylprednisolone were given monthly with gradual recovery. She did not require oxygen supplement after 21 months of this combination. Our report suggested that IVIG and pulse methylprednisolone might have a potential role in the treatment of PAP with chronic interstitial pneumonitis. FAU - Jirarattanasopa, Nongnapa AU - Jirarattanasopa N AD - Division of Allergy and Immunology, Department of Pediatrics, Faculty of Medicine Siriraj Hospital Mahidol University, Bangkok, Thailand. FAU - Tantikul, Chutsumarn AU - Tantikul C FAU - Vichyanond, Pakit AU - Vichyanond P FAU - Pacharn, Punchama AU - Pacharn P FAU - Visitsunthorn, Nualanong AU - Visitsunthorn N FAU - Suttinont, Panthep AU - Suttinont P FAU - Jirapongsananuruk, Orathai AU - Jirapongsananuruk O LA - eng PT - Case Reports PT - Journal Article PL - Thailand TA - Asian Pac J Allergy Immunol JT - Asian Pacific journal of allergy and immunology JID - 8402034 RN - 0 (Immunoglobulins, Intravenous) RN - X4W7ZR7023 (Methylprednisolone) SB - IM MH - Chronic Disease MH - Cyanosis MH - Dyspnea MH - Fatal Outcome MH - Female MH - Humans MH - Hyperbaric Oxygenation MH - Immunoglobulins, Intravenous/administration & dosage MH - Infant MH - Infant, Newborn MH - Lung/immunology/*metabolism/microbiology/pathology MH - Lung Diseases, Interstitial/complications/*diagnosis/physiopathology/therapy MH - Methylprednisolone/administration & dosage MH - Pneumocystis carinii/*immunology/pathogenicity MH - Pulmonary Alveolar Proteinosis/complications/*diagnosis/physiopathology/therapy MH - Respiratory Insufficiency EDAT- 2010/11/03 06:00 MHDA- 2011/02/10 06:00 CRDT- 2010/11/03 06:00 PHST- 2010/11/03 06:00 [entrez] PHST- 2010/11/03 06:00 [pubmed] PHST- 2011/02/10 06:00 [medline] PST - ppublish SO - Asian Pac J Allergy Immunol. 2010 Jun-Sep;28(2-3):200-5.