A 9-year-old girl with respiratory distress, dry cough exacerbated at night and triggered by exercise, and fever for about 48 h before admission was admitted to our department. In her past medical history, she was diagnosed of previous childhood asthma at 3 years of age. Atopy history and skin prick test of aeroallergens in past medical history and records were negative. Asthma control was achieved with inhale corticosteroid and asthma treatment stopped after two years. The patient had neither had an asthma attack nor needed asthma related medication in the last 4 years of her life. Latest pulmonary function test was one year before admission, which revealed FEV1: 85%, FEV1/FVC: 91%, FVC: 93%, and PEF: 78%. The initial physical examination revealed diffuse rales and wheezing. Her vitals revealed tachypnea (respiratory rate: 32), tachycardia (pulse rate: 135), temperature of 38, and oxygen saturation levels of 80% in room air. Chest X-ray revealed perihelia infiltration. The patient was hospitalized primarily based on the impression of being plagued with asthma and pneumonia; thus, specific treatment for asthma and antibiotic therapy for pneumonia was initiated. Seventy-two hours later, antibiotics were changed from Clindamycin to Meropenem plus Vancomycin and Azithromycin. The fever subsided in the patient within 48 h and the symptoms of cough and respiratory distress improved significantly. The asthma symptoms were also improved.
The laboratory findings were as follows: white blood cell count of 10700/mL with 1% eosinophils and IgE level of 1075 IU/ml (normal range: 20-100) (Table 1). Chest CT SCAN revealed mild ground glass appearance, 72 hours later. Skin prick test was negative for aspergillosis. Bronchoscopy was carried out and bronchoalveolar lavage (BAL) secretion was analyzed for gram stain and sent for polymerase chain reaction (PCR) to check for aspergillosis, candida, and tuberculosis that all were negative. In BAL Cytometry, the most dominant cell was macrophage (75%) and less than 5% was eosinophil. The patient was discharged after 7 days with 250 micro fluticasone daily inhaler and oral prednisolone 0.5 mg/kg per day (for 2 days more) by diagnosis of asthma relapse.
Four days later, the patient was readmitted with cough, dyspnea, and diffuse bilateral wheeze. The results obtained from the physical examination were similar to previous findings except for the absence of fever. Laboratory tests revealed WBC: 14700/mul with 30% eosinophil. IgE levels were 1359 IU/mL and 1661 IU/mL in double-checking. The results of further laboratory tests are summarized in Table 1.
On the 2nd day of admission, the patient developed dyspnea and severe subcutaneous emphysema in the anterior and posterior areas of the neck. Spiral chest CT scan revealed severe pneumo-mediastinum and severe emphysema in the chest wall (Figure 1). In addition, ground glass densities and findings in favor of bronchiectasis were also reported in both lungs.
Stool examination was carried out to check for eosinophilia, but the result was negative. According to the high titer of total IgE and eosinophilia, follow-up works were carried out for allergic bronchopulmonary aspergillosis (ABPA), which was negative for specific IgG (18.5mg/ml, cut-off<50) and specific IgE (<0.1IU/ml, cut-off<0/1) of aspergillosis and specific IgG (4.2, ref<113) of Candida but positive for specific IgE (0.74, cut-off <0.1) of Candida. The report of BAL bronchoscopy in previous admissions revealed the presence of Candida albicans. The patient was admitted in the intensive care unit (ICU) because of the decrease in breathing sounds and severe respiratory distress. She was once again placed on Meropenem and Vancomycin medication. As a result of progressive emphysema and decreased O2 saturation, a chest tube was inserted. Intravenous infusion (IV) of methylprednisolone 1 mg/kg/day plus IV fluconazole 6 mg/kg/day in the first day and following 4 mg/kg/day in the following days was administered. After one week, the chest tube was removed and respiratory distress was improved markedly. The patient was transferred to a ward for final diagnosis of ABPM with a high dose of Itraconazole (200mg twice daily) and high doses of oral prednisolone (0.75 mg/kg per day divided twice daily) and was discharged after 10 days. The same doses of Prednisolone and Itraconazole were continued on the patient using the same doses; and Fluticasone plus Salmetrol inhaler spray (250micro/day divided twice daily) and oral Montelukast were also prescribed for relieving severe asthma attack. Oxygen supplement according to oxygen saturation assay was also recommended. In further follow-up, after one month, the patient's general condition improved significantly and the use of oxygen was no longer necessary. The IgE level decreased to 255 IU/mL and the patient had normal social activity and normal lung sounds. After 2 months, by decreasing prednisolone dose to 25%, asthma symptoms worsened; therefore, uptitration of prednisolone was carried out to reach the previous administered doses. After 3 months, prednisolone was tapered by 25% every four weeks; and after 4 months, the patient stopped receiving prednisolone with good asthma control and IgE level of 86 IU/mL. The total eosinophil count decreased to 100/mul in the peripheral blood sample. After 6 months, asthma medication decreased to 125 Fluticasone per day as the doses were needed for mild persistent asthma. Thus, good asthma control was achieved. After passing 6 months, all drugs were stopped and no other respiratory complaint has been reported in the last 4 months.