Pediatric pulmonary and thoracic aspergillosis.

Aspergillus infection can cause significant disease in an immunocompromised host and lungs are the most common site of infection. Various forms of aspergillus involving lung parenchyma have been described including aspergilloma, allergic bronchopulmonary aspergillosis (ABPA), and semi-invasive and invasive pulmonary aspergillosis (IPA). Pulmonary aspergilloma usually occurs in patients with pre-existing cystic lung diseases. The classic radiographic finding is a discrete, round or oval mass in a pulmonary cavity, which moves with gravity. Allergic bronchopulmonary aspergillosis is characterized by clinical asthma, blood and sputum eosinophilia and positive immunologic reaction to aspergillus antigen. Central bronchiectasis in patients with asthma is highly suggestive of ABPA. Invasive pulmonary aspergillosis is usually fatal infection in patients taking immunosuppressants. Infection can also spread to other visceral organs. Multiple and solitary nodular densities as well as diffuse or focal consolidation have been reported. CT halo sign and CT hypodense signs are suggestive of IPA. Other forms of thoracic aspergillosis may include pleural aspergillosis, which can cause empyema. Osteomyelitis of the thoracic cage has been reported particularly in association with chronic granulomatous disease. We would like to demonstrate pertinent clinical and radiographic features of pulmonary and thoracic involvement of aspergillosis including radiographic differential diagnoses.

In-vivo allergenic potency of Siriraj Mite Allergen Vaccine (SMAV) comparing with standardized vaccine in mite-sensitive patients.

Background: The prevalence of allergic diseases, particularly asthma and allergic rhinitis, has increased tremendously in Thailand and worldwide. House dust mite (HDM) is the major IgE sensitizer among allergic children and adults. We have developed local standardized mite allergen extracts, Siriraj Mite Allergen Vaccine (SMAV) from Dermatophagoides pteronyssinus (Dp) and Dermatophagoides farinae (Df) from our source materials which were highly purified (99%). Objective: To compare in-vivo allergenic potency of both SMAV Dp and Df with commercial standardized mite allergen vaccine by using skin prick testing in mite-sensitive individuals. Methods: This was a double-blind, randomized, self controlled study comparing SMAV and commercial standardized mite allergen vaccine (Dp and Df) by using skin prick testing in mite-sensitive adult volunteers, 18 – 60 years of age. Results: The study was performed in 54 adult volunteers (19 males, mean age 26.6 + 5.5 years old) who had positive skin test to commercial Dp and Df. Seventeen of them had no allergic disease. The most common allergic disease among the volunteers was allergic rhinitis (21/37). Mean wheal diameter of SMAV Dp and commercial Dp at the concentration of 10,000 and 5, 000 AU/ml were equivalent but at the concentration of 2,500 AU/ml was inequivalent. Mean wheal diameter of SMAV Dp was significantly larger than commercial Dp at concentration of 2,500 AU/ml (p < 0.05). Mean wheal diameter of SMAV Df and commercial Df at all 3 concentrations were equivalent. There was no systemic side effect in all subjects. Conclusion: The study demonstrated that in mite-sensitive adults, SPT using SMAV Dp (10,000 AU) and Df (10,000 AU) had equivalent allergenic potency to the commercial comparator without any systemic side effect.

A combination of intravenous immunoglobulin and pulse methylprednisolone extended survival in pulmonary alveolar proteinosis with chronic interstitial pneumonitis: a case report.

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.

The accumulation of dust mite allergens on mattresses made of different kinds of materials.

Background: Different mattress materials may affect the accumulation of allergens. Objective: To compare the amount of group 1 dust mite allergens (Der p1 + Der f1) on mattresses made of different kinds of materials before and after use. Methods: Sixty new mattresses made of kapok, synthetic fiber, coconut fiber and sponge-like polyurethane, were placed in the house officers’ dormitory at Siriraj hospital, Thailand. The dust samples were collected before (0), 1, 2, 3, 6, 9 and 12 months after the mattresses were used. Group 1 dust mite allergens were analyzed using two-site monoclonal antibody ELISA. Results: Der f1 made up 86.7 % of group 1 allergens found in the matress dust. After the 2nd month, only the mean level in sponge-like polyurethane mattress was under 2 µg/g dust (sensitized level). At the 6th month, the mean levels were 13.1 in coconut, 21.7 in kapok and 17.3 µg/g dust in synthetic fiber, all of which were more than 10 µg/g dust (symptomatic level). At the 9th month, the level in sponge-like polyurethane mattress was increased to 11.2 µg/g. At 12th month the level in coconut fiber, sponge-like polyurethane synthetic fiber and kapok mattresses were 20.2, 22.4, 28.9 and 32.2 µg/g dust respectively. Conclusions: The accumulation rate in kapok and synthetic mattresses was significantly higher than coconut and sponge-like polyurethane mattresses. The mean level of group 1 mite allergens exceeded 10 µg/g dust after the 6th month of use in coconut fiber, kapok and synthetic fiber and at the 9th month in sponge-like polyurethane mattress.

Comparison between siriraj mite allergen vaccine and Standardized Commercial mite vaccine by skin prick testing in normal Thai adults.

House dust mite is a major cause of allergic asthma and rhinitis in Thai population. Skin prick test (SPT) is a useful tool for the diagnosis of the IgE-mediated reactions. The imported commercial mite vaccine for SPT is available but it is relatively expensive. Aim of this studyis to compare Siriraj Mite Allergen Vaccine (SMAV) with standardized commercial mite allergen vaccine by skin prick testing in normal Thai adults. A double blind, self-controlled study between the SMAV and standardized commercial mite allergen vaccine was performed by SPT in 17 normal Thai adult males and non-pregnant or non-lactating females aged 18-60 years. The study showed that35.29 % of non atopic adults had positive SPT reaction to Dp and Df of both SMAV andstandardized commercial mite allergen vaccine. Mean wheal and flare diameters from SPT of Dp and Df of SMAV showed strong correlation with standardized commercial mite allergen vaccine (r = 0.768 and 0.897 in Dp and Df respectively, p <0.001). The intraclass correlation was also excellent (0.893 and 0.775 in Dp and Df respectively). There was no significant difference in wheal and flare diameter between SMAV andstandardized commercial mite allergen vaccine. No systemic or large local reaction was found in any of the study cases.

Immediate type hypersensitivity to chemotherapeutic agents in pediatric patients.

Nine patients (3 boys and 6 girls) with a median age of 9.5 years, with immediate type hypersensitivi-ty reactions to chemotherapeutic agents were reviewed. The presenting symptoms were urticaria (4/9) and ana-phylaxis (5/9). The causative agents were vincristine (2/9), L-asparaginase (2/9), mesna (1/9), cyclosporine (1/9), carboplatin (2/9) and cyclophosphamide (1/9). Three of the five patients with anaphylaxis were changed to alterna-tive chemotherapeutic agents. In two cases alternative drugs were not available and the patients underwent safe and successful desensitization. Three of the 4 patients with urticaria were successfully exposed to graded chal-lenges with cyclosporine, carboplatin and cyclophosphamide, respectively. In the other case with generalized urti-caria, mesna was withdrawn due to a positive intradermal test. In patients with immediate type hypersensitivity reactions to chemotherapeutic drugs, if effective alternative chemotherapeutic agents are not available and/or the skin test is negative, a careful drug challenge and/or desensitization should be performed.

A novel mutation of the IL12RB1 gene in a child with nocardiosis, recurrent salmonellosis and neurofibromatosis type I: First case report from Thailand.

Genetic defects of interleukin (IL)-12/23-and interferon (IFN)-γ-mediated immunity can cause in-creased susceptibility to intracellular microbes. Among these defects, a mutation of the gene encoding the IL-12 receptor β1 (IL-12Rβ1) is the most common worldwide. A 12-year old Thai boy with pre-existing neurofibromatosis type 1 (NF1) was evaluated for primary immunodeficiency after a history of tuberculous lymphadenitis, recurrent Salmonella infections and nocardiosis. Flow cytometry of phytohemagglutinin (PHA)-stimulated peripheral blood mononuclear cells (PBMCs) revealed a defect in the IL-12Rβ1 surface expression. A genetic study showed a novel nonsense homozygous mutation of the IL12RB1 gene in exon 4 (402C>A), confirming the diagnosis of IL-12Rβ1 deficiency. This is the first case report of a primary IL-12Rβ1 deficiency in Thailand with the interesting finding of a coexisting NF1.

Wheat-dependent, exercise-induced anaphylaxis in Thai children: A report of 5 cases.

Wheat is not an uncommon cause of food-dependent, exercise-induced anaphylaxis. This study aims to describe common clinical characteristics and laboratory manifestations of the disease. Five children, aged 8-14 years were evaluated. An atopic history was found in 20% of the patients. All patients had symptoms which involved the skin and three had hypotension. Serum specific IgE for wheat was measured and showed a positive result in 2 patients. A three-day challenge protocol with an open challenge for wheat on day 1, an exercise chal-lenge test on day 2 and another exercise challenge test on day 3 after a meal containing wheat was performed. Four patients completed the three-day challenge protocol. Anaphylaxis occurred in 2 out of 4 patients who con-sumed more than 100 grams of wheat prior to the exercise. The three-day challenge protocol is a definitive diag-nostic tool to confirm the diagnosis of WDEIA. However, the amount of wheat used for challenging should be at least 100 grams. Abbreviations: WDEIA, wheat-dependent, exercise-induced anaphylaxis; FDEIA, food-dependent, exercise-induced anaphylaxis; SPT, skin prick test.

Successful treatment of disseminated BCG infection in a SCID patient with granulocyte colony stimulating factor.

Severe combined immunodeficiencies (SCID) are disorders with impairment of humoral and cellular immune functions. The prognosis of disseminated bacillus Calmette-Guérin (BCG) infection in immunocompromised host is unfavorable since response to standard therapy is poor. We report a successful treatment of disseminated BCG infection with granulocyte colony stimulating factor (G-CSF) in a patient with severe combined immunodeficiency. The patient failed to response to intensive anti-tuberculous (anti-TB) therapy. After 2 months of G-CSF, in addition to anti-TB treatment, the clinical signs of disseminated BCG infection were improved. Since serious BCG infections in SCID are not uncommon in developing countries, where BCG vaccination is mandatory to all newborns, the combination of G-CSF and anti-TB drugs should be considered in immunocompromised patients with protracted mycobacterial infection.