Application of desaturation index in post-surgery follow-up in children with obstructive sleep apnea syndrome.

Adenotonsillectomy is recommended for children who need surgery for obstructive sleep apnea syndrome (OSAS). Overnight, polysomnography (PSG) is suggested for post-surgery follow-up, but this diagnostic technique is time consuming and inconvenient. Desaturation index (DI) has been reported as a good tool for predicting both the presence and severity of OSAS in children. The purpose of this study was to determine the usefulness of the DI for post-surgery follow-up of children with OSAS. This retrospective study enrolled 42 children, aged 3-12 years, who were snorers diagnosed with OSAS by overnight PSG and who underwent an adenotonsillectomy. Pre- and postoperative PSG parameters, nocturnal pulse oximetry data, and modified Epworth sleepiness scale scores were assessed. Previously determined cut-off DI values (2.05, 3.50, and 4.15 for mild, moderate, and severe OSAS, respectively) were used to predict residual OSAS. Of the 42 children, obvious improvements were observed in apnea-hypopnea index (AHI, decreased 45.5 %), arousal index (decreased 30.5 %), DI (decreased 40.4 %), and snore index (decreased 100.3 %) compared with the preoperative measurements. Among these objective PSG measures, DI had the strongest correlation with AHI both pre- and post-surgeries (r = 0.947 and r = 0.954, respectively; p all <0.001). The DI change, before and after surgery, also had the strongest positive correlation to the AHI change (r = 0.482 and p = 0.001). Using the previously determined DI cut-off values to predict postoperative residual OSAS, there was a good positive predictive value (92.6 %) for mild residual OSAS and a good negative predictive value for moderate and severe residual OSAS (85.2 and 89.7 %, respectively). These findings suggest that DI, as determined using a nocturnal pulse oximeter, may be an alternative tool for postoperative evaluation and follow-up of children with OSAS.

Age-dependent distribution of the atopic phenotype and allergen sensitization among asthmatic children in southern Taiwan.

BACKGROUND: Asthma is divided into atopic and non-atopic phenotypes. The percentages of atopic asthma and allergen sensitization in patients of different ages have not been well studied. OBJECTIVE: To determine the percentage distribution of atopic and non-atopic phenotypes in different age groups of asthmatic children, and investigate the distribution of specific IgE to different allergens when stratified by age group in southern Taiwan. METHOD: We conducted this hospital-based, retrospective, cross-sectional study in southern Taiwan between 2004 and 2006. Asthmatic children aged 3 to 18 years who were diagnosed according to the Global Initiative for Asthma guidelines were enrolled. The MAST-CLA system was used to detect 36 allergen-specific IgEs. RESULTS: A total of 620 asthmatic children were divided into three groups: preschool (3-6 years old, n=360), school-aged (7-12 years old, n=213), and adolescent (13-18 years old, n=41) children. The atopic and non-atopic phenotypes were observed in 54.8% and 45.2% of the asthmatic children, respectively. The atopic phenotype was observed in 45.6%, 65.7%, and 80.5% of the preschool, school-aged and adolescent groups, respectively. The percentages of the atopic phenotype were significantly different when stratified by age group (p<0.001), and there was a positive trend of percentage distribution. The percentages of sensitization to aeroallergens were significantly different and observed in 44.0%, 65.7%, and 80.5% of the preschool, school-aged and adolescent groups, respectively (p<0.001). There were positive trends between age groups and prevalence rates of sensitization to the main aeroallergen and other aeroallergen groups, but not to each allergen of the seafood or other food allergen group. CONCLUSIONS: A trend of an increasing percentage of the atopic phenotype when stratified by age group was found in asthmatic children in southern Taiwan. Aeroallergens contributed more to pediatric asthma than food allergens. The prevalence of sensitization to aeroallergens increased with increasing age when stratified by age group.

Altered chemokine profile in Refractory Mycoplasma pneumoniae pneumonia infected children.

BACKGROUND: Mycoplasma pneumoniae is one of the major pathogens causing community-acquired pneumonia in children. Although usually self-limited, Mycoplasma pneumoniae pneumonia (MPP) may lead to complicated morbidity that can even be life-threatening. Upon MPP infection, alveolar macrophage becomes attracted and activated and will induce subsequent cytokine and chemokine reaction. Refractory Mycoplasma pneumoniae pneumonia (RMPP) is manifested by clinical or radiological deterioration despite proper antibiotic therapy. RMPP is characterized with excessive inflammation and may need subsequent glucocorticoid treatment. AIM: The aim of this study was to investigate the change of plasma chemokines in non-refractory Mycoplasma pneumoniae pneumonia (NRMPP) and RMPP before and after antibiotic or methylprednisolone treatment. METHOD: A total of 42 children with MPP were enrolled in this study. Plasma specimens were collected at admission and one to two weeks after antibiotic or methylprednisolone treatment with declined fever. Plasma specimens were then indicated to chemokines detection. RESULTS: Mycoplasma pneumoniae pneumonia altered the chemokine profile through the observation of decreased plasma M1 related chemokines (CCL2, CCL8 and CXCL10) and increased M2 related chemokines (CCL17 and CCL22) after treatment.When the patients were divided into RMPP and NRMPP groups and the chemokines before treatment were compared, the RMPP group showed higher CXCL10 but lower CCL3 and CCL11 than the NRMPP group. CONCLUSION: Unique changes in macrophage related chemokines is observed in the course of MPP infection. NRMPP and RMPP infection in children showed distinct manifestation in chemokine profiles.

Higher Hospitalization Rate for Lower Airway Infection in Transfusion-Naïve Thalassemia Children.

Few studies have addressed the risk of infection in transfusion-naïve thalassemia patients. We aimed to investigate whether transfusion-naïve thalassemia population has higher hospitalization rates for lower airway infection-related diseases than non-thalassemia population in children. A nationwide population-based retrospective cohort study was conducted using detailed medical records of the Taiwan National Health Insurance Research Database. Transfusion-naïve thalassemia patients were compared with a matched cohort at a ratio of 1:4. Data of the selected patients were adjusted for age, sex, and related comorbidities. We recorded the frequency of admissions or outpatient clinic visits for patients with a diagnosis of pneumonia or acute bronchitis/bronchiolitis. Based on our results, the hospitalization rates and incidence rate ratios of bronchitis/bronchiolitis and pneumonia for transfusion-naïve thalassemia children were all higher than those for non-thalassemia controls. Therefore, we conclude that transfusion-naïve thalassemia children are more likely to experience lower airway infections and have a higher probability of hospitalization for these conditions.

A Multifactorial Evaluation of the Effects of Air Pollution and Meteorological Factors on Asthma Exacerbation.

In the real world, dynamic changes in air pollutants and meteorological factors coexist simultaneously. Studies identifying the effects of individual pollutants on acute exacerbation (AE) of asthma may overlook the health effects of the overall combination. A comprehensive study examining the influence of air pollution and meteorological factors is required. Asthma AE data from emergency room visits were collected from the Taiwan National Health Insurance Research Database. Complete monitoring data for air pollutants (SO2; NO2; O3; CO; PM2.5; PM10) and meteorological factors were collected from the Environmental Protection Agency monitoring stations. A bi-directional case-crossover analysis was used to investigate the effects of air pollution and meteorological factors on asthma AE. Among age group divisions, a 1 °C temperature increase was a protective factor for asthma ER visits with OR = 0.981 (95% CI, 0.971-0.991) and 0.985 (95% CI, 0.975-0.994) for pediatric and adult patients, respectively. Children, especially younger females, are more susceptible to asthma AE due to the effects of outdoor air pollution than adults. Meteorological factors are important modulators for asthma AE in both asthmatic children and adults. When studying the effects of air pollution on asthma AE, meteorological factors should be considered.

Serum lactate dehydrogenase isoenzymes 4 plus 5 is a better biomarker than total lactate dehydrogenase for refractory Mycoplasma pneumoniae pneumonia in children.

Although usually self-limiting, Mycoplasma pneumoniae pneumonia (MPP) may lead to clinical or radiological deterioration despite macrolide antibiotic therapy, resulting in the development of refractory MPP (RMPP). Corticosteroids have been used to treat RMPP with beneficial effects. Serum lactate dehydrogenase (LDH) is a suggested biomarker for the use of steroid therapy. Since serum LDH is a non-specific marker and elevated in many inflammatory processes, this study investigates the predicting level of LDH isoenzymes for RMPP. Fifty-four children with non-refractory M. pneumoniae pneumonia (NRMPP) and 16 children with RMPP were enrolled in this study. In comparison to the NRMPP group, the RMPP group showed significantly higher levels of serum LDH. Concerning LDH isoenzymes, the RMPP group showed significantly lower proportions of LDH1 and LDH2, while higher LDH4 and LDH5 percentage. Receiver operating characteristic curve analysis showed that the area under the curve for the total LDH data was 0.812 with a cut-off of 408 IU/L (sensitivity of 75.0%, specificity of 72.2%). The areas under the curve for LDH4, LDH5, and [LDH4 + LDH5] were estimated to be 0.813, 0.818, and 0.829, respectively. The threshold for [LDH4 + LDH5] was estimated to be 109.4 IU/L (sensitivity, 75.0%; specificity, 87.0%). These results indicate that for the initiation of corticosteroid therapy, serum [LDH4 + LDH5] level is a more sensitive biomarker than total LDH.

Role of Serum Mycoplasma pneumoniae IgA, IgM, and IgG in the Diagnosis of Mycoplasma pneumoniae-Related Pneumonia in School-Age Children and Adolescents.

Mycoplasma pneumoniae is an important causative pathogen of community-acquired pneumonia in children. Rapid and reliable laboratory diagnosis of M. pneumoniae infection is important so that appropriate antibiotic treatment can be initiated to reduce the misuse of drugs and resistance rates. Anti-M. pneumoniae immunoglobulin M (IgM) is an indicator of recent primary infection but can persist for several months after initial infection. It has been suggested that anti-M. pneumoniae immunoglobulin A (IgA) can be a reliable indicator for recent M. pneumoniae infection in adults. We investigated the clinical diagnostic value of M. pneumoniae IgA in school-age children and adolescents with M. pneumoniae-related pneumonia. Eighty children with pneumonia and seropositive for M. pneumoniae IgM or with a 4-fold increase of anti-M. pneumoniae immunoglobulin G (IgG) were enrolled from May 2015 to March 2016. The titers of M. pneumoniae IgA, IgM, and IgG, the clinical features, and laboratory examinations of blood, C-reactive protein, and liver enzymes were analyzed. The initial positivity rates for M. pneumoniae IgM and IgA upon admission to the hospital were 63.6 and 33.8%, respectively. One week after admission, the cumulative positivity rates for M. pneumoniae IgM and IgA increased to 97.5 and 56.3%, respectively. Detection of M. pneumoniae IgM was more sensitive than detection of M. pneumoniae IgA for the diagnosis of M. pneumoniae-related pneumonia in school-age children and adolescents; however, paired sera are necessary for a more accurate diagnosis.

Diagnostic value of bronchoalveolar lavage in children with nonresponding community-acquired pneumonia.

BACKGROUND: Community-acquired pneumonia (CAP) is a common cause of morbidity and mortality in hospitalized children. In CAP, causative agents are seldom identified using noninvasive diagnostic procedures. For those children not responding to empiric antibiotic therapy, it is vital to identify the causative pathogens for further management. METHODS: We aimed to determine the usefulness of identifying the causative agents by bronchoalveolar lavage (BAL) in hospitalized children with nonresponding CAP. Ninety children hospitalized for CAP and treated with empiric antibiotics but having persistent fever ≥48 hours were enrolled, and their BAL data were retrospectively reviewed. RESULTS: Aerobic bacteria were isolated from 38 (42%) of 90 cultures, and anaerobic bacteria were isolated from eight (24%) of 33 cultures. The bacteria isolated most frequently were Streptococcus viridians (26.3%), Pseudomonas aeruginosa (23.7%), and Staphylococcus aureus (15.8%). Streptococcus pneumoniae was isolated from the BALs of only two children, and Haemophilus influenzae from none. For positive aerobic culture results, BAL results guided modifications of antibiotic regimens in 21 episodes (21 of 38, 55.3%). CONCLUSION: BAL results guided a change of antimicrobials in 55% of children with positive aerobic cultures (29% of all children in the study) and contributed to a high rate of successful therapy.

Efficacy of sublingual immunotherapy with high-dose mite extracts in asthma: a multi-center, double-blind, randomized, and placebo-controlled study in Taiwan.

Sublingual immunotherapy (SLIT) has been recommended as a viable alternative to subcutaneous injection therapy in the treatment of airway allergies, though more data is needed from well-controlled studies for documenting its efficacy in different ethnic populations. Ninety-seven children (age range 6-12 years), mild-to-moderate asthma with a single sensitization to mite allergen, were enrolled from 5 medical centers in Taiwan to evaluate the efficacy and safety of SLIT with standardized mite extracts, which contain Dermatophagoides pteronyssinus (D.p.) and Dermatophagoides farinae (D.f.). Patients were double blinded and randomly assigned to either a SLIT or placebo group. Following 24 weeks of study period, symptom and medication scores, lung function tests, skin prick tests, total serum IgE, and specific IgE to D.p. and D.f. were recorded. The results showed that there was statistically significant difference between these two groups in the analysis of daily (P=0.011), nighttime (P=0.028), and daytime (P=0.009) asthmatic scores after 24 weeks of treatment. Patients receiving SLIT improved their forced vital capacity (FVC), forced expiratory volume in 1s (FEV1), and peak expiratory flow (PEF) as compared to baseline (P=0.042, P=0.048, and P=0.001, respectively). No differences were found in skin prick test, total serum IgE and specific IgE to D.p. and D.f. Tolerance with high-dose SLIT was good with few minor adverse events reported. Our results indicated that a 24-week SLIT is of clinical benefit to mite-sensitive asthmatic children in Taiwan.

Complicated parapneumonic effusion and empyema in children.

BACKGROUND AND PURPOSE: Parapneumonic effusion and empyema are recognized complications of bacterial pneumonia. Optimal management in children, especially the duration of parenteral antibiotics and the role of surgery, is controversial. This study analyzed the clinical characteristics, management, outcome, and bacterial etiology of 59 patients with complicated parapneumonic effusion and empyema treated at a single medical center in Kaohsiung from January 1995 to March 2004. METHODS: The diagnosis of complicated parapneumonic effusion was based on the specific characteristics of pleural fluid, computed tomography or ultrasound findings, or direct visualization of loculations during the surgical procedure. RESULTS: Causative agents were culture-confirmed in 42% of the cases. Streptococcus pneumoniae was the leading pathogen in this series (20% of cases). None of the S. pneumoniae isolates were susceptible to penicillin. Mycoplasma pneumoniae accounted for 19% of cases based on immunoglobulin M assay. CONCLUSIONS: An initial combination therapy regimen consisting of cefotaxime or ceftriaxone plus macrolide provided reasonable activity against 80% of the pathogens isolated in this series. This study also revealed that prolonged parenteral antibiotic treatment resulted in longer length of hospital stay.

Clinical analysis of necrotizing pneumonia in children: three-year experience in a single medical center.

Necrotizing pneumonia (NP) is a rare but complex complication of lobar pneumonia in children. It is characterized by massive necrosis and liquification of lung tissues by the presence of multiple cavities rather than a solitary one. To evaluate the clinical features of NP, we enrolled 17 patients at Chang-Gung Memorial Hospital (Kaohsiung) from November 1999 to December 2002. The diagnosis of NP was based upon findings of chest computed tomography. There were six boys and 11 girls, ages ranging from 47 days to seven years. Streptococcus pneumoniae was the most common pathogen responsible for 9 cases in our study. Our patients remained febrile, even with the appropriate antibiotics administered, for a mean of 12.1 +/- 5.2 days and hospitalized for a mean of 22.7 +/- 7.0 days. The right lung was affected more than the left (12:7). On admission, laboratory data showed either leukocytosis, or normal leukocyte count, or leukopenia, but C-reactive protein was abnormally high (mean = 304.8 +/- 96.3 mg/L). Anemia and significant platelet count elevation developed at a mean period of 8.5 +/- 3.5 and 10.9 +/- 4.7 days after their hospitalization, respectively. Surgical intervention was performed in one case only in our series. Chest roentgenography took a mean of 60.3 +/- 15.6 days to resolve completely. Although the clinical course was prolonged, all patients with NP eventually recovered completely with no sequela.

Colistin inhalation monotherapy for ventilator-associated pneumonia of Acinetobacter baumannii in prematurity.

BACKGROUND: Ventilator-associated pneumonia (VAP) caused by Acinetobacter baumannii is increasing. It has a high mortality rate but experience in using inhaled colistin as monotherapy for VAP in children, especially pre-term infants, is limited. This study presents experiences using aerosolized colistin as monotherapy for VAP due to A. baumannii infection in pre-term infants. METHODS: Eight pre-term infants (gestational age 25-36 weeks) admitted to the neonatal intensive care unit (NICU) of Kaohsiung Chang Gung Memorial Hospital in Taiwan from January 2006 to December 2010 who received inhaled colistin as monotherapy for VAP due to A. baumannii infection were retrospectively evaluated. Of the isolated microorganisms, five were multi-drug resistant strains of A. baumannii (MDR-AB) but all were sensitive to colistin. All patients received inhaled colistin at a dose of 1,000,000 IU (33.4 mg) twice daily for an average of 9.1 days (range, 4-22 days). RESULTS: All pre-term infants were cured, with A. baumannii eradicated from airway secretions. There were no clinical or laboratory adverse events related to colistin use. CONCLUSIONS: Aerosolized colistin may be used as monotherapy for VAP due to A. baumannii infection in pre-term infants. A larger controlled study is warranted to corroborate the findings.

Usefulness of desaturation index for the assessment of obstructive sleep apnea syndrome in children.

OBJECTIVE: Obstructive sleep apnea syndrome (OSAS) can result in learning, behavioral, and even growth problems in children. Overnight polysomnography (PSG) is the gold standard for the diagnosis of OSAS, but this diagnostic technique is time consuming and inconvenient. It is well documented that patients with OSAS experience oxygen desaturation during sleep. The purpose of this study was to determine if the level of oxygen desaturation can be used to predict OSAS in children. METHODS: This retrospective study included 148 children aged from 3 to 12 years; all were snorers and were suspected of having OSAS. Objective PSG parameters, nocturnal pulse oximeter data and subjective modified Epworth Sleepiness Scale (ESS) scores were assessed. Mild, moderate, and severe OSAS was classified as apnea-hypopnea index (AHI)≥1 and <5, AHI≥5 and <10, and AHI≥10, respectively. RESULTS: Of the 148 children, 130 (87.8%) were diagnosed with OSAS. There was no correlation between the AHI and the modified ESS score (r = -0.048, p = 0.563). The desaturation index (DI) had the highest correlation with AHI (r = 0.886, p < 0.001). Using the DI cut-off values of 2.05 to predict OSAS in children had good positive predictive value of 98.1%. The optimal DI cut-off values for predicting the occurrence of mild, moderate, and severe OSAS were 2.05 (sensitivity: 77.7%; specificity: 88.9%), 3.50 (sensitivity: 83.8%; specificity: 86.5%) and 4.15 (sensitivity: 89.1%; specificity: 86.0%), respectively. CONCLUSIONS: These findings suggest that DI, as determined using a nocturnal pulse oximeter, may be a good tool for predicting both the presence and the severity of OSAS in children.

Comparison of the Global Initiative for Asthma guideline-based Asthma Control Measure and the Childhood Asthma Control Test in evaluating asthma control in children.

BACKGROUND: Reliable assessment of asthma control is essential for effective treatment. While several validated tools for assessing asthma control in children are currently available, few studies have evaluated the correlations between different asthma control measures in children. This study aimed to determine the correlations between the Childhood Asthma Control Test (C-ACT) and the Global Initiative for Asthma (GINA) guideline-based asthma control measure (ACM) with lung function parameters in children with asthma. METHODS: Sixty-three children aged 6-11 years with mild-intermittent to severe-persistent asthma were evaluated. They completed the C-ACT, the GINA guideline-based assessment and lung function tests with the help of their caregivers. RESULTS: C-ACT scores and GINA guideline-based ACM were positively correlated. The average C-ACT scores for children with controlled, partly controlled and uncontrolled asthma according to the GINA guidelines were 24.4±0.3, 22.8±0.6 and 21.3±1.0 (mean±SE), respectively. High C-ACT scores were also noted in children with uncontrolled asthma based on the GINA guidelines. The GINA guideline-based ACM was correlated with spot spirometry parameters (forced vital capacity, forced expiratory volume in 1 second, and maximal mid-expiratory flow). Conversely, the C-ACT score was not correlated with these spirometry parameters. CONCLUSIONS: The C-ACT may overestimate asthma control in certain circumstances. For children with poorly controlled asthma or poor symptom perception, more frequent visits and serial pulmonary function tests are recommended.

When parents opted not to perform surgery for a long-segment congenital tracheal stenosis child: flexible bronchoscopic balloon tracheoplasty as the primary treatment.

Congenital tracheal stenosis is a rare airway disorder characterized by a fixed tracheal narrowing. Surgical intervention is usually considered as the treatment of choice for long-segment type. However, due to wide spectrum of this disease, treatment modalities need to be individualized. The long-segment type has the most unfavorable outcome because it is often associated with multiple anomalies and, thus, the most difficult to manage. Here we present a case of long-segment congenital tracheal stenosis (LSCTS) that was managed by several sessions of flexible bronchoscopic balloon tracheoplasty as the primary treatment. In this particular situation, we had to deal with the parents' request of a 'non-surgical' approach for an infant who had respiratory distress that also required a way to increase his tracheal diameter simultaneously. Having done this particular experience, we would like to show that this non-surgical approach can be viable for long-segment tracheal stenosis in selected cases.

Tongue-lip adhesion in the management of Pierre Robin sequence with airway obstruction: technique and outcome.

BACKGROUND: Airway obstruction and feeding difficulty can occur in patients born with Pierre Robin sequence. In select patients with pronounced micrognathia, surgical intervention to relieve the airway obstruction is necessary. The surgical indications and appropriate surgical procedure continue to have a great deal of controversy. The purpose of this study was to evaluate our experience of tongue-lip adhesion in the management of upper airway obstruction associated with Pierre Robin sequence. METHODS: From March 1995 through May 2002, a total of 14 patients with Pierre Robin sequence, who were admitted to the pediatric neonatal intensive care unit either with prolong intubation, poor body weight gain, or repeated airway infection, underwent tongue-lip adhesion (TLA). The operation was performed by raising mucosa flaps and approximated the muscles between tongue and lower lip. Retention sutures were used. The patients were evaluated for clinical responses. RESULTS: Our successful rate with tongue-lip adhesion was 70%. Ten of the 14 patients showed clinical improvements including extubation of the endotraccheal tube, body weight gain, return for home care, reduced episodes of respiratory infection, and improvement in O2 saturation and blood gas. The surgical procedure was simple to perform without major complications. CONCLUSIONS: This retrospective review showed that with a thorough preoperative airway evaluation, TLA could be successfully used to treat select patients with Pierre Robin sequence suffering from severe upper airway obstruction. Thus, TLA should be first considered when surgical relief of airway obstruction is indicated and when obstruction is limited to the classic tongue base obstruction type.

Treatment of pediatric idiopathic pulmonary hemosiderosis with low-dose cyclophosphamide.

OBJECTIVE: To report the safety and efficacy of long-term, low-dose cyclophosphamide therapy in a child with idiopathic pulmonary hemosiderosis (IPH). CASE SUMMARY: A 7-year-old boy diagnosed with IPH 4 years previously was initially prescribed prednisolone. Because he only had a transient response to prednisolone, oral cyclophosphamide 2 mg/kg/d was later added. A dramatic improvement was noted during the subsequent follow-up. One year after cyclophosphamide therapy, the patient suddenly developed thrombocytopenia (platelet count 75 x 10(3)/mm(3)), with the platelet count decreasing to 10 x 10(3)/mm(3) over the following 10 months. Cyclophosphamide was tapered to an alternating daily dosage of 1 mg/kg. The tapering resulted in a subsequent increase in the platelet count, which was maintained between 20 and 50 x 10(3)/mm(3) without occurrence of petechiae or spontaneous bleeding. Under this reduced dosing regimen, the disease has remained in remission for >1 year. DISCUSSION: Due to the low prevalence of IPH, only limited data document the safety and efficacy of immunosuppressive therapy in treating this disease. Although our patient showed a good response to low-dose cyclophosphamide, he developed thrombocytopenia with its use. The mechanism is unclear, but it may be similar to that of high-dose cyclophosphamide-induced myelosuppression. Due to the development of thrombocytopenia, the use of cyclophosphamide was maintained under a reduced dosing regimen. The benefit of long-term immunosuppressive therapy is controversial, and more clinical evidence is required to support its continued usage. CONCLUSIONS: Long-term, low-dose cyclophosphamide is effective in treating childhood IPH, but caution should be exercised due to the possible development of thrombocytopenia. Periodic monitoring of the platelet count in long-term treatment is recommended.

Perirenal tailgut cyst (cystic hamartoma).

Tailgut cysts are rare congenital multicystic lesions preferentially occurring in the retrorectal space. They are assumed to arise from the remnant of embryonic tailgut and occasionally exhibit malignant transformation. The authors herein describe the clinical, radiologic, and pathologic features of an unusual perirenal tailgut cyst with an emphasis on its histogenesis and distinction from other intraabdominal cystic lesions.

Anemia as the sole presenting symptom of idiopathic pulmonary hemosiderosis: report of two cases.

Idiopathic pulmonary hemosiderosis (IPH) is a rare disease in children and has an unknown etiology. It is characterized by the triad of hemoptysis, pulmonary infiltrates on chest radiograph (CXR) and iron deficiency anemia. We report two young children, aged 3 and 4 years, were admitted due to pale-looking appearance but without hemoptysis or other respiratory symptoms. Pallor was the sole presenting feature in these 2 children with IPH and which was unusual. CXR obtained on admission led to the suspicion of pulmonary hemorrhage. The diagnosis of IPH was confirmed based on the presence of many hemosiderin-laden macrophages in bronchoalveolar lavage fluid obtained by flexible bronchoscopy. Steroid was initiated after the diagnosis of IPH was established; the both of them have been well and received regular follow-up in our outpatient department. IPH may not be diagnosed because of difficulty in diagnosis. Anemia may be the only presenting feature of IPH, which was due to occult pulmonary hemorrhage. Initial treatment with corticosteroids has been successful in our patients for a period of 6 and 8 months of follow up respectively.