A Pediatric Case of Graves' Hyperthyroidism with Associated Glucose Intolerance Detected by a Urine Glucose Screening Program at School.

We report here a 13-year-old female with Graves' disease, whose diagnostic clue was glycosuria, which was detected by a urine glucose screening program at school. She had had mild general malaise, and a physical examination revealed a slightly enlarged thyroid gland. Hyperthyroidism (thyroid-stimulating hormone (TSH) < 0.01 µU/ml, free triiodothyronine (fT3) 23.57 pg/ml, free thyroxine (fT4) 3.38 ng/dl) and anti-thyroid autoantibodies (TRAb 43.6%) were detected in laboratory tests, and her plasma glucose at 120 minutes was 142 mg/dl in a 75 g oral glucose tolerance test. She was diagnosed as having borderline diabetes. These findings revealed a diagnosis of Graves' hyperthyroidism with associated impaired glucose tolerance. Although it is reported that many adults with hyperthyroidism develop disorders of glucose metabolism, pediatric patients rarely have complications of glucose intolerance or diabetes mellitus, and there are no previous reports of Graves' disease diagnosed by a urine glucose screening program at school. This case suggests a possibility of abnormalities in glucose metabolism even in pediatric cases of Graves' disease. To avoid overlooking the diagnosis of glucose intolerance associated with hyperthyroidism, a careful medical interview and examination should be performed even if the clinical features are mild.

[Evaluation of Clinical Features and Growth Hormone Deficiency in Short Children Born Small For Gestational Age].

Growth hormone (GH) therapy for short children born small for gestational age (SGA) has been approved in Japan. It is important to evaluate GH secretion ability before the initiation of GH therapy because there are some differences in dose and medical expenses between short children born SGA and GH deficiency (GHD). This study was designed to elucidate the incidence of GHD and to find a useful marker for detecting it in short SGA children. We retrospectively reviewed medical records to analyze the clinical features of short children born SGA and with GHD who had started GH therapy before the age of 6 in the University Hospital of Occupational and Environmental Health and Kyushu Rousai Hospital. Nine of 22 SGA subjects (41%) had GHD. There were no significant differences between two groups of short SGA children (GHD, non-GHD) in the median of height and serum insulin-like growth factors (IGF)-1 levels at birth or at the start of GH therapy. The probability of GHD was higher if the height standard deviation scores (SD) of the SGA children were lower than -3.2 (odds ratio, 11.6; 95% confidence interval, 1.52 - 89.1, P = 0.013). This study showed that there is an approximately 40% incidence of GHD in short SGA children needing GH treatment. We should do GH stimulation tests for short SGA children whose height SD is lower than -3 to determine the appropriate GH therapy.

Intralobar sequestration with tuberculous infection confined to the sequestrated lung.

A 22-year-old female was referred to the hospital suffering from a persistent, non-productive cough and repeated exacerbations of a right lower zone infiltrate suggestive of pulmonary sequestration. Angiography revealed an aberrant artery that originated from the left side of the descending aorta, crossed the aorta anteriorly, flowed through the right lower lobe and drained into the right inferior pulmonary vein. Right lower lobectomy was conducted by open thoracotomy. Gross examination revealed mucoid impaction of bronchi in the sequestration. Microscopy demonstrated a clear boundary between sequestrated and normal lung, as well as caseating epitheloid granulomas confined to the sequestration. As the aberrant artery fed both sequestrated and adjacent normal lung and the boundary did not involve the pleura, the lesion was classified as a Pryce's type II intralobar sequestration. No acid-fast bacilli were observed, but homogenates of the sequestration were positive for Mycobacterium tuberculosis by polymerase chain reaction. The patient was treated with isoniazid and rifampicin daily for 9 months, and 6 years later her clinical status remained excellent. Tuberculosis confined to a sequestration is extremely rare and characteristics of the aberrant artery suggested this intralobar sequestration was likely to be congenital in origin.

Decrease of interleukin-10-producing T cells in the peripheral blood of severe unstable atopic asthmatics.

BACKGROUND: Although IL-10 is known as an immunoregulatory cytokine produced by various cells including T cells, its basic profile in atopic asthma remains uncertain. OBJECTIVE: The profiles of IL-10 production in circulating CD4+ T cells of atopic asthmatics were investigated with respect to clinical severity. METHODS: Forty atopic asthmatics were divided into three groups: mild, and severe but stable and severe unstable asthmatics. Eosinophils were counted in the peripheral blood and sputum, and exhaled nitric oxide was assessed. PBMCs were stimulated with or without anti-CD3 and anti-CD28 antibodies and then processed for detecting IL-10-producing CD4+ cells using flow cytometry. RESULTS: There was no difference in the eosinophil count in blood or sputum and in nitric oxide level among the three groups. IL-10-producing CD4+ cells were mainly detected in a CD45RO+ memory population. The frequency of IL-10-producing cells after stimulation was significantly lower in the severe unstable group compared to the mild group. In addition, the frequency of IL-10-producing cells in the severe unstable group was significantly lower than that in the severe stable group despite the fact that both groups received similar treatments with high-dose inhaled corticosteroids. The IL-10 production of CD4+CD45RO+ cells in response to dexamethasone did not differ among the three groups. CONCLUSIONS: IL-10-producing CD4+CD45RO+ cells in the peripheral blood are decreased in severe unstable asthmatics, which is not explained by the effect of high-dose inhaled corticosteroid medication.

[A case of relapsing polychondritis successfully assessed for airway diameters under application of bi-level positive airway pressure (bilevel PAP)].

A 52-year-old man was admitted to our department because of hypercapnic respiratory failure. His illness had begun with scleritis 5 years before, followed by swelling of the fingers and auricular cartilages and saddle nose 3 years before, when a clinical diagnosis of relapsing polychondritis was made. His chest CT after intubation revealed diffuse airway narrowing with complete collapse of both main bronchi on expiration. Though his clinical status was stabilized after initiation of mechanical ventilation and pulse-dose corticosteroid treatment, he was difficult to wean from the ventilator with a conventional on-off trial. We therefore assessed the diameters of the central airways using dynamic CT under application of bilevel PAP, which confirmed an improvement in airway patency. He was then successfully weaned from artificial ventilation using bilevel PAP, initially through tracheotomy and subsequently via a nasal mask. To our knowledge, this is the first report that confirms the advantage of bilevel PAP objectively for a patient with labile airways.

Differential regulation by glucocorticoid of interleukin-13-induced eosinophilia, hyperresponsiveness, and goblet cell hyperplasia in mouse airways.

Interleukin (IL)-13 induces important features of bronchial asthma such as eosinophilic infiltration, airway hyperresponsiveness (AHR), and mucus hypersecretion. Although glucocorticoids suppress airway inflammation and remain the most effective therapy for asthma, the effects of glucocorticoids on the IL-13-dependent features are unknown. We studied the effects of dexamethasone on eotaxin production, eosinophil accumulation, goblet cell hyperplasia, and AHR after IL-13 administration into the airways of mice in vivo. MUC5AC gene expression, a marker of goblet cell hyperplasia, was also analyzed. IL-13 alone dose dependently induced AHR. Treatment with dexamethasone inhibited eotaxin expression and completely abolished eosinophil accumulation, but it did not affect AHR, MUC5AC overexpression, or goblet cell hyperplasia induced by IL-13. The effects of tumor necrosis factor-alpha on IL-13-induced AHR were also examined. Tumor necrosis factor-alpha did not affect AHR despite marked enhancement of eosinophil infiltration in IL-13-treated mice. These findings suggest that glucocorticoid is not sufficient to suppress IL-13-induced AHR or goblet cell hyperplasia and that eotaxin expression and eosinophilic inflammation do not have a causal relationship to the induction of AHR or goblet cell hyperplasia by IL-13. Control of steroid-resistant features induced by IL-13, including AHR and mucus production, may provide new therapeutic modalities for asthma.

[A case report of intralobar sequestration with a Mycobacterium tuberculosis infection limited to the sequestrated lung].

A 22-year-old woman was referred to our hospital suffering from repeated exacerbation of infiltrates confined to the medial portion of the right lower lung lobe, suggestive of pulmonary sequestration. Angiography revealed an anomalous vessel originating from the left side of the descending aorta, flowing behind it into the right lower lobe, the blood being returned to the pulmonary veins. A resection of the right lower lobe was conducted via thoracotomy. However, since there was no clear normal lung-pleura interface, this was classified as Pryce II type intralobar sequestration. Epithelioid granulomas with associated caseation necrosis confined to the sequestrated lung were confirmed by a polymerase chain reaction as Mycobacterium tuberculosis. Following oral antituberculosis drug administration, the patient recovered uneventfully. Superimposition of tuberculosis confined to a sequestrated lung portion is extremely rare. The presence of the aberrant artery led us to conclude that the intralobar sequestration must have been congenital.