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KIYAN, GÜRSU

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    Early pulmonary involvement in Niemann-Pick type B disease: Lung lavage is not useful
    (WILEY, 2005) KARADAĞ, BÜLENT TANER; Uyan, ZS; Karadag, B; Ersu, R; Kiyan, G; Kotiloglu, E; Sirvanci, S; Ercan, F; Dagli, T; Karakoc, F; Dagli, E
    Niemann-Pick disease (NPD) is a rare, autosomal-recessively inherited lipid storage disease which is characterized by intracellular deposition of sphingomyelin in various body tissues. The disease is heterogeneous and classified into six groups. Pulmonary parenchymal involvement may be a feature of several subtypes of NPD, including type B. Progressive pulmonary involvement in NPD type B is a major cause of morbidity and mortality It is usually diagnosed at older ages. Only a few cases with early pulmonary involvement have been reported. In this report, a patient with NPD type B, hospitalized with the diagnosis of pneumonia at age 3 months, is presented. Following treatment for pneumonia, she continued to have persistent respiratory symptoms and became oxygen-dependent. High-resolution computed tomography of the chest revealed diffuse interstitial changes. During follow-up, the patient developed hepatosplenomegaly. Lung, liver, and bone marrow biopsies showed characteristic findings for NPD. Biochemical studies also confirmed the diagnosis, and the sphingomyelinase enzyme level of the patient was low. Unilateral lung lavage was performed in order to decrease lipid storage as a treatment modality However, there was no clinical or radiological improvement. The patient died at age 15 months due to progressive respiratory failure. Pulmonary involvement is a rare entity in early childhood in patients with NPD type B, but should be considered in the differential diagnosis of persistent interstitial lung disease. It may cause progressive respiratory failure, but the treatment options remain limited.
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    An unusual case of chylothorax complicating childhood tuberculosis
    (WILEY, 2008) KARADAĞ, BÜLENT TANER; Cakir, Erkan; Gocmen, Basar; Uyan, Zeynep Seda; Oktem, Sedat; Kiyan, Gursu; Karakoc, Fazilet; Ersu, Refika; Karadag, Bulent; Dagli, Tolga; Dagli, Elif
    Endobronchial tuberculosis (EBTB) and chylothorax are rare clinical disorders. The concurrence of these two disorders as manifestations of childhood pulmonary tuberculosis has not been reported. We report a 4-month-old boy presenting with chylothorax as the initial presentation of tuberculosis that has been successfully treated with octreotide, antituberculosis drugs and steroid therapy.
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    Late diagnosis of foreign body aspiration in children with chronic respiratory symptoms
    (ELSEVIER IRELAND LTD, 2007) KARADAĞ, BÜLENT TANER; Karakoc, F.; Cakir, E.; Ersu, R.; Uyan, Z. S.; Colak, B.; Karadag, B.; Kiyan, G.; Dagli, T.; Dagli, E.
    Background: Children with undiagnosed and retained foreign bodies (FBs) may present with persistent respiratory symptoms. Delayed diagnosis is an important problem in developing countries and several factors affect the delay. Objectives: To investigate, the incidence of clinically unsuspected foreign body aspiration (FBA) in our flexible bronchoscopy procedures, the causes resulting in late diagnosis of FBA, and the incidence of the complications of FBA according to elapsed time between aspiration and diagnosis. Methods: We reviewed the records of all the patients who underwent flexible bronchoscopy between 1997 and 2004 in our clinic. Patients with FBA were identified and their medical records were reviewed. Results: During the study period, 654 children underwent flexible bronchoscopy; 32 cases (4.8%) of FBA were identified. Median age of patients was 29.5 months at presentation with a median symptomatic period of 3 months. None of the patients had a history of FBA. The most common misdiagnosis was bronchitis. Flexible bronchoscopy was performed to these patients within 1 week following presentation. In 87% of the patients (n = 28), FBs were in organic nature. Patients were followed up for 21.0 months after removal of the FBs. Fifty-three percent (n = 17) of the patients had a complete remission after bronchoscopic removal of the FBs. However, nine (28.8%) patients had chronic respiratory problems and six patients (18.8%) developed bronchiectasis. Conclusions: Atypical or prolonged respiratory symptoms should alert the physician and clinical and radiological findings should be carefully evaluated for a possible FBA. Delay in diagnosis and treatment of FBA should be avoided to prevent complications. (C) 2006 Elsevier Ireland Ltd. All rights reserved.