Ahmet ERDİL, Yüksel ATEŞ, Kemalettin YILMAZ, Önder ÖNGÖRÜ, DENİZ DOĞAN, Kemal DAĞALP

İdiopatik pulmoner hemosiderozis ile çölyak hastalığı birlikteliği: Olgu sunumu

İdiopatik pulmoner hemosiderosis tekrarlayan alveolar hemoraji, hemoptizi ve demir eksikliği anemisi ile birlikte seyreden ve nedeni bilinmeyen nadir bir hastalıktır. Hastalık genellikle çocuklarda bulunmasına rağmen, seyrekte olsa erişkinlerde de tespit edilebilmektedir. İdiopatik pulmoner hemosiderosis ile çölyak hastalığının birlikte bulunması da oldukça seyrek görülen bir durumdur. Bu vakada derin anemi ve tekrarlayan hemoptizileri olan 21 yaşındaki bir erkek hastada idiopatik pulmoner hemosiderosis ile birlikte çölyak hastalığı birlikteliği tespit edildi. Hastaya glutensiz diyet verildikten 6 ay sonraki kontrolünde hemoptizisi düzelmişti. Altı ayın sonunda hemoglobini 13.4 g/dl'ye yükseldi ve anti-endomisyal antikor seviyeleri 63,204 IU/ml'ye geriledi.

Anahtar Kelimeler:

İdiopatik pulmoner hemosiderosis, çölyak hastalığı

The combination of idiopathic pulmonary hemosiderosis and celiac disease: Case report

Idiopathic pulmonary hemosiderosis is a rare disease of unknown etiology usually characterized by recurrent episodes of alveolar hemorrhage, hemoptysis and iron deficiency anemia. It occurs most frequently in children but rarely in adults. The combination of idiopathic pulmonary hemosiderosis and celiac disease is also extremely rare. A 21-year-old man with history of recurrent hemoptysis and anemia was diagnosed with the combination of idiopathic pulmonary hemosiderosis and celiac disease. The patient was started on a gluten-free diet and has had no recurrences of hemoptysis over 6 months' follow-up. Hemoglobin at 6 months' follow up was 13.4 g/dL, while antiendomysial antibody titers had declined to 63,204 IU/ml.

Keywords:

Idiopathic pulmonary hemosiderosis, celiac disease,

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Milman N, Pedersen FM. Idiopathic pulmonary haemosiderosis. Epidemiology, pathogenic aspects and diagnosis. Respir Med 1998; 92: 902-7.
Maalej S, Drira I, Fennira H, ve ark. Idiopathic pulmonary hemosi- derosis in adults. Rev Pneumol Clin 2005; 61: 109-11.
Ferrari GF, Fioretto JR, Alves AF, Brandao GS. Idiopathic pulmo- nary hemosiderosis: case report. J Pediatr 2000; 76: 149-52.
Ioachimescu OC, Sieber S, Kotch A. Idiopathic pulmonary hemosi- derosis revisited. Eur Respir J 2004; 24: 162-70.
Farrell RJ, Kelly CP, Diagnosis of Celiac Sprue. Am J Gastroente- rol 2001; 96: 3237-46.
Abdulkarim AS, Murray JA. Review article: The diagnosis of coeli- ac disease. Aliment Pharmacol Ther 2003; 17: 987-95.
Malthora P, Aggarwal R, Aggarwal AN, ve ark. Coeliac disease as a cause of unusually severe anaemia in a young man with idiopat- hic pulmonary haemosiderosis. Resp Med 2005; 99: 451-3.
Bouros D, Panagou P, Rokkas T, Siafakas NM. Bronchoalveolar la- vage findings in a young adult with idiopathic pulmonary haemosi- derosis and coeliac disease. Eur Resp J 1994; 7: 1009-12.
Perelman S, Dupuy C, Bourrillon A. The association of pulmonary hemosiderosis and celiac disease. Apropos of a new case in a child. Ann Pediatr 1992; 39: 185-8.
Wright PH, Menzies IS, Pounder RE, Keeling PW. Adult idiopathic pulmonary haemosiderosis and celiac disease. Q J Med 1981; 50: 95-102.
Reading R, Watson JG, Plat JW, Brid AG. Pulmonary haemosidero- sis and gluten Arch Dis Child 1987; 62: 513-5.
Pacheco A, Casanova C, Fogue L, Sueiro A. Long-term clinical fol- low-up of adult idiopathic haemosiderosis and celiac disease. Chest 1991; 99: 1525-6.
Drut R, Rua EC. The histopathology of pediatric celiac disease: or- der must prevail out of chaos. Int J Surg Pathol 2001; 9: 261-4.