Lane Hamilton Syndrome without gastrointestinal symptoms

Abstract

Objective: Lane-Hamilton syndrome is a rare disease which is coexistence of celiac disease and idiopathic pulmonary hemorrhage. Treatment is based on gluten free diet and immunosuppressive agents however there are no definitive recommendation for the treatment. Case presentation: A 14 year-old boy patient admitted to emergency department with fatigue and minor hemoptysis for 2 months. His hemoglobin was 3.7 g/dl and 2 units of erithrocyte transfusion were given. He had no fever, abdominal pain, nausea, vomiting, diarrhea. Physical examination was normal. There were bilateral interstitial infiltration in lower zones on chest x-ray and common ground-glass opacities were present on Thorax CT. Acute phase reactants, bleeding parametries, immunoglobulins, autoantibodies, serologic and microbiologic tests for tuberculosis, echocardiography were normal. Hemosiderin laden macrophage was present in fasting gastric lavage. Celiac antibodies were positive, endoscopy was performed and cracked mud appearance was present. Duodenum biopsy was compatible with celiac disease. Gluten free diet and methylprednisolone treatment were started. Hemoptysis resolved and hemoglobin increased to 10 g/dl on the second month of the treatment and after then steroid was tapered by weekly. Discussion: Lane-Hamilton syndrome is very rare and it should be kept in mind in patients with pulmonary hemorrhage even without gastrointestinal symptoms. We gave gluten free diet and steroid to our patient because of active hemoptysis and respiratory symptoms and he was improved with the treatment. Although steroid treatment is controversial in the literature, it should be used in patients with active respiratory symptoms.