Marshall University Joan C. Edwards School of Medicine Huntington, WV
Saba Altarawneh, MD, Yasmeen Obeidat, MD, Pramod Pantangi, MD, Yousef Shweihat, MD Marshall University Joan C. Edwards School of Medicine, Huntington, WV
Introduction: Idiopathic pulmonary hemosiderosis (IPH) is a rare cause of hemoptysis in children. The association of IPH with celiac disease is infrequent and has been described in the pediatric population as Lane-Hamilton syndrome (LAH). We present a case of an adult patient with suspected LAH that responded to a gluten-free diet.
Case Description/Methods: A 47-year-old male presented with cough, and shortness of breath, associated with abdominal bloating and diarrhea. Laboratory workup was significant for a low hemoglobin level. High-resolution computed tomography of the chest (HRCT) revealed a diffuse, non-specific inflammatory process with reactive hilar lymphadenopathy. The patient underwent bronchoscopy with endobronchial ultrasound (EBUS), and bronchoalveolar lavage (BAL) was performed; however, an attempt to obtain a biopsy was unsuccessful after the patient had oxygen desaturation during the procedure. BAL showed abundant hemosiderin-laden macrophages, three times more than normal. Tissue transglutaminase and gliadin-A antibodies were positive suggestive of celiac disease. Given the high suspicion of LAH, the patient was started on a gluten-free diet, and his symptoms, hemoglobin level, and CT imaging significantly improved after a 3-month follow-up.
Discussion: The combination of IPH and celiac disease, known as Lane-Hamilton syndrome, is extremely rare. The association between these two conditions remains unclear and is presumed immune-mediated. Very few cases have been reported in adults with limited literature on management. Screening for celiac disease is recommended for suspected IPH, and a gluten-free diet trial is suggested even in the adult population.
Disclosures:
Saba Altarawneh indicated no relevant financial relationships.
Yasmeen Obeidat indicated no relevant financial relationships.
Pramod Pantangi indicated no relevant financial relationships.
Yousef Shweihat indicated no relevant financial relationships.
Saba Altarawneh, MD, Yasmeen Obeidat, MD, Pramod Pantangi, MD, Yousef Shweihat, MD. B0678 - An Unusual Case of Alveolar Hemorrhage Responding to a Gluten-Free Diet, ACG 2022 Annual Scientific Meeting Abstracts. Charlotte, NC: American College of Gastroenterology.