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Published in: Multidisciplinary Respiratory Medicine 1/2013

Open Access 01-12-2013 | Case report

Hemoptysis in patients of celiac disease with disproportionately severe anemia: tip of the iceberg?

Authors: Kamal Kumar Singhal, Ashok K Janmeja, Rakhee Sodhi, Rajpal S Punia

Published in: Multidisciplinary Respiratory Medicine | Issue 1/2013

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Abstract

Idiopathic Pulmonary Hemosiderosis (IPH) is characterized by the triad of iron deficiency anemia, pulmonary infiltrates and haemoptysis with no recognizable cause. Since the first description of its association with Celiac Disease (CD) by Lane and Hamilton in 1971, only a few isolated cases have been reported in literature. Although it has been considered an uncommon association of two disease entities, recent reports indicate that prevalence of celiac disease is as high as one percent. Further, individually both celiac disease and IPH are known to present as refractory anemia only. We are reporting a young adult with Lane Hamilton Syndrome, who realized that he was having significant gastrointestinal complaints only when they disappeared on gluten free diet (GFD). This case report reiterates the fact that celiac disease should be considered in all patients of IPH because of the therapeutic implications. Further on review of literature, we believe that covert hemoptysis may be responsible for disproportionately severe anemia in patients of celiac disease. Thus, prevalence of this association may be more than currently believed. Further research in this regard may improve our understanding of pathogenesis of celiac disease.
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Literature
1.
go back to reference Lane DJ: Idiopathic steatorrhoea and idiopathic pulmonary haemosiderosis. Br Med J. 1971, 10 (2(5753)): 89-90.CrossRef Lane DJ: Idiopathic steatorrhoea and idiopathic pulmonary haemosiderosis. Br Med J. 1971, 10 (2(5753)): 89-90.CrossRef
2.
go back to reference Agarwal R, Aggarwal AN, Gupta D: Lane-Hamilton syndrome: simultaneous occurrence of celiac disease and idiopathic pulmonary haemosiderosis. Intern Med J. 2007, 37 (1): 65-67. 10.1111/j.1445-5994.2006.01226.x.CrossRefPubMed Agarwal R, Aggarwal AN, Gupta D: Lane-Hamilton syndrome: simultaneous occurrence of celiac disease and idiopathic pulmonary haemosiderosis. Intern Med J. 2007, 37 (1): 65-67. 10.1111/j.1445-5994.2006.01226.x.CrossRefPubMed
3.
go back to reference Paksu S, Paksu MS, Kalayci AG, Sancak R: Lane-Hamilton syndrome: association or coincidence. Indian Pediatr. 2012, 49 (3): 243-244.PubMed Paksu S, Paksu MS, Kalayci AG, Sancak R: Lane-Hamilton syndrome: association or coincidence. Indian Pediatr. 2012, 49 (3): 243-244.PubMed
4.
go back to reference Grover PJ, Jayaram R, Madder H: Management of cerebral venous thrombosis in a patient with Lane-Hamilton syndrome and coeliac disease, epilepsy and cerebral calcification syndrome. Br J Neurosurg. 2010, 24 (6): 684-685. 10.3109/02688697.2010.500412.CrossRefPubMed Grover PJ, Jayaram R, Madder H: Management of cerebral venous thrombosis in a patient with Lane-Hamilton syndrome and coeliac disease, epilepsy and cerebral calcification syndrome. Br J Neurosurg. 2010, 24 (6): 684-685. 10.3109/02688697.2010.500412.CrossRefPubMed
5.
go back to reference Hendrickx GF, Somers K, Vandenplas Y: Lane-Hamilton syndrome: case report and review of the literature. Eur J Pediatr. 2011, 170 (12): 1597-1602. 10.1007/s00431-011-1568-5.CrossRefPubMed Hendrickx GF, Somers K, Vandenplas Y: Lane-Hamilton syndrome: case report and review of the literature. Eur J Pediatr. 2011, 170 (12): 1597-1602. 10.1007/s00431-011-1568-5.CrossRefPubMed
6.
go back to reference Keskin O, Keskin M, Guler E, Tutar E, Saygili O, Kucukosmanoglu E, Kor Y, Celik H, Coskun E: Unusual presentation: pulmonary hemosiderosis with celiac disease and retinitis pigmentosa in a child. Pediatr Pulmonol. 2011, 46 (8): 820-823. 10.1002/ppul.21420.CrossRefPubMed Keskin O, Keskin M, Guler E, Tutar E, Saygili O, Kucukosmanoglu E, Kor Y, Celik H, Coskun E: Unusual presentation: pulmonary hemosiderosis with celiac disease and retinitis pigmentosa in a child. Pediatr Pulmonol. 2011, 46 (8): 820-823. 10.1002/ppul.21420.CrossRefPubMed
7.
go back to reference Sethi GR, Singhal KK: Pulmonary diseases and corticosteroids. Indian J Pediatr. 2008, 75 (10): 1045-1056. 10.1007/s12098-008-0209-0.CrossRefPubMed Sethi GR, Singhal KK: Pulmonary diseases and corticosteroids. Indian J Pediatr. 2008, 75 (10): 1045-1056. 10.1007/s12098-008-0209-0.CrossRefPubMed
8.
go back to reference Najada AS, Dahabreh MM: Pulmonary haemosiderosis in a 13-year-old girl with coeliac disease after 3 months on a gluten-free diet: case report and review of the literature. Ann Trop Paediatr. 2010, 30 (3): 249-253. 10.1179/146532810X12786388978841.CrossRefPubMed Najada AS, Dahabreh MM: Pulmonary haemosiderosis in a 13-year-old girl with coeliac disease after 3 months on a gluten-free diet: case report and review of the literature. Ann Trop Paediatr. 2010, 30 (3): 249-253. 10.1179/146532810X12786388978841.CrossRefPubMed
9.
go back to reference Nishino M, Patrick JL, Connors JM: Case 155: lane-Hamilton syndrome. Radiology. 2010, 254 (3): 985-988. 10.1148/radiol.09082062.CrossRefPubMed Nishino M, Patrick JL, Connors JM: Case 155: lane-Hamilton syndrome. Radiology. 2010, 254 (3): 985-988. 10.1148/radiol.09082062.CrossRefPubMed
10.
go back to reference Narula N, Rawal P, Kumar RM, Ram Thapa B: Association of celiac disease with cardiomyopathy and pulmonary hemosiderosis. J Trop Pediatr. 2010, 56 (3): 201-203. 10.1093/tropej/fmp088.CrossRefPubMed Narula N, Rawal P, Kumar RM, Ram Thapa B: Association of celiac disease with cardiomyopathy and pulmonary hemosiderosis. J Trop Pediatr. 2010, 56 (3): 201-203. 10.1093/tropej/fmp088.CrossRefPubMed
11.
go back to reference Hammami S, GhédiraBesbès L, Hadded S, Chouchane S, Ben Meriem C, Gueddiche MN: Co-occurrence pulmonary haemosiderosis with coeliac disease in child. Respir Med. 2008, 102 (6): 935-936. 10.1016/j.rmed.2008.02.014.CrossRefPubMed Hammami S, GhédiraBesbès L, Hadded S, Chouchane S, Ben Meriem C, Gueddiche MN: Co-occurrence pulmonary haemosiderosis with coeliac disease in child. Respir Med. 2008, 102 (6): 935-936. 10.1016/j.rmed.2008.02.014.CrossRefPubMed
12.
go back to reference Mayes DH, Guerrero ML: A few good men: a Marine with hemoptysis and diarrhea. Idiopathic pulmonary hemosiderosis and celiac sprue. Chest. 2008, 134 (3): 644-647. 10.1378/chest.07-2834.CrossRefPubMed Mayes DH, Guerrero ML: A few good men: a Marine with hemoptysis and diarrhea. Idiopathic pulmonary hemosiderosis and celiac sprue. Chest. 2008, 134 (3): 644-647. 10.1378/chest.07-2834.CrossRefPubMed
13.
go back to reference Khemiri M, Ouederni M, Khaldi F, Barsaoui S: Screening for celiac disease in idiopathic pulmonary hemosiderosis. Gastroenterol Clin Biol. 2008, 32 (8–9): 745-748.CrossRefPubMed Khemiri M, Ouederni M, Khaldi F, Barsaoui S: Screening for celiac disease in idiopathic pulmonary hemosiderosis. Gastroenterol Clin Biol. 2008, 32 (8–9): 745-748.CrossRefPubMed
14.
go back to reference Hoca NT, Dayioglu D, Ogretensoy M: Pulmonary hemosiderosis in association with celiac disease. Lung. 2006, 184 (5): 297-300. 10.1007/s00408-005-2596-6.CrossRefPubMed Hoca NT, Dayioglu D, Ogretensoy M: Pulmonary hemosiderosis in association with celiac disease. Lung. 2006, 184 (5): 297-300. 10.1007/s00408-005-2596-6.CrossRefPubMed
16.
go back to reference Pacheco A, Casanova C, Fogue L, Sueiro A: Long-term clinical follow-up of adult idiopathic pulmonary hemosiderosis and celiac disease. Chest. 1991, 99 (6): 1525-1526. 10.1378/chest.99.6.1525.CrossRefPubMed Pacheco A, Casanova C, Fogue L, Sueiro A: Long-term clinical follow-up of adult idiopathic pulmonary hemosiderosis and celiac disease. Chest. 1991, 99 (6): 1525-1526. 10.1378/chest.99.6.1525.CrossRefPubMed
17.
go back to reference Zamani F, Mohamadnejad M, Shakeri R, Amiri A, Najafi S, Alimohamadi SM, Tavangar SM, Ghavamzadeh A, Malekzadeh R: Gluten sensitive enteropathy in patients with iron deficiency anemia of unknown origin. World J Gastroenterol. 2008, 14 (48): 7381-7385. 10.3748/wjg.14.7381.PubMedCentralCrossRefPubMed Zamani F, Mohamadnejad M, Shakeri R, Amiri A, Najafi S, Alimohamadi SM, Tavangar SM, Ghavamzadeh A, Malekzadeh R: Gluten sensitive enteropathy in patients with iron deficiency anemia of unknown origin. World J Gastroenterol. 2008, 14 (48): 7381-7385. 10.3748/wjg.14.7381.PubMedCentralCrossRefPubMed
18.
go back to reference Unsworth DJ, Lock RJ, Harvey RF: Improving the diagnosis of coeliac disease in anaemic women. Br J Haematol. 2000, 111 (3): 898-901. 10.1046/j.1365-2141.2000.02451.x.PubMed Unsworth DJ, Lock RJ, Harvey RF: Improving the diagnosis of coeliac disease in anaemic women. Br J Haematol. 2000, 111 (3): 898-901. 10.1046/j.1365-2141.2000.02451.x.PubMed
19.
go back to reference Çekın AH, Çekın Y, Sezer C: Celiac disease prevalence in patients with iron deficiency anemia. Turk J Gastroenterol. 2012, 23 (5): 490-495.PubMed Çekın AH, Çekın Y, Sezer C: Celiac disease prevalence in patients with iron deficiency anemia. Turk J Gastroenterol. 2012, 23 (5): 490-495.PubMed
20.
go back to reference Uçardağ D, Güliter S, Ceneli O, Yakaryilmaz F, Atasoy P, Cağlayan O: Celiac disease prevalence in patients with iron deficiency anemia of obscure origin. Turk J Gastroenterol. 2009, 20 (4): 266-270.PubMed Uçardağ D, Güliter S, Ceneli O, Yakaryilmaz F, Atasoy P, Cağlayan O: Celiac disease prevalence in patients with iron deficiency anemia of obscure origin. Turk J Gastroenterol. 2009, 20 (4): 266-270.PubMed
Metadata
Title
Hemoptysis in patients of celiac disease with disproportionately severe anemia: tip of the iceberg?
Authors
Kamal Kumar Singhal
Ashok K Janmeja
Rakhee Sodhi
Rajpal S Punia
Publication date
01-12-2013
Publisher
BioMed Central
Published in
Multidisciplinary Respiratory Medicine / Issue 1/2013
Electronic ISSN: 2049-6958
DOI
https://doi.org/10.1186/2049-6958-8-25

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