Abstract
Celiac disease (CD) was diagnosed in a 10-year-old boy who was presented with refractory iron deficiency anemia (IDA), hypertransaminasemia and short stature. Anemia resolved within one month after the institution of gluten-free diet. We performed liver biopsy because of hypergamaglobulinemia and high titers of smooth muscle antibody. On the basis of histologic findings together with clinical and laboratory findings, type 1 autoimmune hepatitis (AIH) was diagnosed. We aimed to report a CD case displaying more than one type of atypical pictures. Concomitant AIH and unusual rapid response of hepatitis to conventional treatment are also emphasized. Turkish Başlık: Atipik Çölyak Hastalığı ve Eşlik Eden Otoimmün Hepatit Anahtar Kelimeler: Çölyak hastalığı; glutensiz diyet; otoimmün hepatit; çocuklar Burada tedaviye dirençli demir eksikliği, transaminaz yüksekliği ve boy kısalığı ile başvuran, çölyak serolojisi pozitif saptanan ve ince bağırsak biyopsisi ile çölyak hastalığı tanısı alan 10 yaşında olgu sunuldu. Anemi sadece glutensiz diyet ile bir ay içerisinde düzeldi. Başvuru anında hipergamaglobülinemi ve anti-düz kas antikor pozitifliği nedeniyle yapılan karaciğer biyopsisinde portal lenfoplazmositer infiltrasyon saptandı. Histolojik, klinik ve laboratuar bulgular ile otoimmün hepatit tanısı aldı ve immünsüpresif tedavi başlandı. Olgu, atipik prezentasyonun yanında eşlik eden otoimmün hepatitin glutensiz diyet altında immunsüpresif tedaviye alışılagelmişin dışında hızlı yanıt vermesi nedeniyle sunuma değer bulunmuştur.
References
- Mäki M, Kallonen K, Lähdeaho ML, Visakorpi JK. Changing pattern of childhood coeliac disease in Finland. Acta Paediatr Scand 1988;77:408-12.
- Logan RF, Tucker G, Rifkind EA, Heading RC, Ferguson A. Changes in clinical features of coeliac disease in adults in Edinburgh and the Lothians 1960-79. Br Med J 1983;286:95-7.
- Ceccarelli M, Caiulo VA, Cortigiani L, Pucci C, Lupetti L, Ughi C. Clinical aspects of celiac dis- ease. Comparison of 2 periods: before and after the introduction of antigliadin antibody determination in clinical practice. Minerva Pediatr 1990;42:263-6. [Abstract]
- Volta U, De Franceschi L, Molinaro N, Cassani F, Muratori L, Lenzi M, et al. Frequency and signifi- cance of anti-gliadin and anti-endomysial antibodies in autoimmune hepatitis. Dig Dis Sci 1998;43:2190-5.
- Vajro P, Fontanella A, Mayer M, De Vincenzo A, Terracciano LM, D'Armiento M, et al. Elevated serum aminotransferase activity as an early mani- festation of gluten-sensitive enteropathy. J Pediatr 1993;122:416-9.
- Alvarez F, Berg PA, Bianchi FB, Bianchi L, Burroughs AK, Cancado EL, et al. International Autoimmune Hepatitis Group Report: review of criteria for diagnosis of autoimmune hepatitis. J Hepatol 1999;31:929-38.
- Shamir R, Levine A, Yalon-Hacohen M, Shapiro R, Zahavi I, Rosenbach Y, et al. Faecal occult blood in children with coeliac disease. Eur J Pediatr 2000;159:832-4.
- Corazza GR, Valentini RA, Andreani ML, D'Anchino M, Leva MT, Ginaldi L, et al. Subclinical coeliac dis- ease is a frequent cause of iron-deficiency anaemia. Scand J Gastroenterol 1995;30:153-6.
- Depla AC, Bartelsman JF, Mulder CJ, Tytgat GN. Anemia: monosymptomatic celiac disease. A report of 3 cases. Hepatogastroenterology 1990;37:90-1.
- Cacciari E, Salardi S, Lazzari R, Cicognani A, Collina A, Pirazzoli P, et al. Short stature and celiac dis- ease: a relationship to consider even in patients with no gastrointestinal tract symptoms. J Pediatr 1983;103:708-11.
- Stenhammar L, Fällström SP, Jansson G, Jansson U, Lindberg T. Coeliac disease in children of short stat- ure without gastrointestinal symptoms. Eur J Pediatr 1986;145:185-6.
- Groll A, Candy DC, Preece MA, Tanner JM, Harries JT. Short stature as the primary manifestation of coeliac disease. Lancet 1980;2:1097-9.
- Carlsson AK, Axelsson IE, Borulf SK, Bredberg AC, Lindberg BA, Sjöberg KG, et al. Prevalence of IgA- antiendomysium and IgA-antigliadin autoantibodies at diagnosis of insulin-dependent diabetes mellitus in Swedish children and adolescents. Pediatrics 1999;103:1248-52.
- Kaukinen K, Collin P, Mykkänen AH, Partanen J, Mäki M, Salmi J. Celiac disease and autoimmune endocri- nologic disorders. Dig Dis Sci 1999;44:1428-33.
- Ventura A, Magazzù G, Greco L. Duration of expo- sure to gluten and risk for autoimmune disor- ders in patients with celiac disease. SIGEP Study Group for Autoimmune Disorders in Celiac Disease. Gastroenterology 1999;117:297-303.
- Iorio R, Sepe A, Giannattasio A, Spagnuolo MI, Vecchione R, Vegnente A. Lack of benefit of gluten- free diet on autoimmune hepatitis in a boy with celiac disease. J Pediatr Gastroenterol Nutr 2004;39:207-10.
- Arvola T, Mustalahti K, Saha MT, Vehmanen P, Partanen J, Ashorn M. Celiac disease, thyrotoxico- sis, and autoimmune hepatitis in a child. J Pediatr Gastroenterol Nutr 2002;35:90-2.
- Vergani D, Mieli-Vergani G. Autoimmune liver dis- ease. In: Walker WA, Durie PR, Hamilton JR, Walker- Smith JA, Watkins JB, editors. Pediatric gastrointes- tinal disease: pathophysiology, diagnosis, manage- ment. 3rd ed. Hamilton Ont: BC Decker; 2000. p. 1007-14.
- Gregorio GV, Portmann B, Reid F, Donaldson PT, Doherty DG, McCartney M, et al. Autoimmune hep- atitis in childhood: a 20-year experience. Hepatology 1997;25:541-7.
Abstract
Burada tedaviye dirençli demir eksikliği, transaminaz yüksekliği ve boy kısalığı ile başvuran, çölyak serolojisi pozitif saptanan ve ince bağırsak biyopsisi ile çölyak hastalığı tanısı alan 10 yaşında olgu sunuldu. Anemi sadece glutensiz diyet ile bir ay içerisinde düzeldi. Başvuru anında hipergamaglobülinemi ve anti-düz kas antikor pozitifliği nedeniyle yapılan karaciğer biyopsisinde portal lenfoplazmositer infiltrasyon saptandı. Histolojik, klinik ve laboratuar bulgular ile otoimmün hepatit tanısı aldı ve immünsüpresif tedavi başlandı. Olgu, atipik prezentasyonun yanında eşlik eden otoimmün hepatitin glutensiz diyet altında immunsüpresif tedaviye alışılagelmişin dışında hızlı yanıt vermesi nedeniyle sunuma değer bulunmuştur.
References
- Mäki M, Kallonen K, Lähdeaho ML, Visakorpi JK. Changing pattern of childhood coeliac disease in Finland. Acta Paediatr Scand 1988;77:408-12.
- Logan RF, Tucker G, Rifkind EA, Heading RC, Ferguson A. Changes in clinical features of coeliac disease in adults in Edinburgh and the Lothians 1960-79. Br Med J 1983;286:95-7.
- Ceccarelli M, Caiulo VA, Cortigiani L, Pucci C, Lupetti L, Ughi C. Clinical aspects of celiac dis- ease. Comparison of 2 periods: before and after the introduction of antigliadin antibody determination in clinical practice. Minerva Pediatr 1990;42:263-6. [Abstract]
- Volta U, De Franceschi L, Molinaro N, Cassani F, Muratori L, Lenzi M, et al. Frequency and signifi- cance of anti-gliadin and anti-endomysial antibodies in autoimmune hepatitis. Dig Dis Sci 1998;43:2190-5.
- Vajro P, Fontanella A, Mayer M, De Vincenzo A, Terracciano LM, D'Armiento M, et al. Elevated serum aminotransferase activity as an early mani- festation of gluten-sensitive enteropathy. J Pediatr 1993;122:416-9.
- Alvarez F, Berg PA, Bianchi FB, Bianchi L, Burroughs AK, Cancado EL, et al. International Autoimmune Hepatitis Group Report: review of criteria for diagnosis of autoimmune hepatitis. J Hepatol 1999;31:929-38.
- Shamir R, Levine A, Yalon-Hacohen M, Shapiro R, Zahavi I, Rosenbach Y, et al. Faecal occult blood in children with coeliac disease. Eur J Pediatr 2000;159:832-4.
- Corazza GR, Valentini RA, Andreani ML, D'Anchino M, Leva MT, Ginaldi L, et al. Subclinical coeliac dis- ease is a frequent cause of iron-deficiency anaemia. Scand J Gastroenterol 1995;30:153-6.
- Depla AC, Bartelsman JF, Mulder CJ, Tytgat GN. Anemia: monosymptomatic celiac disease. A report of 3 cases. Hepatogastroenterology 1990;37:90-1.
- Cacciari E, Salardi S, Lazzari R, Cicognani A, Collina A, Pirazzoli P, et al. Short stature and celiac dis- ease: a relationship to consider even in patients with no gastrointestinal tract symptoms. J Pediatr 1983;103:708-11.
- Stenhammar L, Fällström SP, Jansson G, Jansson U, Lindberg T. Coeliac disease in children of short stat- ure without gastrointestinal symptoms. Eur J Pediatr 1986;145:185-6.
- Groll A, Candy DC, Preece MA, Tanner JM, Harries JT. Short stature as the primary manifestation of coeliac disease. Lancet 1980;2:1097-9.
- Carlsson AK, Axelsson IE, Borulf SK, Bredberg AC, Lindberg BA, Sjöberg KG, et al. Prevalence of IgA- antiendomysium and IgA-antigliadin autoantibodies at diagnosis of insulin-dependent diabetes mellitus in Swedish children and adolescents. Pediatrics 1999;103:1248-52.
- Kaukinen K, Collin P, Mykkänen AH, Partanen J, Mäki M, Salmi J. Celiac disease and autoimmune endocri- nologic disorders. Dig Dis Sci 1999;44:1428-33.
- Ventura A, Magazzù G, Greco L. Duration of expo- sure to gluten and risk for autoimmune disor- ders in patients with celiac disease. SIGEP Study Group for Autoimmune Disorders in Celiac Disease. Gastroenterology 1999;117:297-303.
- Iorio R, Sepe A, Giannattasio A, Spagnuolo MI, Vecchione R, Vegnente A. Lack of benefit of gluten- free diet on autoimmune hepatitis in a boy with celiac disease. J Pediatr Gastroenterol Nutr 2004;39:207-10.
- Arvola T, Mustalahti K, Saha MT, Vehmanen P, Partanen J, Ashorn M. Celiac disease, thyrotoxico- sis, and autoimmune hepatitis in a child. J Pediatr Gastroenterol Nutr 2002;35:90-2.
- Vergani D, Mieli-Vergani G. Autoimmune liver dis- ease. In: Walker WA, Durie PR, Hamilton JR, Walker- Smith JA, Watkins JB, editors. Pediatric gastrointes- tinal disease: pathophysiology, diagnosis, manage- ment. 3rd ed. Hamilton Ont: BC Decker; 2000. p. 1007-14.
- Gregorio GV, Portmann B, Reid F, Donaldson PT, Doherty DG, McCartney M, et al. Autoimmune hep- atitis in childhood: a 20-year experience. Hepatology 1997;25:541-7.
Details
| Primary Language | Turkish |
|---|---|
| Journal Section | Articles |
| Authors | |
| Publication Date | March 1, 2009 |
| Published in Issue | Year 2009 Volume: 2009 Issue: 3 |