Browse > Article
http://dx.doi.org/10.14776/kjpid.2014.21.1.53

A Case of Esophageal Obstruction Complicated in a Patient with Chronic Granulomatous Disease - Esophageal obstruction in Chronic Granulomatous Disease  

Kim, Ji Soo (Department of Pediatrics, Seoul National University College of Medicine)
Hwang, Jinsol (Department of Pediatrics, Seoul National University College of Medicine)
Choi, Young Hun (Department of Radiology, Seoul National University College of Medicine)
Kim, Woo Sun (Department of Radiology, Seoul National University College of Medicine)
Kim, Joong Gon (Department of Pediatrics, Seoul National University College of Medicine)
Publication Information
Pediatric Infection and Vaccine / v.21, no.1, 2014 , pp. 53-58 More about this Journal
Abstract
Chronic granulomatous disease (CGD) is an inherited immunodeficient disease characterized by recurrent infections and granuloma formation. Granulomatous obstruction of esophagus is one of the rare complications of CGD. The use of steroids and antimicrobials for esophageal obstruction by granuloma in CGD patients has been controversial due to the possibility of concomitant inapparent infection. We report a case of esophageal obstruction in an 8-year-old CGD patient showing the poor response to antibiotics therapy. However, dramatic improvement of symptoms and radiologic findings of esophageal obstruction were achieved after steroid therapy. One month after discontinuation of steroid, esophageal obstruction recurred and the patient was re-treated with steroid. After that time, he experienced one more recurrence of esophageal obstruction. This symptom subsided after antibiotics therapy without steroid and he has been followed up to the present without further relapse.
Keywords
Chronic granulomatous disease; Esophageal stenosis; Steroid;
Citations & Related Records
연도 인용수 순위
  • Reference
1 Esophageal involvement in chronic granulomatous disease. Pediatr Radiol 1996;26:169.   DOI
2 Brown JR, Goldblatt D, Buddle J, Morton L, Thrasher AJ. Diminished production of anti-inflammatory mediators during neutrophil apoptosis and macrophage phagocytosis in chronic granulomatous disease (CGD). J Leukoc Biol 2003;73:591-9.   DOI
3 Laskey HL, Gopal L, Gallin JI, Holland SM, Heller T. Twenty-year follow-up of esophageal involvement in chronic granulomatous disease. Am J Gastroenterol 2009;104:2368-70.   DOI
4 Alborzi A, Mostafavi N, Pouladfar GR. Effect of corticosteroid therapy in esophageal stricture of a child with chronic granulomatous disease. Iran J Allergy Asthm 2008;7:235-8.
5 Hiller N, Fisher D, Abrahamov A, Blinder G. Esophageal involvement in chronic granulomatous-disease - casereport and review. Pediatr Radiol 1995;25:308-9.   DOI
6 Renner WR, Johnson JF, Lichtenstein JE, Kirks DR. Esophageal inflammation and stricture - complication of chronic granulomatous-disease of childhood. Radiology 1991;178:189-91.   DOI
7 Chin TW, Stiehm ER, Falloon J, Gallin JI. Corticosteroids in treatment of obstructive lesions of chronic granulomatous- disease. J Pediatr 1987;111:349-52.   DOI
8 Ruiz-Contreras J, Bastero R, Serrano C, Benavent MI, Martinez A. Oesophageal narrowing in chronic granulomatous disease. Eur J Radiol 1998;27:149-52.   DOI
9 Seger RA. Modern management of chronic granulomatous disease. Br J Haematol 2008;140:255-66.   DOI
10 Danziger RN, Goren AT, Becker J, Greene JM, Douglas SD. Outpatient management with oral corticosteroidtherapy for obstructive conditions in chronic granulomatous- disease. J Pediatr 1993;122:303-5.   DOI
11 Rosenzweig SD. Inflammatory manifestations in chronic granulomatous disease (CGD). J Clin Immunol 2008;28:S67-S72.   DOI
12 Marciano BE, Rosenzweig SD, Kleiner DE, Anderson VL, Darnell DN, Anaya-O'Brien S, et al. Gastrointestinal involvement in chronic granulomatous disease. Pediatrics 2004;114:462-8.   DOI
13 AlTawil YS, Abramson SL, Gilger MA, Paul ME. Steroid-responsive esophageal obstruction in a child with chronic granulomatous disease (CGD). J Pediatr Gastr Nutr 1996;23:182-5.   DOI
14 Golioto M, O'Connor JB. Esophageal dysmotility in an adult with chronic granulomatous disease. J Clin Gastroenterol 2001;33:330-2.   DOI