The Journal of Pediatrics
Volume 155, Issue 1 , Page 151, July 2009

Takayasu's aortitis and infliximab

Division of Pediatric Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Stollery Children's Hospital, Alberta, Edmonton, Canada

Article Outline

 

To The Editor:

Filocamo et al reported the use of infliximab as adjunctive therapy for treating Takayasu's disease.1 Recently, we treated a 15-year-old girl who presented with weight loss, abdominal pain, and severe oral and perianal manifestations. The results of initial blood investigations showed high levels of inflammatory markers with a low albumin level. The diagnosis of perianal and ileo-colonic Crohn's disease was confirmed with endoscopy, ileo-colonoscopy, and biopsies. The presence of 1 perianal fistula was confirmed with magnetic resonance imaging. The patient failed to comply with nutritional therapy, and so she was given steroids and azathioprine. However, a few months later, she relapsed, and, after discussion with her and her family, infliximab (5 mg/kg) was started. She did well while taking infliximab and after. Despite recent infliximab maintenance (every 6-8 weeks), a few months later she had fever, headache, vomiting, and abdominal pain. Her blood pressure was in the reference range. She was given intravenous antibiotics, despite negative results on several infection screens. No evidence of the relapse of Crohn's disease was shown with an endoscopy and colonoscopy. Abdominal computed tomography was performed to exclude intra-abdominal collections, and some thickening of the abdominal aorta was shown, which was confirmed with a magnetic resonance imaging angiogram. A diagnosis of Takayasu's disease was confirmed, and the patient responded to systemic intravenous steroids.

In this case, acute manifestations of Takayasu's disease developed while the patient was receiving 5 mg/kg of infliximab every 6 to 8 weeks with azathioprine. Several reports documented the success of infliximab in treating Takayasu's disease with or without other immunosuppressant, even when the patient is resistant to steroids.2, 3 Some reports even used doses of infliximab lower than 3 mg/kg.3 We are not sure whether having Crohn's disease in addition to Takayasu's disease would make the latter difficult to control. There are a number of reports in the literature reporting the association of these 2 conditions,4, 5, 6 but the significance of this association has yet to be defined. Infliximab in this patient did not prevent the development of acute symptoms of Takayasu's disease.

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References 

  1. Filocamo G, Buoncompagni A, Viola S, Loy A, Malattia C, Ravelli A, et al. Treatment of Takayasu's arteritis with tumor necrosis factor antagonists. J Pediatr. 2008;153:432–434
  2. Tanaka F, Kawakami A, Iwanaga N, Tamai M, Izumi Y, Aratake K, et al. Infliximab is effective for Takayasu arteritis refractory to glucocorticoid and methotrexate. Intern Med. 2006;45:313–316
  3. Karageorgaki ZT, Mavragani CP, Papathanasiou MA, Skopouli FN. Infliximab in Takayasu arteritis: a safe alternative?. Clin Rheumatol. 2007;26:984–987
  4. Kellermayer R, Jain AK, Ferry G. Clinical challenges and images in GI. Aortitis as a rare complication of Crohn's disease. Gastroenterology. 2008;134:668–898
  5. Farrant M, Mason JC, Wong NA, Longman RJ. Takayasu's arteritis following Crohn's disease in a young woman: any evidence for a common pathogenesis?. World J Gastroenterol. 2008;14:4087–4090
  6. Reny JL, Paul JF, Lefèbvre C, Champion K, Emmerich J, Bletry O, et al. Association of Takayasu's arteritis and Crohn's disease. Results of a study on 44 Takayasu patients and review of the literature. Ann Med Interne (Paris). 2003;154:85–90

PII: S0022-3476(09)00060-2

doi:10.1016/j.jpeds.2009.01.056

The Journal of Pediatrics
Volume 155, Issue 1 , Page 151, July 2009

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