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Efficacy of Adalimumab in a Girl with Refractory Intestinal Behcet's Disease.

Kaji M, Kishi T, Miyamae T, Nagata S, Yamanaka H, Fujikawa S - Case Rep Rheumatol (2015)

Bottom Line: Methylprednisolone pulse therapy was initiated, after which the symptoms transiently improved, but, during the corticosteroid taper, the abdominal pain recurred.Of importance, the dose of corticosteroids was successfully reduced without exacerbation during 8 months of observation.This is the first reported case in which adalimumab was used for pediatric gastrointestinal Behcet's disease.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatrics, Tokyo Women's Medical University, No. 8-1, Kawadacho, Shinjuku-ku, Tokyo 162-0054, Japan.

ABSTRACT
We describe our experience with a juvenile patient who had refractory intestinal Behcet's disease that responded to adalimumab, a fully humanized antibody against soluble TNF-α and its receptor. The patient, a 13-year-old girl, presented with oral aphthous ulcers, vulvar pain, and rashes on the lower extremities. She gradually developed a low-grade fever, abdominal pain, diarrhea, and hematochezia. Lower gastrointestinal endoscopy revealed ulcers in the terminal ileum, consistent with intestinal Behcet's disease. Methylprednisolone pulse therapy was initiated, after which the symptoms transiently improved, but, during the corticosteroid taper, the abdominal pain recurred. The symptoms resolved soon after the administration of adalimumab. Of importance, the dose of corticosteroids was successfully reduced without exacerbation during 8 months of observation. This is the first reported case in which adalimumab was used for pediatric gastrointestinal Behcet's disease. Adalimumab is a good choice for intestinal Behcet's disease refractory to conventional treatment.

No MeSH data available.


Related in: MedlinePlus

Clinical course of the patient. Corticosteroid pulse therapy with methylprednisolone was administered to the patient. After two courses of corticosteroid pulse therapy, the symptoms and blood test results showed improvement. The abdominal pain relapsed while tapering the dose of corticosteroid. The patient was begun on adalimumab at a dose of 160 mg in week 0, 80 mg in week 2, and 40 mg every other week thereafter. The clinical manifestations of Behcet's disease resolved soon after the initiation of adalimumab.
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fig4: Clinical course of the patient. Corticosteroid pulse therapy with methylprednisolone was administered to the patient. After two courses of corticosteroid pulse therapy, the symptoms and blood test results showed improvement. The abdominal pain relapsed while tapering the dose of corticosteroid. The patient was begun on adalimumab at a dose of 160 mg in week 0, 80 mg in week 2, and 40 mg every other week thereafter. The clinical manifestations of Behcet's disease resolved soon after the initiation of adalimumab.

Mentions: Fasting with total parental nutrition was indicated, and corticosteroid pulse therapy with methylprednisolone (1 g/day for 3 days) was initiated. After two courses of corticosteroid pulse therapy, the abdominal pain and hematochezia resolved. Blood testing showed improvement in inflammatory and fibrinolytic markers. One week after the dosage of corticosteroid was reduced to 20 mg/day, the abdominal pain recurred. Although there was no reactivation of oral and genital ulcers or exacerbation of the rashes, blood testing showed reelevation of CRP, FDP, and D-dimer levels. Because the clinical findings were corticosteroid-dependent, the patient was begun on adalimumab, a fully humanized anti-TNF-α monoclonal antibody. She received subcutaneous injections of adalimumab at a dose of 160 mg in week 0, 80 mg in week 2, and 40 mg every other week thereafter. The symptoms and clinical manifestations resolved soon after the initiation of adalimumab. The circulating inflammatory markers converted to negative. Initiation of the adalimumab made it possible to reduce the corticosteroid dose without any symptoms (Figure 4). Epithelialization of the ulcers was demonstrated on lower gastrointestinal endoscopy, which was performed 1 week after the initiation of adalimumab (Figure 3(b)). Although the oral corticosteroid dose was gradually decreased, the symptoms never recurred. No adverse effects of adalimumab were observed.


Efficacy of Adalimumab in a Girl with Refractory Intestinal Behcet's Disease.

Kaji M, Kishi T, Miyamae T, Nagata S, Yamanaka H, Fujikawa S - Case Rep Rheumatol (2015)

Clinical course of the patient. Corticosteroid pulse therapy with methylprednisolone was administered to the patient. After two courses of corticosteroid pulse therapy, the symptoms and blood test results showed improvement. The abdominal pain relapsed while tapering the dose of corticosteroid. The patient was begun on adalimumab at a dose of 160 mg in week 0, 80 mg in week 2, and 40 mg every other week thereafter. The clinical manifestations of Behcet's disease resolved soon after the initiation of adalimumab.
© Copyright Policy - open-access
Related In: Results  -  Collection

Show All Figures
getmorefigures.php?uid=PMC4644826&req=5

fig4: Clinical course of the patient. Corticosteroid pulse therapy with methylprednisolone was administered to the patient. After two courses of corticosteroid pulse therapy, the symptoms and blood test results showed improvement. The abdominal pain relapsed while tapering the dose of corticosteroid. The patient was begun on adalimumab at a dose of 160 mg in week 0, 80 mg in week 2, and 40 mg every other week thereafter. The clinical manifestations of Behcet's disease resolved soon after the initiation of adalimumab.
Mentions: Fasting with total parental nutrition was indicated, and corticosteroid pulse therapy with methylprednisolone (1 g/day for 3 days) was initiated. After two courses of corticosteroid pulse therapy, the abdominal pain and hematochezia resolved. Blood testing showed improvement in inflammatory and fibrinolytic markers. One week after the dosage of corticosteroid was reduced to 20 mg/day, the abdominal pain recurred. Although there was no reactivation of oral and genital ulcers or exacerbation of the rashes, blood testing showed reelevation of CRP, FDP, and D-dimer levels. Because the clinical findings were corticosteroid-dependent, the patient was begun on adalimumab, a fully humanized anti-TNF-α monoclonal antibody. She received subcutaneous injections of adalimumab at a dose of 160 mg in week 0, 80 mg in week 2, and 40 mg every other week thereafter. The symptoms and clinical manifestations resolved soon after the initiation of adalimumab. The circulating inflammatory markers converted to negative. Initiation of the adalimumab made it possible to reduce the corticosteroid dose without any symptoms (Figure 4). Epithelialization of the ulcers was demonstrated on lower gastrointestinal endoscopy, which was performed 1 week after the initiation of adalimumab (Figure 3(b)). Although the oral corticosteroid dose was gradually decreased, the symptoms never recurred. No adverse effects of adalimumab were observed.

Bottom Line: Methylprednisolone pulse therapy was initiated, after which the symptoms transiently improved, but, during the corticosteroid taper, the abdominal pain recurred.Of importance, the dose of corticosteroids was successfully reduced without exacerbation during 8 months of observation.This is the first reported case in which adalimumab was used for pediatric gastrointestinal Behcet's disease.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatrics, Tokyo Women's Medical University, No. 8-1, Kawadacho, Shinjuku-ku, Tokyo 162-0054, Japan.

ABSTRACT
We describe our experience with a juvenile patient who had refractory intestinal Behcet's disease that responded to adalimumab, a fully humanized antibody against soluble TNF-α and its receptor. The patient, a 13-year-old girl, presented with oral aphthous ulcers, vulvar pain, and rashes on the lower extremities. She gradually developed a low-grade fever, abdominal pain, diarrhea, and hematochezia. Lower gastrointestinal endoscopy revealed ulcers in the terminal ileum, consistent with intestinal Behcet's disease. Methylprednisolone pulse therapy was initiated, after which the symptoms transiently improved, but, during the corticosteroid taper, the abdominal pain recurred. The symptoms resolved soon after the administration of adalimumab. Of importance, the dose of corticosteroids was successfully reduced without exacerbation during 8 months of observation. This is the first reported case in which adalimumab was used for pediatric gastrointestinal Behcet's disease. Adalimumab is a good choice for intestinal Behcet's disease refractory to conventional treatment.

No MeSH data available.


Related in: MedlinePlus