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作 者:Silvana Ancona Sara Signa Chiara Longo Giuliana Cangemi Roberta Carfora Enrico Drago Alessandro La Rosa Marco Crocco Andrea Chiaro Paolo Gandullia Serena Arrigo
机构地区:[1]Dipartimento di Neuroscienze,Riabilitazione,Oftalmologia,Genetica e Scienze Materno-Infantili(DINOGMI),Universitàdegli Studi di Genova,Genova 16126,Italy [2]Pediatric Gastroenterology and Endoscopy Unit,IRCCS Istituto Giannina Gaslini,Genova 16147,Italy [3]Chromatography and Mass Spectrometry Section,Central Laboratory of Analysis,IRCCS Istituto Giannina Gaslini,Genova 16147,Italy
出 处:《World Journal of Gastroenterology》2023年第38期5428-5434,共7页世界胃肠病学杂志(英文版)
摘 要:BACKGROUND Treatment of infantile-onset inflammatory bowel disease(IO-IBD)is often challenging due to its aggressive disease course and failure of standard therapies with a need for biologics.Secondary loss of response is frequently caused by the production of anti-drug antibodies,a well-known problem in IBD patients on biologic treatment.We present a case of IO-IBD treated with therapeutic drug monitoring(TDM)-guided high-dose anti-tumor necrosis factor therapy,in which dose escalation monitoring was used as a strategy to overcome anti-drug antibodies.CASE SUMMARY A 5-mo-old boy presented with a history of persistent hematochezia from the 10th d of life,as well as relapsing perianal abscess and growth failure.Hypoalbuminemia,anemia,and elevated inflammatory markers were also present.Endoscopic assessment revealed skip lesions with deep colic ulcerations,inflammatory anal sub-stenosis,and deep fissures with persistent abscess.A diagnosis of IO-IBD Crohn-like was made.The patient was initially treated with oral steroids and fistulotomy.After the perianal abscess healed,adalimumab(ADA)was administered with concomitant gradual tapering of steroids.Clinical and biochemical steroid-free remission was achieved with good trough levels.After 3 mo,antibodies to ADA(ATA)were found with undetectable trough levels;therefore,we optimized the therapy schedule,first administering 10 mg weekly and subsequently up to 20 mg weekly(2.8 mg/kg/dose).After 2 mo of high-dose treatment,ATA disappeared,with concomitant high trough levels and stable clinical and biochemical remission of the disease.CONCLUSION TDM-guided high-dose ADA treatment as a monotherapy overcame ATA production.This strategy could be a good alternative to combination therapy,especially in very young patients.
关 键 词:Infantile-onset inflammatory bowel disease ADALIMUMAB Loss of response Dose escalation Anti-drug antibodies Case report
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