Thalidomide in Pediatric Inflammatory Bowel Diseases.

NCT ID: NCT00720538

Last Updated: 2012-10-26

Study Results

Results pending

The study team has not published outcome measurements, participant flow, or safety data for this trial yet. Check back later for updates.

Basic Information

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Recruitment Status

COMPLETED

Clinical Phase

PHASE3

Total Enrollment

84 participants

Study Classification

INTERVENTIONAL

Study Start Date

2008-08-31

Study Completion Date

2012-06-30

Brief Summary

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Several open-label studies reported thalidomide efficacy in inducing clinical remission and steroid tapering in refractory Inflammatory Bowel diseases (IBD), both in adults and in children.

This is a randomized placebo controlled (RCT) double blind study, to evaluate the efficacy of thalidomide in inducing clinical remission at 8 weeks in refractory IBD patients aged 2-20 years.

The primary hypotheses of the study is that thalidomide would be more effective than placebo in inducing clinical remission.

The RCT phase is followed by a open-label phase, to further evaluate efficacy and safety of thalidomide in thalidomide responders, with a total follow up of one year.

Detailed Description

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Inflammatory bowel diseases (IBD) are particularly debilitating. They present chronic-recurrent symptoms which often seriously affects the patient's quality of life, acute complications which may need surgical interventions and long-term risks as evolution in cancer for ulcerative colitis.

Although onset is most common in the third decade of life, more than one third of IBD cases are diagnosed before age sixteen, and these younger patients, especially with Crohn's disease, are increasing in Western countries. Recent studies in the United Kingdom, Sweden and the United States report an incidence of 5-7 new cases of IBD every year per 100,000 subjects \< 16 years of age, with an increasing trend over the last two decades.

IBD-related morbidity causes special concern in childhood-onset cases, since the disease can compromise height and weight development. This is even worsened in patients with poor disease control, which need steroid treatment in multiple courses and/or for a long period of time.

There are various drugs and treatments available for the management of IBD, but some patients, during the course of the illness, become resistant to conventional treatment, or develop serious adverse effects to immunosuppressive drugs.

A particular challenge is represented by those patients (especially children) who fail to respond to conventional treatment, or are steroid-dependent, or suffer from serious side effects from steroids use.

Thalidomide was first marketed in the early 1960s as a sedative drug and sadly gained notoriety because of its teratogenic effect. After few years from this tragedy, thalidomide was rediscovered both as an immunomodulator and as an anti-inflammatory drug, with anti tumor necrosis factor-alfa properties.

Thalidomide is now approved for the treatment of multiple myeloma and erythema nodosum leprorum. It has also been tested, with beneficial effects, in a variety of inflammatory diseases, especially those involving the skin and mucous membranes: recurrent aphthous stomatitis, oral and oesophageal ulcers in HIV, oral and genital ulcers and intestinal symptoms in Behcet's syndrome, graft versus host disease, skin manifestations in systemic lupus erythematosus, sarcoidosis, and tuberculosis. In these diseases, especially in those cases resistant to conventional treatments, thalidomide seems to have a promising role as a secondary therapeutic option.

The safety and efficacy of thalidomide in IBD has been recently evaluated in several open-label studies both adults and children, some of which with a long-term follow up. A total of approximately 200 patients with steroid-resistant or steroid-dependent IBD were treated with thalidomide. Overall, the drug was effective in inducing clinical remission in 20-70% of patients (average of 40%). 60-100% patients (average of 70%) had some clinical response. Steroid-tapering was possible in 60-100% of cases.

The most frequent side effect of thalidomide in IBD cases was neuropathy, which mainly reported as cumulative dose-dependent. Other possible side effects are constipation, sedation, dermatitis, mood disturbance, vertigo, cephalea, hypertension, hormonal alterations.

In the experience of our center, thalidomide was effective in achieving clinical remission in several children, adolescents, and young adults with intractable IBD. From 1998 to 2004 twenty-eight patients with refractory moderate-severe IBD (19 CD, 9 UC) received thalidomide 1.5-2.5 mg/kg/day. Remission was achieved with thalidomide in 21 of 28 (75%) patients (17 with CD, 4 with UC). Mean duration of remission was 34.5 months. Sixteen of 20 (80%) patients suspended steroids. Reversible neuropathy occurred in seven of 28 (25%) patients, but only with cumulative doses over 28 g. Other side effects requiring thalidomide suspension were vertigo/somnolence (one case), and agitation/ hallucinations (one case).

The aim of this study is to evaluate the efficacy of thalidomide therapy in children, adolescents and young adults with refractory Crohn's disease (CD) and ulcerative colitis (UD), with a total follow up of one year.

The study is divided in two phases:

* Phase I: randomized placebo controlled double blind study, to evaluate efficacy of thalidomide in refractory IBD patients, at 8 weeks.
* Phase II: open-label study, to evaluate efficacy and safety of thalidomide in patients responders to Thalidomide in Phase I, with a total follow up of one year.

Analysis will be stratified for Crohn's disease and ulcerative colitis. Given the hypotheses that thalidomide will be effective in inducing clinical remission at 8 weeks in 60% of patients with Crohn's disease, and 55% of patients with ulcerative colitis, compared to 20% of patients with placebo, with a significance of 0.05% and a power of 80%, estimated needed sample size is of 124 patients.

Conditions

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Inflammatory Bowel Diseases Crohn's Disease Ulcerative Colitis

Keywords

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Inflammatory bowel diseases. Crohn disease. Ulcerative, colitis. Randomised controlled trial. Therapy. Thalidomide. Children.

Study Design

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Allocation Method

RANDOMIZED

Intervention Model

PARALLEL

Primary Study Purpose

TREATMENT

Blinding Strategy

QUADRUPLE

Participants Caregivers Investigators Outcome Assessors

Study Groups

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1

Thalidomide

Group Type EXPERIMENTAL

Thalidomide

Intervention Type DRUG

Thalidomide 1.5-2.5 mg/kg/die once a day, at evening.

2

Placebo

Group Type PLACEBO_COMPARATOR

placebo

Intervention Type DRUG

placebo capsules

Interventions

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Thalidomide

Thalidomide 1.5-2.5 mg/kg/die once a day, at evening.

Intervention Type DRUG

placebo

placebo capsules

Intervention Type DRUG

Other Intervention Names

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Thalidomide PHARMION Thalidomid CELGENE

Eligibility Criteria

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Inclusion Criteria

* Children, adolescents and young adults aged 2 to 20 with chronic refractory moderate to severe inflammatory bowel disease, referred by the six pediatric gastroenterological centres participating in the study.

Definition of patient with refractory disease:

* Patients with active disease despite steroid therapy (prednisone at a dose 2 mg/kg/die, maximum 60mg/day, or equivalent) for 8 weeks and/or an immunosuppressive of proven efficacy (azathioprine o 6-mercaptopurine for 4 months; methotrexate for 3 months; Infliximab at the dose of 5 mg/kg at week 0,2,6 weeks; cyclosporine at the dose of 2mg/kg/day for 4 weeks or 1 week at 1m/kg/day EV) or patients exhibiting intolerance to these drugs which prevent them from continuing treatment.

Exclusion Criteria

* Patients with ileostomy or colostomy.
* Disease requiring immediate surgical intervention.
* Severe ulcerative colitis or toxic megacolon.
* Contraindications to using thalidomide (on-going pregnancy, neuropathy) .
* Any of the following conditions: active infection, stool culture positive for enteric pathogens, tumors, HIV, transplanted organ, or non-controlled disease of the kidney, liver, endocrine system, heart, blood, nervous system or brain.
* Patients being treated with other drugs as part of an experimental study.
* Patients treated with infliximab in the previous eight weeks.
Minimum Eligible Age

2 Years

Maximum Eligible Age

18 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Ospedale Meyer

OTHER

Sponsor Role collaborator

Pediatric Gastroenterology Unit, IRCCS Gaslini, Genoa.

UNKNOWN

Sponsor Role collaborator

Vittore Buzzi Children's Hospital

OTHER

Sponsor Role collaborator

University of Pisa

OTHER

Sponsor Role collaborator

University of Messina

OTHER

Sponsor Role collaborator

Università degli Studi di Brescia

OTHER

Sponsor Role collaborator

University of Trieste

OTHER

Sponsor Role collaborator

IRCCS Burlo Garofolo

OTHER

Sponsor Role lead

Responsible Party

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Alessandro Ventura

MD

Responsibility Role PRINCIPAL_INVESTIGATOR

Principal Investigators

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Alessandro Ventura, MD

Role: STUDY_CHAIR

IRCCS Burlo Garofolo

Marzia Lazzerini, MD

Role: STUDY_DIRECTOR

IRCCS Burlo Garofolo

Locations

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Anatomy and Hystology, Spedali Civili di Brescia, University of Brescia.

Brescia, Brescia, Italy

Site Status

Department of Pediatrics, Ospedale Meyer.

Florence, Florence, Italy

Site Status

Pediatric Gastroenterology Unit, IRCCS Gaslini.

Genoa, Genoa, Italy

Site Status

Pediatric Gastroenterology Unit, University of Messina.

Messina, Messina, Italy

Site Status

Department of Pediatrics, Ospedali Buzzi.

Milan, Milan., Italy

Site Status

Gastroenterology and Hepatology, University of Pisa.

Pisa, Pisa, Italy

Site Status

Unit of Biochemistry and Pharmacology, University of Triest.

Trieste, Triest, Italy

Site Status

Countries

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Italy

References

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Lazzerini M, Martelossi S, Marchetti F, Scabar A, Bradaschia F, Ronfani L, Ventura A. Efficacy and safety of thalidomide in children and young adults with intractable inflammatory bowel disease: long-term results. Aliment Pharmacol Ther. 2007 Feb 15;25(4):419-27. doi: 10.1111/j.1365-2036.2006.03211.x.

Reference Type BACKGROUND
PMID: 17269997 (View on PubMed)

Lazzerini M, Martelossi S, Magazzu G, Pellegrino S, Lucanto MC, Barabino A, Calvi A, Arrigo S, Lionetti P, Lorusso M, Mangiantini F, Fontana M, Zuin G, Palla G, Maggiore G, Bramuzzo M, Pellegrin MC, Maschio M, Villanacci V, Manenti S, Decorti G, De Iudicibus S, Paparazzo R, Montico M, Ventura A. Effect of thalidomide on clinical remission in children and adolescents with refractory Crohn disease: a randomized clinical trial. JAMA. 2013 Nov 27;310(20):2164-73. doi: 10.1001/jama.2013.280777.

Reference Type DERIVED
PMID: 24281461 (View on PubMed)

Other Identifiers

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TAL-005414-20

Identifier Type: -

Identifier Source: org_study_id