Cyclophosphamide in the Treatment of Associated Acquired Lipodystrophy Syndrome With Type 1 Diabetes

NCT ID: NCT03936829

Last Updated: 2024-08-14

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

RECRUITING

Clinical Phase

NA

Total Enrollment

10 participants

Study Classification

INTERVENTIONAL

Study Start Date

2019-04-28

Study Completion Date

2026-05-31

Brief Summary

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This study evaluates the change of insulin resistance and glucose metabolism of patients with panniculitis associated acquired lipodystrophy syndrome and type 1 diabetes with the treatment of cyclophosphamide.

Detailed Description

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Patients with panniculitis associated acquired lipodystrophy syndrome and type 1 diabetes have difficulty in blood glucose management due to the presence of both severe insulin resistance and complete insulin deficiency. It is often necessary to use insulin doses several times that of other children of the same age with type 1 diabetes.

Since autoimmune response is the main cause of panniculitis associated acquired lipodystrophy syndrome, immunosuppressive agents can suppress immune response, prevent and alleviate the progression of panniculitis and acquired lipodystrophy syndrome, and improve insulin resistance caused by subcutaneous fat deficiency.

Cyclophosphamide is a classic immunosuppressive agent. This study hopes to improve insulin resistance of patients with panniculitis associated acquired lipodystrophy syndrome and type 1 diabetes by cyclophosphamide treatment, thereby reducing insulin dosage and improving glucose metabolism.

Conditions

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Lipodystrophy Acquired Type1diabetes

Study Design

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Allocation Method

NA

Intervention Model

SINGLE_GROUP

Primary Study Purpose

TREATMENT

Blinding Strategy

NONE

Study Groups

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Interventional

Drug:Cyclophosphamide Dosage form: intravenous infusion Dosage: 500 mg/m2 of BSA Frequency: every 4 weeks Duration: 24 weeks

Group Type EXPERIMENTAL

Cyclophosphamide

Intervention Type DRUG

Admitted to the hospital every 4 weeks. Cyclophosphamide was intravenously instilled (500mg mg/m2 of BSA) after contraindications are excluded. ECG monitoring will be taken during the medication. A total of 6 treatments will be performed.

Interventions

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Cyclophosphamide

Admitted to the hospital every 4 weeks. Cyclophosphamide was intravenously instilled (500mg mg/m2 of BSA) after contraindications are excluded. ECG monitoring will be taken during the medication. A total of 6 treatments will be performed.

Intervention Type DRUG

Other Intervention Names

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CYC

Eligibility Criteria

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Inclusion Criteria

1. Meet the diagnostic criteria of type 1 diabetes mellitus: clinical manifestations of typical diabetes mellitus include polyphagia, polyuria, weight loss, or diabetic ketoacidosis, confirmed by blood sugar level, islet function and autoimmune antibody.
2. Meet the diagnostic criteria for panniculitis: fat biopsy suggests inflammatory infiltration.
3. Meet the diagnostic criteria for acquired lipodystrophy syndrome: childhood onset, clinically no nutritional deficiency or catabolism, systemic or partial subcutaneous fat reduction, genetic testing to exclude congenital lipodystrophy syndrome; low leptin level and autoantibodies can aid in diagnosis.

Exclusion Criteria

1. Mature and effective treatment methods are available.
2. HIV, HBV and HCV were positive.
3. A the active period of infection.
4. At the active stage of malignant tumors.
5. Combination of other fatal diseases.
6. Existence of mental and psychological diseases.
Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Children's Hospital of Fudan University

OTHER

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Feihong Luo

Role: STUDY_CHAIR

Children's Hospital of Fudan University

Locations

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Children's Hospital of Fudan University

Shanghai, Shanghai Municipality, China

Site Status RECRUITING

Countries

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China

Central Contacts

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Feihong Luo

Role: CONTACT

+862164931226 ext. +862164931226

Zhenran H Xu

Role: CONTACT

+862164931226 ext. +862164931226

Facility Contacts

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Feihong Luo

Role: primary

+862164931226 ext. +862164931226

Zhenran Xu

Role: backup

+862164931226 ext. +862164931226

References

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Lebastchi J, Ajluni N, Neidert A, Oral EA. A Report of Three Cases With Acquired Generalized Lipodystrophy With Distinct Autoimmune Conditions Treated With Metreleptin. J Clin Endocrinol Metab. 2015 Nov;100(11):3967-70. doi: 10.1210/jc.2015-2589. Epub 2015 Sep 21.

Reference Type BACKGROUND
PMID: 26390101 (View on PubMed)

Kumar R, Pilania RK, Bhatia A, Dayal D. Acquired generalised lipodystrophy and type 1 diabetes mellitus in a child: a rare and implacable association. BMJ Case Rep. 2018 Aug 3;2018:bcr2018225553. doi: 10.1136/bcr-2018-225553.

Reference Type BACKGROUND
PMID: 30076164 (View on PubMed)

Gupta N, Asi N, Farah W, Almasri J, Barrionuevo P, Alsawas M, Wang Z, Haymond MW, Brown RJ, Murad MH. Clinical Features and Management of Non-HIV-Related Lipodystrophy in Children: A Systematic Review. J Clin Endocrinol Metab. 2017 Feb 1;102(2):363-374. doi: 10.1210/jc.2016-2271.

Reference Type BACKGROUND
PMID: 27967300 (View on PubMed)

Carroll M, Mellick N, Wagner G. Dermatomyositis panniculitis: a case report. Australas J Dermatol. 2015 Aug;56(3):224-6. doi: 10.1111/ajd.12172. Epub 2014 Apr 1.

Reference Type BACKGROUND
PMID: 24689899 (View on PubMed)

Li S, Yang H, Guo P, Ao X, Wan J, Li Q, Tan L. Efficacy and safety of immunosuppressive medications for steroid-resistant nephrotic syndrome in children: a systematic review and network meta-analysis. Oncotarget. 2017 Aug 21;8(42):73050-73062. doi: 10.18632/oncotarget.20377. eCollection 2017 Sep 22.

Reference Type BACKGROUND
PMID: 29069848 (View on PubMed)

Brown RJ, Meehan CA, Cochran E, Rother KI, Kleiner DE, Walter M, Gorden P. Effects of Metreleptin in Pediatric Patients With Lipodystrophy. J Clin Endocrinol Metab. 2017 May 1;102(5):1511-1519. doi: 10.1210/jc.2016-3628.

Reference Type BACKGROUND
PMID: 28324110 (View on PubMed)

Chong AY, Lupsa BC, Cochran EK, Gorden P. Efficacy of leptin therapy in the different forms of human lipodystrophy. Diabetologia. 2010 Jan;53(1):27-35. doi: 10.1007/s00125-009-1502-9. Epub 2009 Sep 2.

Reference Type BACKGROUND
PMID: 19727665 (View on PubMed)

Joffe BI, Panz VR, Raal FJ. From lipodystrophy syndromes to diabetes mellitus. Lancet. 2001 May 5;357(9266):1379-81. doi: 10.1016/S0140-6736(00)04616-X. No abstract available.

Reference Type BACKGROUND
PMID: 11356429 (View on PubMed)

Hussain I, Patni N, Garg A. Lipodystrophies, dyslipidaemias and atherosclerotic cardiovascular disease. Pathology. 2019 Feb;51(2):202-212. doi: 10.1016/j.pathol.2018.11.004. Epub 2018 Dec 27.

Reference Type BACKGROUND
PMID: 30595509 (View on PubMed)

Weingartner JS, Zedek DC, Burkhart CN, Morrell DS. Lupus erythematosus panniculitis in children: report of three cases and review of previously reported cases. Pediatr Dermatol. 2012 Mar-Apr;29(2):169-76. doi: 10.1111/j.1525-1470.2011.01544.x. Epub 2011 Nov 8.

Reference Type BACKGROUND
PMID: 22066977 (View on PubMed)

Diker-Cohen T, Cochran E, Gorden P, Brown RJ. Partial and generalized lipodystrophy: comparison of baseline characteristics and response to metreleptin. J Clin Endocrinol Metab. 2015 May;100(5):1802-10. doi: 10.1210/jc.2014-4491. Epub 2015 Mar 3.

Reference Type BACKGROUND
PMID: 25734254 (View on PubMed)

Ale'ed A, Alsonbul A, Al-Mayouf SM. Safety and efficacy of combined cyclophosphamide and rituximab treatment in recalcitrant childhood lupus. Rheumatol Int. 2014 Apr;34(4):529-33. doi: 10.1007/s00296-013-2896-8. Epub 2013 Nov 12.

Reference Type BACKGROUND
PMID: 24218286 (View on PubMed)

Park JY, Chong AY, Cochran EK, Kleiner DE, Haller MJ, Schatz DA, Gorden P. Type 1 diabetes associated with acquired generalized lipodystrophy and insulin resistance: the effect of long-term leptin therapy. J Clin Endocrinol Metab. 2008 Jan;93(1):26-31. doi: 10.1210/jc.2007-1856. Epub 2007 Oct 16.

Reference Type BACKGROUND
PMID: 17940115 (View on PubMed)

Provided Documents

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Document Type: Study Protocol

View Document

Document Type: Statistical Analysis Plan

View Document

Document Type: Informed Consent Form

View Document

Other Identifiers

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C-PAALS&T1D

Identifier Type: -

Identifier Source: org_study_id

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