Cytokine Guided Risk Stratification and Treatment in Pediatric Hemophagocytic Lymphohistiocytosis

NCT ID: NCT05491304

Last Updated: 2022-10-06

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

PHASE4

Total Enrollment

400 participants

Study Classification

INTERVENTIONAL

Study Start Date

2022-09-01

Study Completion Date

2025-12-31

Brief Summary

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Hemophagocytic lymphohistiocytosis (HLH) is a rapidly fatal disease caused by immune-dysregulation characterized by hypercytokinemia, with about 30%-40% of patients suffering death in children. Stratification strategy and individualized treatment is important to improve the survival. In our recent retrospective study, risk stratification based on IL-10 and IFN-γ levels well distinguished patients with different outcomes. In this multicenter prospective study, we will enroll the newly diagnosed pediatric HLH patients and divide them into low, intermediate and high-risk cytokine groups according to IFN-γ and IL-10 levels. The patients'clinical manifestation and laboratory findings will be further evaluated into severe and non-severe groups. For low/intermediate risk and non-severe patients, steroid or ruxolitinib will be used initially; while those with high risk or severe diseases, DXM+VP16±ruxolitinib will be administered. The treatment strategy could be adjusted after evaluation 48-72 hours later.

Detailed Description

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Background and objectives: Hemophagocytic lymphohistiocytosis (HLH) is a rapidly fatal disease caused by immune-dysregulation characterized by hypercytokinemia, with about 30%-40% of patients suffering death in children. Early death is an important issue in HLH treatment, which is greatly caused by hypercytokinemia resulting in multi-organ disfunction and partially due to the treatment toxicities. Thus, stratification strategy and individualized treatment is important to improve the survival. In our recent retrospective study which enrolled 256 pediatric patients, the patients were stratified into low, intermediate and high risk according to their IL-10 and IFN-γ levels. The 8-week mortality for low, intermediate and high-risk patients were 5.4±2.4%, 16.9±3.4% and 48.7±8.0%, and the 5-year OS rate were 82.9±40%, 67.0±4.3% and 51.3±8.0%, respectively. This indicated that IFN-γ and IL-10 are helpful for stratifying HLH patients into different risk groups to receive individualized therapies. However, evidence of cytokine application based on multi-center prospective study is still lacking. The aim of this protocol is to establish a model to early identify the patients with low and high mortality and to guide the precise treatment of pediatric HLH.

Methods and protocol design: A multicenter prospective study in Asian countries is to be launched for children with HLH. The inclusion criterion is newly diagnosed pediatric HLH patients who has not received steroids or etoposide when enrollment. In this study, the patients are identified as low, intermediate and high-risk cytokine groups according to their cytokine levels: (1) low-risk: IFN-γ\<3700pg/mL and IL-10\<200pg/mL; (2) intermediate-risk: IFN-γ\<3700pg/mL and IL-10≥200pg/mL; (3) high-risk: IFN-γ≥3700pg/mL. The patients' clinical manifestation and laboratory findings were evaluated as well and those fulfill either one of the following criteria were considered as "severe": (1) present ≥2 out of 3 ⅰ, albumin\<26.0 g/L; ⅱ, direct bilirubin\>55.0μmol/L; ⅲ, fibrinogen\<0.75g/L. (2) CNS involvement, shock, mechanical ventilation, renal failure.

Based on the cytokine risk and disease severity, different intensity of treatment will be started. For low/intermediate risk and not severe patients, steroid or ruxolitinib will be used initially; while those with high risk or "severe" disease, DXM+VP16±ruxolitinib will be administered. The treatment strategy could be adjusted after evaluation 48-72 hours later based on treatment respose and cytokine levels. A total of 400 pediatric patients under 18 years old are to be recruited. The primary end-point of the study is the 8-week responsive rates and mortality, and one-year overall survival.

Conditions

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Hemophagocytic Lymphohistiocytoses Cytokine Storm

Study Design

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Allocation Method

RANDOMIZED

Intervention Model

PARALLEL

The patients will be divide into different risk groups according to cytokine concentration, clinical manifestation and laboratory findings. For low/intermediate risk and not severe patients, steroid or ruxolitinib will be used initially; while those with high risk or "severe" disease, DXM+VP16±ruxolitinib will be administered.
Primary Study Purpose

TREATMENT

Blinding Strategy

NONE

Open Label

Study Groups

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Non-severe DXM group

Dexamethasone (DXM): week1-2: 10 mg/m2.d, week 3-4: 5 mg/m2.d, week5-6: 2.5 mg/m2.d, week7: 1.25 mg/m2.d, week8: tapering.

Group Type EXPERIMENTAL

Dexamethasone

Intervention Type DRUG

Single drug in non-severe group; combined with etoposide±ruxolitinib in severe group.

Non-severe Ruxo group

Ruxolitinib(Ruxo): body weight (BW)\<10kg: 2.5mg Bid; 10-20kg: 5mg Bid; \>20kg: 10mg Bid; Orally for 4 weeks.

Group Type EXPERIMENTAL

Ruxolitinib

Intervention Type DRUG

Single drug in non-severe group; combined with etoposide and dexamethasone in severe group.

Severe HLH-94 group

DXM: week1-2: 10 mg/m2.d, week 3-4: 5 mg/m2.d, week5-6: 2.5 mg/m2.d, week7: 1.25 mg/m2.d, week8: tapering.

Etoposide (VP16): 100-150mg/m2 twice in the first two weeks, and once every week to week 8.

Group Type EXPERIMENTAL

Dexamethasone

Intervention Type DRUG

Single drug in non-severe group; combined with etoposide±ruxolitinib in severe group.

Etoposide

Intervention Type DRUG

Used in severe group combined with etoposide.

Severe HLH-94 plus ruxolitinib group

DXM: week1-2: 10 mg/m2.d, week 3-4: 5 mg/m2.d, week5-6: 2.5 mg/m2.d, week7: 1.25 mg/m2.d, week8: tapering.

Etoposide (VP16): 100-150mg/m2 twice in the first two weeks, and once every week to week 8.

Ruxolitinib(Ruxo): body weight (BW)\<10kg: 2.5mg Bid; 10-20kg: 5mg Bid; \>20kg: 10mg Bid; Orally for 4 weeks.

Group Type EXPERIMENTAL

Dexamethasone

Intervention Type DRUG

Single drug in non-severe group; combined with etoposide±ruxolitinib in severe group.

Etoposide

Intervention Type DRUG

Used in severe group combined with etoposide.

Ruxolitinib

Intervention Type DRUG

Single drug in non-severe group; combined with etoposide and dexamethasone in severe group.

Interventions

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Dexamethasone

Single drug in non-severe group; combined with etoposide±ruxolitinib in severe group.

Intervention Type DRUG

Etoposide

Used in severe group combined with etoposide.

Intervention Type DRUG

Ruxolitinib

Single drug in non-severe group; combined with etoposide and dexamethasone in severe group.

Intervention Type DRUG

Other Intervention Names

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Steroid Corticosteroid VP16 JAK1/2 inhibitor

Eligibility Criteria

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Inclusion Criteria

* Age from one day to 18 years old;
* Newly diagnosed HLH, fulfilling the HLH criteria;
* To observed the early diagnosis role of cytokines, patients who is suspected to be HLH and fulfill 3 out of 8 criteria can be pre-enrolled.

Exclusion Criteria

* treated with steroids or etoposide within 72 hours before diagnosis.
Minimum Eligible Age

1 Day

Maximum Eligible Age

18 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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The Second Hospital of Anhui Medical University

OTHER

Sponsor Role collaborator

Union hospital of Fujian Medical University

OTHER

Sponsor Role collaborator

Children's Hospital of Fudan University

OTHER

Sponsor Role collaborator

Zunyi Medical College

OTHER

Sponsor Role collaborator

Hunan Provincial People's Hospital

OTHER

Sponsor Role collaborator

Tongji Hospital

OTHER

Sponsor Role collaborator

Children's Hospital of Nanjing Medical University

OTHER

Sponsor Role collaborator

The Affiliated Hospital of Qingdao University

OTHER

Sponsor Role collaborator

Qilu Hospital of Shandong University

OTHER

Sponsor Role collaborator

Shanghai Children's Medical Center

OTHER

Sponsor Role collaborator

Xinhua Hospital, Shanghai Jiao Tong University School of Medicine

OTHER

Sponsor Role collaborator

Shanghai Children's Hospital

OTHER

Sponsor Role collaborator

Shenzhen Children's Hospital

OTHER_GOV

Sponsor Role collaborator

West China Second University Hospital

OTHER

Sponsor Role collaborator

Children's Hospital of Soochow University

OTHER

Sponsor Role collaborator

Second Affiliated Hospital of Wenzhou Medical University

OTHER

Sponsor Role collaborator

Wuhan Children's Hospital

OTHER

Sponsor Role collaborator

Institute of Hematology & Blood Diseases Hospital, China

OTHER

Sponsor Role collaborator

The Third Xiangya Hospital of Central South University

OTHER

Sponsor Role collaborator

First Affiliated Hospital, Sun Yat-Sen University

OTHER

Sponsor Role collaborator

The Children's Hospital of Zhejiang University School of Medicine

OTHER

Sponsor Role lead

Responsible Party

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Xiaojun Xu

chief physician

Responsibility Role PRINCIPAL_INVESTIGATOR

Principal Investigators

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Xiaojun Xu, MD

Role: PRINCIPAL_INVESTIGATOR

The Children's Hospital of Zhejiang University School of Medicine

Yongmin Tang, MD

Role: PRINCIPAL_INVESTIGATOR

The Children's Hospital of Zhejiang University School of Medicine

Locations

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The Children's Hospital of Zhejiang University School of Medicine

Hangzhou, Zhejiang, China

Site Status RECRUITING

Countries

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China

Central Contacts

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Xiaojun Xu, MD

Role: CONTACT

+8657188873617

Zebin Luo, MD

Role: CONTACT

+8618758196529

Facility Contacts

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Aimei Ma, bachelor

Role: primary

+8657180070072

References

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Bergsten E, Horne A, Arico M, Astigarraga I, Egeler RM, Filipovich AH, Ishii E, Janka G, Ladisch S, Lehmberg K, McClain KL, Minkov M, Montgomery S, Nanduri V, Rosso D, Henter JI. Confirmed efficacy of etoposide and dexamethasone in HLH treatment: long-term results of the cooperative HLH-2004 study. Blood. 2017 Dec 21;130(25):2728-2738. doi: 10.1182/blood-2017-06-788349. Epub 2017 Sep 21.

Reference Type BACKGROUND
PMID: 28935695 (View on PubMed)

Xu XJ, Tang YM. Dilemmas in diagnosis and management of hemophagocytic lymphohistiocytosis in children. World J Pediatr. 2020 Aug;16(4):333-340. doi: 10.1007/s12519-019-00299-3. Epub 2019 Sep 10.

Reference Type BACKGROUND
PMID: 31506890 (View on PubMed)

Tang Y, Xu X, Song H, Yang S, Shi S, Wei J, Pan B, Zhao F, Liao C, Luo C. Early diagnostic and prognostic significance of a specific Th1/Th2 cytokine pattern in children with haemophagocytic syndrome. Br J Haematol. 2008 Oct;143(1):84-91. doi: 10.1111/j.1365-2141.2008.07298.x. Epub 2008 Jul 31.

Reference Type BACKGROUND
PMID: 18673367 (View on PubMed)

Xu XJ, Tang YM, Song H, Yang SL, Xu WQ, Zhao N, Shi SW, Shen HP, Mao JQ, Zhang LY, Pan BH. Diagnostic accuracy of a specific cytokine pattern in hemophagocytic lymphohistiocytosis in children. J Pediatr. 2012 Jun;160(6):984-90.e1. doi: 10.1016/j.jpeds.2011.11.046. Epub 2012 Jan 9.

Reference Type BACKGROUND
PMID: 22226576 (View on PubMed)

Xu XJ, Luo ZB, Song H, Xu WQ, Henter JI, Zhao N, Wu MH, Tang YM. Simple Evaluation of Clinical Situation and Subtypes of Pediatric Hemophagocytic Lymphohistiocytosis by Cytokine Patterns. Front Immunol. 2022 Feb 28;13:850443. doi: 10.3389/fimmu.2022.850443. eCollection 2022.

Reference Type BACKGROUND
PMID: 35296096 (View on PubMed)

Wang J, Zhang R, Wu X, Li F, Yang H, Liu L, Guo H, Zhang X, Mai H, Li H, Wang Z. Ruxolitinib-combined doxorubicin-etoposide-methylprednisolone regimen as a salvage therapy for refractory/relapsed haemophagocytic lymphohistiocytosis: a single-arm, multicentre, phase 2 trial. Br J Haematol. 2021 May;193(4):761-768. doi: 10.1111/bjh.17331. Epub 2021 Feb 9.

Reference Type BACKGROUND
PMID: 33559893 (View on PubMed)

Zhang Q, Zhao YZ, Ma HH, Wang D, Cui L, Li WJ, Wei A, Wang CJ, Wang TY, Li ZG, Zhang R. A study of ruxolitinib response-based stratified treatment for pediatric hemophagocytic lymphohistiocytosis. Blood. 2022 Jun 16;139(24):3493-3504. doi: 10.1182/blood.2021014860.

Reference Type BACKGROUND
PMID: 35344583 (View on PubMed)

Ehl S, Astigarraga I, von Bahr Greenwood T, Hines M, Horne A, Ishii E, Janka G, Jordan MB, La Rosee P, Lehmberg K, Machowicz R, Nichols KE, Sieni E, Wang Z, Henter JI. Recommendations for the Use of Etoposide-Based Therapy and Bone Marrow Transplantation for the Treatment of HLH: Consensus Statements by the HLH Steering Committee of the Histiocyte Society. J Allergy Clin Immunol Pract. 2018 Sep-Oct;6(5):1508-1517. doi: 10.1016/j.jaip.2018.05.031. Epub 2018 Jul 4.

Reference Type BACKGROUND
PMID: 30201097 (View on PubMed)

Henter JI, Horne A, Arico M, Egeler RM, Filipovich AH, Imashuku S, Ladisch S, McClain K, Webb D, Winiarski J, Janka G. HLH-2004: Diagnostic and therapeutic guidelines for hemophagocytic lymphohistiocytosis. Pediatr Blood Cancer. 2007 Feb;48(2):124-31. doi: 10.1002/pbc.21039.

Reference Type BACKGROUND
PMID: 16937360 (View on PubMed)

Other Identifiers

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2022-IRB-057

Identifier Type: -

Identifier Source: org_study_id

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