Salvage treatment of ruxolitinib for refractory adenovirus-associated hemophagocytic syndrome post-haploidentical allogeneic stem cell transplantation: a case report

Abstract

Background
Hemophagocytic lymphohistiocytosis (HLH) is a rare complication following hematopoietic stem cell transplantation (HSCT). Currently, there is a lack of consensus recommendations for the treatment of post-transplant HLH. This case report emphasizes the successful utilization of ruxolitinib as a salvage therapy for HLH post-HSCT. The aim is to provide valuable insights into the optimal management of this rare and complex complication.

Case Description
We present a case study of an 11-year-old male patient diagnosed with severe aplastic anemia who received a haploidentical HSCT. On the 86th day post-transplantation, the patient developed recurrent fever, hepatomegaly, hypertriglyceridemia, severe pancytopenia, and elevated levels of inflammatory factors and ferritin. Hemophagocytosis was observed in the bone marrow, and subsequent DNA next-generation sequencing identified adenovirus type C infection, leading to a diagnosis of adenovirus-associated HLH. After unsuccessful treatment attempts with cidofovir, dexamethasone, immunoglobulin, plasmapheresis, and etoposide, ruxolitinib was administered. Remarkably, the patient’s clinical symptoms rapidly improved, and his test results gradually normalized with ruxolitinib therapy. The adenovirus viral load became undetectable by the 180th day. With continuous remission, ruxolitinib was discontinued on the 137th day post-transplantation, and a 15-month follow-up examination showed no relapse.

Conclusions
We present a case of adenovirus-related secondary HLH (sHLH) post-HSCT, which was effectively treated with ruxolitinib. Our case highlights the potential of ruxolitinib as a therapeutic option for patients with viral infections and sHLH. Nonetheless, the safety and efficacy of this innovative treatment should be evaluated in forthcoming large-scale clinical trials.

Full text

© Translational Pediatrics. All rights reserved. Transl Pediatr 2024;13(6):994-1000 | https://dx.doi.org/10.21037/tp-24-27
Case Report
Salvage treatment of ruxolitinib for refractory adenovirus-
associated hemophagocytic syndrome post-haploidentical
allogeneic stem cell transplantation: a case report
Jian Li1,2#, Qizi Wu3#, Xuemei Guo1,2#, Liucheng Rong1,2, Yongjun Fang1,2#
1Department of Hematology and Oncology, Children’s Hospital of Nanjing Medical University, Nanjing, China; 2Key Laboratory of Hematology,
Nanjing Medical University, Nanjing, China; 3Department of Hematology, Nanjing Drum Tower Hospital, Afliated Hospital of Medical School,
Nanjing University, Nanjing, China
Contributions: (I) Conception and design: Y Fang; (II) Administrative support: Y Fang; (III) Provision of study materials or patients: Y Fang;
(IV) Collection and assembly of data: J Li, X Guo, L Rong; (V) Data analysis and interpretation: J Li, Q Wu; (VI) Manuscript writing: All authors;
(VII) Final approval of manuscript: All authors.
#These authors contributed equally to this work.
Correspondence to: Yongjun Fang, MD. Department of Hematology and Oncology, Children’s Hospital of Nanjing Medical University, Code 72,
Guangzhou Road, Nanjing 210008, China; Key Laboratory of Hematology, Nanjing Medical University, Nanjing, China. Email: fyj322@189.cn.
Background: Hemophagocytic lymphohistiocytosis (HLH) is a rare complication following hematopoietic
stem cell transplantation (HSCT). Currently, there is a lack of consensus recommendations for the treatment
of post-transplant HLH. This case report emphasizes the successful utilization of ruxolitinib as a salvage
therapy for HLH post-HSCT. The aim is to provide valuable insights into the optimal management of this
rare and complex complication.
Case Description: We present a case study of an 11-year-old male patient diagnosed with severe aplastic
anemia who received a haploidentical HSCT. On the 86th day post-transplantation, the patient developed
recurrent fever, hepatomegaly, hypertriglyceridemia, severe pancytopenia, and elevated levels of inammatory
factors and ferritin. Hemophagocytosis was observed in the bone marrow, and subsequent DNA next-
generation sequencing identified adenovirus type C infection, leading to a diagnosis of adenovirus-
associated HLH. After unsuccessful treatment attempts with cidofovir, dexamethasone, immunoglobulin,
plasmapheresis, and etoposide, ruxolitinib was administered. Remarkably, the patient’s clinical symptoms
rapidly improved, and his test results gradually normalized with ruxolitinib therapy. The adenovirus viral
load became undetectable by the 180th day. With continuous remission, ruxolitinib was discontinued on the
137th day post-transplantation, and a 15-month follow-up examination showed no relapse.
Conclusions: We present a case of adenovirus-related secondary HLH (sHLH) post-HSCT, which was
effectively treated with ruxolitinib. Our case highlights the potential of ruxolitinib as a therapeutic option
for patients with viral infections and sHLH. Nonetheless, the safety and efcacy of this innovative treatment
should be evaluated in forthcoming large-scale clinical trials.
Keywords: Hemophagocytic lymphohistiocytosis (HLH); hematopoietic stem cell transplantation (HSCT);
adenovirus; ruxolitinib; case report
Submitted Jan 29, 2024. Accepted for publication May 20, 2024. Published online Jun 27, 2024.
doi: 10.21037/tp-24-27
View this article at: https://dx.doi.org/10.21037/tp-24-27
1000

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Introduction
Hemophagocytic lymphohistiocytosis (HLH) is a
potentially life-threatening condition characterized by
immunological dysregulation and an overactive immune
response. Most cases of HLH are triggered by infections,
malignancies, or exacerbations of rheumatologic diseases (1).
However, HLH resulting from concurrent viral infections
after hematopoietic stem cell transplantation (HSCT) is a
relatively rare disease. Moreover, there are no established
guidelines for the diagnosis and treatment of post-
transplant HLH syndrome. Ruxolitinib, a potent inhibitor
of Janus kinase (JAK 1/2), is currently used as both primary
therapy and secondary therapy to treat HLH (2,3). Despite
the efficacy and tolerability of ruxolitinib in treating
secondary hemophagocytic lymphohistiocytosis (sHLH), its
effectiveness and safety in patients with HLH post-HSCT
remain to be investigated. In this report, we present the
case of a boy with adenovirus-associated HLH post-HSCT
who was successfully cured through salvage treatment
concomitant ruxolitinib treatment and management of
the viral infection. We present this case in accordance
with the CARE reporting checklist (available at https://
tp.amegroups.com/article/view/10.21037/tp-24-27/rc).
Case presentation
We present a case of severe aplastic anemia in an 11-year-old
Asian boy diagnosed in March 2022. The patient underwent
a haploidentical HSCT from his father as the donor. A
concise summary of the patient’s clinical information,
treatment, and prognosis is provided in Table 1. All
procedures performed in this study were in accordance with
the ethical standards of the institutional and/or national
research committee(s) and with the Helsinki Declaration (as
revised in 2013). Written informed consent was obtained
from the parents of the patient for publication of this case
report and accompanying images. A copy of the written
consent is available for review by the editorial ofce of this
journal. The conditioning regimen included fludarabine
(30 mg/kg/day from day −11 to day −7), busulfan
(3.2 mg/kg/day from day −9 to day −7) + cyclophosphamide
(60 mg/kg/day from day −6 to day −5), and rabbit
antihuman thymocyte globulin (2.5 mg/kg/day from day −4
to day −2). The patient received 3.87×106 CD34+ cells and
6.5×108 CD3+ cells per kilogram. Graft-versus-host disease
(GvHD) prophylaxis consisted of short-term methotrexate
(MTX), mycophenolate mofetil (MMF), and cyclosporine.
Complete chimerism in peripheral blood was confirmed
on day 13 and neutrophil engraftment was confirmed
on day 14. On T+16 (16 days after transplantation), the
patient developed a persistent fever and rash with pruritus
(>50% body surface area), vomiting, and abdominal
pain, indicating grade III GvHD. Methylprednisolone,
recombinant human c receptor II, and basiliximab were
administered, leading to complete improvement of GvHD
symptoms. However, cytomegalovirus and Epstein-Barr
virus (EBV) activation were detected in peripheral blood
on days 43 and 47, respectively. Immunosuppression was
reduced, antiviral therapy (ganciclovir 10 mg/kg/day and
immunoglobulin 400 mg/kg/day) was administered, and
viral regression was evaluated on days 58 and 70. On
T+75, the patient experienced frequent urination and acute
hematuria, which was diagnosed as hemorrhagic cystitis
with positive results for adenovirus and BK virus in the
urine specimen polymerase chain reaction (PCR) testing. In
terms of patient management, we implemented hydration,
analgesia, and administered scopolamine butylbromide and
oxybutynin upon the emergence of the initial symptoms.
Subsequently, the patient’s symptoms gradually improved
following these interventions.
On T+86, the patient developed a persistent fever (>39 ℃)
along with cytopenia, hyperferritinemia (highest level
of 58,325 ng/mL on T+101, with a pre-HSCT level of
1,038 ng/mL), hypertriglyceridemia (4.95 mmol/L), and
hypofibrinogenemia (1.7 g/L). Bone marrow aspiration
revealed an increased fraction of activated macrophages
(Figure 1A,1B) and high serum levels of sCD25 (>7,500 pg/mL),
Highlight box
Key ndings
• We present a noteworthy case of hematopoietic stem cell
transplantation (HSCT) that was complicated by adenoviral
infection, subsequently leading to hemophagocytic syndrome. This
rare condition was successfully treated using a combination therapy
involving ruxolitinib.
What is known and what is new?
• The rare complication of phagocytic syndrome may occur
subsequent to HSCT.
• Our case highlights the potential of ruxolitinib as a therapeutic
option for patients with viral infections and secondary hemophagocytic
lymphohistiocytosis (sHLH).
What is the implication, and what should change now?
• The safety and efcacy of ruxolitinib treatment in sHLH should be
evaluated in future large-scale clinical trials.

Li et al. Ruxolitinib salvage treatment for sHLH after HSCT
996
© Translational Pediatrics. All rights reserved. Transl Pediatr 2024;13(6):994-1000 | https://dx.doi.org/10.21037/tp-24-27
suggestive of HLH. Next-generation sequencing of peripheral
blood conrmed adenovirus type C infection (Figure 2A,2B).
The patient received cidofovir antiviral therapy (5 mg/kg/
week for two doses, then 5 mg/kg/2 weeks for three doses),
dexamethasone pulse therapy (10 mg/m2/day), and etoposide
(100 mg/m2/week for three doses) (Figure 3A). However,
his fever persisted, leading to transfer to the ICU for
plasma exchange and filtration therapy. After undergoing
plasmapheresis, there has been a temporary decrease in
the patient’s levels of inammatory factors; however, fever
remained unstable. Ruxolitinib (5 mg/day) was initiated on
+113 days, resulting in the stabilization of body temperature
and a decrease in inflammatory cytokine levels, such as
sCD25, interleukin-6 (IL-6), IL-10, TNF-α, and serum
ferritin (Figure 3B-3F). The patient responded well to
treatment, achieving negative adenovirus viral load by day
181 and no recurrence observed after fifteen months of
hospital discharge.
Discussion
In this report, we utilized ruxolitinib to treat a child with
refractory HLH associated with HSCT. Patients who
have viral infections induced by immunodeciency or who
are taking immunosuppressants for GvHD are at risk of
developing HLH following HSCT. However, HLH is a
rare occurrence in the context of HSCT. Diagnosing post-
HSCT HLH can be difcult because it presents clinicians
with various manifestations that can be confused with other
conditions such as capillary leak syndrome, cytokine release
syndrome (CRS), engraftment syndrome (ES), infections,
systemic inflammatory response syndrome (SIRS)/sepsis,
sinusoidal obstruction syndrome (SOS), and thrombotic
microangiopathy (TMA) (4). Furthermore, some of these
conditions can occur at the same time as post-HSCT
HLH. Ferritin levels in HSCT recipients do not strongly
correlate with GvHD, making them a potential biomarker
to differentiate it from sHLH (5). In our case, the patient’s
clinical manifestation fullled the 2004 classication criteria
for familial HLH (fHLH), and his serum ferritin level
exceeded 50,000 ng/mL at a certain juncture. Therefore, it
is reasonable to presume that he experienced a complication
of phagocytic syndrome rather than GvHD.
The rst case of HLH following allogeneic HSCT was
Table 1 Summary of relevant information, treatment, and prognosis of this case of hematopoietic stem cell transplantation
Age/sex Primary
diagnosis
Conditioning
regimen
Type of donor/
graft (HLA match)
GvHD
prophylaxis
Neutrophil
engraftment
Day of onset or
diagnosis of HLH Treatment Outcome
11 years/
male
SAA Bu/Cy + Flu
+ ATG
Haploidentical PB
(father)
CsA/MTX +14 +86 Dex/VP-16/
IVIg/cidofovir
Improved
SAA, severe aplastic anemia; Bu/Cy + Flu + ATG, busulfan + cyclophosphamide + fludarabine + antihuman thymocyte globulin;
HLA, human leukocyte antigen; PB, peripheral blood; GvHD, graft-versus-host disease; CsA/MTX, cyclosporine/methotrexate; HLH,
hemophagocytic lymphohistiocytosis; Dex, dexamethasone; IVIg, intravenous immunoglobulin.
A B
Figure 1 Activated macrophages were observed in the bone marrow at day 104 (Wright-Giemsa stain, ×400). (A) The bone marrow cell
morphology showed activated macrophages exhibiting hemophagocytosis in the bone marrow. (B) Activated macrophages that exhibited
hemophagocytosis in the bone marrow.

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Figure 2 Presents data pertaining to the patient’s adenovirus infection. (A) The genome coverage map of human adenovirus type C. The
horizontal axis represents the reference genome intervals of human adenovirus type C, with each interval representing 100 base pairs. The
coverage of human adenovirus type C in this sample is 46.67%. The primary vertical axis on the left represents the number of sequencing
reads, while the secondary vertical axis on the right represents the average sequencing depth, indicating the average number of times each
base is sequenced. Green denotes the number of sequencing reads within the intervals, and red denotes the average sequencing depth
within the intervals. (B) The real-time quantitative monitoring results of adenovirus in patients eventually achieved successful conversion to
negative. HSCT, hematopoietic stem cell transplantation.
Genome coverage of human mastadenovirus C
(46.67% covered)
Number of map reads
Depth
Copies/mL
60
50
40
30
20
10
0
25
20
15
10
5
0
0T +86
2.47×104
1.37×105
7.87×105
1.38×106
5.00×106
0
T +107 T +114 T +128 T +147 T +181
50 100 150 200 250 300 350
Nucleotide position (×100) genome size: 36,001 Post-HSCT day
Adenovirus
Map reads
Depth
A B
documented by Reardon et al. (6). A study conducted in
Tunisia investigated pediatric and adult cases of sHLH
after HSCT, revealing an overall incidence rate of 4%,
which increased to 8.8% when focusing solely on allogeneic
patients (7). Jaiswal et al. recently reported that sHLH
occurred in 12.2% of haploidentical peripheral-blood
HSCT recipients who received post-cyclophosphamide
(PTCY) as prophylaxis against GvHD, with a median onset
time of 18 days after transplantation (8).
HLH developing after HSCT consists of 2 types: early
and late-onset. The late-onset type is associated with
infection-associated HLH. Adenoviruses are among the
more common pathogens that increase the risk of post-
transplant hematopoietic syndrome, and several reports
have been published on this topic (Table 2). However, there
is no consensus regarding the treatment. In this case, the
patient experienced acute GvHD (grade II–III) shortly
after hematopoietic stem-cell transplantation. Active anti-
GvHD therapy further compromised the patient’s immune
system, leading to the activation of cytomegalovirus and
EBV, as well as the development of hemorrhagic cystitis.
These factors created favorable conditions for subsequent
adenovirus infection.
A case report has documented successful treatment of
adenovirus-associated hemophagocytic syndrome following
HSCT with ribavirin and cidofovir (10,12). However, it
is generally known that HLH caused by viral infection
does not typically respond to specific antiviral therapy,
but rather requires treatment with immunosuppressive
agents or immunomodulators (13). In the present case,
we promptly initiated antiviral treatment with cidofovir
along with glucocorticoids, intravenous immunoglobulin,
and etoposide to suppress the immune response, and
supplemented this with plasma exchange and filtration
support. After undergoing the aforementioned treatments,
the patient’s condition exhibited temporary improvement;
however, his body temperature failed to attain a stable state.
Ruxolitinib, a powerful JAK1/2 inhibitor administered
orally, exhibits favorable pharmacokinetic properties.
It has received approval from the US Food and Drug
Administration (FDA) for the treatment of patients
diagnosed with myeloproliferative neoplasms and steroid-
refractory GvHD (14-16). Ruxolitinib effectively hampers
the activity of both JAK1 and JAK2, which are downstream
of IFN-gamma and various other HLH-related cytokines,
including IL-2, IL-6, IL-10, IL-12, and granulocyte-
macrophage colony-stimulating factor (GM-CSF). Recent
literature has showcased the successful implementation
of ruxolitinib, a Janus-associated kinase inhibitor, in the
management of HLH (17-20). However, its routine usage for
sHLH management in the context of transplantation is not
yet established. Ono et al. recounted the successful treatment

Li et al. Ruxolitinib salvage treatment for sHLH after HSCT
998
© Translational Pediatrics. All rights reserved. Transl Pediatr 2024;13(6):994-1000 | https://dx.doi.org/10.21037/tp-24-27
Figure 3 Presents data regarding the treatment history of the patient’s complications and the alterations in inflammatory factor levels
subsequent to ruxolitinib treatment. (A) The treatment process for complications following patient transplantation. On day 16 post-
transplantation, the patient received 2 mg/kg of methylprednisolone for anti-GvHD therapy. On day 24 post-transplantation, the patient
was administered basiliximab (10 mg/d +1, +3, +8) and etanercept (12.5 mg biw) for combined anti-GvHD therapy. On day 92 post-
transplantation, intensied immunosuppressive therapy was carried out using dexamethasone (10 mg/m2/d) and immunoglobulin (0.2 g/kg
for 5 days). On day 98 post-transplantation, the patient underwent VP-16 chemotherapy (100 mg/m2 in a total of 3 doses) and received
cidofovir (5 mg/kg/week for two doses, then 5 mg/kg/2 weeks for three doses) for antiviral therapy. On day 102 post-transplantation, a
plasma exchange procedure was performed. Lastly, on day 113 post-transplantation, ruxolitinib was added at a dose of 5 mg/d. (B-F) Levels
of inammatory factors post-administration of ruxolitinib. HSCT, hematopoietic stem cell transplantation; GvHD, graft-versus-host disease;
biw, 2 times a week; TNF-α, tumor necrosis factor-α; IL, interleukin.
of two patients with HSCT-associated HLH employing
ruxolitinib (21). Based on the efficacy of ruxolitinib in
the management of refractory phagocytic syndrome and
corticosteroid-resistant GvHD, it was deemed appropriate
to incorporate ruxolitinib into the patient’s therapeutic
regimen at this juncture of their medical course. On day
113 after the transplantation, following the administration
of ruxolitinib (5 mg/d), the patient’s body temperature
gradually stabilized, and the inammatory factor test showed
a gradual decrease in serum ferritin, sCD25, TNF-α, and
IL-6 levels. Ruxolitinib was discontinued on day 137, and
there was no recurrence of HLH thereafter.
Serum cytokine profiles are valuable for tracking the
progression of HLH. However, these profiles may vary
in in cases related to hematopoietic cell transplantation
(HCT). In this study, we observed a significant decrease
in the levels of inflammatory factors like TNF-α and
IL-6 after administering ruxolitinib to the patient under
investigation. This finding provides valuable insight into
the potential mechanisms underlying the therapeutic
effectiveness of ruxolitinib in this specic case. Therefore,
monitoring the dynamic changes in various cytokines can
aid in evaluating illness activity and therapy response in
HLH patients.
One of the limitations of this study was the omission of
adenovirus antibody titration prior to patients undergoing
HSCT. The inclusion of these data would have provided
valuable insights into the patients’ baseline immune status.
Serum ferritin, ng/mLsCD25, U/mL
IL-10, pg/mL
IL-6, pg/mL
IL-6, pg/mL
TNF-α, pg/mL
TNF-α, pg/mL IL-10, pg/mL
sCD25, U/mL
70000
60000
50000
40000
30000
20000
10000
0
20000
18000
16000
14000
12000
10000
8000
6000
4000
2000
0
160
140
120
100
80
60
40
20
0
350
300
250
200
150
100
50
0
900
800
700
600
500
400
300
200
100
0
T+93
T+93
T+93T+93T+93
T+101 T+107
T+107
T+107T+107T+107
T+111
T+111
T+111T+111T+111
T+113
T+113
T+113T+113T+113
T+115
T+115
T+115T+115T+115
T+118
T+118
T+118T+118T+118
T+119
T+119
T+119T+119T+119
T+125
T+125
T+125T+125T+125
Serum ferritin, ng/mL
Ruxolitinib
Ruxolitinib
Ruxolitinib
Ruxolitinib
Ruxolitinib
T, ℃
40
39
38
37
T16
T34
T24
Basiliximab
Basiliximab
VP-16 100 mg/m2
Cidofovir 5 mg/kg
Plasma exchange
Ruxolitinib 5 mg/d
Etanercept 12.5 mg Biw
26 32 45
Methylprednisolone
2 mg/kg/day
T50
T92
T98
T94
T113
101 115
107 110T102
010 20 30 40 50 60 70 80 90 100 110 120 130 140 150 160
Post-HSCT
129 143
102 111
Dexamethasone
10 mg/m2/day
IVIG
200 mg/
kg/day*
5 day
A
D E
B
C
F

Translational Pediatrics, Vol 13, No 6 June 2024 999
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Table 2 Summary of adenovirus infection-associated HLH cases post-HSCT
Author
(years) Age/sex Primary
diagnosis
Conditioning
regimen
Type of donor/
graft (HLA match)
GvHD
prophylaxis
Neutrophil
engraftment
Day of onset
or diagnosis
of HLH
Treatment Outcome
Levy et al.,
1990 (9)
6 years/
female
Relapsed
Wilm’s tumor
Doxorubicin/
local RT/Mel
Autologous
BM
– Primary graft
failure
+60 IVIg Died on T+14
due to graft failure
Reardon
et al., 1991
(6)
8 years/
female
ALL (L1) with
isolated CNS
relapse
Bu/Cy MRD BM
(8/8)
CsA/CS +10 +38 None Died on T+42 due
to unresolved HLH/
multi-organ failure
Iyama et al.,
2005 (10)
51 years/
female
SAA – HLA-identical
sibling
– – – Oral
ribavirin
Improved
Lackner et al.,
2008 (11)
9.5 years/
female
AML (M2) Flu/Mel/ATG Haploidentical
BM (mother, −)
CsA/CS NR +24 CS/IVIg/
ribavarin
Alive 2 years
after HSCT
Nagamatsu
et al., 2021
(12)
65 years/
female
MDS – HLA-DRB1 one
locus mismatched
donor
– – +237 Cidofovir +
mPSL
Improved
HLH, hemophagocytic lymphohistiocytosis; HSCT, hematopoietic stem cell transplantation; HLA, human leukocyte antigen; GvHD,
graft-versus-host disease; RT, radiotherapy; BM, bone marrow; ALL, acute lymphoblastic leukemia; CNS, central nervous system; Bu/
Cy, busulfan + cyclophosphamide; MRD, matched related donor; CsA/CS, cyclosporine/corticosteroid; SAA, severe aplastic anemia;
AML, acute myeloid leukemia; Flu/Mel/ATG, fludarabine/melphalan/antihuman thymocyte globulin; NR, not report; MDS, myelodysplastic
syndrome; mPSL, methylprednisolone; IVIg, intravenous immunoglobulin.
Conclusions
In conclusion, we propose that sHLH should be carefully
considered in the evaluation of acutely or chronically
unwell allogeneic HSCT recipients who present with an
unexplained, culture-negative febrile illness, pancytopenia,
or coagulopathy, particularly those with GvHD, EBV
infection, or other viral reactivation. Furthermore,
ruxolitinib may serve as a viable therapeutic option for
individuals with refractory sHLH following HSCT.
However, extensive clinical trials are necessary to evaluate
the safety and efcacy of this potential treatment.
Acknowledgments
We gratefully acknowledge the pathologists in Children’s
Hospital of Nanjing Medical University, for their expertise
and generous provision of data.
Funding: This research was supported by the Natural
Science Foundation of Jiangsu Province (No. BK20211009)
and Scientific Research Projects of Jiangsu Health
Commission (No. ZDB2020018).
Footnote
Reporting Checklist: The authors have completed the CARE
reporting checklist. Available at https://tp.amegroups.com/
article/view/10.21037/tp-24-27/rc
Peer Review File: Available at https://tp.amegroups.com/
article/view/10.21037/tp-24-27/prf
Conicts of Interest: All authors have completed the ICMJE
uniform disclosure form (available at https://tp.amegroups.
com/article/view/10.21037/tp-24-27/coif). The authors
have no conicts of interest to declare.
Ethical Statement: The authors are accountable for all
aspects of the work in ensuring that questions related
to the accuracy or integrity of any part of the work are
appropriately investigated and resolved. All procedures
performed in this study were in accordance with the ethical
standards of the institutional and/or national research
committee(s) and with the Helsinki Declaration (as revised
in 2013). Written informed consent was obtained from the
parents of the patient for publication of this case report
and accompanying images. A copy of the written consent is
available for review by the editorial ofce of this journal.
Open Access Statement: This is an Open Access article
distributed in accordance with the Creative Commons

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License (CC BY-NC-ND 4.0), which permits the non-
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See: https://creativecommons.org/licenses/by-nc-nd/4.0/.
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Cite this article as: Li J, Wu Q, Guo X, Rong L, Fang Y.
Salvage treatment of ruxolitinib for refractory adenovirus-
associated hemophagocytic syndrome post-haploidentical
allogeneic stem cell transplantation: a case report. Transl
Pediatr 2024;13(6):994-1000. doi: 10.21037/tp-24-27