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Article ; Online: Primary Graft Failure but Treatment Success: A Case of Reversion to Heterozygosity After Allogeneic Hematopoietic Cell Transplantation With Autologous Hematopoietic Recovery in a Child With CBL-related Juvenile Myelomonocytic Leukemia.

Oshrine, Benjamin

Journal of pediatric hematology/oncology

2020 Volume 43, Issue 3, Page(s) e426–e428

Abstract: Juvenile myelomonocytic leukemia (JMML) typically requires allogeneic hematopoietic cell transplantation with full donor chimerism for cure. Certain genetic subtypes, including JMML due to germline mutations in CBL, can have a more indolent course. We ... ...

Abstract	Juvenile myelomonocytic leukemia (JMML) typically requires allogeneic hematopoietic cell transplantation with full donor chimerism for cure. Certain genetic subtypes, including JMML due to germline mutations in CBL, can have a more indolent course. We describe a young male patient with CBL-related JMML who experienced primary graft failure after allogeneic hematopoietic cell transplantation. Despite autologous recovery, the resulting hematopoietic tissue did not harbor the original homozygous CBL mutations, due to reversion of prior loss of heterozygosity of the 11q chromosomal region. The patient remains disease free without further leukemia-directed therapy.
MeSH term(s)	Germ-Line Mutation ; Hematopoietic Stem Cell Transplantation ; Heterozygote ; Humans ; Infant ; Leukemia, Myelomonocytic, Juvenile/genetics ; Leukemia, Myelomonocytic, Juvenile/therapy ; Male ; Proto-Oncogene Proteins c-cbl/genetics ; Transplantation, Homologous ; Treatment Outcome
Chemical Substances	Proto-Oncogene Proteins c-cbl (EC 2.3.2.27) ; CBL protein, human (EC 6.3.2.-)
Language	English
Publishing date	2020-02-03
Publishing country	United States
Document type	Case Reports ; Journal Article
ZDB-ID	1231152-2
ISSN	1536-3678 ; 1077-4114 ; 0192-8562
ISSN (online)	1536-3678
ISSN	1077-4114 ; 0192-8562
DOI	10.1097/MPH.0000000000001740
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Zs.A 1537: Show issues

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Article ; Online: Diagnostic and management roles of FDG PET/CT imaging in post-transplant lympho-proliferation in pediatric heart transplantation.

Brown, Abdel-Karim / Carapellucci, Jennifer / Oshrine, Benjamin / Gomez, Anthony / Meoded, Avner / Asante-Korang, Alfred

Clinical transplantation

2023 Volume 37, Issue 9, Page(s) e15015

Abstract: Background: Post-transplant lymphoproliferative disorder (PTLD) is a serious complication of pediatric heart transplant (PHTx). 18F-FDG PET/CT has been used to differentiate early lympho-proliferation from more advanced PTLD. We report our experience ... ...

Abstract	Background: Post-transplant lymphoproliferative disorder (PTLD) is a serious complication of pediatric heart transplant (PHTx). 18F-FDG PET/CT has been used to differentiate early lympho-proliferation from more advanced PTLD. We report our experience with PET/CT in the management of PTLD following PHTx. Methods: This was a retrospective study of 100 consecutive PHTx recipients at our institution between 2004 and 2018. Patients who underwent PET/CT or conventional CT scans to evaluate for PTLD or high Epstein-Barr viral load were included. Results: Males, eight females. Median age at transplant was 3.5 months (IQR = 1.5-27.5). Median age at PTLD diagnosis was 13.3 years (IQR = 9.2-16.1). Median time between transplant and PTLD diagnosis was 9.5 (IQR = 4.5-15) years. Induction agents were used in 12 patients (50%): Thymoglobulin (N = 9), anti-IL2 (N = 2), and Rituximab (N = 1). Eighteen patients (75%) had PET/CT, of whom 14 had 18FDG-avid PTLD. Six had conventional CT. Nineteen patients (79.2%) had diagnostic biopsy confirmation of PTLD, and 5 (20.8%) had excisional biopsies. Two patients had Hodgkin's lymphoma; nine had monomorphic PTLD; eight had polymorphic PTLD; five were classified as other. Nine patients had monomorphic PTLD, including seven with diffuse large cell lymphoma (DLBC) and one with T cell lymphoma. The majority (16/24) had multi-site involvement at PTLD diagnosis, and PET/CT showed that 31.3% (5/16) had easily accessible subcutaneous nodes. Seventeen patients (overall survival 71%) underwent successful treatment without recurrence of PTLD. Of seven deaths (7/24, 29%), five had DLBC lymphoma, one had polymorphic PTLD and one had T-cell lymphoma. Conclusion: PET-CT allowed simultaneous anatomical and functional assessment of PTLD lesions, while guiding biopsy. In patients with multiple lesions, PET/CT revealed the most prominent and active lesions, improving diagnostic accuracy.
MeSH term(s)	Heart Transplantation/adverse effects ; Humans ; Positron Emission Tomography Computed Tomography ; Fluorodeoxyglucose F18 ; Child ; Adolescent ; Child, Preschool ; Male ; Female ; Biopsy ; Lymphoma/diagnostic imaging ; Lymphoma/etiology ; Lymphoma/pathology
Chemical Substances	Fluorodeoxyglucose F18 (0Z5B2CJX4D)
Language	English
Publishing date	2023-05-26
Publishing country	Denmark
Document type	Journal Article
ZDB-ID	639001-8
ISSN	1399-0012 ; 0902-0063
ISSN (online)	1399-0012
ISSN	0902-0063
DOI	10.1111/ctr.15015
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Zs.A 2204: Show issues

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Article ; Online: Donor characteristics and intraoperative total nucleated cell count influence hematopoietic progenitor cell yield of healthy donor bone marrow grafts.

Kalin, Jacob / Nguyen, Anh Thy H / Oshrine, Benjamin

Pediatric blood & cancer

2021 Volume 68, Issue 4, Page(s) e28902

Abstract: Background: Bone marrow graft cell content impacts engraftment potential after allogeneic hematopoietic cell transplantation (alloHCT). Surrogates, such as intraoperative total nucleated cell count (ioTNC), are of unclear utility in predicting final ... ...

Abstract	Background: Bone marrow graft cell content impacts engraftment potential after allogeneic hematopoietic cell transplantation (alloHCT). Surrogates, such as intraoperative total nucleated cell count (ioTNC), are of unclear utility in predicting final graft characteristics. In addition, demographic and clinical factors may influence graft cellular profile and recipient engraftment. Procedure: We retrospectively reviewed marrow harvests at our institution performed between 2009 and 2019. During this time, an ioTNC was measured after 50% of the projected final graft volume was collected. Regression models were used to assess associations between ioTNC (cells/µL) and final graft CD34+ (cells/mL), and between graft and donor characteristics and final graft CD34+ (cells/mL). Results: Fifty-three marrow harvests and donor-recipient pairs were analyzed. Median (range) donor and recipient ages were 13 (0.7-28) years and 9 (0.2-21) years, respectively. The median ratio of donor/recipient weight was 1.225 kg (range 0.31-7.13). Median total volume of harvested marrow was 15.3 mL/kg (range 4.3-20.4) of donor weight and 19.4 mL/kg (range 4.7-87.4) of recipient weight. Median ioTNC was 20 930/µL (range 6600-44310) or 2.1 × 10 Conclusions: ioTNC and certain donor characteristics correlate moderately well with marrow product CD34+ cells/mL, potentially informing donor selection and marrow procurement strategies.
MeSH term(s)	Adolescent ; Adult ; Antigens, CD34/analysis ; Bone Marrow Cells/cytology ; Cell Count ; Child ; Child, Preschool ; Donor Selection ; Female ; Graft Survival ; Hematopoietic Stem Cell Transplantation/methods ; Hematopoietic Stem Cells/cytology ; Humans ; Infant ; Male ; Transplantation, Homologous/methods ; Young Adult
Chemical Substances	Antigens, CD34
Language	English
Publishing date	2021-01-23
Publishing country	United States
Document type	Journal Article
ZDB-ID	2131448-2
ISSN	1545-5017 ; 1545-5009
ISSN (online)	1545-5017
ISSN	1545-5009
DOI	10.1002/pbc.28902
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Article ; Online: Acute Left Ventricular Dysfunction Following Gemtuzumab Ozogamicin in Two Pediatric AML Patients.

McNerney, Kevin O / Oranges, Katelyn / Seif, Alix E / Oshrine, Benjamin / Ky, Bonnie / Lin, Kimberly Y / Getz, Kelly D / Aplenc, Richard

Journal of pediatric hematology/oncology

2022 Volume 44, Issue 2, Page(s) e507–e511

Abstract: Gemtuzumab ozogamicin (GO) is an anti-CD33 antibody-tumor antibiotic conjugate with proven efficacy in pediatric and adult patients with CD33+ acute myeloid leukemia. Adverse effects commonly associated with GO include hyperbilirubinemia, elevated ... ...

Abstract	Gemtuzumab ozogamicin (GO) is an anti-CD33 antibody-tumor antibiotic conjugate with proven efficacy in pediatric and adult patients with CD33+ acute myeloid leukemia. Adverse effects commonly associated with GO include hyperbilirubinemia, elevated transaminases, and sinusoidal obstruction syndrome. Cardiotoxicity has not been a commonly described adverse event. We describe 2 pediatric patients with relapsed/refractory acute myeloid leukemia who received fractionated GO monotherapy and subsequently developed severe acute left ventricular dysfunction. Both patients achieved remission, recovered cardiac function with medical therapy, and tolerated subsequent stem cell transplantation.
MeSH term(s)	Child ; Gemtuzumab/adverse effects ; Humans ; Leukemia, Myeloid, Acute/drug therapy ; Ventricular Dysfunction, Left/chemically induced
Chemical Substances	Gemtuzumab (93NS566KF7)
Language	English
Publishing date	2022-02-22
Publishing country	United States
Document type	Case Reports ; Research Support, N.I.H., Extramural
ZDB-ID	1231152-2
ISSN	1536-3678 ; 1077-4114 ; 0192-8562
ISSN (online)	1536-3678
ISSN	1077-4114 ; 0192-8562
DOI	10.1097/MPH.0000000000002325
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Article ; Online: Hemophagocytic lymphohistiocytosis in adults.

Bhatt, Neel S / Oshrine, Benjamin / An Talano, Julie

Leukemia & lymphoma

2018 Volume 60, Issue 1, Page(s) 19–28

Abstract: Hemophagocytic lymphohistiocytosis (HLH), a rare but life-threatening condition characterized by uncontrolled inflammation, is increasingly recognized in adults. The management of adult onset HLH is challenging, in part due to gaps in current state of ... ...

Abstract	Hemophagocytic lymphohistiocytosis (HLH), a rare but life-threatening condition characterized by uncontrolled inflammation, is increasingly recognized in adults. The management of adult onset HLH is challenging, in part due to gaps in current state of knowledge on etiology, clinical presentation, diagnosis, and management. HLH secondary to triggers such as infections, autoimmune disorders, and malignancy are more commonly seen in adults although cases of familial form have also been reported. Underlying conditions such as sepsis, or malignancy could pose as major confounders while applying universal diagnostic criteria, and therefore could lead to delay in diagnosis. Despite advent of newer therapeutic agents, outcomes of adults continue to remain poor. Future efforts need to be orchestrated to develop evidence-based tailored therapies to improve outcomes of this under recognized heterogeneous entity.
MeSH term(s)	Adult ; Age of Onset ; Anti-Infective Agents/therapeutic use ; Clinical Trials as Topic ; Disease-Free Survival ; Drug Therapy, Combination ; Evidence-Based Medicine/methods ; Hematopoietic Stem Cell Transplantation/methods ; Humans ; Immunosuppressive Agents/therapeutic use ; Lymphohistiocytosis, Hemophagocytic/diagnosis ; Lymphohistiocytosis, Hemophagocytic/mortality ; Lymphohistiocytosis, Hemophagocytic/therapy ; Myeloablative Agonists/administration & dosage ; Neoplasm Recurrence, Local/mortality ; Neoplasm Recurrence, Local/therapy ; Prognosis ; Remission Induction/methods ; Salvage Therapy/methods ; Transplantation Conditioning/methods ; Transplantation, Homologous/methods
Chemical Substances	Anti-Infective Agents ; Immunosuppressive Agents ; Myeloablative Agonists
Language	English
Publishing date	2018-07-03
Publishing country	United States
Document type	Journal Article ; Review
ZDB-ID	1042374-6
ISSN	1029-2403 ; 1042-8194
ISSN (online)	1029-2403
ISSN	1042-8194
DOI	10.1080/10428194.2018.1482543
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Zs.A 2997: Show issues

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Article ; Online: Early Recovery of Myeloid-Derived Suppressor Cells After Allogeneic Hematopoietic Transplant: Comparison of Post-Transplantation Cyclophosphamide to Standard Graft-Versus-Host Disease Prophylaxis.

Oshrine, Benjamin / Innamarato, Patrick / Branthoover, Holly / Nagle, Luz / Verdugo, Patrick / Pilon-Thomas, Shari / Beatty, Matthew

Transplantation and cellular therapy

2022 Volume 28, Issue 4, Page(s) 203.e1–203.e7

Abstract: Allogeneic hematopoietic cell transplantation (alloHCT) using haploidentical donors (haploHCT) with post-transplantation cyclophosphamide (PTCy) for augmented graft-versus-host disease (GVHD) prophylaxis has emerged as a robust platform to expand donor ... ...

Abstract	Allogeneic hematopoietic cell transplantation (alloHCT) using haploidentical donors (haploHCT) with post-transplantation cyclophosphamide (PTCy) for augmented graft-versus-host disease (GVHD) prophylaxis has emerged as a robust platform to expand donor options with acceptable levels of GVHD and graft failure. The mechanism by which PTCy mitigates GVHD risk is partly explained by preferential cytotoxicity based on aldehyde dehydrogenase levels and up-regulation of regulatory T cells, but is incompletely understood. Myeloid-derived suppressor cells are important mediators of T-cell function and are up-regulated by cyclophosphamide exposure. We hypothesized that this cell type may play a role in GVHD protection in children undergoing haploHCT/PTCy. We prospectively collected samples in the first month after alloHCT from children undergoing standard of care (SOC) alloHCT with matched donors and tacrolimus-based GVHD prophylaxis (N = 11) and PTCy recipients (N = 11). MDSC recovery was compared using flow cytometry, and MDSC suppressive function was assessed at the peak of MDSC quantitative recovery post-alloHCT. Groups were well matched for conditioning regimen and stem cell source. PTCy recipients exhibited more robust MDSC recovery, particularly polymorphonuclear-MDSCs than SOC recipients, with preservation of T-cell suppressive function. This corresponded to significantly lower incidence of Grade II to IV acute GVHD (9.1% versus 27.3%) and moderate/severe chronic GVHD (0% versus 27.3%) in PTCy recipients. Patients who developed GVHD had decreased MDSC-mediated T-cell suppression, as well as higher levels of IL-10, a cytokine closely linked to GVHD biology. Overall, these findings provide support for the role of MDSCs in mediating GVHD protection after PTCy-based haploHCT. © 2022 American Society for Blood and Marrow Transplantation. Published by Elsevier Inc. All rights reserved.
MeSH term(s)	Child ; Cyclophosphamide/pharmacology ; Graft vs Host Disease/prevention & control ; Hematopoietic Stem Cell Transplantation ; Humans ; Myeloid-Derived Suppressor Cells ; Transplantation Conditioning ; United States
Chemical Substances	Cyclophosphamide (8N3DW7272P)
Language	English
Publishing date	2022-01-05
Publishing country	United States
Document type	Journal Article
ZDB-ID	3062231-1
ISSN	2666-6367
ISSN (online)	2666-6367
DOI	10.1016/j.jtct.2021.12.019
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Zs.A 5082: Show issues

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Article: Curative treatment for severe sickle cell disease: allogeneic transplantation.

Oshrine, Benjamin / Talano, Julie-An

Clinical advances in hematology & oncology : H&O

2015 Volume 13, Issue 4, Page(s) 249–256

Abstract: Sickle cell disease is an inherited hematologic disorder that in its severe form can result in substantial morbidity and early mortality. Patients with this disorder can suffer from severe pain, lung disease, and strokes, resulting in chronic ... ...

Abstract	Sickle cell disease is an inherited hematologic disorder that in its severe form can result in substantial morbidity and early mortality. Patients with this disorder can suffer from severe pain, lung disease, and strokes, resulting in chronic debilitating conditions, end organ dysfunction, and organ failure. The health care costs of caring for these chronically ill patients are substantial. Allogeneic transplantation is a modality that has the potential to cure these patients. To date, matched sibling donor transplantation is widely accepted as a standard of care for pediatric patients. Utilizing alternative donors for transplant is still under investigation, as is transplant for adult patients with sickle cell disease. This review focuses on the most recent data for hematopoietic cell transplantation for patients with sickle cell disease.
MeSH term(s)	Anemia, Sickle Cell/therapy ; Cord Blood Stem Cell Transplantation ; Haplotypes ; Hematopoietic Stem Cell Transplantation ; Histocompatibility Testing ; Humans ; Siblings ; Tissue Donors ; Transplantation Conditioning ; Transplantation, Homologous
Language	English
Publishing date	2015-04
Publishing country	United States
Document type	Journal Article ; Review
ZDB-ID	2271951-9
ISSN	1543-0790
ISSN	1543-0790
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Zs.A 6505: Show issues

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Article ; Online: Low-dose azacitidine for relapse prevention after allogeneic hematopoietic cell transplantation in children with myeloid malignancies.

Oshrine, Benjamin R / Shyr, David / Hale, Gregory / Petrovic, Aleksandra

Pediatric transplantation

2019 Volume 23, Issue 4, Page(s) e13423

Abstract: Background: The prognosis of children who relapse after allogeneic hematopoietic cell transplant (alloHCT) for myeloid malignancies remains poor.: Procedure: To describe the safety and feasibility of post-transplant azacitidine for relapse prevention, ...

Abstract	Background: The prognosis of children who relapse after allogeneic hematopoietic cell transplant (alloHCT) for myeloid malignancies remains poor. Procedure: To describe the safety and feasibility of post-transplant azacitidine for relapse prevention, we retrospectively reviewed the charts of 18 children undergoing alloHCT for myeloid malignancies. Results: There were 15 evaluable patients since three patients did not receive planned azacitidine due to early relapse or TRM. Azacitidine (32 mg/m Conclusions: Post-transplant azacitidine in children is safe and feasible, with most patients successfully receiving all planned cycles. Despite the limitations of a small cohort, low relapse incidence suggests a potential benefit in disease control that warrants further investigation.
MeSH term(s)	Adolescent ; Antimetabolites, Antineoplastic/administration & dosage ; Azacitidine/administration & dosage ; Child ; Child, Preschool ; Female ; Hematopoietic Stem Cell Transplantation ; Humans ; Leukemia, Myeloid, Acute/therapy ; Male ; Myelodysplastic Syndromes/therapy ; Neoplasm Recurrence, Local/prevention & control ; Prognosis ; Remission Induction ; Retrospective Studies ; Secondary Prevention ; Tacrolimus ; Transplantation, Homologous ; Treatment Outcome ; Young Adult
Chemical Substances	Antimetabolites, Antineoplastic ; Azacitidine (M801H13NRU) ; Tacrolimus (WM0HAQ4WNM)
Language	English
Publishing date	2019-04-23
Publishing country	Denmark
Document type	Journal Article
ZDB-ID	1390284-2
ISSN	1399-3046 ; 1397-3142
ISSN (online)	1399-3046
ISSN	1397-3142
DOI	10.1111/petr.13423
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Zs.A 4947: Show issues

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Article: Effectiveness and Complication Rate of Percutaneous Endoscopic Gastrostomy Placement in Pediatric Oncology Patients.

Kidder, Molly / Phen, Claudia / Brown, Jerry / Kimsey, Kathryn / Oshrine, Benjamin / Ghazarian, Sharon / Mateus, Jazmine / Amankwah, Ernest / Wilsey, Michael

Pediatric gastroenterology, hepatology & nutrition

2021 Volume 24, Issue 6, Page(s) 546–554

Abstract: Purpose: Malnutrition is a significant issue for pediatric patients with cancer. We sought to evaluate the effectiveness and complication rate of percutaneous endoscopic gastrostomy (PEG) placement in pediatric oncology patients.: Methods: A ... ...

Abstract	Purpose: Malnutrition is a significant issue for pediatric patients with cancer. We sought to evaluate the effectiveness and complication rate of percutaneous endoscopic gastrostomy (PEG) placement in pediatric oncology patients. Methods: A retrospective chart review was performed on 49 pediatric oncology patients undergoing PEG placement at Johns Hopkins All Children's Hospital between 2000 and 2016. Demographic and clinical characteristics, complications, absolute neutrophil count at time of PEG placement and at time of complications, length of stay, and mortality were identified. Weight-for-age Z-scores were evaluated at time of- and six months post-PEG placement. Results: The overall mean weight-for-age Z-score improved by 0.73 ( Conclusion: Consistent with previous studies, our data suggests a relationship between site complications (superficial wound infection, buried bumper syndrome) and neutropenia. Additionally, PEG placement appears to be an effective modality for improving nutritional status in malnourished pediatric oncology patients. However, larger prospective studies with appropriate controls and adjustment for potential confounders are warranted to confirm these findings.
Language	English
Publishing date	2021-11-05
Publishing country	Korea (South)
Document type	Journal Article
ZDB-ID	3032413-0
ISSN	2234-8840 ; 2234-8646
ISSN (online)	2234-8840
ISSN	2234-8646
DOI	10.5223/pghn.2021.24.6.546
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Article ; Online: Allogeneic hematopoietic cell transplantation in chemotherapy-induced aplasia in children with high-risk acute myeloid leukemia or myelodysplasia.

Kussman, Ashleigh / Shyr, David / Hale, Gregory / Oshrine, Benjamin / Petrovic, Aleksandra

Pediatric blood & cancer

2018 Volume 66, Issue 1, Page(s) e27481

Abstract: Relapse remains the most common cause of treatment failure after hematopoietic cell transplantation for acute myeloid leukemia. Inability to achieve hematologic complete remission has been a barrier to transplant for patients with refractory disease. We ... ...

Abstract	Relapse remains the most common cause of treatment failure after hematopoietic cell transplantation for acute myeloid leukemia. Inability to achieve hematologic complete remission has been a barrier to transplant for patients with refractory disease. We describe six children with refractory myeloid disease undergoing transplant in chemotherapy-induced aplasia, as a strategy to facilitate curative therapy in refractory patients. Clofarabine- or high-dose cytarabine-based chemotherapy regimens were used to achieve marrow aplasia, followed by reduced-intensity conditioning and allogeneic transplant before hematologic recovery. Long-term disease control was achieved in five, with one transplant-related mortality, suggesting the feasibility of this approach.
MeSH term(s)	Adolescent ; Antineoplastic Combined Chemotherapy Protocols/adverse effects ; Child ; Child, Preschool ; Cytarabine/administration & dosage ; Female ; Follow-Up Studies ; Graft vs Host Disease/etiology ; Graft vs Host Disease/therapy ; Hematopoietic Stem Cell Transplantation/methods ; Humans ; Leukemia, Myeloid, Acute/pathology ; Leukemia, Myeloid, Acute/therapy ; Male ; Myelodysplastic Syndromes/pathology ; Myelodysplastic Syndromes/therapy ; Neoplasm Recurrence, Local/pathology ; Neoplasm Recurrence, Local/therapy ; Prognosis ; Red-Cell Aplasia, Pure/chemically induced ; Red-Cell Aplasia, Pure/therapy ; Retrospective Studies ; Transplantation Conditioning ; Transplantation, Homologous ; Vidarabine/administration & dosage
Chemical Substances	Cytarabine (04079A1RDZ) ; Vidarabine (FA2DM6879K)
Language	English
Publishing date	2018-10-14
Publishing country	United States
Document type	Journal Article
ZDB-ID	2131448-2
ISSN	1545-5017 ; 1545-5009
ISSN (online)	1545-5017
ISSN	1545-5009
DOI	10.1002/pbc.27481
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