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Outcomes of Referrals in Pediatric Patients With Peripheral Lymphadenopathy. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-04-23 Kathryn A Six, Inmaculada B Aban, Gabriel M Daniels, Julie Wolfson, Elizabeth A Beierle, Matthew A Kutny, Jeffrey Lebensburger, Ana C Xavier
Lymphadenopathy is a common reason for referral to a subspecialist, which may result in significant anxiety for parents. Understanding which patients require a subspecialty referral for lymphadenopathy is key to streamlining health care utilization for this common clinical entity. This is an IRB-approved retrospective study examining pediatric patients consecutively referred to pediatric hematology
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CAR-T Therapy Followed by Hematopoietic Stem Cell Transplantation Can Improve Survival in Children Relapsed/Refractory Philadelphia Chromosome-positive B-cell Acute Lymphoblastic Leukemia. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-04-23 Yao Li, Guan-Hua Hu, Lan-Ping Xu, Xiao-Hui Zhang, Kai-Yan Liu, Pan Suo, Yu Wang, Yi-Fei Cheng, Xiao-Jun Huang
Philadelphia chromosome (Ph)-positive B-cell acute lymphoblastic leukemia (ALL) has a high complete remission (CR) rate, but relapse and prolonged measurable residual disease remain serious problems. We sought to describe the CR rate measurable residual disease negative rate and address the results and safety of pediatric patients who underwent after receiving chimeric antigen receptor (CAR) specific
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Variable Clinical Courses of Varicella Zoster Virus Infection-related or Vaccination-related Bone Marrow Failure. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-04-23 Vasil Toskov, Annamaria Cseh, Alexander Claviez, Beatrice Drexler, Natalia Rotari, Stephan Schwarz-Furlan, Matthias Braun, Peter Bader, Peter Lang, Rita Beier, Bernhard Erdlenbruch, Monika Führer, Miriam Erlacher, Charlotte M Niemeyer, Brigitte Strahm, Ayami Yoshimi
We report 5 children with bone marrow failure (BMF) after primary varicella zoster virus (VZV) infection or VZV vaccination, highlighting the highly variable course. Two patients were treated with intravenous immunoglobulins; one had a slow hematologic recovery, and the other was rescued by allogeneic hematopoietic stem cell transplantation (HSCT). Of the 2 patients treated with immunosuppressive therapy
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Unveiling the Uncommon: A Unique Case of ALPS-Like Syndrome Complicated by Plasma Cell Disorder. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-04-23 Meziane Brizini, Bruno Michon, Marc-Antoine Bédard
Multiple myeloma is a rare disease in pediatrics, where about 30 cases are described under 15 years old. It is even rarer when atypical multiple myeloma occurs in the context of autoimmunity. This case describes a 9-year-old female with autoimmune lymphoproliferative-like disease and combined immune deficiency that developed acute kidney failure with monoclonal peak associated with RAC2 and TNFRSF9
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Very Long-Term Survivorship in Pediatric DIPG: Case Report and Review of the Literature. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-04-04 Evan Dimentberg, Marie-Pier Marceau, Alexandre Lachance, Samuel Bergeron-Gravel, Stephan Saikali, Louis Crevier, Catherine Bourget, Cynthia Hawkins, Nada Jabado, Panagiota Giannakouros, Samuele Renzi, Valérie Larouche
Diffuse intrinsic pontine gliomas are lethal tumors with a prognosis generally less than 1 year. Few cases of survivors of 5 years or more have been reported. This case report highlights the journey of a 9.5-year survivor who underwent 3 rounds of focal radiotherapy; she experienced 6 years of progression-free survival following the first round but ultimately succumbed to her disease. An autopsy revealed
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The Diagnostic Performance of Magnetic Resonance Imaging in the Categorization of Pediatric Neck Lymph Nodes: Radiologic and Pathologic Correlations. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-04-04 Emine Caliskan, Naz Paytoncu, Hanife G Düzkalır, Meral Arifoglu, Neriman Fistikcioglu, Hediye P Gunbey
To present MRI features of neck lymph nodes in benign and malignant conditions in the pediatric population.
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A Single-Center Experience of Radiotherapy in Pediatric Ewing Sarcoma/Primitive Neuroectodermal Tumor of the Chest Wall. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-04-03 Ecem Yigit, Melis Gultekin, Burca Aydin, Tezer Kutluk, Sezin Yuce Sari, Saniye Ekinci, Diclehan Orhan, Ferah Yildiz
To evaluate the treatment results, prognostic parameters, and treatment-related toxicity in patients with Ewing sarcoma (ES)/primitive neuroectodermal tumor (PNET) of the chest wall who underwent surgery, chemotherapy, and radiotherapy (RT) in a tertiary referral center.
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Opioid Use Among Children and Adults With Sickle Cell Disease in North Carolina Medicaid Enrollees in the Era of Opioid Harm Reduction. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-03-28 Nancy Crego, Christian Douglas, Emily Bonnabeau, Kern Eason, Marian Earls, Paula Tanabe, Nirmish Shah
Adults and children with sickle cell disease (SCD) are predominantly African American, with pain-related health disparities. We examined opioid prescription fill patterns in adults and children with SCD and compared factors associated with fills in North Carolina Medicaid enrollees. Our retrospective cohort study included 955 enrollees diagnosed with SCD having at least one opioid fill. Associations
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The Utility of Serum Alpha-fetoprotein for Monitoring for Relapse of Alpha-fetoprotein-Positive Hepatoblastoma. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-03-27 Monica Davini, Caroline Hastings, James Feusner
Hepatoblastoma is the most common liver malignancy in children. Treatment typically involves surgery and cisplatin-based chemotherapy. After therapy completion, children undergo repetitive surveillance imaging to screen for relapse, which occurs in <12% of cases. Monitoring for relapse has gradually shifted to serial determination of serum alpha-fetoprotein (AFP) alone as most cases have AFP elevation
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Delayed Rewarming Thrombocytopenia (DRT): A Suggested Preventable and Treatable Cause of Rewarming Deaths. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-03-07 Ian J Cohen
The lack of a consensus of accepted prognostic factors in hypothermia suggests an additional factor has been overlooked. Delayed rewarming thrombocytopenia (DRT) is a novel candidate for such a role. At body temperature, platelets undergoing a first stage of aggregation are capable of progression to a second irreversible stage of aggregation. However, we have shown that the second stage of aggregation
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Mediastinal Germ-cell Tumors Relapse in a Male With Klinefelter Syndrome. Is Longer Surveillance Needed? J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-03-06 Francesca Stefanachi, Maria Carmen Affinita, Giulia Fichera, Arianna Tagarelli, Federica De Corti, Federico Rea, Gianni Bisogno
Germ cell tumors (GCTs) are a heterogeneous group of pediatric cancers. In up to one-third of male patients, a primary mediastinal location is associated with the presence of Klinefelter syndrome (KS). We describe a case of mediastinal GCT in a patient, with unacknowledged KS, that presented a relapse 7 years from diagnosis, that is, 2 years after the end of the follow-up program usually recommended
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Recurrence Patterns and Surveillance Imaging in Pediatric Brain Tumor Survivors. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-03-05 Chantel Cacciotti, Alicia Lenzen, Chelsea Self, Natasha Pillay-Smiley
Surveillance magnetic resonance imaging (MRI) is routinely used to detect recurrence in pediatric central nervous system (CNS) tumors. The frequency of neuroimaging surveillance varies without a standardized approach. A single-institutional retrospective cohort study evaluated the frequency of recurrences. This study included 476 patients with the majority diagnosed with low-grade glioma (LGG) (n=138
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Nasopharyngeal Carcinoma in Children, Current Treatment Approach. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-03-05 Tal Ben-Ami
Nasopharyngeal carcinoma (NPC) is a rare and locally aggressive form of childhood cancer. Treatment of pediatric NPC includes chemotherapy and radiotherapy. Most studies on the treatment of pediatric NPC are single-arm studies. With current treatment protocols survival rates for patients with nonmetastatic disease exceed 80%, although most children will have long-term treatment-related late effects
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The Incidence of Postoperative Complications Following Lumbar and Bone Marrow Punctures in Pediatric Anesthesia: Insights From APRICOT. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-03-04 Krystelle Dagher, Claudia Benvenuti, Kathy Virag, Walid Habre
Bone marrow aspiration and lumbar puncture are procedures frequently performed in pediatric oncology. We aimed at assessing the incidence and risk factors of perioperative complications in children undergoing these procedures under sedation or general anesthesia.
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Investigating the Impact of Tumor Biology and Social Determinants on Time to Diagnosis and Stage at Presentation of Wilms Tumor. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-03-04 Adele Collins, Elizabeth Molina Kuna, Amy Anderson-Mellies, Carrye Cost, Adam L Green
Delays in diagnosis and time to diagnosis generally are used interchangeably in cancer disparity research, but these terms may have important differences. Although these terms are related, we hypothesize that time to diagnosis is determined by the aggressiveness of the tumor based on intrinsic factors such as tumor biology, whereas delays in diagnosis are caused by extrinsic factors such as socioeconomic
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Use of Maribavir for Multidrug Resistant Cytomegaloviremia in a Pediatric Oncology Patient. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-03-01 James E Fisher, Kevin Mulieri, Elizabeth Finch, Jessica E Ericson
Resistant and refractory cytomegalovirus (CMV) viremia can limit the provision of chemotherapy due to myelosuppression and end-organ dysfunction. Few therapies are available for children with clinically significant CMV viremia. We successfully used maribavir for a 4-year-old patient with lymphoma to complete his chemotherapy course. Resistance to maribavir did result after many months of therapy.
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"Prehabilitation" for Pediatric Patients With Cancer Before Undergoing Hematopoietic Stem Cell Transplantation. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-03-01 Lily Sandblom, Maya Keole, Corinne Winsten, Ray Stanford
Hematopoietic stem cell transplantation (HSCT) is associated with significant physical debility. Studies have suggested that physical activity can improve endurance and strength. However, studies have not yet investigated "prehabilitation," exercise before HSCT, to improve functional outcomes. We describe the impact of exercise therapy on physical debility in children undergoing HSCT to inform the
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Successful Treatment of Acquired Thrombotic Thrombocytopenic Purpura With Caplacizumab Combined With Plasma Exchanges and Immune Suppression in 3 Children. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-29 Irina I Kalinina, Khristina S Antonova, Pavel V Avdonin, Elizaveta E Klebanova, Natalia N Kotskaya, Elena E Kurnikova, Alexandra D Shutova, Victor E Matveev, Alexey A Maschan
Acquired thrombotic thrombocytopenic (aTTP) purpura is a life-threatening condition that can lead to devastating thromboembolic events. Recently, caplacizumab has been shown to rapidly restore platelet numbers and reduce the risk of severe end-organ damage when added to plasma exchanges (PEXs) and immunosuppression (IST). Here, we report the outcomes in 3 children with aTTP who were treated with caplacizumab
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Acute Myeloid Leukemia and Next-Generation Sequencing Panels for Diagnosis: A Comprehensive Review. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-29 Spandan Chaudhary, Pooja Chaudhary, Firoz Ahmad, Neeraj Arora
Acute myeloid leukemia (AML) is a genetically heterogeneous clonal disorder characterized by the accumulation of acquired somatic genetic alterations in hematopoietic progenitor cells, which alter the normal mechanisms of self-renewal, proliferation, and differentiation. Due to significant technological advancements in sequencing technologies in the last 2 decades, classification and prognostic scoring
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Bartonella henselae Infection and Lymphadenopathy in a Patient With T Cell Acute Lymphoblastic Leukemia. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-29 Arhanti Sadanand, Pratik Patel, Erica Riedesel, Frank Berkowitz, Frank G Keller
Patients undergoing therapy for T cell acute lymphoblastic leukemia are at risk of infections during their treatment course. Cat scratch disease caused by Bartonella hensalae can masquerade as leukemic relapse and cause systemic infection. Obtaining a thorough exposure history may aid clinicians in making the diagnosis.
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Hypoglycemia Associated With PEG-asparaginase and 6-MP Therapy During Treatment of Acute Lymphoblastic Leukemia in Pediatric Patients: A Case Series. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-27 Mary R Jiang, Alexandra Ahmet, Sarah Lawrence, Mylene Bassal, Matthew Speckert, Michael T Geraghty, Scott Somerville
Asparaginases are a mainstay treatment for pediatric acute lymphoblastic leukemia (ALL). Recent reports identified hypoglycemia associated with asparaginases. Other reports describe hypoglycemia associated with 6-mercaptopurine (6-MP), another fundamental ALL therapy. Little is known about the risk of hypoglycemia associated with ALL therapy, an adverse event that puts children at risk of decreased
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A Rare Inherited Bone Marrow Failure Syndrome Disclosed by Reanalysis of the Exome Data of a Patient Evaluated for Cytopenia and Dysmorphic Features. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-27 Durmus Durmaz, Ayca Dilruba Aslanger, Zehra Yavas Abali, Yasin Yilmaz, Volkan Karaman, Gozde Yesil Sayin, Guven Toksoy, Aysegul Unuvar, Zehra Oya Uyguner
Multisystemic findings of inherited bone marrow failure syndromes may cause difficulty in diagnosis. Exome sequencing (ES) helps to define the etiology of rare diseases and reanalysis offers a valuable new diagnostic approach. Herein, we present the clinical and molecular characteristics of a girl who was referred for cytopenia and frequent infections.
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Pediatric Plexiform Fibromyxoma: A Case Report. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-26 Patricia Bugeda Gómez, Adrià Costa-Roig, Carolina Montecino Romanini, Ignacio Miró Rubio, Santiago Guindos Rúa, Diana C Lara Cárdenas, Micaela Germani, María Roca Roca, Cleofe Romagosa Pérez Portabella, Marta Garrido Pontnou, Javier Hernández Losa, Luis F Sanchís Solera
The plexiform fibromyxoma is a rare mesenchymal tumor in adults that generally originates in the antrum of stomach, being its occurrence in pediatric patients exceptional. It was classified as a distinct entity by World Health Organization in 2010. No recurrences and metastases have been documented in many of the reported patients to date, being the surgical treatment curative. We report the case of
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Effective Treatment of Anlotinib Combined With Chemotherapy in Children With Desmoplastic Small Round Cell Tumor: A Case Series in a Single-center and Literature Review. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-22 Xiao-Yu Jing, Cheng-Qi Shen, Guo-Qian He, Rong-Rong Xu, Ju Gao, Xia Guo
Desmoplastic small round cell tumor (DSRCT) is a highly aggressive primitive sarcoma with a 5-year survival rate estimated at only 15% to 30%. Although few curative treatment options exist, patients are most often treated with a combination of aggressive chemotherapy, radiation, and surgery. Targeted therapy inhibitors of platelet-derived growth factor A, insulin-like growth factor receptor 1, and
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Comparative Analysis of Skip Metastasis in Pediatric Osteosarcoma: Clinical Features and Outcomes. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-19 Hadeel Halalsheh, Shrouq Amer, Zaid Omari, Munir Shawagfeh, Mohammad Boheisi, Iyad Sultan
Skip metastasis (SM) is a synchronous regional bone metastasis. Using new imaging modalities, the detection of SM is easier and possibly more common. We reviewed patients with SM and compared their characteristics and outcomes to other patients with osteosarcoma treated at our center.
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Superb Microvascular Imaging in Pediatric Focal Nodular Hyperplasia. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-19 Sook Min Hwang, So-Young Yoo, Woo Kyoung Jeong, Min Woo Lee, Tae Yeon Jeon, Ji Hye Kim
To investigate superb microvascular imaging (SMI), a novel Doppler ultrasound technique that can visualize low-velocity microvascular flow, for assessing pediatric focal nodular hyperplasia (FNH).
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Recurrent Nontraumatic Subgaleal Hematomas in a Pediatric Patient With Sickle Cell Disease. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-19 Irtiza N Sheikh, Olayinka Okeleji, Rabya Afzal, Eliana Bonfante, Monica Kodakandla, Neethu M Menon
Spontaneous subgaleal hematoma in pediatric patients with sickle cell disease (SCD) is a rare occurrence that can present with symptoms mimicking ischemic stroke, a known complication of SCD. However, unlike ischemic stroke, subgaleal hematoma is nonlethal and can be managed conservatively without major sequelae. Here, we present the case of an adolescent with SCD who presented with 2 episodes of subgaleal
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B-Lymphoblastic Lymphoma in Children: A Case Series From a Single Institution. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-16 Kaitlin J Devine, Hirva Trivedi, Anne F Reilly
Pediatric B-lymphoblastic lymphoma is an uncommon subtype of non-Hodgkin lymphoma. Studies regarding the biology, clinical course, and approach to relapse are limited.
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Severe Hemolytic Anemia: Atypical Presentation of Cobalamin Deficiency. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-15 Carolina Fraga, Ana Losa, Inês Cascais, Cristina Garrido, Ana Lachado, Isabel Couto Guerra, Anabela Bandeira, Esmeralda Cleto, Emília Costa
Two severe cases of hemolytic anemia are described in different pediatric age groups, both linked to severe cobalamin deficiency from distinct causes. The first case refers to an exclusively breastfed infant with vitamin deficit secondary to maternal impaired absorption. Apart from the neurological deficits present at diagnosis, he also presented with infantile epileptic spasms syndrome a few months
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Central Nervous System Metastases in Pediatric Patients with Ewing Sarcoma. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-02 Leonora R Slatnick, Carrye Cost, Timothy Garrington, Nathan Donaldson, Margaret E Macy
Metastatic central nervous system (CNS) involvement is rare in pediatric primary extracranial Ewing sarcoma (ES). We describe the incidence and course of 6 patients with extracranial ES who developed metastatic CNS lesions treated at a single institution. The median time to CNS disease detection was 16.3 months (10.0-28.3 months). Event-free and overall survival after CNS disease detection were 1.9
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Second Malignant Neoplasms Following Treatment for Hepatoblastoma: An International Report and Review of the Literature. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-02 Angela Trobaugh-Lotrario, Kenichiro Watanabe, Allison F O'Neill, Bozenna Dembowska-Bagińska, Beate Häberle, Andrew Murphy, Eiso Hiyama, Piotr Czauderna, Rebecka L Meyers, Max Langham, James Feusner
Treatment intensification has improved survival in patients with hepatoblastoma (HB); however, these treatments are associated with an increased risk of late effects, including second malignant neoplasms (SMNs). Data is limited regarding SMNs following HB treatment. Cases of SMNs following treatment for HB reported in the literature and from personal communication were analyzed to further assess this
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Clinical Analysis of Pediatric Acute Megakaryocytic Leukemia With CBFA2T3-GLIS2 Fusion Gene. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-02-01 Yu Du, Li Yang, Shanshan Qi, Zhi Chen, Ming Sun, Min Wu, Bin Wu, Fang Tao, Hao Xiong
CBFA2T3-GLIS2 is the most frequent chimeric oncogene identified to date in non-Down syndrome acute megakaryocytic leukemia (AMKL), which is associated with extremely poor clinical outcome. The presence of this fusion gene is associated with resistance to high-intensity chemotherapy, including hematopoietic stem cell transplantation (HSCT), and a high cumulative incidence of relapse frequency. The clinical
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Raccoon Eyes Presentation in a Child Diagnosed With Acute Lymphoblastic Leukemia: A Case Report. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-01-29 Özlem Terzi, Cengiz Bayram, Hüseyin Avni Solgun, Duygu Yildirgan, Ali Ayçiçek
Raccoon's eyes (periorbital ecchymosis) may present as the first sign in patients with skull base/base/facial fractures and tumors. In childhood, orbital metastases of neuroblastoma should be considered in the absence of trauma history. Herein, we report a 3-year-old girl diagnosed with acute lymphoblastic leukemia who presented with periorbital ecchymosis. To the best of our knowledge, this is the
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Intracranial Germinoma in Two Caucasian American Siblings With Autism Spectrum Disorder. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-01-29 Stephanie A Toll, Leigh Anne Flore, Hamza S Gorsi, Neena I Marupudi, Swati Mody, William Kupsky, Zhihong Joanne Wang
Intracranial germ cell tumors (IGCTs) comprise 3% to 5% of all pediatric brain tumors in the West, with a significantly higher prevalence in Asia. Although these tumors are histologically diverse, repeated somatic variants have been demonstrated. Chromosomal aneuploidies, such as Klinefelter and Down syndromes, are associated with IGCTs, but no familial germline tumor syndromes are currently known
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School Attendance Among Pediatric Oncology Patients During the COVID-19 Pandemic in Ontario, Canada. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-01-25 Jacob Joel Kirsh Carson, Helen Coo, Mohammed Al Nuaimi, Angela Punnett, Kirk Leifso, Laura Wheaton
Supporting schooling for current and past pediatric oncology patients is vital to their quality of life and psychosocial recovery. However, no study has examined the perspectives toward in-person schooling among pediatric oncology families during the COVID-19 pandemic. In this online survey study, we determined the rate of and attitudes toward in-person school attendance among current and past pediatric
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Malignancies and Lymphoproliferations in Children With Primary Immune Deficiency-A Single-center Experience. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-01-25 Cigdem Aydogmus, Ozge Turkyilmaz Ucar, Sibel Kaplan Sarikavak, Funda Cipe, Selami Ulas, Isilay Turan, Sezin Naiboglu, Ilke Yildirim, Gulsah Kalay, Nermin Kapci, Serdar Al, Pinar Gokmirza Ozdemir, Mehmet Halil Celiksoy
Primary immune deficiencies (PIDs) are rare genetic disorders characterized by impaired immune function, leading to frequent infections and immune dysregulation. Studies have shown that individuals with PID are at an increased risk of developing malignancies and lymphoproliferative disorders compared with the general population. In this single-center study, we aimed to analyze the occurrence of malignancies
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Normal Erythroid Precursors in Diamond-Blackfan Anemia: A Rare Case Highlighting Challenges That Remain. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-01-25 Daniel Prior, Anna Sowa, Farzana Pashankar
Diamond-Blackfan anemia (DBA) is a rare, inherited bone marrow failure syndrome that is both genetically and clinically heterogeneous. The diagnosis of DBA has changed over time, with advancements in our understanding of the varied genetic etiologies and phenotypic manifestations of the disease. We present a rare case of a patient who never developed erythroid precursor hypoplasia, adding to the understanding
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Allogeneic Hematopoietic Cell Transplantation Ameliorated Asymptomatic Granulomatous and Lymphocytic Interstitial Lung Disease in a Patient With XIAP Deficiency. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-01-22 Mai Oshima, Yukihiro Matsukawa, Yuhachi Ikeda, Kenichi Sakamoto, Takashi Taga, Yoshihiro Maruo
X-linked inhibitor of apoptosis protein (XIAP) deficiency is an inborn error of immunity (IEI). Allogeneic hematopoietic cell transplantation (HCT) is currently the only curative therapy available for XIAP deficiency. Granulomatous and lymphocytic interstitial lung disease (GLILD) is a common immune-related lung complication of IEIs. We present a 6-year-old boy with XIAP deficiency and GLILD. Computed
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Eating Behavior, Nutritional Status, and Taste Perception Alteration in Children with Cancer. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-01-17 Yasemin Fistikçi, Elif Bilsin Kocamaz
The aim of this study was to investigate eating behavior, nutritional status, and taste alterations in children with cancer. The population of the study consisted of children 8 to 18 years of age and their parents who were followed up and received chemotherapy in the pediatric hematology and oncology clinic and outpatient clinic of a University Faculty of Medicine Oncology Hospital. Data were collected
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Characterization of the Use and Efficacy of Isavuconazonium Sulfate in a Pediatric Oncology and Stem Cell Transplant Population: A Single Institution Retrospective Review. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-01-17 Binni Kunvarjee, Molly Siver, Sherry Mathew, Samantha Steiger, Yeon Joo Lee, Barbara Spitzer
Isavuconazonium sulfate (ISA) is a triazole antifungal approved for the treatment of invasive aspergillosis and mucormycosis in adults. This single-center, retrospective review of pediatric oncology and stem cell transplant patients receiving ISA for prophylaxis (n=20) or treatment (n=6) of invasive fungal disease (IFD) aims to characterize real-world clinical efficacy and toxicity of ISA in patients
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Perspectives of Adolescents and Young Adults With Sickle Cell Disease and Clinicians on Improving Transition Readiness With a Video Game Intervention. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-01-15 Paul L Aronson, Sarah A Nolan, Paula Schaeffer, Kimberly D Hieftje, Kortney A Ponce, Cecelia L Calhoun
We aimed to learn the experiences of clinicians and adolescents and young adults with sickle cell disease (AYA-SCD) with managing their disease at home and making medical decisions as they transition from pediatric to adult care, and their perceptions of a video game intervention to positively impact these skills. We conducted individual, semistructured interviews with patients (AYA-SCD ages 15 to
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Bloodstream Infections in Pediatric Oncology Patients: Bacterial Pathogen Distribution and Antimicrobial Susceptibility at the University Hospital Centre Zagreb, Croatia-A 5-year Analysis. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-01-15 Ivana Mareković, Tajana Vujnović, Jasna Radanović, Zoran Herljević, Tomislav Kuliš, Maja Pavlović, Lucija Mucavac, Sara Dejanović-Bekić, Ernest Bilić
The epidemiology of bacterial pathogens causing bloodstream infections (BSIs) in pediatric hematology/oncology patients is changing and resistance to antimicrobial agents is globally spread. We retrospectively assessed demographic, clinical, and microbiologic data of BSIs during a 5-year period at a pediatric hematology/oncology unit from January 1, 2017, to December 31, 2021, at the University Hospital
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Partial Response to Naxitamab for Brain Metastasis in Neuroblastoma. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-01-08 Chinyere Onyeukwu, Autumn Williams, Brian Seyboth, Lorenzo Muñoz, Gladson Scaria, Paul Kent
Neuroblastoma (NBL) is a common pediatric tumor arising from sympathetic ganglion cells. High-risk NBL is based on age, stage, histology, and MYCN amplification, and is associated with a high mortality rate. The combination of naxitamab (NAX) and granulocyte-macrophage (cerebrospinal fluid) is a new treatment for high-risk and relapsed NBL approved for bone or bone marrow disease. NAX is a monoclonal
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Coronavirus Disease 2019 mRNA Vaccination Appears Safe in Pediatric Patients With Hypersensitivity to Polyethylene Glycolated Escherichia Coli L-asparaginase. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2024-01-05 Nicole Wolfset, Amir Reza Pashmineh Azar, Charles A Phillips, Madison Stein, Susan R Rheingold, Jennifer Heimall, Caitlin W Elgarten
Polyethylene glycol-asparaginase (PEGAsp) is an established component of acute leukemia therapy. Hypersensitivity reactions to PEGAsp occur in 10% to 15% of patients, with polyethylene glycol suggested as the antigenic culprit. As coronavirus disease 2019 (COVID-19) mRNA vaccines contain polyethylene glycol, the safety of administration of these vaccines to patients with prior PEGAsp hypersensitivity
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Treatment of Monomorphic Posttransplant Lymphoproliferative Disorder in Pediatric Solid Organ Transplant: A Multicenter Review. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-12-25 Catherine Mark, Georgina Martin, Bjorn Baadjes, Ashley V Geerlinks, Angela Punnett, Lucie Lafay-Cousin
Posttransplant lymphoproliferative disorder (PTLD) is the most common posttransplant malignancy in children. We reviewed data from 3 Canadian pediatric centers to determine patient characteristics, treatment approaches, and outcomes for children with monomorphic PTLD. There were 55 eligible children diagnosed between January 2001 to December 2021. Forty-eight patients (87.2%) had B-cell PTLD: Burkitt
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Iron Overload in Children With Leukemia; Identification of a Cutoff Value for Serum Ferritin Level. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-12-22 Omid Reza Zekavat, Farima Fallah Tafti, Mohammadreza Bordbar, Shirin Parand, Sezaneh Haghpanah
To determine the prevalence of iron overload in children with acute lymphoblastic leukemia (ALL) after treatment cessation and establish a cutoff value for serum ferritin level as an indicator of iron overload.
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Patterns and Outcomes of Acute Central Nervous System Complications During Treatment of Childhood Acute Lymphoblastic Leukemia: A Single-center Experience. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-12-22 Asmaa Hamoda, Mohamed Bedair, Samah Fathy Semary, Ayda Youssef, Hanafy Hafez
Central nervous system (CNS) complications are considered adverse events during the treatment of pediatric acute lymphoblastic leukemia (ALL). This study aimed to assess the incidence, types, clinical and radiologic patterns, risk factors, and the fate of different CNS complications during the treatment of pediatric ALL. A retrospective study included 390 patients with pediatric ALL, treated according
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Increased Incidence of TdT-negative Pre-B Acute Lymphoblastic Leukemia Associated With Poor Prognostic Features Among Mexican Children in Central Mexico. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-12-22 Meagan Vacek, Laura C Zárraga Vargas, Elizabeth González Domínguez, Lilia Garcia Rodriguez, Omar A Tello Teran, Carlos Mojica Cardoso, Luisa V Ocampo Roosens, Juan J Acevedo Fernandez, Weijie Li, Brian Lee, Jesús S Olalla Tapia, Terrie Flatt
Mexican and Hispanic children in Mexico and the United States, respectively, have the highest incidence and worst outcomes of pre-B acute lymphoblastic leukemia (ALL) compared with other racial/ethnic groups. Terminal deoxynucleotidyl transferase (TdT) is an intranuclear DNA polymerase normally present on immature lymphocytes (TdT-positive) and distinguishes ALL from mature lymphoid malignancies. We
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Noonan Syndrome-related Myeloproliferative Disorder Occurring in the Neonatal Period: Case Report and Literature Review. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-12-22 Yuki Hoshino, Kunihiko Moriya, Kanako Mitsui-Sekinaka, Yu Hashimoto, Satoshi Nakayama, Daichi Sajiki, Hideki Muramatsu, Hidetoshi Hagiwara, Shuichi Suzuki, Yujin Sekinaka, Hajime Wakamatsu, Hiroyuki Kawaguchi, Kohsuke Imai
Noonan syndrome-related myeloproliferative disorder (NS/MPD) and juvenile myelomonocytic leukemia (JMML) are rare MPDs that occur in young children. We herein report a case of NS/MPD with neonatal onset. The patient had a characteristic appearance and high monocyte count in the peripheral blood and bone marrow. Genetic testing showed the E139D mutation in PTPN11; however, the patient did not meet all
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Hematopoietic Bone Marrow Transplant to Treat Systemic EBV-positive T-cell Lymphoma of Childhood. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-12-20 Nicholas Manini, Kimberly Davidow, Katrina Conard, Jonathan Powell
Systemic Epstein-Barr virus-positive T-cell lymphoma of childhood (S-EBV-TCL) is a rare disease for which there is no standard of care. S-EBV-TCL is often associated with hemophagocytic lymphohistiocytosis and is generally thought of on the spectrum of EBV-related disease. For the few reported cases of cure in the literature, hematopoietic stem cell transplant has been required because it is the only
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Coexistence of Bloom Syndrome and Kostmann Disease and a Novel Mutation. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-12-18 Esra Pekpak Sahinoglu, Ayse Ceyda Oren, Bahtiyar Sahinoglu, Ugur Gumus, Sinan Akbayram
Bloom syndrome (BS) is a rare autosomal recessive inherited disorder. Patients with BS have photosensitivity, telangiectatic facial erythema, and stunted growth. They usually have mild microcephaly, and distinctive facial features such as a narrow, slender face, micrognathism, and a prominent nose. Kostmann disease (KD) is a subgroup of severe congenital neutropenias. The diagnosis of severe congenital
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Mediastinal Radiotherapy-induced Early-onset Valvulopathy in a 6-year-old Boy With Hodgkin Lymphoma. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-12-18 Mustafa Doğan, Uğur Demirsoy, Eviç Zeynep Başar, Kadir Babaoğlu
Mediastinal radiotherapy for childhood cancers, particularly Hodgkin disease, has numerous potential adverse effects, including coronary artery disease, pericarditis, cardiomyopathy, valvular disease, and conduction abnormalities. The prevalence of valvular stenosis is relatively low, and regurgitation is more common. Mediastinal radiotherapy-induced valvular disease develops more than 10 years after
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Successful Crizotinib-targeted Therapy of Pediatric Unresectable ERC1::ALK Fusion Sarcoma. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-12-15 Megan L Wood, Julie C Fanburg-Smith, James M Brian, Jason C White, Jonathan L Powell, Andrew S Freiberg
Anaplastic lymphoma kinase (ALK)-fusion sarcomas are rare part of the emerging theoretically targetable tyrosine kinase RAS::MAPK pathway fusion myopericytic-ovoid sarcomas. We report our clinicopathologic and treatment experience with an ALK fusion sarcoma. A novel ELKS/RAB6-interacting/CAST family member 1-unaligned ALK fusion infiltrative nonmetastatic low-grade sarcoma of the right hand of a 15-month-old
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Hybrid Email and Outpatient Clinics to Optimize Maintenance Therapy in Acute Lymphoblastic Leukemia. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-12-12 Tushar Mungle, Ananya Mahadevan, Parag Das, Amit K Mehta, Manash P Gogoi, Bishwaranjan Jana, Niharendu Ghara, Debjani Ghosh, Vaskar Saha, Shekhar Krishnan
Acute lymphoblastic leukemia treatment includes an outpatient (OP)-based 2-year maintenance therapy (MT). Over an 8-year period, patients were transited from only OP to a hybrid e-clinic/OP-clinic model. Electronic and patient-held medical records of acute lymphoblastic leukemia patients aged 1 to 18 years during MT were used to analyze demographics, drug doses, treatment response and cost. A survey
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Six Years of Disease-free Survival After a Second Cord Blood Transplantation for Recurrent Acute Lymphocytic Leukemia in a Child With Down Syndrome. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-12-08 Momoka Aoshima, Hiroshi Yagasaki, Katsuyoshi Shimozawa, Koji Kanezawa, Masaru Ueno, Ichiro Morioka
Outcomes are extremely poor in Down syndrome-associated acute lymphocytic leukemia, particularly in recurrent cases. A 2-year-old boy with Down syndrome-associated acute lymphocytic leukemia achieved complete remission after standard chemotherapy. However, he experienced recurrence twice in the bone marrow and central nervous system. Salvage treatments included whole-brain/whole-spine irradiation.
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Successful Treatment for Hepatoblastoma in Trisomy 18: A Case Report. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-12-08 Kazuki Shirane, Ai Yoshimi, Takayuki Masuko, Daigo Kajikawa, Miki Toma, Hiroto Idesawa, Yugo Tsukada, Yusuke Yano, Keisuke Kato, Keiichi Motoyama, Nobuyoshi Asai, Koji Hirono, Tatsuo Kono, Haruo Otani, Junko Shiono, Isho Izumi, Toshihiro Yanai
Children with trisomy 18 tend to develop hepatoblastoma. Since the introduction of appropriate management for organ malfunction, individuals with trisomy 18 have come to have a longer life expectancy. However, the predisposition to hepatoblastoma becomes a significant issue for the quality of a case. Here, we present a rare multifocal hepatoblastoma involving predominantly Couinaud segments 5 and 7
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Sirolimus-induced Hypertriglyceridemia Leads to Acute Pancreatitis and Diabetic Ketoacidosis Post Stem Cell Transplant for Sickle Cell Disease. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-11-30 Subhasish Paul, Garima Nirmal, Vaibhav Chadha, Rahul Sharma, Ips Kochar, Namith Jerath, Rohit Vohra, Gaurav Kharya
Sirolimus (mammalian target of rapamycin inhibitor) is a potent immunosuppressive agent, used in patients receiving hematopoietic stem cell transplant (HSCT) for Graft vs Host disease prophylaxis. Compared to calcineurin inhibitors, sirolimus has no neurotoxicity or nephrotoxicity, but sirolimus causes dose-dependent thrombocytopenia, leukopenia, delayed wound healing, hyperlipidemia, and hypertriglyceridemia
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Case of Hemolytic Disease of the Fetus and Newborn Treated Without Blood Products. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-11-30 Leah Hirschman, Ashley Munchel
Hemolytic disease of the fetus and newborn (HDFN) affects 3/100,000 to 80/100,000 patients yearly and can cause severe anemia and hyperbilirubinemia. Recombinant human erythropoietin has been used as an adjunct therapy in patients with HDFN and hypo-regenerative anemia in the setting of receiving intrauterine blood transfusions. This case describes a patient with HDFN, in which the family were Jehovah
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Bevacizumab in the Treatment of Refractory Brain Edema in High-grade Glioma. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-11-30 Aysha K Alsahlawi, Claudie Michaud-Couture, Alexandre Lachance, Samuel Bergeron-Gravel, Mélanie Létourneau, Catherine Bourget, Peter V Gould, Panagiota Giannakouros, Emily M Nakada, Damien Faury, Louis Crevier, Éric Bouffet, Nada Jabado, Valérie Larouche, Samuele Renzi
We report the case of a 14-year-old boy with a steroid-dependent refractory tumor whose longstanding dexamethasone treatment was successfully discontinued after a course of bevacizumab. The use of bevacizumab despite the absence of clear evidence of radionecrosis allowed a significant decrease in the amount of the brain edema.
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Optical Genome Mapping Reveals Novel Structural Variants in Lymphoblastic Lymphoma. J. Pediatr. Hematol. Oncol. (IF 1.2) Pub Date : 2023-11-29 Hanli Xu, Huixia Gao, Chanjuan Wang, Xiyu Cheng, Zhigang Li, Cui Lei, XiaoTong Huang, Weijing Li, Zhixia Yue, Shuo Tian, Xiaoxi Zhao, Tianlin Xue, Tianyu Xing, Jun Li, Ying Wang, Yanlong Duan, Tianyou Wang, Ruidong Zhang
Accurate histologic and molecular genetic diagnosis is critical for the pathogenesis study of pediatric patients with lymphoblastic lymphoma (LBL). Optical genome mapping (OGM) as all-in-one process allows the detection of most major genomic risk markers, which addresses some of the limitations associated with conventional cytogenomic testing, such as low resolution and throughput, difficulty in ascertaining