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Journal of Pediatric Hematology/oncology

Jaikumar Ramamoorthy, Amita Trehan, Jasmina Ahluwalia, Paramjeet Singh, Rohit Manoj, Deepak Bansal
Independence from regular transfusions is the hallmark of nontransfusion-dependent thalassemia (NTDT). However, the associated complications need anticipation and screening. One such complication is a hypercoagulable state predisposing to development of thrombosis. We evaluated children with NTDT >10 years of age for prevalence of neuroimaging abnormalities (NIA) and identified associated risk factors. In total, 29 patients were evaluated. Blood counts, serum ferritin, protein C, protein S, antithrombin III, brain magnetic resonance imaging, and angiography was done in all patients...
February 12, 2019: Journal of Pediatric Hematology/oncology
Tiraje Celkan, Hande Kizilocak, Melike Evim, Adalet Meral Güneş, Namik Y Özbek, Neşe Yarali, Ekrem Ünal, Türkan Patiroğlu, Deniz Yilmaz Karapinar, Nazan Sarper, Emine Zengin, Serap Karaman, Ülker Koçak, Emin Kürekçi, Canan Özdemir, Deniz Tuğcu, Ezgi Uysalol, Gürcan Dikme, İbrahim Adaletli, Sebuh Kuruoğlu, Rejin Kebudi
BACKGROUND: Invasive fungal infections, including hepatosplenic fungal infections (HSFI), cause significant morbidity and mortality in children with leukemia. There are not enough data to support for the best approach to diagnosis of HSFI in children, nor for the best treatment. PROCEDURE: In this multicentric study, we assessed the demographic data, clinical and radiologic features, treatment, and outcome of 40 children with leukemia and HSFI from 12 centers. RESULTS: All cases were radiologically diagnosed with abdominal ultrasound, which was performed at a median of 7 days, of the febrile neutropenic episode...
February 5, 2019: Journal of Pediatric Hematology/oncology
Suvir Singh, Chepsy C Philip, Manthanathu J John
Subcutaneous panniculitis-like T-cell lymphoma (SPTCL) is a distinct subtype of peripheral T-cell lymphoma associated with aggressive clinical behavior. Since its original description, it has continued to be a rare disease, and <200 cases have been reported in literature. We report an 11-year-old boy who presented with SPTCL and hemophagocytic lymphohistiocytosis (HLH) and responded to high-dose multiagent chemotherapy. He presented with steroid refractory erythematous, raised plaques over his face, trunk, and limbs over a period of 15 months treated elsewhere...
February 5, 2019: Journal of Pediatric Hematology/oncology
Eu Jeen Yang, Kyung Mi Park, Yoo-Mi Kim, Ki Sun Jung, Young Tak Lim, Chong Kun Cheon
May-Hegglin anomaly (MHA) is a rare autosomal dominant disorder caused by a mutation in the myosin heavy chain 9 (MYH9) gene. MHA patients have variable clinical manifestations including thrombocytopenia, renal injury, hearing impairment, and cataracts. We describe a 25-year-old man with isolated thrombocytopenia initially. He experienced recurrent seizures with stable thrombocytopenia after the first seizures related to intracranial hemorrhage. He was identified a novel c.3452C>T mutation by targeted exome sequencing...
February 1, 2019: Journal of Pediatric Hematology/oncology
Luisa Barón González de Suso, Marta Olmedilla Jódar, Vanesa Pérez Alonso, Ana Melgar Bonis
INTRODUCTION: Enterocolitis is a relatively common disease in neonatal period that can be a result of many underlying pathologies. One of them, which is an unusual disorder especially in neonatal age and with gastrointestinal involvement, is Langerhans cell histiocytosis (LCH). This case shows a severe neonatal LCH with digestive involvement which required intensive care and had an abnormal presentation, being hard to diagnose attributable to the diversity of symptoms. CASE REPORT: Eleven-day-old newborn presented for excessive weight loss followed by deterioration to shock, abdominal distension, digestive bleeding, and purpuric exanthema...
February 1, 2019: Journal of Pediatric Hematology/oncology
Crystal S Lim, Cynthia Karlson, Sara N Edmond, Josie S Welkom, Ifeyinwa Osunkwo, Lindsey L Cohen
Sickle cell disease (SCD) is associated with pain and decreased health-related quality of life (HRQOL). Coping strategies influence pain but have not been evaluated as mediating the relation between pain and HRQOL in pediatric SCD. The current study examined whether pain-related coping mediates the association between pain and HRQOL in children and adolescents with SCD. In total, 104 children and adolescents aged 8 to 18 years (Mage=12.93 y) with SCD attending outpatient clinics completed pain intensity, HRQOL, and pain-related coping measures...
February 1, 2019: Journal of Pediatric Hematology/oncology
Aarthi Jayanthan, Bradley Hofmann, Vanessa Meier-Stephenson, Maneka Perinpanayagam, Sandra E Dunn, Jessica Boklan, Tanya M Trippett, Tony H Truong, Aru Narendran
BACKGROUND: Recent studies have shown that cell cycle events are tightly controlled by complex and shared activities of a select group of kinases. Among these, polo-like kinases (Plks) are regulatory mitotic proteins that are overexpressed in several types of cancer and are associated with poor prognosis. MATERIALS AND METHODS: We have evaluated, in preclinical in vitro studies, the activity of a panel of Plk inhibitors against cell lines derived from refractory pediatric leukemia, as well as primary leukemia cells, in culture...
January 29, 2019: Journal of Pediatric Hematology/oncology
Jian Wang, Hong-Man Xue, Yan-Ru Chen, Hong-Gui Xu, Shao-Fen Lin, Xi-Kang Tang, Chun Chen
OBJECTIVE: Hyperglycemia increases the risk of early recurrence and high mortality in some adult blood cancers. In response to increased glucose levels, insulin is secreted, and several studies have shown that insulin-induced AKT signaling can regulate tumor cell proliferation and apoptosis. The AKT pathway is aberrantly activated in adult acute lymphoblastic leukemia (ALL), but the mechanisms underlying this activation and its impact in pediatric patients with ALL are unclear. MATERIALS AND METHODS: We evaluated the insulin-induced chemoresistance and AKT pathway activation by measuring cell proliferation, apoptosis, and other parameters in ALL cell lines (Jurkat and Reh cells), as well as in primary pediatric leukemic cell samples, after culture with insulin, the chemotherapeutic drugs daunorubicin (DNR), vincristine (VCR), and L-asparaginase (L-Asp), or anti-insulin-like growth factor-1 receptor (IGF-1R) monoclonal antibody...
January 25, 2019: Journal of Pediatric Hematology/oncology
Valérie Lemay, Maxime Caru, Mariia Samoilenko, Simon Drouin, Nathalie Alos, Geneviève Lefebvre, Emile Levy, Sarah Lippé, Valérie Marcil, Serge Sultan, Laurence Bertout, Maja Krajinovic, Caroline Laverdière, Marie-Josée Raboisson, Daniel Sinnett, Gregor Andelfinger, Daniel Curnier
BACKGROUND: Most childhood acute lymphoblastic leukemia (ALL) survivors develop chronic treatment-related adverse effects several years after the end of therapy. A regular practice of physical activity and a good cardiorespiratory fitness have the potential to reduce the risk of chronic disease and improve quality of life. The aim of this study was to evaluate in a cohort of ALL survivors, the association between a good cardiorespiratory fitness or the respect of physical activity guidelines and major long-term health outcomes...
January 25, 2019: Journal of Pediatric Hematology/oncology
Hiromasa Fujii, Kanya Honoki, Takashi Ishihara, Toshiaki Shinomiya, Shinji Tsukamoto, Akira Kido, Yumiko Kondoh, Shingo Kishi, Midori Shima, Yasuhito Tanaka
Few studies have examined the relationship between functional outcome and sociooccupational or psychological status in adolescent and young adults (AYA) generation and childhood sarcoma patients. We retrospectively analyzed clinical (prognostic and functional) and sociooccupational outcomes in 50 patients; 22 children aged under 14 years and 28 AYAs generation (15 to 29 y). There were 35 cases of bone sarcomas and 15 of soft tissue sarcomas. Limb-sparing surgery was performed in 30 of 37 extremity cases. The most prevalent problems among patients were as follows: limited activities; drop-out or delayed studies among high school and college students; limitation in job searching; and changes in social relationships...
January 25, 2019: Journal of Pediatric Hematology/oncology
Naoki Hirano, Kazuo Nishikawa, Takahiro Hiratsuka, Souichi Suenobu, Kenji Ihara
Childhood cancer survivors (CCSs) from high-grade malignancies, such as high-risk neuroblastoma, have been increased, and second malignant neoplasm, becomes a serious problem for CCSs. However, detailed reports about rare types of second cancer such as gastric cancer remain limited. We herein reported a female patient who developed diffuse type gastric carcinoma after 21 years from completion of treatment to high-risk neuroblastoma. We reviewed the previous cohort studies for second gastrointestinal cancer in CCSs and the case reports with second gastric carcinoma for CCSs...
January 25, 2019: Journal of Pediatric Hematology/oncology
Sultan Aydin Köker, Bengü Demirağ, Neryal Tahta, Nuri Bayram, Yeşim Oymak, Tuba H Karapinar, Salih Gözmen, Mine Düzgöl, Hazer Erçan Bozyer, Canan Vergin, İlker Devrim
BACKGROUND: Acute viral respiratory infections are common causes of febrile episodes in children. There are still limited data about distribution of acute viral respiratory infections in children with cancer. OBJECTIVE: The first aim of this study was to evaluate the viral etiology and seasonality of acute viral respiratory infection in pediatric patients with cancer in a 3-year study. Our second aim was to evaluate the impact of viral infections on delaying the patients' chemotherapy or radiotherapy...
January 25, 2019: Journal of Pediatric Hematology/oncology
Mohamed Fawzy, Hossam El Zomor, Salma El Menawi, Naglaa Elkinaai, Gehad Ahmed, Amal Refaat, Mai Elahmadawy, Mohamed Zaghloul
Neuroblastoma (NBL) in infants has the potential to regress/mature spontaneously. The literature showed some cases, subjected to initial observation, with reasonable outcome. Deferring/avoiding active treatment was investigated in selected favorable NBL cases. Patients enrolled on the watch and see strategy (W&S) had small primary tumor, localized stages 1 to 2, uncomplicated stage 4s, or stage 3. Tissue biopsy was not mandatory for infants below 6 months with localized mass. On progression, active intervention was indicated according to disease stage and risk after biological characterization...
January 22, 2019: Journal of Pediatric Hematology/oncology
Nobuyoshi Kusano, Naoki Sakata, Keisuke Sugimoto, Tomoki Miyazawa, Satoshi Ueda, Munehiro Okano, Ken-Ichi Imadome, Akihiro Hoshino, Hirokazu Kanegane, Masatomo Kimura, Takao Sato, Mitsuru Okada, Tsukasa Takemura
X-linked lymphoproliferative syndrome type 1 (XLP1) is a rare congenital immunodeficiency disease. We report the case of an 18-year-old male who developed hemophagocytic lymphohistiocytosis (HLH) with neurological complications after primary Epstein-Barr virus (EBV) infection and subsequently developed EBV-related central nervous system lymphoma (CNSL). Given the vulnerability to EBV, he was finally diagnosed with XLP1 and treated with whole-brain irradiation along with chemotherapy and subsequent allogeneic hematopoietic stem cell transplantation from a SH2D1A wild-type sibling donor...
January 22, 2019: Journal of Pediatric Hematology/oncology
Amanda Scheuermann, Meghan Belongia, Michael W Lawlor, Mariko Suchi, Bruce Kaufman, Varshini Vasudevaraja, Jonathan Serrano, Matija Snuderl, Jeffrey Knipstein
Congenital tumors account for 2% to 4% of all pediatric central nervous system tumors. Glioblastoma multiforme (GBM) represents a small subset of these tumors. Despite harboring histologic features similar to older patients, infants with GBM exhibit improved survival and respond more favorably to surgery and chemotherapy. To highlight this tumor's unique behavior, we report the case of a survivor of infantile GBM who developed a recurrent tumor in the surgical bed 6 months after diagnosis. The tumor was ultimately resected and was a ganglioglioma...
January 22, 2019: Journal of Pediatric Hematology/oncology
Elena Soto-Vega, Luis S Carrillo-Vicente, Juan C Vázquez, María C Pérez de Celis Herrero, María J Muñoz-Pérez
Cancer treatments are associated with short and long-effects. Epidemiological reports have revealed clinical features of metabolic syndrome (MS), obesity or overweight in young cancer survivors. The aim of the study was to examine the prevalence of unhealthy weight status and risk factors associated with MS related to chemotherapy. We study 52 pediatric cancer patients and analyze cholesterol, triglycerides, glycosylated hemoglobin, body mass index, waist circumference (WC), FINDRISC test. All the parameters were analyzed according to the percentile corresponding to sex and age of each child...
January 22, 2019: Journal of Pediatric Hematology/oncology
Miriam Kim, Seema Rao, Jens C Eickhoff, Kenneth B DeSantes, Christian M Capitini
Hepatic sinusoidal obstruction syndrome (SOS) remains a serious complication of hematopoietic stem cell transplantation (HSCT). In this single institution retrospective case series, 18 children developed SOS after HSCT. Patients were treated with antithrombin III (ATIII), defibrotide, or ATIII followed by defibrotide. Twelve of 13 patients who were treated with ATIII therapy alone had complete resolution of SOS, including 4 of 5 children with severe SOS. In this limited cohort, ATIII was safe and successfully prevented progression of hepatic SOS following HSCT in the majority of children at our center...
January 22, 2019: Journal of Pediatric Hematology/oncology
Dona S Raju, Aadharsa Sugunan, Pavithran Keechilattu, Arun Philip, Remya Reghu
Trichosporon asahii is a rare opportunistic fungal pathogen that causes fatal systemic infection in immunocompromised patients. Neutropenia developing due to malignancies is an important risk factor for fungal infection. Invasive infections due to T. asahii can be divided into disseminated and localized forms. The disseminated form is more common and usually occurs in neutropenic patients. The patient typically has an acute febrile illness that progresses rapidly to multiorgan failure. Here, we are presenting a case of fungal sepsis by invasive T...
January 22, 2019: Journal of Pediatric Hematology/oncology
Mohsen Mirzakhani, Mohammad J Tarrahi, Atefe Baghersad, Mohammad R Maracy
BACKGROUND AND AIM: Thalassemia screening instructions in Iran categorizes couples with mean corpuscular volume (MCV)=75 to 80, mean corpuscular hemoglobin (MCH)=26 to 27, hemoglobin A2 (HbA2)<3.5, and hemoglobin fetal (HbF)<3 indices as low-risk couples, and therefore further genetic testing is not obligatory. This study examined the possibility of classifying couples with MCH<26 and MCV≥80 indices in the low-risk group when their HbF was <3 and HbA2 was <3.2. METHODOLOGY: This was a cross-sectional study...
January 22, 2019: Journal of Pediatric Hematology/oncology
Grace Egan, Jill Hamilton, Tara McKeown, Eric Bouffet, Uri Tabori, Peter Dirks, Ute Bartels
Low-grade gliomas (LGG) represent the most common form of primary central nervous system tumor arising in childhood. There is growing evidence to support the role of the mitogen-activated protein kinase pathway in driving tumor growth and MEK inhibitors are being investigated in clinical trials for refractory and unresectable LGGs. As MEK inhibitors progress through clinical trials, drug toxicities have been identified. We report on 2 pediatric patients with LGG and known diabetes insipidus who developed severe hyponatraemia associated with significant decreases in desmopressin doses after starting trametinib...
January 22, 2019: Journal of Pediatric Hematology/oncology
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