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Purpose@#Renal tumors account for approximately 7% of all childhood cancers. These include Wilms tumor (WT), clear cell sarcoma of the kidney (CCSK), malignant rhabdoid tumor of the kidney (MRTK), renal cell carcinoma (RCC), congenital mesoblastic nephroma (CMN) and other rare tumors. We investigated the epidemiology of pediatric renal tumors in Korea. @*Materials and Methods@#From January 2001 to December 2015, data of pediatric patients (0–18 years) newly-diagnosed with renal tumors at 26 hospitals were retrospectively analyzed. @*Results@#Among 439 patients (male, 240), the most common tumor was WT (n=342, 77.9%), followed by RCC (n=36, 8.2%), CCSK (n=24, 5.5%), MRTK (n=16, 3.6%), CMN (n=12, 2.7%), and others (n=9, 2.1%). Median age at diagnosis was 27.1 months (range 0-225.5) and median follow-up duration was 88.5 months (range 0-211.6). Overall, 32 patients died, of whom 17, 11, 1, and 3 died of relapse, progressive disease, second malignant neoplasm, and treatment-related mortality. Five-year overall survival and event free survival were 97.2% and 84.8% in WT, 90.6% and 82.1% in RCC, 81.1% and 63.6% in CCSK, 60.3% and 56.2% in MRTK, and 100% and 91.7% in CMN, respectively (p < 0.001). @*Conclusion@#The pediatric renal tumor types in Korea are similar to those previously reported in other countries. WT accounted for a large proportion and survival was excellent. Non-Wilms renal tumors included a variety of tumors and showed inferior outcome, especially MRTK. Further efforts are necessary to optimize the treatment and analyze the genetic characteristics of pediatric renal tumors in Korea.
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Hepatocellular adenoma is an uncommon, benign liver tumor usually occurring in patients using estrogen or anabolic androgens and in those with a genetic disease, including glycogen storage disease. Hepatocellular adenomas can sometimes induce pain. However, it is usually asymptomatic. Moreover, few studies have reported cases of hepatocellular adenomas presenting with iron deficiency anemia. Herein, we report a pediatric case of a large hepatocellular adenoma, presenting with iron therapy-refractory iron deficiency anemia. A 14-year-old boy was diagnosed with hepatocellular adenoma during an anemia work-up. Improvement in iron deficiency anemia was observed after tumor resection.
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Background@#The incorporation of a reduced-intensity conditioning (RIC) regimen in hematopoietic cell transplantation (HCT) for patients with hemophagocytic lymphohistiocytosis (HLH) has decreased early mortality but is associated with a high rate of mixed chimerism and graft failure. Here, we present a successful single-center experience using busulfan and a fludarabine-based RIC regimen for the treatment of HLH. @*Methods@#The medical records of pediatric patients with HLH who underwent HCT using a busulfan/fludarabine-based RIC regimen between January 2008 and December 2017 were reviewed retrospectively. @*Results@#Nine patients received HCT with a busulfan/fludarabine-based RIC regimen. Three patients had primary HLH, and the other six patients had secondary HLH with multiple reactivations. All three patients with primary HLH had UNC13D mutations. All patients achieved neutrophil and platelet engraftment at a median of 11 days (range, 10‒21) and 19 days (range, 13‒32), and all eight evaluable patients had sustained complete donor chimerism at the last follow-up. Two patients (22%) experienced grade 2 acute graft-versus-host disease (GVHD). Two patients (22%) developed chronic GVHD, and one died from chronic GVHD. One patient (11%) experienced reactivation 4 months after HCT from a syngeneic donor and died of the disease. The 8-year overall survival and event-free survival rates were 78%. No early treatment-related mortality within 100 days after HCT was observed. @*Conclusion@#Our experience suggests that a busulfan/fludarabine-based RIC regimen is a viable option for pediatric patients with HLH who require HCT.
ABSTRACT
Hemophagocytic lymphohistiocytosis (HLH) is a fatal disease unless timely and effective treatment is given. Immunochemotherapy including etoposide, with or without hematopoietic stem cell transplantation (HCT), has improved the outcomes for patients with HLH. In patients with familial or refractory HLH, HCT is now routinely performed and is the only curative treatment. Conditioning regimens play an important role in the success of HCT for pediatric patients with HLH and other nonmalignant diseases and have improved dramatically in recent decades. The initial HCT approach using myeloablative conditioning significantly improved the outcomes of patients with HLH but was associated with considerable transplant-related mortality. A subsequent strategy using reduced-intensity conditioning (RIC) remarkably reduced the incidence of T RM. However, the high level of mixed chimerism associated with RIC has prompted the search for improved conditioning regimens. Recently, treosulfan has replaced busulfan as a component of a reduced toxicity conditioning regimen. Both its myeloablative and immunosuppressive properties, as well as a favorable toxicity profile, make treosulfan a potential candidate for use as part of conditioning regimen prior to HCT. Indeed, treosulfan-based conditioning regimens are being increasingly used in pediatric patients with various non-malignant diseases. We here review the recent progress in HCT for pediatric HLH with a focus on treosulfan-based conditioning regimens, including our own experience.
ABSTRACT
Hemophagocytic lymphohistiocytosis (HLH) is a fatal disease unless timely and effective treatment is given. Immunochemotherapy including etoposide, with or without hematopoietic stem cell transplantation (HCT), has improved the outcomes for patients with HLH. In patients with familial or refractory HLH, HCT is now routinely performed and is the only curative treatment. Conditioning regimens play an important role in the success of HCT for pediatric patients with HLH and other nonmalignant diseases and have improved dramatically in recent decades. The initial HCT approach using myeloablative conditioning significantly improved the outcomes of patients with HLH but was associated with considerable transplant-related mortality. A subsequent strategy using reduced-intensity conditioning (RIC) remarkably reduced the incidence of T RM. However, the high level of mixed chimerism associated with RIC has prompted the search for improved conditioning regimens. Recently, treosulfan has replaced busulfan as a component of a reduced toxicity conditioning regimen. Both its myeloablative and immunosuppressive properties, as well as a favorable toxicity profile, make treosulfan a potential candidate for use as part of conditioning regimen prior to HCT. Indeed, treosulfan-based conditioning regimens are being increasingly used in pediatric patients with various non-malignant diseases. We here review the recent progress in HCT for pediatric HLH with a focus on treosulfan-based conditioning regimens, including our own experience.
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Background@#Hodgkin's lymphoma (HL) constitutes 10%–20% of all malignant lymphomas and has a high cure rate (5-year survival, around 90%). Recently, interest has increased concerning preventing secondary complications (secondary cancer, endocrine disorders) in long-term survivors. We aimed to study the epidemiologic features and therapeutic outcomes of HL in children, adolescents, and young adults in Korea. @*Methods@#We performed a multicenter, retrospective study of 224 patients aged < 25 years diagnosed with HL at 22 participating institutes in Korea from January 2007 to August 2016. @*Results@#A higher percentage of males was diagnosed at a younger age. Nodular sclerosis histopathological HL subtype was most common, followed by mixed cellularity subtype.Eighty-one (36.2%), 101 (45.1%), and 42 (18.8%) patients were classified into low, intermediate, and high-risk groups, respectively. Doxorubicin, bleomycin, vinblastine, dacarbazine was the most common protocol (n = 102, 45.5%). Event-free survival rate was 86.0% ± 2.4%, while five-year overall survival (OS) rate was 96.1% ± 1.4%: 98.7% ± 1.3%, 97.7% ± 1.6%, and 86.5% ± 5.6% in the low, intermediate, and high-risk groups, respectively (P = 0.021). Five-year OS was worse in patients with B-symptoms, stage IV disease, highrisk, splenic involvement, extra-nodal lymphoma, and elevated lactate dehydrogenase level.In multivariate analysis, B-symptoms and extra-nodal involvement were prognostic factors for poor OS. Late complications of endocrine disorders and secondary malignancy were observed in 17 and 6 patients, respectively. @*Conclusion@#This is the first study on the epidemiology and treatment outcomes of HL in children, adolescents, and young adults in Korea. Future prospective studies are indicated to develop therapies that minimize treatment toxicity while maximizing cure rates in children, adolescents, and young adults with HL.
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Purpose@#This study presents the process of designing workbooks for advance care planning appropriate for the Korean cultural setting and describes actual case studies. @*Methods@#This study focused on single inductive case studies of the utilization of an advance care planning workbook and recruited individual participants. @*Results@#The workbook for adolescents contained six sessions and the workbook for children contained seven sessions. The workbook sessions led to four major discoveries: 1) considering the Korean cultural context, discussions on life and death must be held indirectly; 2) the role of the counselor as a supporter is crucial for the workbook to be effective; 3) the workbook must be accessible regardless of the seriousness of the illness; and 4) patients must be able to make their own choice between the workbook versions for children and adolescents. Six facilitating factors improved engagement: 1) the role of the counselor as a supporter; 2) building trust with the patient; 3) affirming freedom of expression on topics the patient wished to avoid talking about; 4) having discussions on what private information to keep secret and to whom the information can be disclosed; 5) discovering and regularly discussing relevant topics; and 6) regular communication and information-sharing with the patient’s medical service providers. Conclusion: It is necessary to build on actual case studies regarding workbooks for children and adolescents in order to expand the usage of these workbooks to all relevant medical institutions in Korea.
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Purpose@#This study presents the process of designing workbooks for advance care planning appropriate for the Korean cultural setting and describes actual case studies. @*Methods@#This study focused on single inductive case studies of the utilization of an advance care planning workbook and recruited individual participants. @*Results@#The workbook for adolescents contained six sessions and the workbook for children contained seven sessions. The workbook sessions led to four major discoveries: 1) considering the Korean cultural context, discussions on life and death must be held indirectly; 2) the role of the counselor as a supporter is crucial for the workbook to be effective; 3) the workbook must be accessible regardless of the seriousness of the illness; and 4) patients must be able to make their own choice between the workbook versions for children and adolescents. Six facilitating factors improved engagement: 1) the role of the counselor as a supporter; 2) building trust with the patient; 3) affirming freedom of expression on topics the patient wished to avoid talking about; 4) having discussions on what private information to keep secret and to whom the information can be disclosed; 5) discovering and regularly discussing relevant topics; and 6) regular communication and information-sharing with the patient’s medical service providers. Conclusion: It is necessary to build on actual case studies regarding workbooks for children and adolescents in order to expand the usage of these workbooks to all relevant medical institutions in Korea.
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BACKGROUND: Precursor T-cell acute lymphoblastic leukemia (T-ALL) has worse prognosis than B-cell ALL. We aimed to evaluate prognostic variables in pediatric T-ALL. METHODS: Medical records of 36 T-ALL patients (27 males and 9 females; median age at diagnosis, 10.6 years) diagnosed and treated at Asan Medical Center from 2001 to 2017 were reviewed. Six patients (16.7%) had early T-cell precursor ALL (ETP-ALL). Most patients received the Children's Cancer Group-1882 (CCG1882) or Korean multicenter high risk ALL (ALL0601) protocols and prophylactic cranial irradiation. Clinical features at presentation, response to therapy, and treatment outcomes were analyzed. RESULTS: The six patients with ETP-ALL and 17 of 30 with non-ETP-ALL received CCG1882 or ALL0601 chemotherapy. Three patients, including two with ETP-ALL, did not achieve complete remission after induction. Rapid early response during induction was achieved by 26 patients. Five year overall survival (OS) and event free survival (EFS) rates were 71.4% and 70.2%, respectively. ETP-ALL and slow early response during induction were significant adverse prognostic factors, while hyperleukocytosis at diagnosis was not. CCG1882/ALL0601 chemotherapy resulted in superior survival (OS: 78.9%, EFS: 73.3%) compared with CCG1901 chemotherapy (OS: 64.3%, EFS: 64.3%), and patients undergoing prophylactic cranial irradiation had superior EFS to non-radiated patients. CONCLUSION: A high risk ALL protocol with intensified post-remission therapy, including prophylactic cranial irradiation, conferred T-ALL survival outcomes comparable with those of Western studies. Further treatment intensification should be considered for patients with ETP-ALL and slow induction responders. Additionally, CNS-directed treatment intensification, without prophylactic cranial irradiation, is needed.
Subject(s)
Female , Humans , Male , B-Lymphocytes , Cranial Irradiation , Diagnosis , Disease-Free Survival , Drug Therapy , Medical Records , Precursor Cell Lymphoblastic Leukemia-Lymphoma , Precursor Cells, T-Lymphoid , Precursor T-Cell Lymphoblastic Leukemia-Lymphoma , Prognosis , T-LymphocytesABSTRACT
Isolated pulmonary Langerhans cell histiocytosis (LCH) is a very rare disease in childhood. We report a case of a 5-month-old girl with isolated pulmonary LCH, who was transferred due to incidental chest x-ray finding of multiple cystic lesions without any clinical symptoms. Chest computed tomography (CT) finding suggested that pulmonary LCH was likely, but evaluations including lung biopsy were negative. At a follow-up visit three months later, we performed bronchoalveolar lavage (BAL) fluid analysis and confirmed the presence of CD1a-positive cells, thereby confirming diagnosis of pulmonary LCH. After completing eight months of chemotherapy, yearly follow-up evaluations were performed and there has been no evidence of reactivation of the disease for four years. Based on our case, we suggest that BAL with immunohistochemical staining can be a valuable modality to eliminate the possibility of infection and other infiltrating disorders, and diagnose pulmonary LCH in case of suspicious pulmonary lesions.
Subject(s)
Female , Humans , Infant , Biopsy , Bronchoalveolar Lavage , Diagnosis , Drug Therapy , Follow-Up Studies , Histiocytosis, Langerhans-Cell , Lung , Rare Diseases , ThoraxABSTRACT
Isolated pulmonary Langerhans cell histiocytosis (LCH) is a very rare disease in childhood. We report a case of a 5-month-old girl with isolated pulmonary LCH, who was transferred due to incidental chest x-ray finding of multiple cystic lesions without any clinical symptoms. Chest computed tomography (CT) finding suggested that pulmonary LCH was likely, but evaluations including lung biopsy were negative. At a follow-up visit three months later, we performed bronchoalveolar lavage (BAL) fluid analysis and confirmed the presence of CD1a-positive cells, thereby confirming diagnosis of pulmonary LCH. After completing eight months of chemotherapy, yearly follow-up evaluations were performed and there has been no evidence of reactivation of the disease for four years. Based on our case, we suggest that BAL with immunohistochemical staining can be a valuable modality to eliminate the possibility of infection and other infiltrating disorders, and diagnose pulmonary LCH in case of suspicious pulmonary lesions.
Subject(s)
Female , Humans , Infant , Biopsy , Bronchoalveolar Lavage , Diagnosis , Drug Therapy , Follow-Up Studies , Histiocytosis, Langerhans-Cell , Lung , Rare Diseases , ThoraxABSTRACT
BACKGROUND: Among pediatric non-Hodgkin lymphomas, there are 4 major subtypes: Burkitt lymphoma, lymphoblastic lymphoma, diffuse large B-cell lymphoma, and anaplastic large-cell lymphoma. Understanding of other rare subtypes derives only from small pediatric case series. We report our institutional experience with rare pediatric NHLs.METHODS: Thirty-six cases of rare NHL subtypes diagnosed at the Asan Medical Center from 1995 to 2015 were evaluated. We retrospectively reviewed the clinical and pathologic features and outcomes of these patients, excluding peripheral T-cell lymphoma, not otherwise specified (PTCL, NOS), and extranodal NK/T-cell lymphoma (ENKL), on which we have previously reported.RESULTS: There were 23 cases of T-cell lineage (13 PTCL, 6 ENKL, 2 subcutaneous panniculitis-like T-cell lymphoma, 1 primary cutaneous CD4+2016-11-22 small/medium sized T-cell lymphoma, 1 enteropathy-associated T-cell lymphoma) and 13 cases of B-cell lineage lymphoma (5 marginal zone lymphoma, 6 primary mediastinal large B-cell lymphoma, 2 immunoblastic and plasmablastic lymphoma). All patients were treated with chemotherapy with or without surgery, except 4 out of 5 patients with marginal zone lymphoma who received surgery only. Two patients died and 6 patients relapsed. One patient with primary mediastinal large B-cell lymphoma received autologous peripheral blood stem cell transplantation. The 5-year overall survival and event-free survival rates of rare pediatric NHL excluding PTCL, NOS, and ENKL was 80.0% and 72.0%, respectively.CONCLUSION: Children diagnosed with rare pediatric NHL other than PTCL, NOS, and ENKL showed variable incidence and treatment outcomes. Multicenter studies in larger cohorts are needed for better understanding of these rare NHL subtypes in childhood.
Subject(s)
Child , Humans , B-Lymphocytes , Burkitt Lymphoma , Cohort Studies , Disease-Free Survival , Drug Therapy , Incidence , Lymphoma , Lymphoma, B-Cell , Lymphoma, Large-Cell, Anaplastic , Lymphoma, Non-Hodgkin , Lymphoma, T-Cell , Lymphoma, T-Cell, Peripheral , Peripheral Blood Stem Cell Transplantation , Precursor Cell Lymphoblastic Leukemia-Lymphoma , Retrospective Studies , T-LymphocytesABSTRACT
BACKGROUND: Among pediatric non-Hodgkin lymphomas, there are 4 major subtypes: Burkitt lymphoma, lymphoblastic lymphoma, diffuse large B-cell lymphoma, and anaplastic large-cell lymphoma. Understanding of other rare subtypes derives only from small pediatric case series. We report our institutional experience with rare pediatric NHLs. METHODS: Thirty-six cases of rare NHL subtypes diagnosed at the Asan Medical Center from 1995 to 2015 were evaluated. We retrospectively reviewed the clinical and pathologic features and outcomes of these patients, excluding peripheral T-cell lymphoma, not otherwise specified (PTCL, NOS), and extranodal NK/T-cell lymphoma (ENKL), on which we have previously reported. RESULTS: There were 23 cases of T-cell lineage (13 PTCL, 6 ENKL, 2 subcutaneous panniculitis-like T-cell lymphoma, 1 primary cutaneous CD4+2016-11-22 small/medium sized T-cell lymphoma, 1 enteropathy-associated T-cell lymphoma) and 13 cases of B-cell lineage lymphoma (5 marginal zone lymphoma, 6 primary mediastinal large B-cell lymphoma, 2 immunoblastic and plasmablastic lymphoma). All patients were treated with chemotherapy with or without surgery, except 4 out of 5 patients with marginal zone lymphoma who received surgery only. Two patients died and 6 patients relapsed. One patient with primary mediastinal large B-cell lymphoma received autologous peripheral blood stem cell transplantation. The 5-year overall survival and event-free survival rates of rare pediatric NHL excluding PTCL, NOS, and ENKL was 80.0% and 72.0%, respectively. CONCLUSION: Children diagnosed with rare pediatric NHL other than PTCL, NOS, and ENKL showed variable incidence and treatment outcomes. Multicenter studies in larger cohorts are needed for better understanding of these rare NHL subtypes in childhood.
Subject(s)
Child , Humans , B-Lymphocytes , Burkitt Lymphoma , Cohort Studies , Disease-Free Survival , Drug Therapy , Incidence , Lymphoma , Lymphoma, B-Cell , Lymphoma, Large-Cell, Anaplastic , Lymphoma, Non-Hodgkin , Lymphoma, T-Cell , Lymphoma, T-Cell, Peripheral , Peripheral Blood Stem Cell Transplantation , Precursor Cell Lymphoblastic Leukemia-Lymphoma , Retrospective Studies , T-LymphocytesABSTRACT
BACKGROUND AND PURPOSE: To determine how cognitive function is related to epilepsy classification and comorbid attention deficit hyperactivity disorder (ADHD) in children with newly diagnosed epilepsy of genetic or unknown etiology. METHODS: The medical records of children aged 6-16 years with newly diagnosed epilepsy of genetic or unknown etiology were reviewed retrospectively. The Korean Education Development Institute-Wechsler Intelligence Scale for Children and the Comprehensive Attention Test were used to evaluate intelligence and attention/executive function, respectively. RESULTS: The data of a total of 149 children, 103 with focal seizures and 46 with generalized seizures, were reviewed. The prevalence of ADHD was 49.2% (59 out of 120 examined patients), and ADHD patients exhibited significantly worse auditory selective attention, flanker test results, and spatial working memory. Patients with generalized seizures exhibited significantly worse auditory selective and sustained attention than patients with focal seizures. In patients with generalized seizures, sustained attention, flanker test findings, and spatial working memory were found to be affected by ADHD, and auditory selective and sustained attention were significantly worse in patients with benign childhood epilepsy with centrotemporal spikes and ADHD than in their counterparts without ADHD. CONCLUSIONS: Cognitive processes are affected by seizure type and comorbid ADHD. Proper characterization of these neuropsychiatric impairments may allow earlier intervention during the disease course.
Subject(s)
Child , Humans , Attention Deficit Disorder with Hyperactivity , Classification , Cognition , Education , Epilepsy , Intelligence , Medical Records , Memory, Short-Term , Prevalence , Retrospective Studies , SeizuresABSTRACT
PURPOSE: Bronchial hyperresponsiveness (BHR) is typically measured by bronchial challenge tests that employ direct stimulation by methacholine or indirect stimulation by adenosine 5'-monophosphate (AMP). Some studies have shown that the AMP challenge test provides a better reflection of airway inflammation, but few studies have examined the relationship between the AMP and methacholine challenge tests in children with asthma. We investigated the relationship between AMP and methacholine testing in children and adolescents with atopic asthma. METHODS: The medical records of 130 children with atopic asthma (mean age, 10.63 years) were reviewed retrospectively. Methacholine and AMP test results, spirometry, skin prick test results, and blood tests for inflammatory markers (total IgE, eosinophils [total count, percent of white blood cells]) were analyzed. RESULTS: The concentration of AMP that induces a 20% decline in forced expiratory volume in 1 second [FEV1] (PC20) of methacholine correlated with the PC20 of AMP (r2=0.189, P<0.001). No significant differences were observed in the levels of inflammatory markers (total eosinophil count, eosinophil percentage, and total IgE) between groups that were positive and negative for BHR to methacholine. However, significant differences in inflammatory markers were observed in groups that were positive and negative for BHR to AMP (log total eosinophil count, P=0.023; log total IgE, P=0.020, eosinophil percentage, P<0.001). In contrast, body mass index (BMI) was significantly different in the methacholine positive and negative groups (P=0.027), but not in the AMP positive and negative groups (P=0.62). The PC20 of methacholine correlated with FEV1, FEV1/forced vital capacity (FVC), and maximum mid-expiratory flow (MMEF) (P=0.001, 0.011, 0.001, respectively), and the PC20 of AMP correlated with FEV1, FEV1/FVC, and MMEF (P=0.008, 0.046, 0.001, respectively). CONCLUSIONS: Our results suggest that the AMP and methacholine challenge test results correlated well with respect to determining BHR. The BHR to AMP more likely implicated airway inflammation in children with atopic asthma. In contrast, the BHR to methacholine was related to BMI.
Subject(s)
Adolescent , Child , Humans , Adenosine , Asthma , Body Mass Index , Bronchial Provocation Tests , Eosinophils , Forced Expiratory Volume , Hematologic Tests , Immunoglobulin E , Inflammation , Medical Records , Methacholine Chloride , Retrospective Studies , Skin , Spirometry , Vital CapacityABSTRACT
Systemic lupus erythematosus (SLE) is an autoimmune disorder involving multiple organs. Neuropsychiatric symptoms are frequently associated in SLE, which is referred to as neuropsychiatric SLE (NPSLE). NPSLE contains both central and peripheral nervous systems, which includes transverse myelitis, and Guillain-Barre syndrome (GBS). We report our experience of concurrent manifestation of transverse myelitis and GBS as an initial presentation of SLE, which suggests the common immune-mediated mechanisms of diseases. We here report the case of a 14-year-old boy with SLE who first presented with features of GBS. The patient developed ascending weakness starting from low extremities, experienced difficulty voiding, and had a facial rash. An initial diagnosis of GBS was made on the basis of clinical findings and nerve conduction studies. But he did not respond to intravenous immunoglobulin therapy and following spine MRI displayed T2 weighted high signal intensities from the cervical to thoracic region of the spinal cord, and serological analysis revealed the presence of anti-dsDNA, anti-smAb, anti nuclear antibody with decreased level of complements. The diagnosis was revised to GBS and acute transverse myelitis resulting from SLE. Additional methylprednisolone pulse therapy led to rapid clinical improvement. This was followed by oral prednisolone and cyclophosphamide pulse therapy. This is the first case of concurrent manifestation of GBS and transverse myelitis as initial presentation of SLE. The cross-reactivity of autoantibodies and increased susceptibility to infection owing to immunologic changes associated with lupus may form the basis of the association. Clinicians should consider a diagnosis of SLE as an etiology of GBS or transverse myelitis.