Journal of Yeungnam Medical Science

Case report

Delayed treatment-free response after romiplostim discontinuation in pediatric chronic immune thrombocytopenia: Hyun Ji Lim, Young Tae Lim, Jeong Ok Hah, Jae Min Lee; Yeungnam Univ J Med. 2021;38(2):165-168. Published online August 7, 2020; DOI: https://doi.org/10.12701/yujm.2020.00493

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We report the case of a 16-month-old patient with chronic immune thrombocytopenia (ITP) patient who experienced delayed treatment-free response (TFR) after romiplostim treatment. He received intravenous immunoglobulin every month to maintain a platelet count above 20,000/μL for 2 years. Thereafter, he received rituximab and cyclosporine as second-line therapy, with no response, followed by romiplostim. After 4 weeks of treatment, the platelet count was maintained above 50,000/μL. Following 7 months of treatment, he discontinued romiplostim, and the platelet count decreased. His platelet counts remained above 50,000/μL, without any bleeding symptoms, 2 years after romiplostim discontinuation. This is the first report of TFR after romiplostim treatment in pediatric chronic ITP.

Citations

Citations to this article as recorded by

A cost–utility analysis of thrombopoietin receptor agonists for treating pediatric immune thrombocytopenia purpura after failure of first‐line therapies
Huimin Du, Jiamin Wang, Joel Livingston, Ziyad Alrajhi, Melanie Kirby‐Allen, Brian Chan, Rebecca Hancock‐Howard, Peter C. Coyte
Pediatric Blood & Cancer.2023;[Epub] CrossRef
Generic romiplostim for children with persistent or chronic immune thrombocytopenia: Experience from a tertiary care centre in North India
Chandana Mareddy, Manas Kalra, Anupam Sachdeva
British Journal of Haematology.2022; 197(5): 618. CrossRef
Tapering of the thrombopoietin receptor agonist in paediatric patients with chronic immune thrombocytopenia: Is it possible?
María Solsona, Rubén Berrueco, Elena Sebastián, Áurea Cervera, Ana Sastre, Itziar Astigarraga, Bienvenida Argilés, María Ángeles Dasí, José Luís Dapena, Emilio Monteagudo
British Journal of Clinical Pharmacology.2022; 88(9): 4220. CrossRef

Case Reports

Atypical Cerebellar Medulloblastoma Originating from Tentorium: Case Report.: Seong Ho Kim, Chang Hwan Kim, Oh Lyong Kim, Chul Hoon Chang, Sang Woo Kim, Byung Yon Choi, Soo Ho Cho, Jeong Ok Hah; Yeungnam Univ J Med. 2007;24(2):311-314. Published online December 31, 2007; DOI: https://doi.org/10.12701/yujm.2007.24.2.311

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Abstract PDF

The extraaxial presentation of a medulloblastoma is rare. This article describes the case of a 12-year-old boy who presented with severe headache, nausea, and vomiting. The tumor developed in the left tentorium; it was misdiagnosed as a meningioma based on the radiology examination. We review the literature and discuss the atypical presentation of medulloblastoma.

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New Developments in the Pathogenesis, Therapeutic Targeting, and Treatment of Pediatric Medulloblastoma
Francia Y. Fang, Jared S. Rosenblum, Winson S. Ho, John D. Heiss
Cancers.2022; 14(9): 2285. CrossRef

Intrathecal Methotrexate Induced Neurotoxicity in Children with Acute Lymphoblastic Leukemia: Jae Min Lee, Han Ku Moon, Jeong Ok Hah; Yeungnam Univ J Med. 2007;24(2 Suppl):S761-769. Published online December 31, 2007; DOI: https://doi.org/10.12701/yujm.2007.24.2S.S761

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Abstract PDF: Central nervous system (CNS) prophylaxis is an essential component of the treatment in childhood acute lymphoblastic leukemia (ALL). Methotrexate (MTX) is an dispensable antimetabolite for treatment of ALL. High-dose (HD) MTX and intrathecal (IT) MTX have improved the prognosis and reduced the rate of CNS relapse. However, the drug also has a significant toxic effect on the CNS and can potentially lead to severe neurologic morbidity. The overall incidence of acute MTX induced neurotoxicity has been estimated to be 0.8∼10% of treated children, depending on the amounts of MTX and leucovorin in the treatment protocol. Acute neurotoxicity generally develops within 5∼14 days after IT MTX or HD MTX and may include headache, nausea, emesis, lethargy, altered mental status, blurred vision, aphasia, hemiparesis, and seizure. Diffusion weighted MRI shows restricted diffusion of water in brains of patients with ALL who experienced stroke-like event after IT MTX. We report the cinical and imaging findings of acute neurotoxicity in two patients after intrathecal administration of MTX for CNS prophylaxes of ALL.

Original Article

Etiology of Treatment Related Mortality after Hematopoietic Stem Cell Transplantation: Yong Jik Lee, Jeong Ok Hah; Yeungnam Univ J Med. 2007;24(2 Suppl):S569-579. Published online December 31, 2007; DOI: https://doi.org/10.12701/yujm.2007.24.2S.S569

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Abstract PDF: Bactground：The etiologies of treatment related mortality (TRM) after hematopoietic stem cell transplantation (HSCT) have been variable according to the disease status or the centers. We evlauated the etiologies of TRM for the pediatric patients at Yeungnam University Hospital (YNUH). Materials and Methods：The records of 66 patients, 19 years of age or younger, who had HSCT at YNUH from September 1995 to August 2007 were reviewed.
Results
：Among 66 patients, allogeneic bone marrow transplantation (Allo-BMT) was done in 21 (19 related, 2 unrelated), allogeneic peripheral blood stem cell transplantation (Allo- PBSCT) in 1, cord blood transplantation (CBT) in 12 (1 related, 11 unrelated), autologous peripheral blood stem cell transplantation (Auto-PBSCT) in 32 patients. The TRM rates of Allo-BMT, CBT, and Auto-PBSCT were 19%, 33.3%, and 12.5%, respectively. Among four patients who had TRM after Allo-BMT, two were related transplantation and the others were unrelated. All four patients developed severe acute GVHD of at least grade Ⅲ. Sepsis developed in three patients, acute renal failure (ARF) in two, veno-occlusive disease (VOD) and thrombotic microangiopathy (TMA) in one patient each. All four patients who had TRM after CBT had two mismatches in HLA-A, B, DR, and engraftment syndrome developed in three. Sepsis developed in all four patients, VOD in two, encephalopathy in two, TMA and ARF in one patient each. All four patients who had TRM after Auto-PBSCT developed sepsis and ARF in two, VOD and TMA in one patient each.
Conclusion
：Although the number of cases were not large enough for firm conclusion, sepsis was the most common TRM after HSCT. Therefore, prevention and control of sepsis are very important in reducing TRM after HSCT. Outcomes of severe acute GVHD after Allo-BMT and engraftment syndrome after CBT are very poor and contribute for TRM. Continuous effort to reduce the incidence of GVHD and engraftment syndrome are needed.

Case Report

A Case of Superior Mesenteric Venous Thrombosis after Endoscopic Sclerotherapy.: Won Duck Kim, Kwang Hae Choi, Jeong Ok Hah; Yeungnam Univ J Med. 2001;18(2):297-301. Published online December 31, 2001; DOI: https://doi.org/10.12701/yujm.2001.18.2.297

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Abstract PDF: The efficacy of injection sclerotherapy for treatment of acute esophageal variceal bleeding is well established. But several complications of endoscopic sclerotherapy have been reported. One of the complications is mesenteric venous thrombosis which develops when vasopressin is user for the sclerotherapy. We report a case of superior mesenteric venous thrombosis with developed after endoscopic sclerotherapy for control of esophageal variceal bleeding.

Original Articles

Indication of Bone Marrow Aspiration in Acute Idiopathic Thrombocytopenic Purpura in Children.: Won Duck Kim, Jeong Ok Hah; Yeungnam Univ J Med. 2001;18(2):239-245. Published online December 31, 2001; DOI: https://doi.org/10.12701/yujm.2001.18.2.239

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Abstract PDF: BACKGROUND
Acute idiopathic thrombocytopenic purpura(ITP) is one of the common hematologic disorders in children. Bone marrow aspiration(BMA) is often performed in children with acute ITP to rule out leukemia, aplastic anemia or other hematologic diseases. However, whether BMA is needed in children with typical clinical and hematological features of acute ITP have been questioned. This study was performed to examine the proper indication of BMA in acute childhood ITP. MATERIALS AND METHODS: The medical records and BMA reports of children with the provisional diagnosis of acute ITP were reviewed from January 1984 to December 2000. Patients were divided into two groups, one with typical and another with atypical clinical and hematological features of acute ITP. Typical acute ITP group was characterized by the history of previous viral infection, well being appearance, no hepatosplenomegaly, no lymphadenopathy, normal Hb, WBC, neutrophil count and peripheral blood smear except thrombocytopenia. A platelet count of 50x109/L or lower was the cutoff level. RESULTS: Total 120 children with the provisional diagnosis of acute ITP were included. One hundred eighteen of them were confirmed to have acute ITP by BMAs. Of these, 66 had typical and 54 had atypical features. All of typical features and 52 of 54 with atypical features of acute ITP were confirmed to have acute ITP by BMAs. Two patients with atypical features of acute ITP were diagnosed as aplastic anemia and myelodyspalstic syndrome, respectively, by BMAs. CONCLUSION: This study concludes that BMA is not needed for the children with typical features of acute ITP but it is needed for the children with atypical features of acute ITP to rule out other hematologic disorders.

Splenectomy in Hereditary Spherocytosis in Childhood.: Young Soo Heo, Chang Sig Kim, Byung Soo Do, Bo Yang Suh, Jeong Ok Hah; Yeungnam Univ J Med. 1994;11(1):42-48. Published online June 30, 1994; DOI: https://doi.org/10.12701/yujm.1994.11.1.42

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Abstract PDF

Among the erythrocyte membrane defects, hereditary spherocytosis is the most common. The erythrocyte membrane defect results from a deficiency of spectrin, the most important structural protein in red cell. Hereditary spherocytosis often presents with hemolytic anemia, jaundice, moderate splenomegaly. Diagnosis is established by the presence of spherocytes in the peripheral blood, reticulocytosis, an increased osmotic fragility, and a negative Coombs test. In children, splenectomy is usually performed after age 6 years but can be done at a younger age if warranted by the severity of the anemia and the need for frequent transfusions. In the period December 1987 to Agust 1993, 9 patients with hereditrary spherocytosis underwent splenectomy and the following results were obtained. 1. Nine patients were comprised of five males and four females. 2. Five patients(55.6%) had been admitted to our hospital during age 6-10 years. 3. Four of the nine patients had autosomal dominant inheritance with variable expression. The other five patients had no known inheritance. 4. The diagnosis of the spherocytosis was based on the increased osmotic fragility and increased autohemolysis of the erythrocytes, as well as on the appearance of spherocytes in the peripheral blood smear. 5. In all cases splenectomy was performed. Two patients had concomitant gall stones and choledocholithiasis, respectively. One patient with concomitant gall stones underwent simultaneous cholecystectomy and splenectomy. The other patients associated with choledocholithiasis underwent splenectomy, cholecystectomy, choledocholithotomy, and T-tube drainage. 6. Complete hematologic recovery was obtained by the splenectomy in all cases. 7. Postoperative complication was not occurred.

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A histopathological study of spectrum of splenic lesions- An eleven year analysis of clinical and pathological aspects of splenectomy specimens in a tertiary care hospital
Namratha R, Vijaya B, Karthika Bhadran
Indian Journal of Pathology and Oncology.2022; 9(3): 220. CrossRef

Effect of Recombinant Human Erythropoietin in the Anemia of Prematurity : a Pilot Study.: Kyung Ah Lee, Son Moon Shin, Yong Hoon Park, Jeong Ok Hah; Yeungnam Univ J Med. 1994;11(1):115-126. Published online June 30, 1994; DOI: https://doi.org/10.12701/yujm.1994.11.1.115

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Abstract PDF: The recent availability of recombinant human erythropoietin has opened new perspectives in the management of a variety of anemias. Clinical trials have been initiated in several countries using different approaches and methodology. We randomly assigned twelve premature infants(gestational age < 32 week) at high risk of requiring erythrocyte transfusion for anemia of prematurity with either subcutaneous recombinant human erythropoietin or a placebo. Treatment with rHuEPO was initiated at a dose of 100 units/kg day for 3 days a week. All patients were given supplemental oral iron therapy at a dose of 3 mg/kg per day, as tolerated and oral vitamin E at a dose of 25 units per day. Treated and control babies did not differ with respect to weight, hematocrit, overall mean reticulocyte count or rate of growth respectively. However, reticulocyte counts increased earlier in patients given rHuEPO. We conclude that rHuEPO administration is safe and feasible at the dose studied.

Meaning of anti hepatitis C virus antibody detection in patients treated with intravenous immunoglobulin: Myong Gyun Lee, Young Hwan Lee, Son Moon Shin, Jeong Ok Hah; Yeungnam Univ J Med. 1993;10(2):306-312. Published online December 31, 1993; DOI: https://doi.org/10.12701/yujm.1993.10.2.306

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Abstract PDF: To evaluate the meaning of anti-HCV detection in patients treated with IVIG, serum levels of aspartate aminotranstferase(AST), alanine aminotransterase(ALT), HCV Ab titer were measured after treatment with IVIG in 36 patients diagnised of Kawasaki disease or neonatal sepsis. Also polymerase chain reaction (PCR) for the detection of HCV was done in 8 patients with persistent HCV Ab positivity at 3 months after IVIG treatment. The results were as follows 1) HCV Ab was positive in all 36 patients at 1 week after IVIG treatment, but in only 8 cases it was positive at 3 months after IVIG treatment. 2) AST, ALT were elevated in 9 cases at 1 week after IVIG treatment, but they were normalized in all cases at 3 months after IVIG treatment. 3) PCR for the detection of HCV was done in 8 patients with persistent HCV Ab positivity at 3 months after IVIG treatment, but HCV was not isolated in any cases. These results suggested that detection of anti-HCV was merely transitory phenominon of HCV Ab transmission, did not show any evidence of HCV infection due to HCV transmission.

A study of cisplatin nephrotoxicity.: Young Hee Hwang, Kyoung A Lee, Son Moon Shin, Young Hoon Park, Jeong Ok Hah, Chun Dong Kim, Young Hwan Lee; Yeungnam Univ J Med. 1992;9(2):327-333. Published online December 31, 1992; DOI: https://doi.org/10.12701/yujm.1992.9.2.327

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Abstract PDF: To evaluate the nephrotoxicity of cisplatin, serum levels of sodium, potassium, chloride, calcium, phosphorous, magnesium, BUN, creatinine and creatinine clearance were measured before and after administration of cisplatin in 18 cases of patients with malignant neoplasm. The results were as follows: 1) Serum calcium, magnesium, potassium and BUN levels were changed after cisplatin administration, but those changes were not statistically significant. 2) The mean value of creatinine clearance was not decreased significantly after treatment with cisplatin. 3) Acute renal failure was developed in one case, and four cases of hypocalcemia, hypomagnesemia were also detected after administration of cisplatin.

Treatment of Acute Lymphoblastic Leukemia in children.: Jeong Ok Hah; Yeungnam Univ J Med. 1992;9(1):10-22. Published online June 30, 1992; DOI: https://doi.org/10.12701/yujm.1992.9.1.10

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Abstract PDF: No abstract available.

A clinical study on children with delayed language development.: Jeong Ho Kim, Han Ku Moon, Jeong Ok Hah; Yeungnam Univ J Med. 1991;8(2):24-34. Published online December 31, 1991; DOI: https://doi.org/10.12701/yujm.1991.8.2.24

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Delayed emergence of speech or language are frequent causes for concern about development during early childhood. Delay in evaluation and proper management until school entry in more likely to result in frustration, anxiety and school failure. Many language disturbances and their attendant behavior disorders respond to intensive language therapy. Authors analyzed the medical records of 52 children with delayed language development evaluated during 30 months from January 1986 through June 1988. The results were as follows: 1. The majority of cases were evaluated at the age of 2-2.9 year old (16 cases, 30.8%) and 3-3.9 year old (11 cases, 21.2%) 2. Male to female ratio was 3:1 3. The most common cause was mental retardation (53.8%), followed by developmental language disorder (23.1%) and autism (13.5%) 4. The most common associated condition was dysarticulation (17.3%), followed by strabismus (9.6%) and seizures (7.7%) 5. Special education was recommended in cases of 23 (44.2%), speech therapy in 12 cases (23.1%) and consultation to child psychiatry in 7 cases (13.5%) Making diagnosis of underlying disorders in not simple because assessment of intelligence in young children in difficult and only a few tests are standardized. More detailed study on children with delayed language development and development of psychometric tests for handicapped children are necessary, especially in Korea.

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The usefulness of diagnostic tests in children with language delay
Seung Taek Oh, Eun Sil Lee, Han Ku Moon
Korean Journal of Pediatrics.2009; 52(3): 289. CrossRef

Low volume peritoneal dialysis in newborns and infants.: Young Hoon Park, Soo Ho Ahn, Son Moon Shin, Jeong Ok Hah; Yeungnam Univ J Med. 1991;8(2):128-137. Published online December 31, 1991; DOI: https://doi.org/10.12701/yujm.1991.8.2.128

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Abstract PDF: Peritoneal dialysis has been widely considered to be the dialytic treatment of choice for acute renal failure in infants and young children, because the technique is simple, safe and easily adapted for these patients. Also peritoneal dialysis in infants might have more effective ultrafiltration and clearance than in adults. In certain circumstances associated with hemodynamic instability, ordinary volume peritoneal dialysis (30-50 ml/kg body weight per exchange) or hemodialysis may not be suitable unfortunately. But frequent cycled, low volume, high concentration peritoneal dialysis may be more available to manage the acute renal failure of newborns and infants. Seven infants underwent peritoneal dialysis for hemodynamically unstable acute renal failure with low exchange volume (14.2±4.2 ml/kg), short exchange time (30 to 45 minutes) and hypertonic glucose solution (4.25% dextrose). Age was 1.9±1.3 months and body weight was 4.6±1.6 kg. Etiology of acute renal failure was secondary to sepsis with or without shock (5 cases) and postcardiac operation (2 cases). Catheter was inserted percutaneously with pigtail catheter or Tenkhoff catheter by Seldinger method. Dialysate was commercially obtained Peritosol which contained sodium, chloride, potassium, magnesium, lactate and calcium. Net ultrafiltration (ml/min) showed no difference between low volume dialysis and control (0.27±0.09 versus 0.29±0.09). Blood BUN decreased from 95.7±37.5 to 75.7±25.9 mg/dl and blood pH increased from 7.122±0.048 to 7.326±0.063 after 24 hours of peritoneal dialysis. We experienced hyperglycemia which were controlled by insulin (2 episodes), leakage at the exit site (2), mild hyponatremia (1) and Escherichia coli peritonitis (1). Two children of low volume dialysis died despite the treatment. In our experience, low volume and high concentration peritoneal dialysis with frequent exchange may have sufficient ultrafiltration and clearance without significant complications in the certain risked acute renal failure of infants.

Estimation of Glomerular Filtration Rate from Plasma Creatinine and Height in Children.: Jeong Lan Kim, Yong Hoon Park, Jeong Ok Hah; Yeungnam Univ J Med. 1988;5(1):93-100. Published online June 30, 1988; DOI: https://doi.org/10.12701/yujm.1988.5.1.93

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Abstract PDF: In clinical practice, creatinine clearance (Ccr) remains the most commonly used laboratory assessment of glomerular function despite methodological and technical problems of urine collection. Schwartz et al. in 1976, reported that an accurate estimate of glomerular filtration rate (GFR) could be obtained from the simple determinations of plasma creatinine (Per) and body length (L): GFR (ml/min/1.73m2)=kL (cm)/Per (mg/100 ml), (k=constant). The subject of this study were 81 children admitted to our pediatric department from July, 1985 to June, 1987 and they were divided into three groups; group I, from 1 to 5 years old, group II, from 6 to 10 years old, group III, from 11 to 15 years old. The results were as following: 1) Measured creatinine clearance Ccr(M), ml/min/1.73m²) were 109.73±9.97 in group I, 108.26±9.02 in group II, 96.20±4.72 in group III and 105.48±5.23 in all age group. 2) Measured k(k(M)) obtained from CcrM=k Ht/Pcr were 0.49±0.03 in group I, 0.48±0.02 in group II, 0.43±0.02 in group III, and 0.47±0.02 in all age group (Ht; height). 3) Linear equations and correlation coefficient between Ht/Pcr (x) and Ccr (y) were y=0.82x-65.63 (r=0.99) in group I, y=0.61x-23.46(r=0.72) in group II, y=0.18x+54.44 (r=0.54) in group III and y=0.58x-22.13 (r=0.81) in all age group. 4) Ccr(E) was again estimated from linear equations between Ht/Pcr and Ccr(M) and k(E) was calculated with Ht/Pcr and Ccr(E) were 0.48±0.01 in group I, 0.49±0.01 in group II, 0.43±0.01 in group III and 0.47±0.00 in all age group. 5) Consistent values of k(E) and k(M) were highly significant as 95~97.5% in group I and II, 90~95% in group III and 97.5~99% in all age group. In summary, we could estimate GFR with height, plasma creatinine and measured k(k(M)) according to the age in easy and rapid way.

A Clinical Study on Low Birth Weight Infants.: Han Ku Moon, Jung Ok Kim, Jae Honng Park, Son Moon Shin, Jeong Ok Hah; Yeungnam Univ J Med. 1987;4(2):97-103. Published online December 31, 1987; DOI: https://doi.org/10.12701/yujm.1987.4.2.97

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Abstract PDF: Neonatal and obstetrical problems related to the low birth weight infants were evaluated by a retrospective review of the medical records of the 186 low birth weight infants born at Yeungnam University Hospital during 3 years and 8 months from May 1, 1983 to February 28, 1987. The results were as follows: 1. The incidence of the low birth weight infants was 4.98% among 3,803 live births. 2. Male to female ratio was 1.02:1. 3. The incidence of the low birth weight infants was lowest in mothers of 25 to 29 years, increased in mothers of 19 years of less and 30 to 39 years significantly. 4. There was no difference in the incidence of low birth weight infants between primiparous and multiparous mothers. 5. Common obstetrical complications associated with low weight infants were multiple pregnancy, toxemia and premature rupture of membrane in order of frequency. 6. Common neonatal problems in low birth weight infants were jaundice, idiopathic respiratory, distress syndrome, asphyxia and metabolic derangement such as hypocalcemia and hypoglycemia in order of frequency.

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