Agnogenic myeloid metaplasia in childhood: A report of two cases and efficiency of intravenous high dose methylprednisolone treatment

Çetingül, Nazan; Yener, Erdal; Öztop, Senay; Nişli, Güngör; Soydan, Saliha

doi:10.1111/j.1442-200x.1994.tb03273.x

Cited by 6 publications

(5 citation statements)

References 21 publications

(1 reference statement)

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“…Fewer than 25 cases of paediatric IMF, none of which had the extent of long‐term follow‐up detailed here, have been reported ( Lau et al , 1981 ; Cetingul et al , 1994 ; Sekhar et al , 1996; Domingues et al , 1998 ). Furthermore, among a large series of childhood myelodysplastic syndrome–myeloproliferative syndrome cases, IMF was not identified ( Luna‐Fineman et al , 1999 ).…”

Section: Discussionmentioning

confidence: 78%

“…Interestingly, younger patients (< 64 years) were included in the favourable survival group ( Cervantes et al , 1997 ; Manoharan, 1998). In children, the outcome appears to be even more favourable ( Reid et al , 1988 ; Cetingul et al , 1994 ; Sekhar et al , 1996 ). Here, we describe the clinical characteristics of IMF in three infants, two of whom are long‐term survivors.…”

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confidence: 99%

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Long‐term survival of infants with idiopathic myelofibrosis

Altura

Head²,

Wang

2000

Br J Haematol

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Summary. Idiopathic myelofibrosis can develop in children as well as adults. However, the disease appears to be much more aggressive in adults, being characterized by poor survival rates and a high frequency of malignant transformation. Here, we describe three cases of idiopathic myelofibrosis in infants, two of whom were followed for 16 and 22 years after diagnosis. Neither of these patients required more than minimal supportive care, and both have had spontaneous erythropoietic recovery as early as 2± 3 years after diagnosis. There have been no indications of malignant transformation or clinical deterioration. Thus, idiopathic myelofibrosis may have a different pathogenesis and clinical course in infants from adults, requiring a more conservative approach to management.

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Section: Discussionmentioning

confidence: 78%

mentioning

confidence: 99%

Long‐term survival of infants with idiopathic myelofibrosis

Altura

Head²,

Wang

2000

Br J Haematol

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“…17 Both children with IMF are of Indian/Sri Lankan descent, similar to early reports of pediatric IMF. [18][19][20] They underwent investigations to rule out secondary causes. Hepatosplenomegaly was present in one patient.…”

Section: Resultsmentioning

confidence: 99%

A Basic Classification and a Comprehensive Examination of Pediatric Myeloproliferative Syndromes

Gassas

Doyle

Weitzman

et al. 2005

Journal of Pediatric Hematology/Oncology

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Myeloproliferative syndromes (MPSs) are clonal stem cell disorders resulting in excessive proliferation of one or more cell lineages. Since MPSs in children occur much less commonly than adults, one can argue that the biology and the categories of the various pediatric MPSs seem to be different from adults. Furthermore, confusion exists between pediatric MPS and other overlapping conditions, such as myelodysplastic syndrome. The authors' objectives were to develop a classification system with a list of disorders relevant to children and to characterize pediatric cases of MPS that were devised according to this classification. Based on the predominant proliferating cell lineage, the authors established a classification system for childhood MPS. Primary MPS was classified into granulocytic proliferation--chronic myelogenous leukemia (CML); monocytic--juvenile myelomonocytic leukemia (JMML); megakaryocytic--essential thrombocythemia (ET), familial thrombocytosis, transient myeloproliferative disorder of Down syndrome (TMD); erythrocytic--polycythemia vera, familial erythrocytosis; fibroblastic--idiopathic myelofibrosis (IMF); eosinophilic--idiopathic hypereosinophilic syndrome (IHES); and mast cells--mastocytosis. Secondary MPS was classified as non-clonal proliferation (eg, infections, drugs, toxins, autoimmune, non-hematologic neoplasm, and trauma), and these were excluded from the study. Next, the classification system was applied to the patient population at the authors' institution. One hundred two cases with primary MPS were identified between 1970 and 2001. Patients were evaluated for clinical manifestations, blood and bone marrow parameters, cytogenetics, and survival following different treatment modalities. Significant proportions of cases of childhood MPS (60%) were unique to the pediatric population and not seen in adults. The most common disorders were JMML (n = 31), TMD of Down syndrome (n = 30), and CML (n = 30); the other disorders were rare: four cases of ET, two of IMF, two of IHES, two of mastocytosis, and one primary erythrocytosis. In contrast to adults, MPS in children is more frequently treated with hematopoietic stem cell transplantation (HSCT), the only available curative option for most of these diseases. HSCT was particularly successful in the more recent cases due to more advanced techniques for HSCT. The authors found that all the cases could be easily classified. MPS in children is different from adult-type MPS in terms of biology, categories, classification, and prognosis.

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“…Thus, several cases are reported in the literature to undergo spontaneous remission, to be successfully treated symptomatically or with intravenous high dose of methylprednisolone. [2,4,6,7] Nevertheless, the only curative treatment is HSCT, which leads to complete resolution of myelofibrosis with normal hematopoiesis. [3] According to the 2017 guidelines of PMF in adults, observation alone is a reasonable treatment strategy for asymptomatic low-or intermediate-1-risk disease, especially in the absence of high-risk mutations, and all the other patients should be considered for HSCT.…”

Section: Discussionmentioning

confidence: 99%

Childhood Primary Myelofibrosis Presented with Headache, Splenomegaly, and Severe Thrombocytosis: A Case Report

Tragiannidis

Apsemidou

Liampas

et al. 2018

Indian Journal of Medical and Paediatric Oncology

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Primary myelofibrosis (PMF) is a clonal disorder of a multipotent hematopoietic progenitor cell that occurs predominantly in the elderly age group. We report here an 11-year-old girl who presented with headache, fever, and splenomegaly. Full blood cell count revealed severe thrombocytosis. Laboratory and radiology examinations excluded the diagnosis of essential/reactive thrombocytosis. Bone marrow biopsy showed megakaryocytic hyperplasia, reticulin and collagen fibrosis, and erythroid and myeloid hypoplasia, findings compatible to PMF. The patient was put symptomatically on hydroxyurea and hydration due to thrombocytosis and platelet number decreased. Hematopoietic stem cell transplantation was scheduled to avoid delaying definitive therapy and secondary complications such as infections and transfusion dependency. To the best of our knowledge, this is the first reported case of PMF in childhood in Greece.

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Agnogenic myeloid metaplasia in childhood: A report of two cases and efficiency of intravenous high dose methylprednisolone treatment

Cited by 6 publications

References 21 publications

Long‐term survival of infants with idiopathic myelofibrosis

Long‐term survival of infants with idiopathic myelofibrosis

A Basic Classification and a Comprehensive Examination of Pediatric Myeloproliferative Syndromes

Childhood Primary Myelofibrosis Presented with Headache, Splenomegaly, and Severe Thrombocytosis: A Case Report

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