Literature DB >> 18759084

Paroxysmal nocturnal hemoglobinuria in childhood and adolescence--a retrospective analysis of 18 cases.

Rahul Naithani1, Manoranjan Mahapatra, Pankhi Dutta, Rajat Kumar, Hara Prasad Pati, Ved Prakash Choudhry.   

Abstract

OBJECTIVE: To assess the clinical and hematological profile of PNH in children.
METHODS: Clinical and laboratory features of children with PNH diagnosed in the past six years at our centre were reviewed. Various investigations done included a complete blood count and peripheral smear examination, plasma hemoglobin, urine hemosiderin, acid ham test, sucrose lysis test, immunophenotyping of erythrocytes by sephadex column gel card and of granulocytes by flow cytometry. There were 18 children with a marked male predominance (M 14: F 4).
RESULTS: Pallor, jaundice, dark urine and bleeding manifestations were the major presenting complaints. One girl suffered an arterial stroke. All children had cytopenia in at least one cell line. Children were treated with danazol, stanazolol, prednisolone and cyclosporin A variously. Overall response rate was 61%. Children with classical PNH performed slightly better with response rates of 66% (6/9) as compared to aplastic anemia-PNH group which has a response rate of 55% (5/9). Amongst various variables only danazol correlated with better response (p=0.029).
CONCLUSION: PNH is an uncommon disease in children and should be included in the differential diagnosis of children presenting with cytopenia.

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Year:  2008        PMID: 18759084     DOI: 10.1007/s12098-008-0111-9

Source DB:  PubMed          Journal:  Indian J Pediatr        ISSN: 0019-5456            Impact factor:   1.967


  23 in total

1.  Flowcytometric detection of PNH defect in Indian patients with aplastic anemia and myelodysplastic syndromes.

Authors:  N Varma; G Garewal; S Varma; H Vohra
Journal:  Am J Hematol       Date:  2000-11       Impact factor: 10.047

2.  The hematopoietic defect in PNH is not due to defective stroma, but is due to defective progenitor cells.

Authors:  Jun-ichi Nishimura; Russell E Ware; Angela Burnette; Andrew L Pendleton; Kiyoshi Kitano; Toshiyuki Hirota; Takashi Machii; Teruo Kitani; Clay A Smith; Wendell F Rosse
Journal:  Blood Cells Mol Dis       Date:  2002 Sep-Oct       Impact factor: 3.039

3.  Syngeneic bone marrow transplantation without conditioning in a patient with paroxysmal nocturnal hemoglobinuria: in vivo evidence that the mutant stem cells have a survival advantage.

Authors:  M Endo; P G Beatty; T M Vreeke; C T Wittwer; S P Singh; C J Parker
Journal:  Blood       Date:  1996-07-15       Impact factor: 22.113

4.  Paroxysmal nocturnal haemoglobinuria: long-term follow-up and prognostic factors. French Society of Haematology.

Authors:  G Socié; J Y Mary; A de Gramont; B Rio; M Leporrier; C Rose; P Heudier; H Rochant; J Y Cahn; E Gluckman
Journal:  Lancet       Date:  1996-08-31       Impact factor: 79.321

5.  Paroxysmal nocturnal hemoglobinuria in childhood and adolescence. Clinical and erythrocyte metabolic studies in two cases.

Authors:  D R Miller; R L Baehner; L K Diamond
Journal:  Pediatrics       Date:  1967-05       Impact factor: 7.124

6.  Paroxysmal nocturnal haemoglobinuria in Thailand with special reference to as association with aplastic anaemia.

Authors:  M Kruatrachue; P Wasi; S Na-Nakorn
Journal:  Br J Haematol       Date:  1978-06       Impact factor: 6.998

7.  Screening for paroxysmal nocturnal hemoglobinuria (PNH) clone in Egyptian children with aplastic anemia.

Authors:  S Rizk; I Youssry Ibrahim; I M Mansour; D Kandil
Journal:  J Trop Pediatr       Date:  2002-06       Impact factor: 1.165

8.  Paroxysmal nocturnal hemoglobinuria arising from Fanconi anemia.

Authors:  Linda Wainwright; Robert A Brodsky; Linda K Erasmus; Stelios Poyiadjis; Gita Naidu; Diane MacKinnon
Journal:  J Pediatr Hematol Oncol       Date:  2003-02       Impact factor: 1.289

Review 9.  Stem cell transplantation for paroxysmal nocturnal haemoglobinuria in childhood.

Authors:  Christian Flotho; Brigitte Strahm; Udo Kontny; Ulrich Duffner; Anke M J Peters; Wolfgang Dupuis; Charlotte M Niemeyer
Journal:  Br J Haematol       Date:  2002-07       Impact factor: 6.998

10.  Clinical significance of a minor population of paroxysmal nocturnal hemoglobinuria-type cells in bone marrow failure syndrome.

Authors:  Hongbo Wang; Tatsuya Chuhjo; Shizuka Yasue; Mitsuhiro Omine; Shinji Nakao
Journal:  Blood       Date:  2002-08-08       Impact factor: 22.113

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  5 in total

1.  Different clinical characteristics of paroxysmal nocturnal hemoglobinuria in pediatric and adult patients.

Authors:  Álvaro Urbano-Ispizua; Petra Muus; Hubert Schrezenmeier; Antonio M Almeida; Amanda Wilson; Russell E Ware
Journal:  Haematologica       Date:  2016-11-24       Impact factor: 9.941

Review 2.  Paroxysmal nocturnal haemoglobinuria.

Authors:  Anita Hill; Amy E DeZern; Taroh Kinoshita; Robert A Brodsky
Journal:  Nat Rev Dis Primers       Date:  2017-05-18       Impact factor: 52.329

3.  Cerebral Stroke in a Teenage Girl with Paroxysmal Nocturnal Hemoglobinuria.

Authors:  Francesco Gervasi; Lucia D'Amelio; Antonino Trizzino; Fabrizia Ferraro; Delia Russo; Giuseppe Santangelo; Francesca Cardella; Angela Trizzino; Floriana Di Marco; Piero Farruggia
Journal:  Hematol Rep       Date:  2017-06-01

4.  Fatigue, symptom burden, and health-related quality of life in patients with myelodysplastic syndrome, aplastic anemia, and paroxysmal nocturnal hemoglobinuria.

Authors:  Carmen P Escalante; Stephanie Chisolm; Juhee Song; Marsha Richardson; Ellen Salkeld; Etsuko Aoki; Guillermo Garcia-Manero
Journal:  Cancer Med       Date:  2019-01-11       Impact factor: 4.452

5.  Diagnostic and Treatment Challenges of Paroxysmal Nocturnal Hemoglobinuria in Uganda.

Authors:  Boniface Amanee Elias Lumori; Daniel Muyanja
Journal:  Case Rep Hematol       Date:  2019-01-31
  5 in total

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