Literature DB >> 8185357

Early diagnosis of severe combined immunodeficiency syndrome.

R A Hague1, S Rassam, G Morgan, A J Cant.   

Abstract

Infants with severe combined immunodeficiency syndrome (SCIDS) have a greatly improved prognosis if diagnosed and treated before they develop overwhelming infection. Clinical and laboratory data on 45 patients with SCIDS were retrospectively reviewed to assess the value of absolute lymphocyte counts in making an early diagnosis. Ninety infants matched for age, sex, and presenting symptoms were used as controls. Thirteen (29%) infants with SCIDS were diagnosed at birth as previous siblings had been affected; 32 (71%) were diagnosed after the development of symptoms. Eighteen (56%) of these remained undiagnosed until after 6 months of age. The first symptoms occurred at a median of 5 weeks (range 1 day to 8 months) and the first admission to hospital was at 4 months (range 1 week to 16 months). Symptoms included respiratory infection (91%), vomiting and diarrhoea (81%), failure to thrive (88%), candidiasis (50%), and skin lesions (28%). The mean lymphocyte count was 1.71 x 10(9)/l compared with 7.2 x 10(9)/l in controls. Excluding one child with Omenn's syndrome (lymphocyte count 23.3 x 10(9)/l, all symptomatic infants with SCIDS had lymphocyte counts less than 2.8 x 10(9)/l at presentation. The median delay between the first abnormal lymphocyte count and diagnosis was seven weeks (range one day to 13 months). Twenty eight (88%) of 32 infants would have been diagnosed before 6 months of age if investigated after the first low lymphocyte count. These data indicate that low lymphocyte counts are predictive of SCIDS. Paediatricians are urged to pay attention to the absolute lymphocyte counts in all infants in whom a full blood count is performed. Those with lymphocyte counts persistently less than 2.8 x 10(9)l should be investigated for SCIDS.

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Year:  1994        PMID: 8185357      PMCID: PMC1029775          DOI: 10.1136/adc.70.4.260

Source DB:  PubMed          Journal:  Arch Dis Child        ISSN: 0003-9888            Impact factor:   3.791


  14 in total

1.  Prevalence of lymphocytopenia in severe combined immunodeficiency.

Authors:  D L Gossage; R H Buckley
Journal:  N Engl J Med       Date:  1990-11-15       Impact factor: 91.245

2.  Cutaneous lesions as a clue to severe combined immunodeficiency.

Authors:  L De Raeve; M Song; J Levy; F Mascart-Lemone
Journal:  Pediatr Dermatol       Date:  1992-03       Impact factor: 1.588

3.  SCID continues to point the way.

Authors:  E W Gelfand
Journal:  N Engl J Med       Date:  1990-06-14       Impact factor: 91.245

4.  Vertical transmission of HIV: a prospective study.

Authors:  J Y Mok; R A Hague; P L Yap; F D Hargreaves; J M Inglis; J M Whitelaw; C M Steel; O B Eden; S Rebus; J F Peutherer
Journal:  Arch Dis Child       Date:  1989-08       Impact factor: 3.791

5.  Epidemiology and treatment of hypogammaglobulinemia.

Authors:  C S Hosking; D M Roberton
Journal:  Birth Defects Orig Artic Ser       Date:  1983

6.  Severe combined immunodeficiency disease. Characterization of the disease and results of transplantation.

Authors:  M M Bortin; A A Rimm
Journal:  JAMA       Date:  1977-08-15       Impact factor: 56.272

7.  Assessment of a clinical scoring system for detection of immunodeficiency in children with recurrent infections.

Authors:  E G Lyall; O B Eden; R Dixon; R Sutherland; A Thomson
Journal:  Pediatr Infect Dis J       Date:  1991-09       Impact factor: 2.129

8.  NK cell function in severe combined immunodeficiency (SCID): evidence of a common T and NK cell defect in some but not all SCID patients.

Authors:  H H Peter; W Friedrich; R Dopfer; W Müller; C Kortmann; W J Pichler; F Heinz; C H Rieger
Journal:  J Immunol       Date:  1983-11       Impact factor: 5.422

9.  Severe combined immunodeficiency with T lymphocytes retaining functional activity.

Authors:  G Fontán; M C García Rodriguez; S Carrasco; J M Zabay; E G de la Concha
Journal:  Clin Immunol Immunopathol       Date:  1988-03

10.  European experience of bone-marrow transplantation for severe combined immunodeficiency.

Authors:  A Fischer; P Landais; W Friedrich; G Morgan; B Gerritsen; A Fasth; F Porta; C Griscelli; S F Goldman; R Levinsky
Journal:  Lancet       Date:  1990-10-06       Impact factor: 79.321

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  15 in total

Review 1.  Severe combined immunodeficiency--molecular pathogenesis and diagnosis.

Authors:  H B Gaspar; K C Gilmour; A M Jones
Journal:  Arch Dis Child       Date:  2001-02       Impact factor: 3.791

Review 2.  Diagnosis of severe combined immunodeficiency.

Authors:  A R Gennery; A J Cant
Journal:  J Clin Pathol       Date:  2001-03       Impact factor: 3.411

3.  Fiscal implications of newborn screening in the diagnosis of severe combined immunodeficiency.

Authors:  Catherine Kubiak; Soma Jyonouchi; Caroline Kuo; Maria Garcia-Lloret; Morna J Dorsey; John Sleasman; Arthur S Zbrozek; Elena E Perez
Journal:  J Allergy Clin Immunol Pract       Date:  2014-08-28

4.  Clinical characteristics and mutation analysis of X-linked severe combined immunodeficiency in China.

Authors:  Cui Zhang; Zhi-Yong Zhang; Jun-Feng Wu; Xue-Mei Tang; Xi-Qiang Yang; Li-Ping Jiang; Xiao-Dong Zhao
Journal:  World J Pediatr       Date:  2011-11-21       Impact factor: 2.764

5.  Cost-Effectiveness/Cost-Benefit Analysis of Newborn Screening for Severe Combined Immune Deficiency in Washington State.

Authors:  Yao Ding; John D Thompson; Lisa Kobrynski; Jelili Ojodu; Guisou Zarbalian; Scott D Grosse
Journal:  J Pediatr       Date:  2016-02-11       Impact factor: 4.406

6.  Engraftment of human iPS cells and allogeneic porcine cells into pigs with inactivated RAG2 and accompanying severe combined immunodeficiency.

Authors:  Kiho Lee; Deug-Nam Kwon; Toshihiko Ezashi; Yun-Jung Choi; Chankyu Park; Aaron C Ericsson; Alana N Brown; Melissa S Samuel; Kwang-Wook Park; Eric M Walters; Dae Young Kim; Jae-Hwan Kim; Craig L Franklin; Clifton N Murphy; R Michael Roberts; Randall S Prather; Jin-Hoi Kim
Journal:  Proc Natl Acad Sci U S A       Date:  2014-05-05       Impact factor: 11.205

7.  Severe combined immunodeficiency in Serbia and Montenegro between years 1986 and 2010: a single-center experience.

Authors:  Srdjan Pasic; Dragana Vujic; Dobrila Veljković; Bojana Slavkovic; Marija Mostarica-Stojkovic; Predrag Minic; Aleksandra Minic; Goran Ristic; Silvia Giliani; Anna Villa; Cristina Sobacchi; Desa Lilić; Mario Abinun
Journal:  J Clin Immunol       Date:  2014-02-01       Impact factor: 8.317

8.  The respiratory presentation of severe combined immunodeficiency in two Mennonite children at a tertiary centre highlighting the importance of recognizing this pediatric emergency.

Authors:  Simon Lam; Fotini Dimitriou Kavadas; Seemab Haider; Mary Elizabeth Noseworthy
Journal:  Can Respir J       Date:  2013-11-28       Impact factor: 2.409

9.  Clinical characteristics and genetic profiles of 44 patients with severe combined immunodeficiency (SCID): report from Shanghai, China (2004-2011).

Authors:  Chun-Mei Yao; Xiao-Hua Han; Yi-Dan Zhang; Hui Zhang; Ying-Ying Jin; Rui-Ming Cao; Xi Wang; Quan-Hua Liu; Wei Zhao; Tong-Xin Chen
Journal:  J Clin Immunol       Date:  2012-12-19       Impact factor: 8.317

Review 10.  Clinical immunology review series: an approach to the patient with recurrent infections in childhood.

Authors:  M A Slatter; A R Gennery
Journal:  Clin Exp Immunol       Date:  2008-03-28       Impact factor: 4.330

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