Literature DB >> 3746838

Microcephaly, short stature, and developmental delay associated with a chemotactic defect and transient hypogammaglobulinaemia in two brothers.

B Say, N Barber, G C Miller, S E Grogg.   

Abstract

Two brothers presented with unusual facial features, microcephaly, developmental delay, and severe postnatal growth retardation. They both developed eczema in infancy and have had recurrent infections. Additional physical findings in both boys included hypogonadism, flexion contractures, hypoplastic patellae, and scoliosis. Their facial similarity was striking with sloping foreheads, beaked noses, large, protruding ears, and micrognathia. Low levels of serum gammaglobulins and defective chemotaxis were present in both boys in infancy. The hypogammaglobulinaemia was transient and improved, reaching normal levels by 3 1/2 years and 15 months, respectively. Defective chemotaxis and recurrent infections have persisted to the present. Both parents were normal. The mode of inheritance was not clear, as both X linked and autosomal recessive patterns were possible. Although patients with congenital malformations who also had immunodeficiency have previously been reported, immune system abnormalities, especially those of a transient nature, may frequently go unrecognised.

Entities:  

Mesh:

Year:  1986        PMID: 3746838      PMCID: PMC1049705          DOI: 10.1136/jmg.23.4.355

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  6 in total

1.  Ear anomalies associated with renal dysplasia and immunodeficiency disease. A histopathological study.

Authors:  J R Lindsay; R Hinojosa
Journal:  Ann Otol Rhinol Laryngol       Date:  1978 Jan-Feb       Impact factor: 1.547

2.  Association of birth defects and immunodeficiency.

Authors:  B Say; G C Miller; N Barber; S Grogg
Journal:  J Pediatr       Date:  1979-05       Impact factor: 4.406

3.  Hereditary lymphopenic agammaglobulinemia associated with a distinctive form of short-limbed dwarfism and ectodermal dysplasia.

Authors:  R A Gatti; N Platt; H H Pomerance; R Hong; L O Langer; H E Kay; R A Good
Journal:  J Pediatr       Date:  1969-10       Impact factor: 4.406

4.  Deficiency of humoral immunity and hypoparathyroidism associated with the Hallerman-Streiff syndrome.

Authors:  R K Chandra; S Joglekar; Z Antonio
Journal:  J Pediatr       Date:  1978-11       Impact factor: 4.406

5.  Immunodeficiency in children with severe craniofacial anomalies.

Authors:  G C Miller; B Say
Journal:  South Med J       Date:  1982-01       Impact factor: 0.954

6.  Transient hypogammaglobulinemia of infancy: review of the literature, clinical and immunologic features of 11 new cases, and long-term follow-up.

Authors:  T L Tiller; R H Buckley
Journal:  J Pediatr       Date:  1978-03       Impact factor: 4.406

  6 in total
  7 in total

1.  Bilateral choanal atresia and paranasal sinus hypoplasia in an adult patient with hypogammaglobulinaemia.

Authors:  Hamid El-Sawy; M Azher Siddiq; Aubu Anbarasu
Journal:  Eur Arch Otorhinolaryngol       Date:  2006-07-15       Impact factor: 2.503

2.  Microcephaly-cardiomyopathy: a new autosomal recessive phenotype?

Authors:  I M Winship; D L Viljoen; P M Leary; M M De Moor
Journal:  J Med Genet       Date:  1991-09       Impact factor: 6.318

Review 3.  Microcephaly, characteristic facies, joint abnormalities, and deficient leucocyte chemotaxis: a further case of the syndrome of Say et al.

Authors:  C Perandones; R I Cerretini; R M Vargas Vera; E I Aranda; L G Alba; O H Pivetta
Journal:  J Med Genet       Date:  1996-03       Impact factor: 6.318

4.  A case of Barber-Say syndrome in a male Japanese newborn.

Authors:  Kenichi Suga; Miki Shono; Aya Goji; Sato Matsuura; Miki Inoue; Masami Kawahito; Michiyo Kinoshita; Misa Takeda; Kazuhiro Mori
Journal:  Clin Case Rep       Date:  2014-09-04

5.  Barber-say syndrome: a confirmed case of TWIST2 gene mutation.

Authors:  Mulakkan David Yohannan; Jennifer Hilgeman; Katlin Allsbrook
Journal:  Clin Case Rep       Date:  2017-06-02

6.  General anesthesia of a Japanese infant with Barber-Say syndrome: a case report.

Authors:  Eisuke Hamaguchi; Yasuo M Tsutsumi; Katsuyoshi Kume; Yoko Sakai; Nami Kakuta; Yuta Uemura; Shinji Kawahito; Katsuya Tanaka
Journal:  JA Clin Rep       Date:  2016-06-02

Review 7.  Skeletal malocclusion: a developmental disorder with a life-long morbidity.

Authors:  Nishitha Joshi; Ahmad M Hamdan; Walid D Fakhouri
Journal:  J Clin Med Res       Date:  2014-09-09
  7 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.