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PubMed1986

Phagocyte defects.

Although inherited forms of phagocyte defects affect a small proportion of the general population, their clinical course can be altered dramatically by a physician's awareness of these diseases and modifications of the approach to and treatment of affected patients. The most common syndromes are chronic granulomatous disease of childhood (CGD), the Chediak-Higashi syndrome (CHS), the hyperimmunoglobulin-E-recurrent infection (Job's) syndrome (HIE), and myeloperoxidase (MPO) deficiency. CGD patients have defects in the oxidative metabolism involved in killing catalase-positive organisms. CHS patients have giant granules defective in fusing with phagosomes and subsequent killing of ingested organisms. HIE patients have abnormal chemotaxis and elevated IgE levels and are susceptible to skin infections with Staphylococcus aureus and recurrent sinopulmonary infections. MPO-deficient patients often go undetected since they rarely have recurrent infections unless they have a concomitant disease such as diabetes mellitus. Patients with a recently described syndrome, C3bi receptor deficiency, have recurrent bacterial infections and persistent leukocytosis, and their neutrophils have abnormal adherence and phagocytosis. The absence of specific granules is a more rare entity but these patients also have recurrent infections thought to be secondary to a chemotactic defect and a minor abnormality of microbial killing exhibited by their neutrophils. This review will focus on the clinical presentation and management of these patients.

Chediak-Higashi Syndrome↗
PubMed1987

Evolution of the hyperimmunoglobulin E and recurrent infection (HIE, JOB's) syndrome in a young girl.

The hyperimmunoglobulin E and recurrent infection syndrome is difficult to diagnose in children with markedly elevated IgE and recurrent superficial Staphylococcus aureus infections who have not presented with a severe infection. The patient, the child of a woman with HIE, had elevated cord blood IgE. In early infancy, she had cutaneous colonization with S. aureus followed by frank impetiginous lesions. Anti-S. aureus IgE was easily detected with a highly specific ELISA assay at 2 years of age (2 years before her presentation with a S. aureus subcutaneous abscess). Thus, the measurement of anti-S. aureus IgE by this technique may be a useful laboratory test for the diagnosis of HIE before the appearance of a severe infection.

Abscess↗
PubMed1987

Elevated urinary histamine in the hyperimmunoglobulin E and recurrent infection (Job's) syndrome: association with eczematoid dermatitis and not with infection.

To test the hypothesis that IgE-mediated release of histamine may be, in part, responsible for the abnormal inflammatory response observed in the hyperimmunoglobulin E (HIE) and recurrent infection syndrome, urine and plasma histamine levels were measured. Twenty-four-hour urinary histamine concentrations from 15 patients with HIE (23.7 +/- 6.9 micrograms/24 hr) were significantly elevated (p less than 0.001) compared to a large historical control population (10.5 +/- 0.7 micrograms/24 hr; n = 97). However, urinary histamine levels in HIE were much less abnormal (p less than 0.01) than in five patients with biopsy-proven systemic mastocytosis (159 +/- 62 micrograms/24 hr) and were not significantly elevated when levels were compared to 13 concurrently studied normal subjects (10.1 +/- 1.7 micrograms/24 hr) and nine patients with chronic granulomatous disease (8.1 +/- 1.2 micrograms/24 hr). Overall, there was no clear relationship between urine histamine values and the presence of infection as well as no significant correlation between urine histamine and total IgE or anti-Staphylococcus aureus IgE. However, urine histamine levels in a subgroup of six patients with HIE with chronic eczematoid dermatitis (42.4 +/- 12.5 micrograms/24 hr) were elevated compared with values from the historical control subjects (p less than 0.001), the concurrent control subjects (p less than 0.01), the patients with chronic granulomatous disease (p less than 0.01), and five patients with HIE who did not have skin manifestations (4.6 +/- 1.1 micrograms/24 hr; p less than 0.05).(ABSTRACT TRUNCATED AT 250 WORDS)

Dermatitis, Contact↗
PubMed1988

Possible treatment of AIDS patients with live lactobacteria.

The enhancement of antimicrobial resistance and immunomodulatory action, and the anabolic effect caused by the consumption of live lactobacteria as a dietary adjunct are proposed by the author as sufficient reasons to test lactobacterial preparations in patients with AIDS. The problem of dosage is discussed and a practical solution presented.

Acetylmuramyl-Alanyl-Isoglutamine↗
PubMed1987

Hyperimmunoglobulinemia E and pregnancy: a case report.

Hyperimmunoglobulinemia E is characterized by recurrent bacterial sinopulmonary and skin infections from birth or early childhood, with IgE levels at least 10 times greater than the upper limits of normal. The following case describes a young black woman with hyperimmunoglobulinemia E syndrome who had an uneventful pregnancy and delivery. The infant has been diagnosed as suffering from hyperimmunoglobulinemia E syndrome as well.

Adult↗
PubMed1990

Impairment of neutrophil chemotactic and bactericidal function in children infected with human immunodeficiency virus type 1 and partial reversal after in vitro exposure to granulocyte-macrophage colony-stimulating factor.

Because polymorphonuclear neutrophils are the most important component of host defense against bacteria, we assessed their function in 13 children with asymptomatic and 12 with symptomatic infection with human immunodeficiency virus type 1 (HIV-1), and compared their values with healthy adult control values. The functions assessed were (1) chemotaxis, (2) bacterial phagocytosis, (3) superoxide generation, and (4) bactericidal activity. Chemotaxis of polymorphonuclear neutrophils toward the chemoattractant N-formylmethionyl leucyl phenylalanine (FMLP) was significantly decreased in symptom-free infected children compared with control subjects (p less than 0.0001), but was increased in children with symptomatic infection (p less than 0.025). Bactericidal activity of the neutrophils against Staphylococcus aureus was defective in 8 of 12 children with asymptomatic infection (p = 0.016), and in 8 of 9 children with symptomatic infection (p less than 0.00001). Superoxide generation by polymorphonuclear neutrophils on stimulation with FMLP and phagocytosis of S. aureus were normal. Serum from patients with symptomatic HIV-1 infection was not as efficient in low concentrations as normal serum in the ability to opsonize S. aureus. The in vitro bactericidal defect was partially corrected by granulocyte-macrophage colony-stimulating factor (GM-CSF). The results suggest that both cellular (neutrophils) and humoral defects contribute to the increased incidence of bacterial infections in HIV-1-infected children, and that GM-CSF may improve the defective bactericidal activity of polymorphonuclear neutrophils in these patients.

Adolescent↗