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PubMed1976

Dumbbell granulomatous abscess of the chest wall following needle biopsy of the pleura.

A 38-year-old woman who had a Cope needle biopsy of the pleura was treated for plural tuberculosis on the basis of a positive PPD-S skin test and presence of caseating granulomas in the pleural biopsy. Ten months later she developed a tender, subcutaneous nodule in the area of the previous needle biopsy. Surgical exploration revealed a dumbbell abscess through the chest wall communicating with an area of consolidation in the right middle lobe. En bloc surgical resection of the abscess and peripheral portion of the right middle lobe was curative, although all pathologic and cultural studies of the resected tissue were non-diagnostic.

Abscess↗
PubMed1990

Spinal anaesthesia in a child with Job's syndrome, pneumatoceles and empyema.

We present a case of acute bowel obstruction in an immunocompromised child, who also had lobar pneumonia and a giant unilateral pneumatocele. She was successfully managed with subarachnoid anaesthesia for exploratory laparotomy to relieve a colonic obstruction. This proved to be a safe alternative to general anaesthesia with tracheal intubation in this patient and should be considered in infants and children in selected cases whenever a contraindication to general anaesthesia exists.

Anesthesia, Spinal↗
PubMed1975

Chronic granulomatous disease and the Kell blood groups.

Fifteen antigenic determinants are known to be related to the Kell blood group. Some boys with X-linked chronic granulomatous disease have the very rare McLeod or Ko phenotype on their red cells. Serological studies of the McLeod type suggest that the weak Kell antigens that are present differ qualitatively and quantitatively from those on red cells of common Kell type. A new antigen, Kx, has been characterized and shown to be present on red cells and neutrophil leucocytes. Lack of red-cell Kx is associated with the McLeod phenotype, lack of leucocyte Kx is associated with chronic granulomatous disease.

Antigen-Antibody Reactions↗
PubMed1991

Role of leucocyte integrins in phagocyte responses to granulocyte-macrophage colony stimulating factor (GM-CSF): in vitro and in vivo studies on leucocyte adhesion deficiency neutrophils.

The role of leucocyte integrins in phagocyte function has been studied by comparing normal neutrophils with those from a patient with partial leucocyte adhesion deficiency (LAD), in whom the levels of CD11b and CD11c were 10% of controls, whereas CD11a levels were normal. Unstimulated LAD neutrophils exhibited defective adhesion to plastic (4.4 +/- 1.5% cf. 14.4 +/- 3.8% in controls), but not to human umbilical vein endothelial cells (HUVECs). The adhesion to HUVECs could be further upregulated by granulocyte-macrophage colony stimulating factor (GM-CSF), but not by 12-O-tetradecanoylphorbol 13-acetate (TPA) which, in normal cells, is a more potent 'pro-adhesive agonist'. The normal neutrophil-endothelial interaction induced by GM-CSF in LAD neutrophils was confirmed in vivo when administration of GM-CSF resulted in rapid phagocyte margination. Neutrophil migration and phagocytosis/killing were defective in LAD neutrophils, and some improvement in phagocytosis/killing was seen following in vivo administration of GM-CSF. These studies illustrate that the degree to which the leucocyte integrins mediate adherence-related phagocyte functions varies not only with the particular function, but also with the conditions of stimulation. High levels of CD11b and CD11c expression appear not to be required for unstimulated or GM-CSF-stimulated neutrophil-endothelial interactions, either in vitro or in vivo. Other neutrophil functions, on the other hand, such as migration and phagocytosis/killing are much more dependent on the leucocyte integrins.

Antigens, CD↗
PubMed1976

Diffuse chronic granulomatous mucocutaneous candidiasis.

A 3-year-old Thai boy with diffuse chronic granulomatous mucocutaneous candidiasis, recurrent bacterial skin infection and adrenal insufficiency is reported. Candida albicans was demonstrated in the dermal granuloma. He had a defect in cell-mediated immunity and was anemic. Although therapy with topical clotrimazole, oral iron, systemic antibiotic and low-dose of prednisone gave a dramatic result, he died of disseminated cryptococcosis.

Adrenal Insufficiency↗
PubMed1976

Frequency of the carrier state for X-linked chronic granulomatous disease among females with lupus erythematosus.

Carriers for chronic granulomatous disease (CGD) and patients with lupus erythematosus (LE) share several characteristics: They are mostly females, they reduce nitroblue tetrazolium (NBT) poorly in their neutrophils, and, in some cases, they have similar skin lesions. We thus investigated 19 female LE patients for the presence of laboratory findings characteristic of the carrier state for CGD. None of these patients turned out to have the combined abnormality of neutrophil bactericidal activity and neutrophil NBT-reduction that is diagnostic of CGD carrier state in the X-linked form. An increased frequency of CGD carriers among female LE patients thus appear to be unlikely. Why some CGD carriers develop skin lesions typical of LE remains unexplained.

Adult↗
PubMed1976

Kx antigen, the McLeod phenotype, and chronic granulomatous disease: further studies.

Leukocytes of nine unrelated boys with X-linked chronic granulomatous disease lack Kx antigen. In three of these cases, the red cells also lack Kx and have the McLeod phenotype and abnormal morphology. X-linked chronic granulomatous disease CGD can thus be separated into two types. Type I cases have an antigenic deficiency that is restricted to the phagocytic leukocytes while in type II, the deficiency involves both leukocytes and red cells. Red cells of type II CGD patients have enhanced i antigen activity suggesting that they are under hemopoietic stress. Normal Kx synthesis is directed by an X-linked gene named X1k. Three variants, X2k, X3k, and X4k order the different permutations of leukocyte and red cell Kx antigen production that have been recognized.

Blood Group Antigens↗