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Interleukin-12 and Interleukin-23 Blockade in Leukocyte Adhesion Deficiency Type 1

by Zerbe, Christa S , Hajishengallis, George , Wild, Teresa , Notarangelo, Lucia D , Uzel, Gulbu , Holland, Steven M , Notarangelo, Luigi D , Moutsopoulos, Niki M , Lisco, Andrea , Dutzan, Nicolas , DiPasquale, Giovanni , Axelrod, Karen C , Brenchley, Laurie

in Allergies / Animal models / Antibiotics / Biopsy / Cytokines / Disease / Gingiva - pathology / Humans / Hypotheses / Infections / Infectious diseases / Inflammation / Injections, Subcutaneous / Interleukin 12 / Interleukin 17 / Interleukin 23 / Interleukin-12 - antagonists & inhibitors / Interleukin-17 - metabolism / Interleukin-23 - antagonists & inhibitors / Interleukin-23 - metabolism / Leukocyte-Adhesion Deficiency Syndrome - complications / Leukocyte-Adhesion Deficiency Syndrome - drug therapy / Leukocytes / Male / Monoclonal antibodies / Neutropenia / Neutrophils / Patients / Periodontal Diseases - drug therapy / Periodontal Diseases - etiology / Periodontal Diseases - pathology / Periodontitis / Pneumonia / Psoriasis / RNA, Messenger - metabolism / Sacrum / Skin Ulcer - drug therapy / Skin Ulcer - etiology / Skin Ulcer - pathology / Teeth / Ulcers / Ustekinumab - adverse effects / Ustekinumab - therapeutic use / Young Adult

2017

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Interleukin-12 and Interleukin-23 Blockade in Leukocyte Adhesion Deficiency Type 1

2017

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2017

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Journal Article

Interleukin-12 and Interleukin-23 Blockade in Leukocyte Adhesion Deficiency Type 1

Zerbe, Christa S,

Hajishengallis, George,

Wild, Teresa,

Notarangelo, Lucia D,

Uzel, Gulbu,

Holland, Steven M,

Notarangelo, Luigi D,

Moutsopoulos, Niki M,

Lisco, Andrea,

Dutzan, Nicolas,

DiPasquale, Giovanni,

Axelrod, Karen C,

Brenchley, Laurie

2017

Overview

A 19-year-old man with leukocyte adhesion deficiency type 1 had severe periodontitis and a chronic open sacral wound that were corrected by periodic administration of ustekinumab, an antibody to interleukin-12 and interleukin-23 signaling. LAD1 is a primary immunodeficiency resulting from mutations in ITGB2, which encodes the common CD18 subunit of the β2 integrins; the β2 integrins are required for the adhesion of neutrophils to endothelium and the transmigration of neutrophils into tissues. Patients with LAD1 typically have recurrent skin infections that can be chronic and refractory. Recurrent oral ulcers cause severe pain and difficulty eating, and severe periodontitis leads to complete loss of adult teeth in almost all patients. 1 The mucocutaneous lesions in LAD1 were widely assumed to be due to infections that were a result of the relative tissue neutropenia caused by . . .

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Publisher

Massachusetts Medical Society

Subject

/ Biopsy

/ Disease

/ Gingiva - pathology