Intraoral and maxillofacial abnormalities in patients with autosomal dominant hyper-IgE syndrome

Ildikó Tar; Márta Szegedi; Ewa Krasuska-Sławińska; Edyta Heropolitańska-Pliszka; Ewa A. Bernatowska; Elif Öncü; Sevgi Keles; Sukru N. Guner; Ismail Reisli; Nevena Gesheva; Elissaveta Naumova; Lydie Izakovicova-Holla; Jiri Litzman; Igor Savchak; Larysa Kostyuchenko; Melinda Erdős

doi:10.5114/ceji.2023.130874

eISSN: 1644-4124
ISSN: 1426-3912

Central European Journal of Immunology

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3/2023
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abstract:

Clinical immunology

Intraoral and maxillofacial abnormalities in patients with autosomal dominant hyper-IgE syndrome

Ildikó Tar

¹

,

Márta Szegedi

¹

,

Ewa Krasuska-Sławińska

²

,

Edyta Heropolitańska-Pliszka

³

,

Ewa A. Bernatowska

³

,

Elif Öncü

⁴

,

Sevgi Keles

⁵

,

Sukru N. Guner

⁵

,

Ismail Reisli

⁵

,

Nevena Gesheva

⁶

,

Elissaveta Naumova

⁶

,

Lydie Izakovicova-Holla

⁷

,

Jiri Litzman

⁸

,

Igor Savchak

⁹

,

Larysa Kostyuchenko

⁹

,

Melinda Erdős

¹⁰

Faculty of Dentistry, University of Debrecen, Debrecen, Hungary

Dental Surgical Clinic for Children, Children’s Memorial Health Institute, Warsaw, Poland

Department of Immunology, Children’s Memorial Health Institute, Warsaw, Poland

Department of Periodontology, Lokman Hekim University, Ankara, Turkey

Division of Pediatric Allergy and Immunology, Necmettin Erbakan University, Konya, Turkey

Department of Clinical Immunology and Stem Cell Bank, University Hospital “Aleksandrovska”, Sofia, Bulgaria

Department of Stomatology, St Anne’s University Hospital, Faculty of Medicine, Masaryk University, Brno, Czech Republic

Department of Clinical Immunology and Allergology, St Anne’s University Hospital, Faculty of Medicine, Masaryk University, Brno, Czech Republic

Department of Pediatric Immunology and Rheumatology, Western Ukrainian Specialized Children’s Medical Center, Lviv, Ukraine

10.

J Project Education and Research Network, Debrecen, Hungary

Cent Eur J Immunol 2023; 48 (3): 228-236

DOI: https://doi.org/10.5114/ceji.2023.130874

Online publish date: 2023/09/05

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AMA

Tar I, Szegedi M, Krasuska-Sławińska E, et al. Intraoral and maxillofacial abnormalities in patients with autosomal dominant hyper-IgE syndrome. Central European Journal of Immunology. 2023;48(3):228-236. doi:10.5114/ceji.2023.130874.

APA

Tar, I., Szegedi, M., Krasuska-Sławińska, E., Heropolitańska-Pliszka, E., Bernatowska, E.,  & Öncü, E. et al. (2023). Intraoral and maxillofacial abnormalities in patients with autosomal dominant hyper-IgE syndrome. Central European Journal of Immunology, 48(3), 228-236. https://doi.org/10.5114/ceji.2023.130874

Chicago

Tar, Ildikó, Márta Szegedi, Ewa Krasuska-Sławińska, Edyta Heropolitańska-Pliszka, Ewa A. Bernatowska, Elif Öncü,  and Sevgi Keles et al. 2023. "Intraoral and maxillofacial abnormalities in patients with autosomal dominant hyper-IgE syndrome". Central European Journal of Immunology 48 (3): 228-236. doi:10.5114/ceji.2023.130874.

Harvard

Tar, I., Szegedi, M., Krasuska-Sławińska, E., Heropolitańska-Pliszka, E., Bernatowska, E., Öncü, E., Keles, S., Guner, S., Reisli, I., Gesheva, N., Naumova, E., Izakovicova-Holla, L., Litzman, J., Savchak, I., Kostyuchenko, L.,  and Erdős, M. (2023). Intraoral and maxillofacial abnormalities in patients with autosomal dominant hyper-IgE syndrome. Central European Journal of Immunology, 48(3), pp.228-236. https://doi.org/10.5114/ceji.2023.130874

MLA

Tar, Ildikó et al. "Intraoral and maxillofacial abnormalities in patients with autosomal dominant hyper-IgE syndrome." Central European Journal of Immunology, vol. 48, no. 3, 2023, pp. 228-236. doi:10.5114/ceji.2023.130874.

Vancouver

Tar I, Szegedi M, Krasuska-Sławińska E, Heropolitańska-Pliszka E, Bernatowska E, Öncü E et al. Intraoral and maxillofacial abnormalities in patients with autosomal dominant hyper-IgE syndrome. Central European Journal of Immunology. 2023;48(3):228-236. doi:10.5114/ceji.2023.130874.

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Autosomal dominant hyper-IgE syndrome (AD-HIES) is an inborn error of immunity (IEI) caused by a dominant-negative mutation in the signal transducer and activator of transcription 3 (STAT 3). This disease is characterized by chronic eczematoid dermatitis, recurrent staphylococcal skin abscesses, pneumonia, pneumatoceles, and extremely high serum IgE levels. Loss-of-function STAT3 mutations may also result in distinct non-immunologic features such as dental, facial, skeletal, and vascular abnormalities, central nervous system malformations and an increased risk for bone fractures. Prophylactic treatment of Candida infections and prophylactic antimicrobial therapy for staphylococcal skin infections and sinopulmonary infections are essential. An awareness of the oral and maxillofacial features of HIES may facilitate early diagnosis with genetic counselling and may improve future patient care. This study describes oral, dental, and maxillofacial manifestations in 14 patients with genetically defined AD-HIES. We also review the literature and propose recommendations for the complex care of patients with this rare primary immunodeficiency.

keywords:

Intraoral and maxillofacial abnormalities in patients with autosomal dominant hyper-IgE syndrome

dentist, hyper-IgE syndrome, maxillofacial, intraoral