Experience in the treatment of orofacial granulomatosis in the form of Misher’s macrocheilitis with cyclosporine A

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Abstract

Background. Orofacial granulomatosis (OFG) is a chronic inflammatory disease that currently includes two syndromes: Melkersson-Rosenthal syndrome and Miescher’s granulomatous cheilitis. The etiopathogenesis of the disease has not been studied enough, the metals used in the manufacture of dental implants, crowns and fillings are considered to be possible factors leading to its development.

Description of the clinical case. The article describes a case of OFG in the form of Misher’s macrocheilitis in a young female patient, which caused difficulties in diagnosis and treatment. The diagnosis was established 2.5 years after the onset of the disease; hydroxychloroquine therapy was ineffective. A good clinical effect was obtained during therapy with cyclosporine A at an initial dose of 350 mg/day (3.8 mg per kg). The patient continues the recommended treatment is under the supervision of a dermatologist of the Outpatient Department of the Clinic of Dermatovenereology of the Pavlov University.

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About the authors

Tatiana V. Melnikova

Pavlov University

Author for correspondence.
Email: tatmel2007@yandex.ru
ORCID iD: 0000-0001-9983-0327

Head of the Outpatient Department of the Clinic of Dermatovenereology

Russian Federation, St. Petersburg

N. Yu. Burtseva

Pavlov University

Email: tatmel2007@yandex.ru
ORCID iD: 0000-0003-1800-1610
Russian Federation, St. Petersburg

M. A. Milyutina

Pavlov University

Email: tatmel2007@yandex.ru
ORCID iD: 0009-0001-8687-765X
Russian Federation, St. Petersburg

K. N. Monakhov

Pavlov University

Email: tatmel2007@yandex.ru
ORCID iD: 0000-0002-8211-1665
Russian Federation, St. Petersburg

E. V. Sokolovsky

Pavlov University

Email: tatmel2007@yandex.ru
ORCID iD: 0000-0001-7610-6061
SPIN-code: 6807-7137
Russian Federation, St. Petersburg

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Supplementary files

Supplementary Files
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1. JATS XML
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2. Fig.1. Appearance of patient G. with FG upon admission (source T.V. Melnikova et al., 06/20/2022)

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3. Fig.2. Dynamics of the clinical picture of FG during treatment (source T.V. Melnikova et al., 07/18/2022)

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4. Fig.3. Dynamics of the clinical picture of FG during treatment (source T.V. Melnikova et al., 10.10.2022)

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5. Fig.4. Dynamics of the clinical picture of FG during treatment (source T.V. Melnikova et al., 10/26/2022)

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6. Fig.5. Dynamics of the clinical picture of FG during treatment (source T.V. Melnikova et al., 12/13/2022)

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7. Fig.6. Dynamics of the clinical picture of FG during treatment (source T.V. Melnikova et al., 04/24/2023)

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8. Fig.7. Hyperkeratosis, interpapillary and uniform acanthosis, intercellular edema in the epidermis, focal lymphocytic-histiocytic infiltrates in the upper part of the dermis, individual granulomas of histiocytes, epithelial cells, lymphocytes

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