A case of Melkersson-Rosenthal syndrome with temporomandibular joint osteoarthritis: multidisciplinary treatment and autoimmune etiological hypothesis

Antong Wu; Ying Zhang; Wei Cao; Xinhong Wang; Zhiqiang Song; Richard T. Jaspers; Lu Chen; Janak L. Pathak; Qingbin Zhang

doi:10.1186/s12903-024-04723-7

A case of Melkersson-Rosenthal syndrome with temporomandibular joint osteoarthritis: multidisciplinary treatment and autoimmune etiological hypothesis

Antong Wu
, Ying Zhang
, Wei Cao
, Xinhong Wang
, Zhiqiang Song
, Richard T. Jaspers
, Lu Chen
, Janak L. Pathak^*
, Qingbin Zhang^*

^*Corresponding author for this work

Research output: Contribution to Journal › Article › Academic › peer-review

Abstract

Background: Melkersson-Rosenthal syndrome (MRS) is a rare neuro-mucocutaneous disorder characterized by recurrent edema, facial palsies, and nerve dysfunctions often associated with the plicata tongue. Although the etiology of MRS is not well understood, there is growing evidence suggesting an autoimmune involvement. Case presentation: This paper presents a case report of a 25-year-old male with MRS as the initial symptom, followed by temporomandibular joint osteoarthritis (TMJ-OA). A comprehensive diagnosis and multidisciplinary treatment approach including surgery, local injections, and oral medication were implemented, resulting in a favorable prognosis. Conclusions: These findings support the hypothesis that MRS is a systemic granulomatous disease caused by autoimmunity, which may also influence the occurrence and development of TMJ-OA through immune-related mechanisms. This study emphasizes the significance of systemic immune regulation in the treatment of patients with MRS and TMJ-OA comorbid conditions.

Original language	English
Article number	935
Pages (from-to)	1-8
Number of pages	8
Journal	BMC Oral Health
Volume	24
Early online date	13 Aug 2024
DOIs	https://doi.org/10.1186/s12903-024-04723-7
Publication status	Published - 2024

Bibliographical note

Publisher Copyright:
© The Author(s) 2024.

Funding

This research was funded by the Guangzhou Science and Technology Key R&D Program Fund (No. 202206010004 and 202201020203), the Guangzhou Elite Scholarship Council (No. JY202221), the Research Capability Enhancement Program of Guangzhou Medical University (Research Start-up Project: No. 2024SRP162), and the Medical Science and Technology Research Foundation of Guangdong Province: (No. A2022205; No. A2024532).

Funders	Funder number
Guangzhou Science and Technology Key R&D Program Fund	202201020203, 202206010004
Guangzhou Medical University	2024SRP162
Medical Science and Technology Foundation of Guangdong Province	A2024532, A2022205
Guangzhou Elite Scholarship Council	JY202221

Keywords

Melkersson-Rosenthal syndrome
Systemic immune regulation
Temporomandibular joint osteoarthritis

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10.1186/s12903-024-04723-7

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@article{804b772be5ca4600a66d35416e4b2651,

title = "A case of Melkersson-Rosenthal syndrome with temporomandibular joint osteoarthritis: multidisciplinary treatment and autoimmune etiological hypothesis",

abstract = "Background: Melkersson-Rosenthal syndrome (MRS) is a rare neuro-mucocutaneous disorder characterized by recurrent edema, facial palsies, and nerve dysfunctions often associated with the plicata tongue. Although the etiology of MRS is not well understood, there is growing evidence suggesting an autoimmune involvement. Case presentation: This paper presents a case report of a 25-year-old male with MRS as the initial symptom, followed by temporomandibular joint osteoarthritis (TMJ-OA). A comprehensive diagnosis and multidisciplinary treatment approach including surgery, local injections, and oral medication were implemented, resulting in a favorable prognosis. Conclusions: These findings support the hypothesis that MRS is a systemic granulomatous disease caused by autoimmunity, which may also influence the occurrence and development of TMJ-OA through immune-related mechanisms. This study emphasizes the significance of systemic immune regulation in the treatment of patients with MRS and TMJ-OA comorbid conditions.",

keywords = "Melkersson-Rosenthal syndrome, Systemic immune regulation, Temporomandibular joint osteoarthritis",

author = "Antong Wu and Ying Zhang and Wei Cao and Xinhong Wang and Zhiqiang Song and Jaspers, \{Richard T.\} and Lu Chen and Pathak, \{Janak L.\} and Qingbin Zhang",

note = "Publisher Copyright: {\textcopyright} The Author(s) 2024.",

year = "2024",

doi = "10.1186/s12903-024-04723-7",

language = "English",

volume = "24",

pages = "1--8",

journal = "BMC Oral Health",

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T1 - A case of Melkersson-Rosenthal syndrome with temporomandibular joint osteoarthritis

T2 - multidisciplinary treatment and autoimmune etiological hypothesis

AU - Wu, Antong

AU - Zhang, Ying

AU - Cao, Wei

AU - Wang, Xinhong

AU - Song, Zhiqiang

AU - Jaspers, Richard T.

AU - Chen, Lu

AU - Pathak, Janak L.

AU - Zhang, Qingbin

PY - 2024

Y1 - 2024

N2 - Background: Melkersson-Rosenthal syndrome (MRS) is a rare neuro-mucocutaneous disorder characterized by recurrent edema, facial palsies, and nerve dysfunctions often associated with the plicata tongue. Although the etiology of MRS is not well understood, there is growing evidence suggesting an autoimmune involvement. Case presentation: This paper presents a case report of a 25-year-old male with MRS as the initial symptom, followed by temporomandibular joint osteoarthritis (TMJ-OA). A comprehensive diagnosis and multidisciplinary treatment approach including surgery, local injections, and oral medication were implemented, resulting in a favorable prognosis. Conclusions: These findings support the hypothesis that MRS is a systemic granulomatous disease caused by autoimmunity, which may also influence the occurrence and development of TMJ-OA through immune-related mechanisms. This study emphasizes the significance of systemic immune regulation in the treatment of patients with MRS and TMJ-OA comorbid conditions.

AB - Background: Melkersson-Rosenthal syndrome (MRS) is a rare neuro-mucocutaneous disorder characterized by recurrent edema, facial palsies, and nerve dysfunctions often associated with the plicata tongue. Although the etiology of MRS is not well understood, there is growing evidence suggesting an autoimmune involvement. Case presentation: This paper presents a case report of a 25-year-old male with MRS as the initial symptom, followed by temporomandibular joint osteoarthritis (TMJ-OA). A comprehensive diagnosis and multidisciplinary treatment approach including surgery, local injections, and oral medication were implemented, resulting in a favorable prognosis. Conclusions: These findings support the hypothesis that MRS is a systemic granulomatous disease caused by autoimmunity, which may also influence the occurrence and development of TMJ-OA through immune-related mechanisms. This study emphasizes the significance of systemic immune regulation in the treatment of patients with MRS and TMJ-OA comorbid conditions.

KW - Melkersson-Rosenthal syndrome

KW - Systemic immune regulation

KW - Temporomandibular joint osteoarthritis

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SN - 1472-6831

VL - 24

SP - 1

EP - 8

JO - BMC Oral Health

JF - BMC Oral Health

M1 - 935

ER -

A case of Melkersson-Rosenthal syndrome with temporomandibular joint osteoarthritis: multidisciplinary treatment and autoimmune etiological hypothesis

Abstract

Bibliographical note

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Keywords

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