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

BMC Oral Health volume 24, Article number: 935 (2024) Cite this article

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.

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Background

Melkersson-Rosenthal syndrome (MRS) is a rare disorder of triad of facial palsy [1], lingua plicata (fissured tongue), and orofacial edema [2] affecting 0.08% of the world’s population [3]. Recurrent orofacial edema, recurrent peripheral facial palsy, and fissured tongue are typical clinical manifestations of MRS [4]. For the majority of MRS patients with single or few symptoms, the complete triad is present in only 8–25% of cases [5]. For the clinical diagnosis, the identification of non-caseating granuloma on mucocutaneous biopsies as histopathological evidence can lead to a diagnosis of monosymptomatic patients [6]. The hypothesis of an autoimmune involvement in the pathogenesis of this disease has been suggested by its association with other autoimmune diseases, such as autoimmune thyroiditis and multiple sclerosis [7, 8].

Temporomandibular joint osteoarthritis (TMJ-OA) is one of the common diseases in the oral and maxillofacial region [9]. It mainly affects young and middle-aged adults, with up to one-third of adults reporting one or more symptoms, which include jaw or neck pain, headache, and clicking or grating within the joint [10, 11]. Autoimmune diseases often affect the thyroid, blood vessels, kidneys, skin, joints, and other tissues and organs [12]. The etiology of TMJ-OA has not been fully elucidated, and immune factors are considered to be one of the most important causes. TMJ can be affected by autoimmune diseases. Common autoimmune diseases such as rheumatoid arthritis (RA), ankylosing spondylitis (AS), and systemic lupus erythematosus (SLE) affect TMJ [13,14,15,16]. TMJ joint ankylosis is also associated with the joint-related autoimmune disease RA [13].

To the best of our knowledge, the presented case is the first report of MRS with TMJ-OA. Based on the patient's medical history and examination, we made a comprehensive diagnosis and developed a multidisciplinary treatment strategy. Thereafter, we discussed the potential pathological basis of various complex manifestations of MRS and TMJ-OA.

Case description

Patient description

A 25-year-old male experienced recurrent facial swelling in the last 2 years. Pain in the right preauricular region and limitations in mouth opening were experienced with right facial paralysis in the last 6 months. The patient had no family history of similar disease and there was no history of hereditary disease, congenital disease, or other infectious diseases in the family. At the first outpatient visit, the patient's mental state was depressed.

Pre-treatment physical examination

Physical examination revealed facial asymmetry, buccal swelling, loss of mandibular contour, right facial paralysis, limitations in right eyebrow-raising, dry crusted lips, swollen, tough upper lip, swollen tongue, and poor mobility. The plae lingual margin, tooth marks, and the cracked back of the tongue were observed (Fig. 1A-B). TMJ clinical examination showed restricted mandibular movements accompanied by pain. A numeric pain rating scale (NRS) was used to quantify the pain [17]. Pain occurred during the opening and closing of the mouth, lateral movement, and protrusion movement with right TMJ NRS 4 and left TMJ NRS 3. Bilateral TMJ has compression pain with a right NRS 5 and left NRS 1. No sounds in joints were noted while opening and closing the mouth. Chewing muscles i.e., masseter muscle, temporal muscle, lateral and medial pterygoid muscles did not have pressing pain. Maximum mouth opening (MMO, 25 mm), jaw protrusion (4 mm), jaw left lateral movement (3 mm), and right lateral movement (3 mm) were noted. Helkimo clinical test dysfunction index (HCDI) was used for TMD symptom quantification [18]. Pre-treatment HCDI evaluation was stage III (11 points) (Table 1).

Fig. 1
figure 1

Pre-treatment and post-treatment photos of the patient's face. The patient's photos show triple symptoms (Recurrent orofacial edema, recurrent peripheral facial palsy, and fissured tongue) of MRS (A) (C) Overall photos of maxillofacial (B) (D) Partial photos of lips and tongue. The red horizontal line represents the height of the eyebrows

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Table 1 Evaluation of symptoms of patients with TMD according to Helkimo clinical test dysfunction index (HCDI)
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Pre-treatment imaging examination

Pre-treatment TMJ CBCT showed extensive bilateral TMJ condylar bone damage with higher damage in the right TMJ. Bilateral TMJ tubercle articular eminence bone destruction was also observed, and the damage was mainly on the posterior slope of the articular tubercle. (Fig. 2A). Bilateral TMJ articular disc perforation was possible, and the anterior and posterior bands of the articular disc were displaced forward and backward, respectively. Bilateral TMJ condyle and fossa bone resorption with synovitis. The soft tissue and muscle space around bilateral TMJ joints showed mild edema, indicating inflammatory changes. Bilateral TMJ movement was limited (Fig. 3A). Pre-treatment endoscopy (No. MZ73520036): Terminal ileum and colorectal mucosa showed no abnormality. B ultrasound (No. US2302180016, US230220013, and US002140) showed no abnormality in the kidneys, heart, bladder, liver, gallbladder, spleen, pancreas, and prostate. Head MRI and MRA (No. MR052744) indicated no abnormality. Chest X-ray (No. DX242384) also showed normal heart, lung, or mediastinum.

Fig. 2
figure 2

CBCT images (Transverse, coronal, and sagittal) of the patient's bilateral TMJ showed cortical irregularities in the mandibular condyle and temporal fossa. A Pre-treatment; (B) Posttreatment. The red arrow represents the bone defect

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Fig. 3
figure 3

MRI images of the patient's bilateral TMJ show compression deformation and position changes in the TMJ disc. A Pre-treatment; B Post-treatment. The red arrow and red irregular shape represent the TMJ disc position and shape

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Pre-treatment laboratory tests

Allergen detection tests for syphilis, hepatitis B and C, and human immunodeficiency virus were negative. The results of the investigations (blood routine examination, coagulation function, electrolytes, angiotensin-converting enzyme, lupus anticoagulant factor, rheumatism factor, antistreptolysin-O, C-reaction protein) were within the normal range. IgG level (24.6 g/L) was higher than normal (7.0 -16.0 g/L) [19].

Diagnosis and treatment plan

We excluded Crohn's disease (CD), sarcoidosis, SLE, RA, AS, and tumors. We conducted a comprehensive analysis of the patient's clinical, imaging, and laboratory examinations combined with the medical history that diagnosed MRS with TMJ-OA. The treatment plan was as follows: (1) Surgical treatment for TMJ-OA i.e., the adhesion tissue of the articular fossa was removed and pathological examination was conducted. Then the anterior articular disc attachment and external pterygoid muscle were released, and the remaining articular disc tissue was sutured (2) Symptomatic treatment for MRS i.e., local injection of triamcinolone and lidocaine hydrochloride combined with oral medication loratadine, multivitamin, and methylprednisolone.

Expected outcome of the treatment plan

TMJ Pain and mouth opening movements will be improved and edema of the face and lips will improve.

Actual outcome

Post-treatment pathological examination

First, we performed a histological examination In the tissue of the upper lip, the epithelial acanthosis of mucosal tissue showed high edema. Edema was shown in the lamina propria with chronic perineural inflammatory cell infiltration, and the local non-caseous granuloma was observed (Fig. 4A-B). Non-caseous granuloma is the gold standard for MRS, indicating that our diagnosis was consistent with the original diagnosis. The left TMJ fossa synovial tissue showed chronic inflammatory markers, focal synovial cell proliferation, increased interstitial capillaries, local edema or hyaline degeneration of interstitial fibrous connective tissue, and perivascular lymphocyte infiltration (Fig. 4C). The right TMJ fossa synovial tissue and fibrous connective tissue showed lymphocyte infiltration and most noteworthy, there focal non-caseous granuloma formation in the synovial interstitium and perivascular area (Fig. 4D).

Fig. 4
figure 4

Histopathological examination of Mucosal and TMJ synovial tissue (H&E staining). A Mucosal tissue of the upper lip; B The mucosal tissue of the upper lip showed pathological changes of non-caseous granuloma; C Left TMJ synovial tissue; D Right TMJ synovial tissue showed pathological changes of non-caseous granuloma. The red arrow and black circle represent the non-caseous granuloma area

Full size image

Post-treatment physical examination

After 2 months of treatment, TMJ clinical examination showed the MMO: 30 mm, jaw protrusion (6 mm), jaw left lateral movement (5 mm), and right lateral movement (6 mm). No pain occurred during the opening and closing of the mouth, lateral movement, or protrusion movement (NRS: 0). No pressure pain was observed in TMJ. Chewing muscles did not have pressing pain. The treatment effect was quantified according to the HDCI score. The pre-treatment evaluation was stage III (11 points) and the post-treatment evaluation was stage (1 point) (Table 1). Reduced facial swelling, tongue back swelling, texture, furrow, and lip swelling were observed but the right eyebrow lifting was still limited (Fig. 1C-D).

Post-treatment imaging examination

According to TMJ CBCT after treatment, bone destruction of the bilateral TMJ condylar was increased compared to that within the pre-treatment situation (Fig. 2B). According to TMJ MRI after treatment, the posterior disc band of the left TMJ was at the 2 o'clock direction of the condyle, and the anterior disc band was at the 1 o'clock direction. The shape of the right TMJ disc was more continuous than that in the pre-treatment. The posterior disc band was located at the 2 o'clock direction of the condyle, and the anterior disc band was located at the 1 o'clock direction (Fig. 3B).

Post-treatment patient’s mental condition

The patient's overall health improves, negative emotions are eliminated, and self-confidence is fully restored.

Discussion

MRS was first described and named after E. Melkersson and C. Rosenthal in 1928 and 1931 respectively. It is a neuro-mucocutaneous disorder that mostly occurs in young adults and has non-caseous granuloma as the main pathological manifestation [20]. Some studies have shown that the prevalence of MRS is close to or slightly higher in males than in females. However, there are also reports from different countries in the world that there are no racial or gender differences in the incidence of MRS [21,22,23]. There is a significant abnormal immune function, immune deregulation, and allergic tendencies in patients with MRS [24, 25]. Short courses of immunosuppressants are often used in the treatment of MRS [26]. Corticosteroids are currently the main treatment for MRS. Therapy with corticosteroids leads to improvement in 50–80% of patients and has been shown to reduce relapse frequency by 60–75% [27]. Typically, oral corticosteroids are used for 1 week and tapered over 2 weeks. High-dose pulse methylprednisolone has been used in severe cases [26]. Local injections of lidocaine and triamcinolone acetonide are effective ways to treat facial swelling [28]. In the presented case, the patient had a long-term history of urticaria before visiting our department and had been taking anti-allergic drugs (loratadine, cetirizine hydrochloride, ebastine) for a long time, but did not follow the treatment principles of peripheral facial paralysis, that is, use adrenocortical hormones, supplement B vitamins, antiviral drugs, etc., thus missing the opportunity to treat peripheral facial paralysis. Therefore, adrenocortical hormonal therapy administered according to our second-stage treatment protocol was only successful in improving the patient's facial swelling but was not effective in lingual grooves and facial paralysis. In the future, we can consider surgical treatment of grooved tongue and peripheral facial paralysis according to the patient's wishes.

Some TMJ diseases are associated with autoimmune diseases and classical TMJ diseases are often pathologically indistinguishable [15]. Synovitis is the most important pathological change of TMJ damage caused by autoimmune diseases, and synovitis caused by different autoimmune diseases such as RA, SLA, and AS are similar [29]. However, in this report, we discovered a phenotype different from synovitis caused by other immune diseases after histopathological analysis of the synovial tissue of this patient's TMJ-OA. We found non-caseous granuloma lesions in the joint tissue of patients with TMJ-OA. This finding helps us further understand the etiological and pathological mechanisms of TMJ-OA. In general, the higher concentration of IgG complexes (e.g. IgG RF) in the synovium of patients with autoimmune diseases makes TMJ cartilage or bone tissue more susceptible to involvement by formed synovium granulation tissue [29]. In this case, the patient's serum showed higher IgG expression (24.6 g/L) levels than the typical values of serum IgG reported in adults (i.e. 7.0–16.0 g/L) [19]. To better clarify the condition and develop corresponding treatment plans, scholars have conducted a series of studies on articular disc displacement and the staging of TMJ-OA. In 1989, WILKES classified articular disc displacement into early stage (stage I), early to middle stage (stage II), middle stage (stage III), and middle to late stage (stage IV) and late stage (stage V) based on clinical manifestations, imaging manifestations, and intraoperative findings [30]. Non-surgical and surgical treatments are the main ways to treat TMJ-OA. At present, non-surgical treatment methods include health education, psychological counseling, drug treatment, occlusal treatment, physical therapy, local injection treatment, etc. [31, 32]. Among drug treatments commonly used are antirheumatic drugs, non-steroidal anti-inflammatory drugs, or immunosuppressive drugs. Advanced TMJ disorders, such as serious OA, disc perforation, or rupture are commonly used in TMJ replacement, disc extraction, and other operations [33,34,35,36]. All the above different treatments can improve clinical symptoms and achieve good therapeutic effects. Early use of anti-rheumatic immune drugs can delay the progression of the disease and reduce or prevent joint damage. The most commonly used drugs are non-steroidal anti-inflammatory drugs and anti-rheumatic drugs (such as methotrexate), and steroidal anti-inflammatory drugs are still necessary to reduce symptoms [37].

In this case, the treatment successfully improved the patient's mouth opening and relieved pain, but the CBCT after treatment showed significant condylar bone resorption. Considering MRS in this patient, it is particularly important to regulate the systemic immune system before surgery. When facing patients with TMJ-OA accompanied by autoimmune diseases such as RA, SLE, and AS (including MRS in this case), clinicians can consider using systemic immunomodulatory drugs (such as glucocorticoids) in advance or at the same time, which may obtain a good prognosis. Glucocorticoids have good analgesic and anti-inflammatory effects, which can have a good effect on immune diseases and TMJ-OA. Surgical trauma could be the main cause of postoperative bone destruction. The secondary causes of postoperative condylar bone destruction were disc rupture and the defect caused by non-caseous changes of the disc and synovium, and it was difficult to repair and restore the disc during the operation. Joint replacement is the only option when the damage progresses to severe condylar breakdown or rigidity. Autoimmune diseases are contraindicated for autogenous bone replacement and may still damage regenerated tissue unless inflammation is controlled. Therefore, TMJ replacement with autoimmune diseases should use artificial joints as much as possible to reduce the possibility of recurrence and stabilize the long-term curative effect. According to this case study, we believe that the TMJ lesions related to immune diseases should be identified as early as possible through clinical and imaging findings, and the selection of follow-up treatment should be guided. Timely diagnosis can inhibit the further development of the disease, prevent more serious structural and functional loss, and improve the prognosis of patients. Once it reaches the advanced stage, the effect of artificial joint replacement is more effective and stable. It is worth noting that TMD surgical treatment is considered the last option, regardless of the underlying cause of the disease. Except for rare conditions such as neoplasms and TMJ ankylosis, the potential damage often outweighs the benefits of surgical intervention.

Conclusion

This case report highlights the multidisciplinary treatment and autoimmune etiological hypothesis in a patient with MRS and TMJ-OA. The findings suggest that MRS is a systemic granulomatous disease with an autoimmune basis which may impact the occurrence and development of TMJ-OA through immune-related mechanisms. Clinicians should prioritize the regulation of systemic immune regulation in the treatment of patients with TMJ-OA and concomitant conditions. Further research is needed to explore the complex pathogenesis of these disorders and develop optimal treatment strategies.

Availability of data and materials

The data that support the findings of this study are available from the Affiliated Stomatology Hospital of Guangzhou Medical University, but restrictions apply to the availability of these data, which were used under license for the current study, and so are not publicly available. Data without patients' names and initials however are available from the corresponding author upon reasonable request and with permission of the Affiliated Stomatology Hospital of Guangzhou Medical University.

Abbreviations

MRS:

Melkersson-rosenthal syndrome

TMJ-OA:

Temporomandibular joint osteoarthritis

RA:

Rheumatoid arthritis

AS:

Ankylosing spondylitis

SLE:

Systemic lupus erythematosus

MMO:

Maximum mouth opening

CD:

Crohn's disease

NRS:

Numeric pain rating scale

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Acknowledgements

We would like to acknowledge Prof. dr. Astrid D. Bakker from the Department of Oral Cell Biology, Academic Centre for Dentistry Amsterdam (ACTA), University of Amsterdam and Vrije Universiteit Amsterdam, The Netherlands for her guidance in this research.

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).

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Author notes

  1. Antong Wu, Ying Zhang and Wei Cao contributed equally to this work.

Authors and Affiliations

  1. Department of Temporomandibular Joint, School and Hospital of Stomatology, Guangdong Engineering Research Center of Oral Restoration and Reconstruction, Guangzhou Key Laboratory of Basic and Applied Research of Oral Regenerative Medicine, Guangzhou Medical University, Guangzhou, China

    Antong Wu, Ying Zhang, Wei Cao, Zhiqiang Song, Richard T. Jaspers, Janak L. Pathak & Qingbin Zhang

  2. Department of Pathology, School and Hospital of Stomatology, Guangdong Engineering Research Center of Oral Restoration and Reconstruction, Guangzhou Key Laboratory of Basic and Applied Research of Oral Regenerative Medicine, Guangzhou Medical University, Guangzhou, China

    Xinhong Wang & Lu Chen

  3. Department of Oral Mucosa, School and Hospital of Stomatology, Guangdong Engineering Research Center of Oral Restoration and Reconstruction, Guangzhou Key Laboratory of Basic and Applied Research of Oral Regenerative Medicine, Guangzhou Medical University, Guangzhou, China

    Xinhong Wang

  4. Laboratory for Myology, Department of Human Movement Sciences, Faculty of Behavioural and Movement Sciences, Vrije Universiteit Amsterdam, Amsterdam Movement Science, Amsterdam, The Netherlands

    Antong Wu & Richard T. Jaspers

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Contributions

A.W. and J.L.P. wrote the original manuscript; J.L.P, W.C., X.W., R.T.J. and Q.Z. reviewed the manuscript; A.W., Y.Z., W.C., Z.S., X.W. and Q.Z. were involved in the patient’s treatment and care; A.W., Y.Z., W.C., Z.S. and L.C. recorded the patient’s medical history and examination information; L.C. and X.W. performed histological examination. All authors reviewed the manuscript.

Corresponding authors

Correspondence to Janak L. Pathak or Qingbin Zhang.

Ethics declarations

Ethics approval and consent to participate

This study was conducted in accordance with the Declaration of Helsinki, and the protocol was approved by the Medical Research Ethics Committee of the Affiliated Stomatology Hospital of Guangzhou Medical University (LCYJ2023027). The patient signed a written consent stating their approval for participation in this report.

Consent for publication

Written informed consent for publication of their clinical details and/or clinical images was obtained from the patient. A copy of the consent form is available for review by the Editor of this journal.

Competing interests

The authors declare no competing interests.

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Wu, A., Zhang, Y., Cao, W. et al. A case of Melkersson-Rosenthal syndrome with temporomandibular joint osteoarthritis: multidisciplinary treatment and autoimmune etiological hypothesis. BMC Oral Health 24, 935 (2024). https://doi.org/10.1186/s12903-024-04723-7

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BMC Oral Health

ISSN: 1472-6831

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