Recurrent Facial Palsy with Orofacial Swelling in a Young Female: A Case of Melkersson–Rosenthal Syndrome

Authors

  • Zeeshan Khan Saidu Group of Teaching Hospitals, Swat, Pakistan
  • Amjad Iqbal Saidu Group of Teaching Hospital, Swat, Pakistan
  • Sabir Rehman Saidu Medical College/Saidu Group of Teaching Hospitals, Swat, Pakistan
  • Wasim Ajaz Saidu Group of Teaching Hospital, Swat, Pakistan
  • Salman Khan Saidu Group of Teaching Hospital, Swat, Pakistan
  • Fazal Wahab DHQ Daggar, Buner, Pakistan
  • Zai Ullah Shifa Hospital, Swat, Pakistan

DOI:

https://doi.org/10.52206/jsmc.2026.16.1.1235

Abstract

Background: Melkersson–Rosenthal syndrome (MRS) is a rare neuro-mucocutaneous disorder characterized by a classical triad of recurrent facial nerve palsy, orofacial edema, and fissured tongue. Owing to its variable and often incomplete clinical presentation, the condition poses a diagnostic challenge and is frequently underrecognized. Diagnosis is primarily clinical and relies on exclusion of other neurological, infectious, and granulomatous disorders.

Case Presentation: We report the case of a 23-year-old female who presented with a three-year history of recurrent left-sided lower motor neuron facial palsy associated with intermittent, painful swelling of the upper and lower lips. The episodes occurred every few months and lasted approximately two to three weeks. Clinical examination revealed unilateral facial weakness, recurrent orofacial edema, and a fissured tongue. Extensive laboratory investigations, autoimmune screening, and magnetic resonance imaging of the brain were unremarkable, excluding alternative etiologies. Based on the characteristic clinical features and exclusion of other causes, a diagnosis of Melkersson–Rosenthal syndrome was established.

Results: The patient was initially managed with oral corticosteroids during acute episodes, resulting in significant symptomatic improvement. Due to the recurrent nature of the disease, long-term immunosuppressive therapy with azathioprine was initiated. At six-week follow-up, the patient demonstrated a marked reduction in the frequency and severity of facial palsy episodes, significant resolution of orofacial swelling, and improvement in tongue-related discomfort, leading to enhanced quality of life.

Conclusion: This case highlights the importance of clinical awareness and early recognition of Melkersson–Rosenthal syndrome in patients presenting with recurrent facial palsy and orofacial swelling. Prompt diagnosis and appropriate immunomodulatory therapy can effectively control symptoms, reduce recurrence, and improve long-term outcomes in affected individuals.

Keywords: Fissured Tongue, Immunosuppressive Therapy, Melkersson–Rosenthal Syndrome, Orofacial Edema, Recurrent Facial Palsy.

References

Pinna M, Orrù G, Denotti G, Murgia MS, Casu C. Melkersson–Rosenthal syndrome: A case report. Clinical Case Reports. 2024 Feb;12(2):e8075. https://doi.org/10.1002/ccr3.8075

Wenda N, Stuhrmann NC, Messerschmid A, Märker?Hermann E, Gosepath J. Melkersson?Rosenthal?syndrome?A Rare Case of Laryngeal Involvement. The Laryngoscope. 2022 Dec;132(12):2442-4 . Epub 2022 Jul 23. https://doi.org/10.1002/lary.30311

Yu S, Li Q. Orofacial edema and facial paralysis: Melkersson-Rosenthal syndrome. JCR: Journal of Clinical Rheumatology. 2021 Aug 1;27(5):e190-1. https://doi.org/10.1097/RHU.0000000000001339

Liu R, Yu S. Melkersson–Rosenthal syndrome: a review of seven patients. Journal of clinical neuroscience. 2013 Jul 1;20(7):993-5. https://doi.org/10.1016/j.jocn.2012.10.009

Ibrahim A, Ibrahim M, Al Adawi M, Oweis L, Bahou Y. Think Melkersson-Rosenthal Syndrome: A Fissured Tongue With Facial Paralysis. Cureus. 2020 Jul;12(7). https://doi.org/10.7759/cureus.9480

Torabi M, Afshar MK, Barati H. Melkersson-rosenthal syndrome: a case report of the classic triad. Journal of Dentistry. 2020 Dec;21(4):335. PMID: 33344685.

Savasta S, Rossi A, Foiadelli T, Licari A, Elena Perini AM, Farello G, et al. Melkersson–Rosenthal syndrome in childhood: report of three paediatric cases and a review of the literature. International Journal of Environmental Research and Public Health. 2019 Apr;16(7):1289. https://doi.org/10.3390/ijerph16071289

Gerressen M, Ghassemi A, Stockbrink G, Riediger D, Zadeh MD. Melkersson-Rosenthal syndrome: case report of a 30-year misdiagnosis. Journal of oral and maxillofacial surgery. 2005 Jul 1;63(7):1035-9. https://doi.org/10.1016/j.joms.2005.03.021

Cancian M, Giovannini S, Angelini A, Fedrigo M, Bendo R, Senter R, et al. Melkersson–Rosenthal syndrome: a case report of a rare disease with overlapping features. Allergy, Asthma & Clinical Immunology. 2019 Dec;15:1-5. https://doi.org/10.1186/s13223-018-0316-z

Kanerva M, Moilanen K, Virolainen S, Vaheri A, Pitkäranta A. Melkersson-rosenthal syndrome. Otolaryngology–Head and Neck Surgery. 2008 Feb;138(2):246-51. https://doi.org/10.1016/j.otohns.2007.11.015

Dhawan SR, Saini AG, Singhi PD. Management strategies of Melkersson-Rosenthal syndrome: a review. International Journal of General Medicine. 2020 Feb 26:61-5. https://doi.org/10.2147/IJGM.S186315

Critchlow WA, Chang D. Cheilitis granulomatosa: a review. Head and Neck Pathology. 2014 Jun;8:209-13. https://doi.org/10.1007/s12105-013-0488-2

Tonkovic?Capin V, Galbraith SS, Rogers III RS, Binion DG, Yancey KB. Cutaneous Crohn's disease mimicking Melkersson–Rosenthal syndrome: treatment with methotrexate. Journal of the European Academy of Dermatology and Venereology. 2006 Apr;20(4):449-52. https://doi.org/10.1111/j.1468-3083.2006.01458.x

Additional Files

Published

04-02-2026

How to Cite

1.
Khan Z, Iqbal A, Rehman S, Ajaz W, Khan S, Wahab F, Ullah Z. Recurrent Facial Palsy with Orofacial Swelling in a Young Female: A Case of Melkersson–Rosenthal Syndrome. J Saidu Med Coll [Internet]. 2026 Feb. 4 [cited 2026 Nov. 10];16(1):222–225. Available from: http://jsmc.pk/index.php/jsmc/article/view/1235

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