Presentation of bilateral facial paralysis in Melkersson–Rosenthal syndrome
Introduction. Melkersson–Rosenthal syndrome (MRS) is a neuromucocutaneous disorder characterized by the following classic symptom triad: peripheral facial paralysis, orofacial edema, and scrotal or fissured tongue. It is rare, and since most of the patients are oligo- or monosymptomatic, it makes it...
- Autores:
-
Gaitan Quintero, Gustavo
Camargo, Loida
López Velásquez, Norman Darío
González, Miguel
- Tipo de recurso:
- Article of journal
- Fecha de publicación:
- 2021
- Institución:
- Corporación Universidad de la Costa
- Repositorio:
- REDICUC - Repositorio CUC
- Idioma:
- spa
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- oai:repositorio.cuc.edu.co:11323/8899
- Acceso en línea:
- https://hdl.handle.net/11323/8899
https://doi.org/10.1155/2021/6646115
https://repositorio.cuc.edu.co/
- Palabra clave:
- Facial paralysis
Melkersson–Rosenthal
Syndrome
- Rights
- openAccess
- License
- CC0 1.0 Universal
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Presentation of bilateral facial paralysis in Melkersson–Rosenthal syndrome |
| title |
Presentation of bilateral facial paralysis in Melkersson–Rosenthal syndrome |
| spellingShingle |
Presentation of bilateral facial paralysis in Melkersson–Rosenthal syndrome Facial paralysis Melkersson–Rosenthal Syndrome |
| title_short |
Presentation of bilateral facial paralysis in Melkersson–Rosenthal syndrome |
| title_full |
Presentation of bilateral facial paralysis in Melkersson–Rosenthal syndrome |
| title_fullStr |
Presentation of bilateral facial paralysis in Melkersson–Rosenthal syndrome |
| title_full_unstemmed |
Presentation of bilateral facial paralysis in Melkersson–Rosenthal syndrome |
| title_sort |
Presentation of bilateral facial paralysis in Melkersson–Rosenthal syndrome |
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Gaitan Quintero, Gustavo Camargo, Loida López Velásquez, Norman Darío González, Miguel |
| dc.contributor.author.spa.fl_str_mv |
Gaitan Quintero, Gustavo Camargo, Loida López Velásquez, Norman Darío González, Miguel |
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Facial paralysis Melkersson–Rosenthal Syndrome |
| topic |
Facial paralysis Melkersson–Rosenthal Syndrome |
| description |
Introduction. Melkersson–Rosenthal syndrome (MRS) is a neuromucocutaneous disorder characterized by the following classic symptom triad: peripheral facial paralysis, orofacial edema, and scrotal or fissured tongue. It is rare, and since most of the patients are oligo- or monosymptomatic, it makes it difficult to diagnose. Clinical Case. We present a 26-year-old male patient with a history of sickle cell trait, untreated snoring, and left peripheral facial paralysis when he was 11 years old. +is was an overall 20- day clinical profile that started with left peripheral facial paralysis, which was accompanied by moderate-intensity occipital pulsatile headaches. Additionally, the patient experienced paresthesias in the tongue and feelings of labial edema. After one week, he manifested peripheral facial paralysis on the right side. Physical examination revealed bilateral peripheral facial paralysis, mild labial edema, and a scrotal or fissured tongue. +e patient received corticosteroids, which resulted in improvement of the edema and facial paralysis. Discussion. MRS is a rare disorder that predominantly affects women, typically starting in their 20s or 30s. +e etiology is unknown. However, a multifactorial origin that involves environmental factors and a genetic predisposition has been proposed, which causes a dysfunction of the local immune system and autonomic nervous system (ANS) and an appearance of granulomatous inflammation in the lips and tongue. Facial paralysis usually appears later on; however, it can occur from its clinical debut. +ere are no curative treatments. +erapy is focused on modulating the patient’s immune response, and relapses are frequent. |
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2021 |
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[1] A. Basman, M. Gumusok, S. Degerli, M. Kaya, and M. Toraman Alkurt, “Melkersson-rosenthal syndrome: a case report,” Journal of Istanbul University Faculty of Dentistry, vol. 51, no. 1, pp. 42–45, 2017. [2] M. Cancian, S. Giovannini, A. Angelini et al., “Melkersson–Rosenthal syndrome: a case report of a rare disease with overlapping features,” Allergy, Asthma & Clinical Immunology, vol. 15, no. 1, p. 1, 2019. [3] S. Feng, J. Yin, J. Li, Z. Song, and G. Zhao, “MelkerssonRosenthal syndrome: a retrospective study of 44 patients,” Acta Oto-Laryngologica, vol. 134, no. 9, pp. 977–981, 2014. [4] W. A. Critchlow and D. Chang, “Cheilitis granulomatosa: a review,” Head and Neck Pathology, vol. 8, no. 2, pp. 209–213, 2014. [5] S. R. Dhawan, A. G. Saini, and P. D. Singhi, “Management strategies of Melkersson-rosenthal syndrome,” A Review, vol. 13, p. 61, 2020. [6] S. Aydın, S. Ozt¨urk, A. Faras¸o ¨ glu, T. Çakıl, and S. J. S. Çoruk, ˘ “Melkersson–Rosenthal syndrome,” A Case Report, vol. 29, no. 2, 2018. [7] O. B. Ozgursoy, S. Karatayli Ozgursoy, O. Tulunay, O. Kemal, A. Akyol, and G. Dursun, “Melkersson-Rosenthal syndrome revisited as a misdiagnosed disease,” American Journal of Otolaryngology, vol. 30, no. 1, pp. 33–37, 2009. [8] E. Brozek-Ma˛dry, W. Jaremek-Ochniak, M. Straburzy ˙ nski, ´ and A. Krzeski, “Lower lip Oedema-Melkersson-Rosenthal Syndrome or Cheilitis Granulomatosa,” New American Journal of Otolaryngology Research, vol. 1, pp. 1-2, 2019. [9] X. Xu, L. Guan, Y. Lv et al., “Exome sequencing identifies FATP1 mutation in Melkersson-Rosenthal syndrome,” vol. 31, no. 5, Article ID e230, 2017. [10] Y. Pei, G. M. Beaman, D. Mansfield, J. Clayton-Smith, M. Stewart, and W. G. Newman, “Clinical and genetic heterogeneity in Melkersson-rosenthal syndrome,” European Journal of Medical Genetics, vol. 62, no. 6, Article ID 103536, 2019. [11] S. Bohra, P. B. Kariya, S. D. Bargale, and S. Kiran, “Clinicopathological significance of Melkersson-Rosenthal syndrome,” BMJ Case Reports, vol. 2015, 2015. [12] J.-J. Tang, X. Shen, J.-J. Xiao, and X.-P. Wang, “Retrospective analysis of 69 patients with Melkersson-Rosenthal syndrome in mainland China,” vol. 9, no. 2, pp. 3901–3908, 2016. [13] R. A. Gaudin, N. Jowett, C. A. Banks, C. J. Knox, and T. A. Hadlock, “Bilateral facial paralysis: a 13-year experience,” Plastic and Reconstructive Surgery, vol. 138, no. 4, pp. 879–887, 2016. [14] W. Zeng, S. Geng, X. Niu, and J. Yuan, “Complete Melkersson–Rosenthal syndrome with multiple cranial nerve palsies,” vol. 35, no. 3, pp. 272–274, 2010. [15] M. U. Aluclu, U. Keklikci, A. Guzel, K. Unlu, and M. Tatli, “Melkersson-Rosenthal syndrome with partial oculomotor nerve palsy,” Annals of Saudi Medicine, vol. 28, no. 2, pp. 135–137, 2008. [16] R. Liu and S. Yu, “Melkersson-Rosenthal syndrome: a review of seven patients,” Journal of Clinical Neuroscience, vol. 20, no. 7, pp. 993–995, 2013. [17] Z. Chu, Y. Liu, H. Zhang, W. Zeng, and S. Geng, “MelkerssonRosenthal syndrome with genitalia involved in a 12-year-old boy,” Annals of Dermatology, vol. 28, no. 2, pp. 232–236, 2016. [18] T. Ogawa, M. N. Sotto, and M. P. Hoang, “Granulomatous dermatitis and others,” in Hospital-Based Dermatopathology: An Illustrated Diagnostic Guide, M. P. Hoang and M. A. Selim, Eds., pp. 137–198, Springer International Publishing, Cham, Switzerland, 2020. [19] G. Shalom, L. Bodner, and S. Halevy, “Melkersson-Rosenthal Syndrome: the possible role of comorbidities in the etiopathogenesis. Giornale italiano di dermatologia e venereologia: organo ufficiale, Societa italiana di dermatologia e sifilografia,” vol. 154, no. 3, pp. 347–351, 2019. [20] F. Kayhan, F. ˙Ilik, and A. Kayhan, “Obsessive–compulsive disorder concurrent with Melkersson-Rosenthal Syndrome: a case report,” General Hospital Psychiatry, vol. 37, no. 5, p. 497, 2015. [21] M. K. Elias, F. J. Mateen, and C. R. Weiler, “+e Melkersson–Rosenthal syndrome: a retrospective study of biopsied cases,” Journal of Neurology, vol. 260, no. 1, pp. 138–143, 2013. [22] A. G. Saini, N. Sankhyan, H. Padmanabh, A. Das, and P. Singhi, “Recurrent facial palsy and electrophysiological findings in oligosymptomatic Melkersson rosenthal syndrome,” 2e Indian Journal of Pediatrics, vol. 83, no. 10, pp. 1188–1190, 2016. [23] L. Oudrhiri, S. Chiheb, F. Marnissi, S. Zamiati, and H. Benchikhi, “Successful treatment of Miescherʼs cheilitis in Melkersson-Rosenthal syndrome with betamethasone injections and doxycycline,” vol. 13, 2012. [24] C. Bacci and M. L. Valente, “Successful treatment of cheilitis granulomatosa with intralesional injection of triamcinolone,” vol. 3, no. 24, pp. 363-364, 2010. [25] I. Espinoza, J. Navarrete, J. Benedetto, A. Borzutzky, P. Roessler, and A. J. A. Ortega-Pinto, “Orofacial granulomatosis and diet therapy: a review of the literature and two clinical cases,” vol. 93, no. 1, pp. 80–85, 2018. [26] S. Raˇskovic, J. Bolpa ´ ci ˇ c, A. Peri ´ c-Popadi ´ c, ´ Z. Jovici ˇ c, ´R. Miˇskovic, and M. Bogi ´ c, “Clinical characteristics and ´treatment of Melkersson-Rosenthal syndrome: overview of six patients,” vol. 68, no. 11-12, pp. 401–404, 2015. [27] J. Wang, P. Li, X. Jin, Y. Xu, and X. J. Zhang, “Outcomes of recurrent facial palsy in Melkersson Rosenthal syndrome,” Annals of Otology, Rhinology & Laryngology, vol. 124, no. 3, pp. 232–234, 2015. [28] Z. Tan, Y. Zhang, W. Chen, W. Gong, J. Zhao, and X. Xu, “Recurrent facial palsy in Melkersson Rosenthal syndrome: total facial nerve decompression is effective to prevent further recurrence,” American Journal of Otolaryngology, vol. 36, no. 3, pp. 334–337, 2015. |
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Gaitan Quintero, GustavoCamargo, LoidaLópez Velásquez, Norman DaríoGonzález, Miguel2021-11-23T19:19:39Z2021-11-23T19:19:39Z20212090-66682090-6676https://hdl.handle.net/11323/8899https://doi.org/10.1155/2021/6646115Corporación Universidad de la CostaREDICUC - Repositorio CUChttps://repositorio.cuc.edu.co/Introduction. Melkersson–Rosenthal syndrome (MRS) is a neuromucocutaneous disorder characterized by the following classic symptom triad: peripheral facial paralysis, orofacial edema, and scrotal or fissured tongue. It is rare, and since most of the patients are oligo- or monosymptomatic, it makes it difficult to diagnose. Clinical Case. We present a 26-year-old male patient with a history of sickle cell trait, untreated snoring, and left peripheral facial paralysis when he was 11 years old. +is was an overall 20- day clinical profile that started with left peripheral facial paralysis, which was accompanied by moderate-intensity occipital pulsatile headaches. Additionally, the patient experienced paresthesias in the tongue and feelings of labial edema. After one week, he manifested peripheral facial paralysis on the right side. Physical examination revealed bilateral peripheral facial paralysis, mild labial edema, and a scrotal or fissured tongue. +e patient received corticosteroids, which resulted in improvement of the edema and facial paralysis. Discussion. MRS is a rare disorder that predominantly affects women, typically starting in their 20s or 30s. +e etiology is unknown. However, a multifactorial origin that involves environmental factors and a genetic predisposition has been proposed, which causes a dysfunction of the local immune system and autonomic nervous system (ANS) and an appearance of granulomatous inflammation in the lips and tongue. Facial paralysis usually appears later on; however, it can occur from its clinical debut. +ere are no curative treatments. +erapy is focused on modulating the patient’s immune response, and relapses are frequent.Gaitan Quintero, Gustavo-will be generated-orcid-0000-0003-1785-5585-600Camargo, Loida-will be generated-orcid-0000-0003-0056-6832-600López Velásquez, Norman-will be generated-orcid-0000-0003-1272-7429-600González, Miguelapplication/pdfspaCorporación Universidad de la CostaCC0 1.0 Universalhttp://creativecommons.org/publicdomain/zero/1.0/info:eu-repo/semantics/openAccesshttp://purl.org/coar/access_right/c_abf2Case Reports in Neurological Medicinehttps://www.hindawi.com/journals/crinm/2021/6646115/Facial paralysisMelkersson–RosenthalSyndromePresentation of bilateral facial paralysis in Melkersson–Rosenthal syndromeArtículo de revistahttp://purl.org/coar/resource_type/c_6501http://purl.org/coar/resource_type/c_2df8fbb1Textinfo:eu-repo/semantics/articlehttp://purl.org/redcol/resource_type/ARTinfo:eu-repo/semantics/acceptedVersion[1] A. Basman, M. Gumusok, S. Degerli, M. Kaya, and M. Toraman Alkurt, “Melkersson-rosenthal syndrome: a case report,” Journal of Istanbul University Faculty of Dentistry, vol. 51, no. 1, pp. 42–45, 2017.[2] M. Cancian, S. Giovannini, A. Angelini et al., “Melkersson–Rosenthal syndrome: a case report of a rare disease with overlapping features,” Allergy, Asthma & Clinical Immunology, vol. 15, no. 1, p. 1, 2019.[3] S. Feng, J. Yin, J. Li, Z. Song, and G. Zhao, “MelkerssonRosenthal syndrome: a retrospective study of 44 patients,” Acta Oto-Laryngologica, vol. 134, no. 9, pp. 977–981, 2014.[4] W. A. Critchlow and D. Chang, “Cheilitis granulomatosa: a review,” Head and Neck Pathology, vol. 8, no. 2, pp. 209–213, 2014.[5] S. R. Dhawan, A. G. Saini, and P. D. Singhi, “Management strategies of Melkersson-rosenthal syndrome,” A Review, vol. 13, p. 61, 2020.[6] S. Aydın, S. Ozt¨urk, A. Faras¸o ¨ glu, T. Çakıl, and S. J. S. Çoruk, ˘ “Melkersson–Rosenthal syndrome,” A Case Report, vol. 29, no. 2, 2018.[7] O. B. Ozgursoy, S. Karatayli Ozgursoy, O. Tulunay, O. Kemal, A. Akyol, and G. Dursun, “Melkersson-Rosenthal syndrome revisited as a misdiagnosed disease,” American Journal of Otolaryngology, vol. 30, no. 1, pp. 33–37, 2009.[8] E. Brozek-Ma˛dry, W. Jaremek-Ochniak, M. Straburzy ˙ nski, ´ and A. Krzeski, “Lower lip Oedema-Melkersson-Rosenthal Syndrome or Cheilitis Granulomatosa,” New American Journal of Otolaryngology Research, vol. 1, pp. 1-2, 2019.[9] X. Xu, L. Guan, Y. Lv et al., “Exome sequencing identifies FATP1 mutation in Melkersson-Rosenthal syndrome,” vol. 31, no. 5, Article ID e230, 2017.[10] Y. Pei, G. M. Beaman, D. Mansfield, J. Clayton-Smith, M. Stewart, and W. G. Newman, “Clinical and genetic heterogeneity in Melkersson-rosenthal syndrome,” European Journal of Medical Genetics, vol. 62, no. 6, Article ID 103536, 2019.[11] S. Bohra, P. B. Kariya, S. D. Bargale, and S. Kiran, “Clinicopathological significance of Melkersson-Rosenthal syndrome,” BMJ Case Reports, vol. 2015, 2015.[12] J.-J. Tang, X. Shen, J.-J. Xiao, and X.-P. Wang, “Retrospective analysis of 69 patients with Melkersson-Rosenthal syndrome in mainland China,” vol. 9, no. 2, pp. 3901–3908, 2016.[13] R. A. Gaudin, N. Jowett, C. A. Banks, C. J. Knox, and T. A. Hadlock, “Bilateral facial paralysis: a 13-year experience,” Plastic and Reconstructive Surgery, vol. 138, no. 4, pp. 879–887, 2016.[14] W. Zeng, S. Geng, X. Niu, and J. Yuan, “Complete Melkersson–Rosenthal syndrome with multiple cranial nerve palsies,” vol. 35, no. 3, pp. 272–274, 2010.[15] M. U. Aluclu, U. Keklikci, A. Guzel, K. Unlu, and M. Tatli, “Melkersson-Rosenthal syndrome with partial oculomotor nerve palsy,” Annals of Saudi Medicine, vol. 28, no. 2, pp. 135–137, 2008.[16] R. Liu and S. Yu, “Melkersson-Rosenthal syndrome: a review of seven patients,” Journal of Clinical Neuroscience, vol. 20, no. 7, pp. 993–995, 2013.[17] Z. Chu, Y. Liu, H. Zhang, W. Zeng, and S. Geng, “MelkerssonRosenthal syndrome with genitalia involved in a 12-year-old boy,” Annals of Dermatology, vol. 28, no. 2, pp. 232–236, 2016.[18] T. Ogawa, M. N. Sotto, and M. P. Hoang, “Granulomatous dermatitis and others,” in Hospital-Based Dermatopathology: An Illustrated Diagnostic Guide, M. P. Hoang and M. A. Selim, Eds., pp. 137–198, Springer International Publishing, Cham, Switzerland, 2020.[19] G. Shalom, L. Bodner, and S. Halevy, “Melkersson-Rosenthal Syndrome: the possible role of comorbidities in the etiopathogenesis. Giornale italiano di dermatologia e venereologia: organo ufficiale, Societa italiana di dermatologia e sifilografia,” vol. 154, no. 3, pp. 347–351, 2019.[20] F. Kayhan, F. ˙Ilik, and A. Kayhan, “Obsessive–compulsive disorder concurrent with Melkersson-Rosenthal Syndrome: a case report,” General Hospital Psychiatry, vol. 37, no. 5, p. 497, 2015.[21] M. K. Elias, F. J. Mateen, and C. R. Weiler, “+e Melkersson–Rosenthal syndrome: a retrospective study of biopsied cases,” Journal of Neurology, vol. 260, no. 1, pp. 138–143, 2013.[22] A. G. Saini, N. Sankhyan, H. Padmanabh, A. Das, and P. Singhi, “Recurrent facial palsy and electrophysiological findings in oligosymptomatic Melkersson rosenthal syndrome,” 2e Indian Journal of Pediatrics, vol. 83, no. 10, pp. 1188–1190, 2016.[23] L. Oudrhiri, S. Chiheb, F. Marnissi, S. Zamiati, and H. Benchikhi, “Successful treatment of Miescherʼs cheilitis in Melkersson-Rosenthal syndrome with betamethasone injections and doxycycline,” vol. 13, 2012.[24] C. Bacci and M. L. Valente, “Successful treatment of cheilitis granulomatosa with intralesional injection of triamcinolone,” vol. 3, no. 24, pp. 363-364, 2010.[25] I. Espinoza, J. Navarrete, J. Benedetto, A. Borzutzky, P. Roessler, and A. J. A. Ortega-Pinto, “Orofacial granulomatosis and diet therapy: a review of the literature and two clinical cases,” vol. 93, no. 1, pp. 80–85, 2018.[26] S. Raˇskovic, J. Bolpa ´ ci ˇ c, A. Peri ´ c-Popadi ´ c, ´ Z. Jovici ˇ c, ´R. Miˇskovic, and M. Bogi ´ c, “Clinical characteristics and ´treatment of Melkersson-Rosenthal syndrome: overview of six patients,” vol. 68, no. 11-12, pp. 401–404, 2015.[27] J. Wang, P. Li, X. Jin, Y. Xu, and X. J. Zhang, “Outcomes of recurrent facial palsy in Melkersson Rosenthal syndrome,” Annals of Otology, Rhinology & Laryngology, vol. 124, no. 3, pp. 232–234, 2015.[28] Z. Tan, Y. Zhang, W. Chen, W. Gong, J. Zhao, and X. Xu, “Recurrent facial palsy in Melkersson Rosenthal syndrome: total facial nerve decompression is effective to prevent further recurrence,” American Journal of Otolaryngology, vol. 36, no. 3, pp. 334–337, 2015.PublicationORIGINALPresentation of Bilateral Facial Paralysis in Melkersson–Rosenthal Syndrome.pdfPresentation of Bilateral Facial Paralysis in Melkersson–Rosenthal Syndrome.pdfapplication/pdf1517249https://repositorio.cuc.edu.co/bitstreams/6e22fe9a-5251-4210-a2ca-ec09495d213d/download7b6cc229f6f75bcc7575f54d1b42d406MD51CC-LICENSElicense_rdflicense_rdfapplication/rdf+xml; charset=utf-8701https://repositorio.cuc.edu.co/bitstreams/26f091d0-e2e0-4197-8c06-cbc4cd5a2a17/download42fd4ad1e89814f5e4a476b409eb708cMD52LICENSElicense.txtlicense.txttext/plain; charset=utf-83196https://repositorio.cuc.edu.co/bitstreams/e6c74158-e1bb-4e72-9e22-0e1192c7bcb5/downloade30e9215131d99561d40d6b0abbe9badMD53THUMBNAILPresentation of Bilateral Facial Paralysis in Melkersson–Rosenthal 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