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...

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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
id RCUC2_83c39a4fa3ede279a89cdf085f716126
oai_identifier_str oai:repositorio.cuc.edu.co:11323/8899
network_acronym_str RCUC2
network_name_str REDICUC - Repositorio CUC
repository_id_str
dc.title.spa.fl_str_mv 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
dc.creator.fl_str_mv 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
dc.subject.spa.fl_str_mv 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.
publishDate 2021
dc.date.accessioned.none.fl_str_mv 2021-11-23T19:19:39Z
dc.date.available.none.fl_str_mv 2021-11-23T19:19:39Z
dc.date.issued.none.fl_str_mv 2021
dc.type.spa.fl_str_mv Artículo de revista
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dc.identifier.issn.spa.fl_str_mv 2090-6668
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dc.identifier.uri.spa.fl_str_mv https://hdl.handle.net/11323/8899
dc.identifier.doi.spa.fl_str_mv https://doi.org/10.1155/2021/6646115
dc.identifier.instname.spa.fl_str_mv Corporación Universidad de la Costa
dc.identifier.reponame.spa.fl_str_mv REDICUC - Repositorio CUC
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Corporación Universidad de la Costa
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url https://hdl.handle.net/11323/8899
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https://repositorio.cuc.edu.co/
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dc.relation.references.spa.fl_str_mv [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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spelling Gaitan Quintero, Gustavobe70fcd3e154b53507fc38cae90ff5b3Camargo, Loidac2ac212e95132aeef3bb340b6680bd6dLópez Velásquez, Norman Darío1765fb512946a9b5a55e7d3377ced18fGonzález, Miguelcf012044d1e00fa1fa33d59feaca0ba42021-11-23T19:19:39Z2021-11-23T19:19:39Z20212090-66682090-6676https://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.application/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.ORIGINALPresentation of Bilateral Facial Paralysis in Melkersson–Rosenthal Syndrome.pdfPresentation of Bilateral Facial Paralysis in Melkersson–Rosenthal Syndrome.pdfapplication/pdf1517249https://repositorio.cuc.edu.co/bitstream/11323/8899/1/Presentation%20of%20Bilateral%20Facial%20Paralysis%20in%20Melkersson%e2%80%93Rosenthal%20Syndrome.pdf7b6cc229f6f75bcc7575f54d1b42d406MD51open accessCC-LICENSElicense_rdflicense_rdfapplication/rdf+xml; charset=utf-8701https://repositorio.cuc.edu.co/bitstream/11323/8899/2/license_rdf42fd4ad1e89814f5e4a476b409eb708cMD52open accessLICENSElicense.txtlicense.txttext/plain; charset=utf-83196https://repositorio.cuc.edu.co/bitstream/11323/8899/3/license.txte30e9215131d99561d40d6b0abbe9badMD53open accessTHUMBNAILPresentation of Bilateral Facial 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