Melkersson Rosenthal Syndrome: A Case Report

Introduction: Melkersson-Rosenthal Syndrome (MRS) is a rare neuro-mucocutaneous condition. It mainly includes three triads as orofacial swelling, facial palsy and fissured tongue. All of the classical triad may not be seen at the same time, as symptoms may appear in different interval of time and has a recurrence. MRS is diagnosed based on oligosymptomatic when two or more clinical features are present or monosymptomatic cases when there evidence of a non-necrotising, granulomatous cheilitis in patients with isolated facial or lip edema can be noted in biopsy. Case Presentation: In this case report a 18 year old male patient all the three classic clinical characteristic features as granulomatous cheilitis, unilateral facial muscle palsy and a fissured tongue are seen. The clinical features of this rare condition along with treatment procedure are presented. Conclusion: Oral corticosteroids are the mainstay in the treatment of MRS. Intralesional triamcinolone acetonide used in oro-facial edema. In recurrent cases of MRS need second-line immunosuppressants like methotrexate and thalidomide. In refractory recurrent facial nerve palsies Surgical decompression of the facial nerve may be considered.

Melkersson-Rosenthal syndrome: a classical case report / Síndrome de Melkersson-Rosenthal: relato de caso clássico

ABSTRACT The Melkersson-Rosenthal syndrome constitutes a rare manifestation characterized by a triad of signs and symptoms: recurrent orofacial edema, fissured tongue, and recurrent facial paralysis. The difficulty in diagnosing Melkersson-Rosenthal syndrome is that orofacial edema is common to various diseases besides the lack of awareness of the syndrome by health professionals and the frequent metachronous manifestation of its symptomatology. The aim of this report is to present a classical case of Melkersson-Rosenthal syndrome and its clinical and therapeutic approach. A patient who sought for assistance at the Stomatology Clinic presented a synchronous manifestation of the triad: a left lip and cheek nonpitting edema accompanied by facial paralysis on the same side and fissured tongue. Melkersson-Rosenthal syndrome was diagnosed due to the presence of the triad of signs and symptoms after initially ruling out Crohn's disease, Sarcoidosis, and tuberculosis due to a lack of intestinal or respiratory complaints and absence of other clinical evidence. The treatment administered was steroids, the most common treatment with a satisfied prognosis we found in the literature for Melkersson-Rosenthal syndrome patients. We recommend its implementation intralesional injections of betamethasone dipropionate as after four infiltrations the edema subsided by 80% with no further relapses within one-year follow-up.

RESUMO A síndrome de Melkersson-Rosenthal constitui uma manifestação rara caracterizada por uma tríade de sinais e sintomas: edema orofacial recorrente, língua fissurada e paralisia facial recorrente. A dificuldade no diagnóstico da síndrome de Melkersson-Rosenthal é que o edema orofacial é comum a diversas doenças, além do desconhecimento da síndrome pelos profissionais de saúde e da frequente manifestação metacrônica de sua sintomatologia. O objetivo deste relato é apresentar um caso clássico de síndrome de Melkersson-Rosenthal e sua abordagem clínica e terapêutica. Paciente procurou atendimento no Ambulatório de Estomatologia apresentando manifestação sincrônica da tríade: edema não depressível em lábio e bochecha esquerdos acompanhado de paralisia facial do mesmo lado e língua fissurada. A síndrome de Melkersson-Rosenthal foi diagnosticada devido à presença da tríade de sinais e sintomas após a exclusão inicial de doença de Crohn, sarcoidose e tuberculose por ausência de queixas intestinais ou respiratórias e ausência de outras evidências clínicas. O tratamento administrado foi o esteroide, sendo o tratamento mais comum com prognóstico satisfatório que encontramos na literatura para pacientes com SRM. Recomendamos a aplicação de injeções intralesionais de dipropionato de betametasona, pois após quatro infiltrações o edema cedeu em 80% sem novas recidivas em um ano de seguimento.

Prevalence of Oral Normal Variance in Healthy Elderly Patients: A Descriptive Study on Oral Pseudo-Lesions

Abstract Introduction The aging process in the elderly can change the condition of the oral cavity, regarding both clinical appearance and function. The oral mucosa becomes more susceptible to mechanical damage and irritant agents. Any changes in the oral mucosa, including normal variance, should be examined. This is especially true for the elderly, to improve oral health and prevent any abnormalities. Objective To describe the prevalence of oral normal variance in healthy elderly patients. Methods An observational descriptive study with a cross-sectional design with total sampling conducted from March to April 2020. Results Among the 117 elderly patients who participated in this study, 58 were described as healthy without any comorbidities. From those subjects, there were 117 cases of normal variance in the oral mucosa. The three most prevalent issues were fissured tongue (93.1%), coated tongue (58.62%), and crenated tongue (27.59%). Conclusion Fissured tongue is a normal variance of the oral mucosa that is commonly found in healthy elderly patients.

An Overview on Developmental Disturbances of the Tongue

Tongue is a vital organ present in the oral cavity. Besides the important functions of speech and deglutition, it may act as an index for underlying systemic diseases. Although it is easy to examine the various developmental anomalies of the tongue clinically still some anomalies may present a diagnostic and therapeutic dilemma

Alergias alimentarias en pacientes con glositis migratoria benigna / Food allergies in patients with benign migratory glossitis

Introducción: la glositis migratoria benigna, también conocida como lengua geográfica, es una variante normal, que presenta zonas superficiales circulares. Según la literatura revisada, dentro de sus causas figuran los procesos alérgicos, en los que están las alergias alimentarias. Se decidió realizar esta investigación para determinar la prevalencia de las alergias alimentarias frecuentes en estos pacientes mediante pruebas cutáneas. Materiales y métodos: se realizó un estudio descriptivo, longitudinal y prospectivo. La muestra estuvo compuesta por 27 pacientes en el período de enero hasta agosto de 2018 en portadores de glositis migratoria benigna, a los cuales se les realizaron pruebas alérgicas de tipo cutáneas para la determinación de alergias alimentarias. Resultados: el 59,3 % fue mujer y el antecedente alérgico más frecuente fue la rinitis alérgica. El antecedente de alergia alimentaria más frecuente fue los lácteos. Respecto a los resultados de las pruebas para la leche y los mariscos, estas dieron positivas en la mayoría de los pacientes, seguidos por el pescado, los huevos y los frutos secos. La prevalencia de positividad para por lo menos una prueba fue de 85,2%. Discusión: actualmente no existe suficiente literatura que hable sobre las posibles etiologías de la glositis migratoria benigna. Dentro de los antecedentes alérgicos, la rinitis alérgica fue la más frecuente, seguida de la alergia alimentaria. Hasta ahora existen pocas investigaciones que estudien esta prevalencia de alergias alimentarias en los pacientes de forma específica. Conclusión: la lengua geográfica no tiene factor epidemiológico predominante. El antecedente alérgico puede ser un factor predisponente, la alergia alimentaria puede ser desencadenante de la presentación de la lengua geográfica.

Introduction: The benign migratory glossitis, also known as geographic tongue, is a normal variant, which presents circular or irregular depapillated superficial areas. According to the literature reviewed, its causes include allergic processes, among these are food allergies. It was decided to carry out this investigation to determine the prevalence of frequent food allergies in these patients using skin tests. Materials and methods: A descriptive, longitudinal, prospective study was carried out. The sample consisted of 27 patients in the period January-August 2018, carriers of geographic tongue who underwent skin-type allergic tests to determine food allergies. Results: 59.3% were women without statistically significant predominance, the most frequent allergic history was allergic rhinitis. The most frequent history of food allergy was to dairy. Regarding the results of the tests for milk and shellfish, they were positive in 55.6%, followed by fish 14.8%, eggs and nuts with 11.1% respectively. The prevalence of positivity for at least one test was 85.2% with a statistically significant predominance over the negative result. Discussion: There is currently not enough literature that discusses the possible etiologies of benign migratory glossitis. Within the allergic history, allergic rhinitis was the most frequent, followed by food allergy. Until now there are few investigations that study this prevalence of food allergies in patients in a specific way. Conclusion: Geographic tongue does not have a predominant epidemiological factor, an allergic history can be a predisposing factor, food allergy can be a trigger for presentation of geographic language.

Síndrome de Miescher-Melkersson-Rosenthal: Presentación de tres casos y revisión de la literatura / Miescher-Melkersson-Rosenthal Syndrome: Three cases reports and literature review

RESUMEN El síndrome de Miescher-Melkerson-Rosenthal es una condición clínica poco frecuente caracterizada por un edema orofacial episódico, paresia facial periférica recurrente, lengua geográfica (lingua plicata) y granulomas en biopsia de piel o labial. Se ha reportado más frecuentemente en mujeres, y su incidencia es probablemente subestimada por el desconocimiento del síndrome, su presentación clínica incompleta y la plétora de diagnósticos diferenciales. Se debe considerar en pacientes que presenten al menos dos de las cuatro características mencionadas. Su tratamiento, con esteroides vía oral, está dirigido a los episodios de edema orofacial y/o paresia facial. A continuación se describen tres casos del síndrome valorados en el servicio de neurología de la Fundación Cardioinfantil y se presenta una breve revisión de la literatura.

SUMMARY Miescher-Melkerson-Rosenthal syndrome is an uncommon clinical condition characterized by episodic orofacial oedema, recurrent peripheral facial paralysis, geographic tongue (lingua plicata) and granulomas in skin or labial biopsy. It is reported more frequently in women, and its incidence is probably underestimated due to lack of knowledge of the syndrome, incomplete clinical presentation and the plethora of differential diagnosis. It should be considered in patients with at least two of the four mentioned characteristics. Its treatment, with oral steroids, is directed at the episodes of orofacial oedema and/or the facial paralysis. We described three cases of the syndrome seen at the Fundacion Cardioinfantil's neurology service and present a short review of the literature.

Síndrome de Melkersson-Rosenthal / Melkersson-Rosenthal syndrome

A síndrome de Melkersson-Rosenthal é uma doença rara que pode se apresentar como uma tríade clássica de edema orofacial, paralisia facial e língua fissurada ou, mais frequentemente, com características oligo/monossintomáticas. Relatamos um caso que aportou a um alergista para o diagnóstico de um angioedema, e que na avaliação se configurou como a síndrome completa. Diagnósticos diferenciais com angioedema alérgico, hereditário, idiopático e com outras patologias devem ser considerados. Apesar de o diagnóstico ser clínico, a biópsia cutânea foi relevante. Objetivamos alertar o especialista que se depara com angioedema crônico recorrente sobre essa patologia.

Melkersson-Rosenthal syndrome is a rare disease that may present as the classic triad of orofacial edema, facial paralysis, and fissured tongue or, more often, with oligosymptomatic/ monosymptomatic features. We report a case referred to an allergist for the diagnosis of angioedema that was identified as a complete syndrome during evaluation. Differential diagnoses with allergic, hereditary, and idiopathic angioedema and with other diseases should be considered. Although the diagnosis is clinical, skin biopsy is relevant. Our goal is to alert specialists who deal with chronic recurrent angioedema to the possibility of this syndrome.

Investigation of the psychological factors associated with fissured tongue / Investigação dos fatores psicológicos associados à língua fissurada

ABSTRACT Objective: This study aimed to investigate the correlation between FT and anxiety. Methods: The sample consisted of 95 participants categorized into two groups: a) G-FT, 48 patients with FT and b) CG, 47 healthy participants. All patients were submitted to complete oral and dermatological examinations. The anxiety levels were measured using the State-Trait Anxiety Inventory (STAI). A p-value of < 0.05 was considered significant. Results: Fourteen (30%) patients reported awareness of having FT and 9 (64%) were women. Symptoms like burning feeling were reported by 3 (6%) only patients, all of then women. None of the control group patients presented these symptoms. In both groups, control and FT, the average scores of trait-anxiety were slightly higher than state-anxiety. There were no statistically significant differences between the FT and the control group related to the state-anxiety (p=0.724) and the trait-anxiety (p=0.680) scores. Study limitations: This study was limited by the number of subjects. Conclusion: Although moderate state-anxiety and trait-anxiety scores were determined, anxiety, as an environmental factor may not play a significant role in the development of FT. Considering FT as a multifactorial disease, probably a genetic component is necessary for expression.

RESUMO Objetivo: Investigar a correlação entre língua fissurada e ansiedade. Métodos: A amostra consistiu em 95 participantes divididos em dois grupos: a) G-LF, 48 pacientes com língua fissurada e b) CG, 47 participantes saudáveis. Os pacientes foram submetidos aos exames orais e dermatológicos. Os níveis de ansiedade foram medidos utilizando o Inventário de Ansiedade de Traçado do Estado (IDATE). Um valor de p <0,05 foi considerado significativo. Resultados: Quatorze (30%) pacientes relataram ter consciência de língua fissurada e 9 (64%) eram mulheres. Sintomas como sensação de queimação foram relatados por apenas 3 (6%) pacientes. Nenhum dos pacientes do grupo controle apresentou esses sintomas. Nos dois grupos, controle e língua fissurada, os escores médios de ansiedade-tração foram ligeiramente superiores ao estado-ansiedade. Não houve diferenças estatisticamente significativas entre grupo língua fissurada e o grupo controle, relacionado ao estado-ansiedade (p = 0,724) e os achados traço-ansiedade (p = 0,680). Limitação: Este estudo é limitado devido o tamanho da amostra. Conclusão: Embora tenham sido determinados valores moderados de ansiedade de estado e traço-ansiedade, a ansiedade, como fator ambiental, pode não desempenhar um papel significativo no desenvolvimento de língua fissurada. Considerando língua fissurada como uma doença multifatorial, é necessário um componente genético para a expressão.

Tongue lesions and anomalies in a sample of Yemeni dental patients: a cross-sectional study

Background: Tongue examination helps considerably in diagnosing the underlying health state of the patient, especially in the cases of chronic diseases. The aim of this study is to determine the prevalence and association of tongue lesions with risk factors among Yemeni dental patients. Materials and methods: An oral medicine specialist examined all 713 patients attending the dental polyclinics at the University of Sciences and Technology (Sana’a, Yemen). The examination sheet was designed to include information related to patient characteristics, medical history, dental history, habits, and tongue lesions. Results: The prevalence of tongue lesions among the examined participants was 76.5 percent. The prevalence rate was 83.4 percent for males and 69.2 percent for females. Fissured tongue was the most common condition. Logistic regression analysis indicated that older age (p<0.001), gender (p=0.007), khat chewing (p<0.001), and smoking (p=0.001) were associated with fissured tongue; gender (p<0.001), khat chewing (p<0.001), and smoking (p<0.001) were associated with hairy tongue; and older age (p<0.001), khat chewing (p=0.001), and smoking (p=.021) were associated with coated tongue. Conclusion: The prevalence of tongue lesions among this sample of Yemeni population was 76.5 percent; fissured tongue and hairy tongue were the most prevalent lesions. Khat chewing, smoking, and older age were the associated risk factors for many of studied lesions and anomalies.

Avaliação do traço e estado de ansiedade em pacientes com Glossite Migratória Benigna / Evaluation of trait and state anxiety in patients with Benign Migratory Glossitis

Objetivo: avaliar estado e traço de ansiedade em pacientes com glossite migratória benigna (GMB). O estudo consistiu de 78 pacientes com GMB, apresentando ou não ocorrência simultânea de língua fissurada. Um grupo controle de 48 pacientes, sem lesões orais e/ou cutâneas, também foi incluído no estudo. Foi aplicado o Inventário de Traço-Estado de Ansiedade (STAI). Resultados: estado moderado e pontuações de ansiedade-traço foram observados nos grupos. Não houve diferenças estatisticamente significativas nos escores de ansiedade entre os grupos GMB e controle. Conclusão: a ansiedade não parece desempenhar um papel significativo na GMB, apesar dos níveis moderados observados em pacientes com GMB. No entanto, o estado emocional pode ser um fator determinante para as diferentes formas de manifestações de GMB.

Objective: To assess state and trait anxiety in patients with benign migratory glossitis (GMB). The study consisted of 78 patients with GMB, presenting or not simultaneous occurrence of fissured tongue. A control group of 48 patients without oral lesions and / or skin, was also included in the study. Inventory -Trait State Anxiety (STAI) was applied. Results: moderate state and trait anxiety scores were observed in groups. There were no statistically significant differences in anxiety scores between the GMB and control groups. Conclusion: The anxiety seems to play a significant role in the GMB, despite moderate levels observed in patients with GMB. However, the emotional state can be a determining factor for the different forms of manifestations of GMB.

The diagostic dilemma of oral psoriasis: a review

Psoriasis is chronic immunologically mediated inflammatory skin disorder affecting 1–3% of Swedish population. It is associated with impairments quality of life even in mild cases, while in severe cases it excess mortality .Oral manifestations of psoriasis are rare and has various clinical presentations which are often difficult to diagnose. Oral psoriasis is a rare entity that might be confused with other oral mucous membrane dermatoses; hence, it should be considered under differential diagnosis of oral mucous membrane disorders and confirmed histo-pathologically. The occurrences of cutaneous lesions along with oral lesions that are diagnosed histo-pathologically give definite diagnosis for oral psoriasis.

Prevalencia de alteraciones linguales y factores relacionados en niños que consultan a la Universidad de Cartagena, Colombia / Prevalence of tongue alterations and related factors in children attending the University of Cartagena, Colombia

Objetivo: Describir la prevalencia de alteraciones linguales y factores relacionados en niños que asisten a las clínicas odontológicas de la universidad de Cartagena, Colombia. Métodos: Estudio descriptivo de corte transversal, se examinaron aleatoriamente 134 niños que consultaron en el segundo semestre del año 2011. Se realizó examen clínico al niño, revisión de su historia clínica médica y una encuesta a la madre, para evaluar la presencia de alteraciones linguales, clasificación, características sociodemográficas y los posibles factores relacionados (ingesta de medicamentos, anemia, problemas nutricionales, déficit de vitamina B, alergias, estrés, psoriasis e higiene bucal). Se utilizaron proporciones, intervalos de confianza y la prueba estadística χ², asumiendo una significancia menor a 0.05. Resultados: La prevalencia de alteraciones linguales fue de 79.9%, siendo las lesiones más frecuentes la lengua saburral con el 74.6% y anquiloglosia con el 5.2%. No hubo relación estadísticamente significativa entre la presencia de alteraciones linguales y los factores evaluados. Conclusión: Las alteraciones linguales en niños tuvieron una alta prevalencia en especial la lengua saburral, en este sentido se deben implementar protocolos de manejo para incentivar en los niños y los padres conductas de higiene bucal saludables enfatizando en la superficie dorsal de la lengua.

Aim: The aim of the present study was to describe prevalence of tongue alterations and related factors observed in child patients attending the University of Cartagena, Colombia. Method: The present study was of a cross-sectional, descriptive nature. 134 children were randomly examined after attending consultation during the second semester of 2011. Selected children were subjected to clinical examination, their medical history was reviewed and their mothers were interviewed, in order to ascertain presence of tongue alterations. A classification was undertaken and socio-demographic characteristics were noted as well as possibly related factors (drug intake, anemia, nutritional problems, vitamin B deficiencies, allergies, stress, psoriasis and oral hygiene. Proportions, confidence intervals and χ2 statistical test were used, assuming significance lesser than 0.05. Results: Prevalence of tongue alterations was 79.9%. Most frequent lesions were coated tongue (74.6%) and ankyloglossia (5.2%). No statistically significant relationship was found between presence of tongue alterations and assessed factors. Conclusion: Tongue alterations in children were highly prevalent, especially coated tongue cases. Bearing this in mind parents and children should be encouraged to implement healthy oral hygiene habits stressing hygiene of the tongue's dorsal surface.

Tetralogy of Fallot: a case report and dental considerations / Tetralogia de Fallot: relato de caso com considerações dentárias

OBJETIVO: Apresentar um caso de Tetralogia de Fallot, uma doença cardíaca congênita grave, que pode causar um impacto nos tecidos dentários, os quais necessitam de tratamento. DISCUSSÃO E CONCLUSÃO: O tratamento adequado de pacientes com problemas cardíacos depende do conhecimento do clínico a respeito da condição. Este relato enfatiza o manejo odontológico de pacientes com Tetralogia de Fallot.

OBJECTIVE: To present a case of Tetralogy of Fallot, a serious congenital heart disease, which can have an impact upon the dental tissue and which needs dental care. DISCUSSION AND CONCLUSION: Successful management of cardiac patients depends upon, knowledge about the condition. This paper highlights on dental management of Tetralogy of Fallot patients.

A Case of Pyogenic Granuloma on the Fissured Tongue / 대한피부과학회지

Pyogenic granuloma is a benign vascular tumor related to trauma, infection, or hormonal changes. When it affects the oral cavity, the most frequent site is the gingiva and involvement of the tongue is very rare. Occurrence of pyogenic granuloma on the fissured tongue has not been reported yet. We present a rare case of pyogenic granuloma on the tongue in a 64 year old female patient in which the fissured tongue seemed to play important roles in the pathogenesis of occurrence of the pyogenic granuloma.

A Case of Melkersson-Rosenthal syndrome

Melkersson-Rosenthal syndrome (MRS) is a rare form of hereditary angioedema characterized by a triad of orofacial swelling, relapsing facial paralysis, and a fissured tongue. However, the classic triad is not frequently seen in its complete form, and monosymptomatic or oligosymptomatic forms are more common. Case: A 20-year-old man presented with recurrent labial swelling 9 months ago and recently with a fissured tongue. The surface of the tongue showed deep furrows characteristic of lingua plicata, and an edematous enlargement was observed on the lower lips. Upon neurologic examination, a left sided facial palsy of a peripheral type was noted without abnormalities in the taste sense or lacrimation. An electromyography of the left frontal muscle showed positive sharp waves and fibrillation potentials. Biopsies performed on the lower lips revealed the congested vessels and perivascular inflammatory cells. We report a 20-year-old man with a classical triad of symptoms of Melkersson-Rosenthal syndrome.

Two Cases of Combined Geographic Tongue and Fissured Tongue in Generalized Pustular Psoriasis / 대한피부과학회지

Generalized pustular psoriasis is a rare disease, even psoriasis involving oral mucosa is very rare. There are many reports of tongue lesions in patients with psoriasis that are clinically and histologically indistinguishable from geographic tongue, and there are many reports of fissured tongue in association with psoriasis. It has been debated whether oral psoriasis exists or not, or occurs as part of generalized pustular psoriasis. We experienced two cases of combined geographic tongue and fissured tongue in patient with generalized pustular psoriasis, and report these cases with review of the literature about the oral lesions in psoriasis, geographic tongue, and fissured tongue.