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La morfea es una rara enfermedad fibrosante de piel y tejidos subyacentes que aparece a cualquier edad, con más frecuencia en mujeres. Es de etiología autoinmune, benigna, con remisión espontánea o recidivante crónica; y se caracteriza por la aparición de áreas de piel esclerosadas, únicas o múltiples, redondeadas o lineales, asintomáticas, de evolución crónica y sin afección sistémica. Se presentó un paciente en edad pediátrica con esta enfermedad, cuya clínica, examen diagnóstico e histología fueron compatibles con la enfermedad de morfea lineal que, a pesar del retraso en su diagnóstico una vez iniciado el tratamiento inmunosupresor presentó una evolución favorable, simultáneamente se le realizó fisioterapia.

Morphea is a rare fibrosing disease of the skin and underlying tissues which is more common in women and can appear at any age. It is an autoimmune and benign disease with spontaneous remission or chronic relapsing course; it is characterized by the appearance of single or multiple, rounded or linear, asymptomatic, chronically evolving sclerosed skin areas without systemic involvement. We present a female pediatric patient with this disease, whose symptoms, diagnostic examination and histology were compatible with linear morphea that, despite the delay in its diagnosis, she had a favorable evolution once immunosuppressive treatment was started, and physiotherapy was simultaneously performed.

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Introdução: a hemiatrofia facial progressiva, também conhecida como síndrome de Parry Romberg, é uma forma rara de esclerodermia, cujo manejo de sequelas é desafiador. Objetivo: diante desse desafio, o objetivo deste estudo foi avaliar pela primeira vez na literatura a eficácia do preenchimento da hidroxiapatita de cálcio (CaHa) na reconstrução da mandíbula de um paciente com síndrome de Parry Romberg. Relato do caso: paciente do sexo feminino, 15 anos, com atrofia progressiva do lado direito da face por síndrome de Parry Romberg. Após o controle da doença, a principal queixa da paciente era assimetria facial, principalmente na região mandibular. Discussão: a CaHa é um preenchedor injetável biocompatível, que é gradualmente reabsorvido e substituído por estroma fibrovascular, formado principalmente por novo colágeno, em um processo que ocorre sem qualquer reação imunológica. Este perfil de segurança torna a CaHa uma boa escolha para a correção de sequelas de esclerodermia estável. Conclusão: este relato permite concluir que a biocompatibilidade do preenchimento de CaHa e a adequação para correção da mandíbula também são aplicáveis em procedimentos reconstrutivos para esclerodermia estável, de forma segura e minimamente invasiva, com ótimos resultados estéticos. O procedimento deve ser programado caso a caso, e um acompanhamento regular também é recomendado.

Introduction: Progressive Facial Hemiatrophy, also known as Parry Romberg Syndrome, is a rare form of linear scleroderma. The management of facial atrophy sequelae is challenging. Objective: This study aims to evaluate for the first time in literature the effectiveness of the calcium hydroxylapatite (CaHa) filler in the jawline reshaping of a patient with Parry Romberg Syndrome. Case report: A 15-year-old woman with progressive atrophy of the right side of the face due to Parry Romberg Syndrome. After disease control, the patient's main complaint was facial asymmetry, mainly in the jawline region. Discussion: CaHa is a biocompatible injectable filler that is gradually resorbed and replaced by fibrovascular stroma, mainly formed for new collagen, in a process that occurs without any immunological reaction. This safety profile makes CaHa a good choice for correcting stable scleroderma defects. Conclusion: This report concludes that CaHa filler biocompatibility and suitability for the jawline correction are also applicable in reconstructive procedures for stable scleroderma, safely and minimally invasively, with optimal aesthetic results. The method must be programmed case-by-case, and a regular follow-up is also recommended.

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ABSTRACT Taxanes are a class of chemotherapeutic agents with common associated dermatologic adverse events, such as skin hyperpigmentation, hand-foot skin syndrome, paronychia and onycholysis. Taxane-induced scleroderma is rare. Few cases with skin findings resembling systemic sclerosis, have been reported after the administration of these agents. We report two cases with stage IV breast cancer, aged 66 and 71 years, who developed sclerodermic skin lesions in their extremities after starting treatment with placlitaxel and nabplaclitaxel respectively.

Los taxanos son agentes quimioterapéuticos cuyo uso se asocia a problemas dermatológicos tales como hiperpigmentación, síndrome manos-pies, paroniquia y onicolisis. La esclerodermia inducida por taxanos es rara, con pocos casos informados en la literatura. Informamos los casos de dos pacientes con cáncer de mama en estado IV, de 66 y 71 años, que desarrollaron lesiones esclerodérmicas en las extremidades después de ser tratadas con placlitaxel y nabplaclitaxel, respectivamente.

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Abstract Intravascular large B-cell lymphoma is a rare, non-mass-forming, extranodal large B-cell lymphoma subtype characterized by the presence of tumor cells in the lumens of vessels. It is divided into two major types: classical and Asian. Patients presenting only with skin involvement are mostly female, at a younger age than classical intravascular large B-cell lymphoma patients, and have a better prognosis. Since the diagnosis of cases with isolated skin involvement is difficult, keeping this entity in mind, performing a careful microscopic examination, and applying new, effective treatment regimens will make it possible to achieve better clinical outcomes in these cases.

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Intravascular large B-cell lymphoma is a rare, non-mass-forming, extranodal large B-cell lymphoma subtype characterized by the presence of tumor cells in the lumens of vessels. It is divided into two major types: classical and Asian. Patients presenting only with skin involvement are mostly female, at a younger age than classical intravascular large B-cell lymphoma patients, and have a better prognosis. Since the diagnosis of cases with isolated skin involvement is difficult, keeping this entity in mind, performing a careful microscopic examination, and applying new, effective treatment regimens will make it possible to achieve better clinical outcomes in these cases.

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Abstract In kidney biopsies reviews, scleroderma renal crisis (SRC) is characterized by vascular endothelial injuries, C4d deposits on peritubular vessels, and acute and chronic injuries coexisting on the same biopsy. The clinical signs of thrombotic microangiopathy (TMA) are described in systemic sclerosis (SSc), nevertheless, it has not been related to acute injuries described on kidney biopsies. We report a case of SRC in a patient with scleroderma-dermatomyositis overlap syndrome, which also showed clinical and histopathological data of TMA. On fundus examination, a severe acute hypertensive retinopathy was found. The kidney biopsy showed severe endothelial damage with widening of mucoid cells at the level of the intima, focal concentric proliferation on most small arterioles, and C3, C4d, and IgM deposits along the capillary walls. The genetic study of complement only showed the presence of membrane cofactor protein (MCP) risk haplotypes, without other genetic complement disorders. We understand that in a patient with TMA and SSc, the kidney damage would be fundamentally endothelial and of an acute type; moreover, we would observe clear evidence of complement activation. Once further studies correlate clinical-analytical data with anatomopathological studies, it is likely that we will be forced to redefine the SRC concept, focusing on the relationship between acute endothelial damage and complement activation.

Resumo Nas revisões de biópsias renais, a crise renal esclerodérmica (CRE) é caracterizada por lesões endoteliais vasculares, depósitos de C4d em vasos peritubulares e lesões agudas e crônicas que coexistem na mesma biópsia. Os sinais clínicos de microangiopatia trombótica (MAT) são descritos na esclerose sistêmica (ES); no entanto, não foram relacionados às lesões agudas descritas nas biópsias renais. Relatamos um caso de CRE em um paciente com síndrome de superposição de esclerodermia-dermatomiosite, que também apresentou dados clínicos e histopatológicos de MAT. No exame de fundo do olho, foi encontrada uma retinopatia hipertensiva aguda grave. A biópsia renal mostrou lesão endotelial grave com alargamento das células mucoides ao nível da íntima, proliferação concêntrica focal na maioria das pequenas arteríolas e depósitos de C3, C4d e IgM ao longo das paredes dos capilares. O estudo genético do complemento mostrou apenas a presença de haplótipos de risco da proteína cofator de membrana (PCM), sem outros distúrbios genéticos do complemento. Entendemos que em um paciente com MAT e ES, o dano renal seria fundamentalmente endotelial e do tipo agudo; além disso, observaríamos evidências claras de ativação do complemento. Uma vez que novos estudos correlacionam dados clínico-analíticos com estudos anatomopatológicos, é provável que sejamos forçados a redefinir o conceito de CRE, enfocando a relação entre dano endotelial agudo e ativação do complemento.

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Abstract: Atrophoderma of Pasini and Pierini is a skin disorder affecting dermal collagen and is clinically characterized by well-defined plaques of depressed skin. Histopathological changes are subtle, and in most cases, the diagnosis requires a comparative study with healthy skin from the same anatomical site. High frequency ultrasound is a useful imaging method for diagnosis of atrophic skin changes. A case is presented in which ultrasound can support the clinical and the histopathological diagnosis of atrophoderma of Pasini and Pierini.

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Abstract: A 63-year-old black female patient with blisters and exulcerations on the face, neck, upper limbs, and subsequent evolution with hypochromic sclerotic areas and alopecia, is reported. Chronic hepatitis C and presence of high levels of porphyrins in urine were demonstrated. There was complete remission with the use of hydroxychloroquine, photoprotection, and treatment of hepatitis. Significant sclerodermoid involvement of the skin as a manifestation of porphyria cutanea tarda secondary to hepatitis C emphasizes the importance of diagnostic suspicion regarding skin manifestation in order to indicate the appropriate therapy, and to minimize the hepatic morbidity.

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ABSTRACT CONTEXT: Localized scleroderma (morphea) is characterized by fibrosis of skin and subcutaneous tissue. Granuloma annulare is a relatively common disease that is characterized by dermal papules and arciform plaques. CASE REPORT: Here, we present the case of a 42-year-old woman who developed granuloma annulare on the dorsum of her feet and abdominal region, and morphea on the anterior side of her lower limbs. We also discuss the etiological and pathogenetic processes that may cause the rare coexistence of these two diseases. CONCLUSION: Only a few cases in the literature have described coexistence of morphea and granuloma annulare.

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Abstract: Background: Localized scleroderma is a chronic inflammatory skin disease characterized by sclerosis of the dermis and subcutaneous tissue. Platelets play an important role in inflammation. Following activation, platelets rapidly release numerous mediators and cytokines, which contribute to inflammation. Objectives: To evaluate whether there was any relation between localized scleroderma and platelet parameters. Methods: Forty-one patients with localized scleroderma were enrolled in the study. The control group consisted of 30 healthy subjects. Results: The mean platelet volume level in the patient group was 9.9 ± 1.3 fl and in the control group was 7.6 ± 1.1 fl. This difference was statistically significant (p< 0.001). The plateletcrit values are minimally higher in the patient group as compared to the control group. It was statistically significant (p<0.001). There was no significant difference in the platelet counts between the two groups (p= 0.560) In the patient group, there was no significant relation between the mean platelet volume levels and clinical signs of disease (p=0.09). However, plateletcrit values are higher in generalized than localized forms of disease (p=0.01). Study Limitations: The limited number of patients and the retrospective nature of the study were our limitations. Conclusions: This study suggests that platelets might play a role in the pathogenesis of scleroderma. Platelet parameters may be used as markers for evaluating disease severity and inflammatory processes. Thus, there is a need for more detailed and prospective studies.

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La esclerodermia localizada es la forma localizada cutánea de esclerodermia, caracterizada por una fibrosis que se manifiesta en forma de placas o bandas cutáneas escleróticas infiltradas al tacto. La prevalencia se estima en aproximadamente 1-9/100.000, por tal razón se presenta el caso de una paciente con manifestaciones de una esclerodermia cutánea localizada cuya lesión apareció en una mama dos meses antes de asistir a consulta en su área de salud. La principal lesión que presentaba era una placa áspera, que no se dejaba pellizcar, de color carmelita, bordes irregulares, de aproximadamente seis centímetros de diámetro, localizada en mitad inferior de la mama derecha, incluyendo pezón y areola. Se detectaron las alteraciones histológicas propias de la enfermedad de base lo que corroboró el diagnóstico planteado y se descartó mediante otros exámenes cualquier presencia de proceso maligno.

Located scleroderma in the located for of cutaneous scleroderma, characterized by a fibrosis which shows in plaque form of cutaneous sclerotic bands infiltrated to touch. The prevalence is estimated in approximately 1-9/100.000, this is the reason why a case of a patient is presented with signs of a located cutaneous scleroderma. The lesion presented in a breast two months before visiting the doctor in her area. The main lesion was a tough plaque, which was not possible to pinch, brown colored, irregular edges, of approximately six centimeters, located in the mid-inferior right breast, including the nipple and areola. Histological changes which characterize the base disease were found, which corroborated the diagnosis and any malignant process was ruled out by other performed exams.

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Abstract: Werner syndrome is a rare autosomal recessive disorder, caused by mutations in the WRN gene. Clinical findings include: senile appearance, short stature, grey hair, alopecia, bird-like face, scleroderma-like skin changes, skin ulcers, voice abnormalities, cataracts, osteoporosis, type 2 diabetes mellitus, ischemic heart disease and hypogonadism. The syndrome begins to become apparent in adolescence but it is usually diagnosed in the third or fourth decade of life. Since the patients usually die by the age of 40-50 years related to malignant neoplasms or atherosclerotic complications, they should be closely followed and treated for complications

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La esclerodermia pertenece a un grupo de enfermedades autoinmunes del tejido conectivo, que produce: inflamación, disfunción vascular y fibrosis excesiva del tejido conectivo de soporte de la piel y los órganos viscerales. A nivel bucal se evidencian afecciones como xerostomía, microstomía, caries y enfermedad periodontal. El tratamiento odontológico requiere un manejo integral por parte de los profesionales, que comprenda las patologías orales presentes y los síntomas permanentes característicos de la enfermedad sistémica de base. El objetivo de este artículo es presentar dos casos de esclerodermia resaltando sus manifestaciones orales presentes.

Scleroderma belongs to a group of autoimmune connective tissue diseases that causes inflammation, vascular dysfunction and excessive fibrosis of the connective tissue supporting the skin and visceral organs. A level oral conditions such as xerostomia, microstomia, caries and periodontal disease are evident. Dental treatment requires a comprehensive management by professionals who understand oral pathologies and permanent characteristic symptoms of systemic disease. The aim of this paper is to present two cases of scleroderma highlighting present oral manifestations.

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Abstract: Lichen sclerosus is an uncommon inflammatory dermatosis with preferential involvement of the urogenital region. The extragenital involvement is uncommon and is characterized by small rounded macules or papules, pearly white in color. The coexistence of lichen sclerosus and scleroderma plaques in most cases with extragenital location has been reported in the literature. We report a case of lichen sclerosus associated with scleroderma in children, highlighting the importance of dermoscopy in diagnosis.

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Abstract The scleroderma en coup de sabre is a variant of localized scleroderma that occurs preferentially in children. The disease progresses with a proliferative and inflammatory phase and later atrophy and residual deformity, which are treated with surgical techniques such as injectable fillers, transplanted or autologous fat grafting and resection of the lesion. Among the most widely used fillers is hyaluronic acid. However, there are limitations that motivate the search for alternatives, such as polymethylmethacrylate, a permanent filler that is biocompatible, non-toxic, non-mutagenic and immunologically inert. In order to illustrate its application, a case of scleroderma en coup de sabre in a 17-year-old patient, who was treated with polymethylmethacrylate with excellent aesthetic results, is reported.

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A large number of diseases may cause Atrophic skin disorders are caused by a large number of diseases, some of them idiopathic and others inflammatory, in which there is loss of volume of body segments. Localized scleroderma is a rare inflammatory dermatosis, manifested by atrophic skin and subcutaneous tissue alterations. Lipoatrophy may be genetically inherited or acquired as a result of panniculitis, HIV infections or aging. Many treatments have been proposed. Results vary in the acute inflammatory phase and are scarce when sclerosis and atrophy have already been established. This article describes four cases of localized facial scleroderma and one of facial idiopathic lipoatrophy treated with implantation of autologous fat globules extracted from the infragluteal groove, without utilization of cannula aspiration, with lasting results.

Os distúrbios atróficos da pele abrangem inúmeras doenças, algumas idiopáticas e outras, inflamatórias, em que há perda do volume de segmentos do corpo. A esclerodermia localizada é uma dermatose inflamatória, rara, que pode manifestar-se com alterações atróficas da pele e tecido subcutâneo. A lipoatrofia pode ser herdada geneticamente ou adquirida relacionada a paniculites, infeccção pelo HIV, ou envelhecimento. Muitos tratamentos são propostos. Os resultados são variáveis na fase inflamatória aguda e apresentam pouca resposta quando a esclerose e atrofia já estão instaladas. Descreve-se o tratamento de quatro casos de esclerodermia localizada e um de lipoatrofia idiopática, na face, tratados com enxerto de fragmentos de lóbulos de gordura autóloga extraídos sem a utilização da cânula aspirativa, com resultados duradouros.

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Nodular sclerosis is a rare form of presentation related to both systemic and localized forms of scleroderma. We describe the case of a patient with nodular sclerosis in order to alert the medical community to recognize this entity.

Esclerose nodular é uma forma de apresentação cutânea rara relacionada tanto com a forma sistêmica como com a forma localizada da esclerodermia. Descrevemos aqui o caso de uma paciente esta forma nodular de esclerose, no sentido de alertar a comunidade médica para o reconhecimento dessa entidade.

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BACKGROUND: Scleroderma is a chronic autoimmune disease characterized by progressive connective tissue sclerosis and microcirculatory changes. Localized scleroderma is considered a limited disease. However, in some cases atrophic and deforming lesions may be observed that hinder the normal development. Literature reports indicate phototherapy as a therapeutic modality with favorable response in cutaneous forms of scleroderma. OBJECTIVES: This study had the purpose of assessing the phototherapy treatment for localized scleroderma. METHODS: Patients with localized scleroderma were selected for phototherapy treatment. They were classified according to the type of localized scleroderma and evolutive stage of the lesions. Clinical examination and skin ultrasound were used to demonstrate the results thus obtained. RESULTS: Some clinical improvement was observed after an average of 10 phototherapeutic sessions. All skin lesions were softer at clinical palpation with scores reduction upon pre and post treatment comparison. The ultrasound showed that most of the assessed lesions presented a decrease in dermal thickness, and only five maintained their previous measure. Treatment response was similar regardless of the type of phototherapeutic treatment employed. CONCLUSIONS: The proposed treatment was effective for all lesions, regardless of the phototherapeutic modality employed. The improvement was observed in all treated skin lesions and confirmed by clinical evaluation and skin ultrasound.

FUNDAMENTOS: A esclerodermia é uma doença autoimune caracterizada pela esclerose progressiva do tecido conjuntivo e alterações da microcirculação. A forma cutânea é considerada uma doença autolimitada. No entanto, em alguns casos, ocorrem lesões atróficas, deformantes, que dificultam o desenvolvimento normal. Relatos da literatura apontam a fototerapia como uma modalidade terapêutica com resposta favorável nas formas cutâneas da esclerodermia. OBJETIVOS: Este trabalho teve como objetivo avaliar o tratamento da esclerodermia cutânea com fototerapia. MÉTODOS: Foram selecionados pacientes com diagnóstico de esclerodermia cutânea para o tratamento com fototerapia, os quais foram classificados de acordo com o tipo clínico e o estágio evolutivo das lesões. Utilizou-se o exame clínico e a ultrassonografia da pele como metodologia para demonstrar os resultados obtidos com o tratamento proposto. RESULTADOS: Foi observado o início da melhora clínica das lesões com média de 10 sessões de fototerapia. A palpação clínica mostrou amolecimento em todas as lesões estudadas, com redução nos escores de avaliação estabelecidos. No exame de ultrassom, a maioria das lesões avaliadas mostrou diminuição da espessura da derme, e apenas cinco mantiveram sua medida. Não se observou diferença na resposta ao tratamento de acordo com o tipo de fototerapia instituída. CONCLUSÕES: O tratamento proposto foi efetivo em todas as lesões, independentemente do tipo de fototerapia realizada. A melhora foi observada em todas as lesões tratadas e comprovada pela avaliação clínica e pelo exame de ultrassom da pele.

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Introdução - Os pacientes com esclerodermia podem apresentar complicações como ulcerações em terço distal de membros inferiores. A exata incidência dessa complicação recorrente é desconhecida, mas sabe-se que são lesões de mau prognóstico. O laser de baixa potência é amplamente explorado no tratamento de úlceras crônicas, mas até o momento não foram encontrados estudos publicados sobre a utilização do laser em úlceras secundárias à esclerodermia. Este estudo visa relatar os efeitos do laser de baixa potência no tratamento de úlceras cutâneas decorrentes de esclerodermia em paciente do sexo feminino. Materiais e Métodos - Realizou-se aplicação do laser de baixa potência nas feridas secundárias à esclerodermia, em paciente do sexo feminino com quinze anos de lesão. O laser utilizado apresentava comprimento de onda de 660 nm e foi utilizada a técnica pontual com contato na margem das lesões e pontual sem contato no leito das mesmas. A distância entre os pontos era de 1 cm. A energia aplicada por ponto foi de 200 mJ aplicada em 4 rajadas de 50 mJ, duas vezes por semana, durante 10 semanas. As áreas das lesões foram mensuradas através do programa Image J antes e após o tratamento. Resultados - A paciente apresentou diminuição significativa da área de todas as lesões de pele. Além disso, observou-se melhora no aspecto vascular e na sensibilidade dos membros inferiores, assim como, uma diminuição da área de hiperestesia em úlcera na região lateral do membro inferior esquerdo (MIE). Conclusão - O presente estudo documenta o sucesso de uma terapêutica menos invasiva do que cirurgias atualmente eleitas como tratamento adequado e propõe a continuidade de estudos sobre esse tipo de ferida.

Introduction - Ulcers in lower limbs extremity are a frequent problem in patients with systemic sclerosis; but the exact incidence is not defined, whereas these damages are difficult prognostic.The low-level laser is widely studied in treatment of venous ulcers and its utilization is well understood, but there are no studies about laser in ulcers secondary to scleroderma published yet. This study aims to report the low-level laser treatment efficacy over skin ulcers originated by scleroderma in a female patient. Materials and Methods - Low-level laser was applied on wounds secondary to scleroderma in a female patient who presented these injuries during fifteen years. The low-level laser applied had 660 nm wavelength, and it was used the punctual technique with contact around the ulcer, and inside the ulcer without contact. The distance between points was 1 cm. The energy applied per point was 200 mJ applied in 4 discharges of 50 mJ, twice a week, during ten weeks. The wound areas were measured through Image J software before and after treatment. Results - As a result of the treatment it was observed that wound areas significantly decreased. Additionally, there was an improvement of vascular aspect and sensibility in lower-limbs, as well a decrease of an area of hyperesthesia in ulcer at lateral region of left low-limb. Conclusion - This research presents the benefits of an alternative therapy less invasive than surgeries currently considered as adequate treatment, and it suggests continuity of studies about this type of wound.

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A esclerodermia localizada (EL) é uma entidade caracterizada por infiltração e espessamento da pele. Apresenta distribuição assimétrica, sem envolvimento de órgãos internos, mas que pode levar, em alguns casos, ao comprometimento estético da paciente. O objetivo do trabalho foi demonstrar a contribuição da Fisioterapia no tratamento da esclerodermia localizada, através de um caso clínico com uma paciente de 10 anos de idade, com comprometimento de membro superior, tronco e membro inferior, tendo apresentado os sintomas iniciais aos 8 anos de idade. No tratamento, utilizou-se eletrotermofototerapia, cinesioterapia, massoterapia e hidroterapia, tendo, como resultados, a melhora na recuperação funcional, possibilitando à paciente exercer suas atividades com maior facilidade e manutenção da amplitude de movimento (ADM) em todos os graus das articulações avaliadas, segundo dados colhidos na goniometria. Desta forma, é possível concluir que a utilização de recursos fisioterapêuticos nesta patologia é satisfatória, sendo de extrema importância o conhecimento das técnicas pelo profissional.

The localized scleroderma is an entity characterized by the infiltration and the denseness of the skin. It has asymmetrical distribution, without the involvement of the internal organs, but which can lead to, in some cases, to an important jeopardizing of the patient esthetics. The target of this present work had been to demonstrate the contribution of the physiotherapy over the treatment of the localized scleroderma, by means of the accompaniment of a specific clinical case, taking in account a female patient with 10 years old, beaming the superior members, lower trunk and members jeopardized, which has presented the first symptoms by the 8 years old. In her treatment were employed the following physiotherapeutic resources: electro thermo phototherapy, cinesiotherapy,mass therapy and hydrotherapy, wherein was achieved the improvement on the functional recovering, making possible to the patient to carry out or perform her activities with readiness, together with the keeping of the movements amplitude or range (ADM), over all degrees of the evaluated articulations according to the data gathered from the goniometry. As a matter of fact, is possible to conclude that to make use of physiotherapeutic resources in this pathology is satisfactory, it being extremely important the knowledge of the techniques by professional.

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