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1.
Clin Nucl Med ; 44(12): 961-963, 2019 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-31689277

RESUMO

A 57-year-old woman with a history of uterine endometrial carcinoma underwent PET/CT examinations for initial staging and posttreatment survey. Multiple patchy accumulations were noted in the muscles, particularly in both thighs. These accumulations resolved spontaneously 6 months after the follow-up examination. However, 3.5 years after the surgery, the multiple patchy accumulations reappeared in the muscle of the upper and lower extremities showing an increase in signal intensity from previous examination. A biopsy of the right thigh revealed epithelioid cell granuloma without necrosis. We therefore consider that this case might be "idiopathic" granulomatous myositis.


Assuntos
Fluordesoxiglucose F18 , Granuloma/complicações , Miosite/complicações , Miosite/diagnóstico por imagem , Tomografia Computadorizada com Tomografia por Emissão de Pósitrons , Neoplasias do Endométrio/complicações , Neoplasias do Endométrio/diagnóstico por imagem , Neoplasias do Endométrio/patologia , Neoplasias do Endométrio/terapia , Feminino , Humanos , Pessoa de Meia-Idade , Miosite/patologia , Estadiamento de Neoplasias
3.
World Neurosurg ; 130: e150-e159, 2019 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-31203060

RESUMO

BACKGROUND: Xanthogranuloma is a chronic inflammatory mass characterized by cholesterol crystal deposition, which is rarely seen in the sellar region. The objective of this study is to identify the clinical features and cause of sellar xanthogranulomas. METHODS: We retrospectively analyzed manifestation, radiographic, and endocrinologic presentation in 9 patients (7 women and 2 men) whom we had previously treated. RESULTS: The patients were between 26 and 73 years of age (median, 56 years). The chief symptoms were visual symptoms in 3, polyuria in 3, headache in 4, and tiredness in 4 patients. Perimetry found visual field deficit in 6 patients. Anterior pituitary provocation tests disclosed impairment of ≥1 hormone in all patients: growth hormone in 8 patients and adrenocorticotropic hormone-cortisol axis in 8 patients. The lesions were suprasellar in 2 patients, intrasellar in 2 patients, and intrasuprasellar region in 5 patients. Three of the lesions were solid and 6 were single cystic to multicystic. Very low intensity area on T2-weighted magnetic resonance imaging was observed in 4 lesions. Postcontrast study performed in 7 lesions showed enhancement in solid parts or cyst walls. Surgical decompression improved visual disturbance in half of the patients but rarely improved hormonal deficits. Follow-up (median, 47 months) found no recurrence of the lesion. In addition to these 9 cases, we found 2 xanthogranulomatous lesions pathologically associated with ciliated epithelia, which also presented with severe hypopituitarism. CONCLUSIONS: Xanthogranuloma seems to be the last stage of the chronic inflammation affecting Rathke cleft cyst or craniopharyngioma presenting with severe anterior pituitary insufficiency.


Assuntos
Granuloma/complicações , Granuloma/patologia , Hipopituitarismo/complicações , Neoplasias Hipofisárias/complicações , Sela Túrcica/patologia , Xantomatose/complicações , Xantomatose/patologia , Adulto , Idoso , Feminino , Granuloma/diagnóstico por imagem , Humanos , Hipopituitarismo/diagnóstico , Hipopituitarismo/patologia , Masculino , Pessoa de Meia-Idade , Adeno-Hipófise/diagnóstico por imagem , Adeno-Hipófise/patologia , Neoplasias Hipofisárias/diagnóstico por imagem , Neoplasias Hipofisárias/patologia , Estudos Retrospectivos , Sela Túrcica/diagnóstico por imagem , Xantomatose/diagnóstico por imagem
5.
Medicine (Baltimore) ; 98(16): e14718, 2019 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-31008920

RESUMO

Xanthogranuloma of choroid plexus is an extremely rare, benign, and mostly asymptomatic intracranial lesion. We report a case of symptomatic lateral ventricular xanthogranuloma resected via a neuronavigator-guided ventriculoscopic approach. Then we review recent English medical literature and notice that craniotomies have been the most popular treatment. But our choice of a ventriculoscopic approach possesses unique advantages such as minimized neural tissue damage, shortened operative time, less blood loss, and safer access to central structures over conventional open surgeries. Informed consent has been obtained from the patient and his immediate family regarding this case report.


Assuntos
Encefalopatias/diagnóstico , Plexo Corióideo , Granuloma/diagnóstico , Ventrículos Laterais , Xantomatose/diagnóstico , Adulto , Encefalopatias/complicações , Encefalopatias/diagnóstico por imagem , Encefalopatias/cirurgia , Granuloma/complicações , Granuloma/diagnóstico por imagem , Granuloma/cirurgia , Cefaleia/etiologia , Humanos , Imagem por Ressonância Magnética , Masculino , Neuroendoscopia , Exame Neurológico , Neuronavegação , Xantomatose/complicações , Xantomatose/diagnóstico por imagem , Xantomatose/cirurgia
6.
J Eur Acad Dermatol Venereol ; 33(7): 1412-1420, 2019 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-30869812

RESUMO

BACKGROUND: Paediatric cutaneous granuloma with primary immunodeficiency (PID) is a rare condition. The physiopathology is unclear, and treatment is challenging. We report on 17 paediatric cases and review the literature. OBJECTIVES: To make dermatologists and dermatopathologists aware of the diagnostic value of skin granulomas in paediatric PID. METHODS: We collected data on 17 patients with cutaneous granulomas and PID registered with us and also reviewed 33 cases from the literature. RESULTS: Cutaneous granuloma was the presenting feature of the PID in 15 of the 50 collated cases. The lesions presented as red-brownish nodules and infiltrated ulcerative plaques, predominantly on the face and limbs. Scleroderma-like infiltration on a single limb was observed in 10% of the cases. The associated PID was ataxia-telangiectasia (52%), combined immunodeficiency (24%), cartilage-hair hypoplasia (6%) and other subtypes (18%). The granulomas were mostly sarcoidal, tuberculoid, palisaded or undefined subtypes. In some patients, several different histopathologic granulomatous patterns were found in the same biopsy. Some granulomas were associated with the presence of a vaccine strain of rubella virus. CONCLUSION: Cutaneous granulomas associated with a PID have a variable clinical presentation. A PID can be suspected when crusty, brownish lesions are found on the face or limbs. The concomitant presence of several histological subtypes in a single patient is suggestive of a PID.


Assuntos
Granuloma/diagnóstico , Granuloma/patologia , Dermatopatias/diagnóstico , Dermatopatias/patologia , Anormalidades Múltiplas/diagnóstico , Ataxia Telangiectasia/etiologia , Criança , Pré-Escolar , Feminino , Granuloma/complicações , Cardiopatias Congênitas/complicações , Cardiopatias Congênitas/diagnóstico , Humanos , Hidrocolpos/complicações , Hidrocolpos/diagnóstico , Lactente , Masculino , Polidactilia/complicações , Polidactilia/diagnóstico , Imunodeficiência Combinada Severa/complicações , Imunodeficiência Combinada Severa/diagnóstico , Dermatopatias/complicações , Úlcera Cutânea/etiologia , Doenças Uterinas/complicações , Doenças Uterinas/diagnóstico
7.
Sanid. mil ; 75(1): 52-62, ene.-mar. 2019. ilus
Artigo em Espanhol | IBECS | ID: ibc-183706

RESUMO

En 1914 el infierno se afloró en Europa y se extendió a una buena parte del mundo. La capacidad destructiva del hombre para con el hombre llegó a niveles insospechados. Centenares de miles de bajas precisaban atención sanitaria urgente en cada campaña. Esta enorme demanda hizo que en apenas cinco años la Medicina avanzara lo que en otra situación hubiera necesitado muchas décadas. El tratamiento quirúrgico de las heridas, el tratamiento protésico, la cirugía plástica, la radiología y la anestesia fueron los campos donde se avanzó más. Sin embargo, probablemente uno de los mayores hitos alcanzados en este periodo, y seguramente uno de los de más trascendencia, fue el desarrollo de la terapia transfusional. En pocos años pasó de ser una técnica de «científicos extraños» a ser una herramienta terapéutica indispensable. Aquí se describe el camino hacia la consecución y sus protagonistas


In 1914, hell broke out in Europe and spread to much of the world. The destructive capacity of man against man reached unsuspected levels. Hundreds of thousands of casualties needed urgent health care in each campaign. This enormous demand meant that in just five years, medicine would advance what in another situation would have needed many decades. Surgical treatment of wounds, orthopedic treatment, plastic surgery, radiology and anesthesia were the fields where more progress was made. However, probably one of the major milestones reached in this period of time, and certainly one of the most transcendent, was the development of blood transfusion. In a few years it went from being a technique of "strange scientists" to become essential. Here we describe the path to achievement and its main characters


Assuntos
Humanos , Lactente , Dermatite de Contato/complicações , Dermatite de Contato/epidemiologia , Dermatite das Fraldas/complicações , Hidrocortisona/administração & dosagem , Dermatite de Contato/diagnóstico , Dermatite de Contato/terapia , Diagnóstico Diferencial , Granuloma/complicações , Granuloma/tratamento farmacológico , Higiene , Permanganato de Potássio/administração & dosagem
9.
Arch Soc Esp Oftalmol ; 94(5): 248-251, 2019 May.
Artigo em Inglês, Espanhol | MEDLINE | ID: mdl-30712950

RESUMO

Orbital adult-onset xanthogranuloma is a rare idiopathic inflammatory disorder included in the group of orbital xanthogranulomatous diseases with possible systemic associations. A 43 year-old woman presented with a deep fast-growing mass on the right upper eyelid. She had a past medical history of bilateral non-ulcerative nodules on the upper eyelid and two cosmetic upper lid blepharoplasties. An excisional biopsy was performed and the histopathology analysis confirmed the diagnosis of adult histiocytic xanthogranuloma. The systemic work up was negative and the patient is asymptomatic without treatment. Orbital xanthogranulomatous diseases are rare, but potentially life-threatening disorders. The ophthalmologist must be aware of this disease in order to make an early diagnosis using the anamnesis, clinical examination, and histology of the periocular lesions that may resemble palpebral xanthelasmas.


Assuntos
Blefarite/etiologia , Granuloma/complicações , Xantomatose/complicações , Adulto , Feminino , Granuloma/patologia , Humanos , Recidiva , Xantomatose/patologia
10.
J Clin Immunol ; 39(1): 81-89, 2019 01.
Artigo em Inglês | MEDLINE | ID: mdl-30607663

RESUMO

The association of immunodeficiency-related vaccine-derived rubella virus (iVDRV) with cutaneous and visceral granulomatous disease has been reported in patients with primary immunodeficiency disorders (PIDs). The majority of these PID patients with rubella-positive granulomas had DNA repair disorders. To support this line of inquiry, we provide additional descriptive data on seven previously reported patients with Nijmegen breakage syndrome (NBS) (n = 3) and ataxia telangiectasia (AT) (n = 4) as well as eight previously unreported patients with iVDRV-induced cutaneous granulomas and DNA repair disorders including NBS (n = 1), AT (n = 5), DNA ligase 4 deficiency (n = 1), and Artemis deficiency (n = 1). We also provide descriptive data on several previously unreported PID patients with iVDRV-induced cutaneous granulomas including cartilage hair hypoplasia (n = 1), warts, hypogammaglobulinemia, immunodeficiency, myelokathexis (WHIM) syndrome (n = 1), MHC class II deficiency (n = 1), Coronin-1A deficiency (n = 1), X-linked severe combined immunodeficiency (X-SCID) (n = 1), and combined immunodeficiency without a molecular diagnosis (n = 1). At the time of this report, the median age of the patients with skin granulomas and DNA repair disorders was 9 years (range 3-18). Cutaneous granulomas have been documented in all, while visceral granulomas were observed in six cases (40%). All patients had received rubella virus vaccine. The median duration of time elapsed from vaccination to the development of cutaneous granulomas was 48 months (range 2-152). Hematopoietic cell transplantation was reported to result in scarring resolution of cutaneous granulomas in two patients with NBS, one patient with AT, one patient with Artemis deficiency, one patient with DNA Ligase 4 deficiency, one patient with MHC class II deficiency, and one patient with combined immunodeficiency without a known molecular etiology. Of the previously reported and unreported cases, the majority share the diagnosis of a DNA repair disorder. Analysis of additional patients with this complication may clarify determinants of rubella pathogenesis, identify specific immune defects resulting in chronic infection, and may lead to defect-specific therapies.


Assuntos
Reparo do DNA/genética , Granuloma/complicações , Granuloma/virologia , Síndromes de Imunodeficiência/complicações , Vírus da Rubéola/patogenicidade , Dermatopatias/etiologia , Dermatopatias/virologia , Adolescente , Ataxia Telangiectasia/genética , Ataxia Telangiectasia/virologia , Criança , Pré-Escolar , Feminino , Granuloma/genética , Cabelo/anormalidades , Cabelo/virologia , Transplante de Células-Tronco Hematopoéticas/métodos , Doença de Hirschsprung/genética , Doença de Hirschsprung/virologia , Humanos , Síndromes de Imunodeficiência/genética , Síndromes de Imunodeficiência/virologia , Masculino , Síndrome de Quebra de Nijmegen/genética , Síndrome de Quebra de Nijmegen/virologia , Osteocondrodisplasias/congênito , Osteocondrodisplasias/genética , Osteocondrodisplasias/virologia , Rubéola (Sarampo Alemão)/genética , Rubéola (Sarampo Alemão)/virologia , Pele/virologia , Dermatopatias/genética , Doenças por Imunodeficiência Combinada Ligada ao Cromossomo X/genética , Doenças por Imunodeficiência Combinada Ligada ao Cromossomo X/virologia
11.
Asian J Psychiatr ; 39: 98-100, 2019 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-30599452

RESUMO

Lesion-based investigations of psychopathology have preceded contemporary network-neuroscience initiatives. However, brain-lesions detected in routine psychiatric practice are often considered incidental and therefore ignored. Here, we illustrate a strategy to combine individual subject-level lesion information with open-source normative functional-connectomics data to make putative, neuroscience-informed symptom interpretation. Specifically, we report a patient with left precuneus granulomatous lesion and seizures followed by two distinct symptoms - kinetopsia and delusions of nihilism and guilt - which had a differential treatment response. The lesion-based brain-mapping approach could identify correlated (default-mode) and anti-correlated (temporo-parieto-occipital) networks, which enabled a neurobiological formulation of these diverse clinical manifestations.


Assuntos
Neoplasias Encefálicas/complicações , Transtorno Depressivo Maior/complicações , Granuloma/complicações , Imagem por Ressonância Magnética/métodos , Lobo Parietal/diagnóstico por imagem , Esquizofrenia Paranoide/complicações , Anticonvulsivantes/uso terapêutico , Antidepressivos de Segunda Geração/uso terapêutico , Antipsicóticos/uso terapêutico , Mapeamento Encefálico/métodos , Neoplasias Encefálicas/diagnóstico por imagem , Neoplasias Encefálicas/terapia , Transtorno Depressivo Maior/psicologia , Transtorno Depressivo Maior/terapia , Diagnóstico Diferencial , Eletroconvulsoterapia , Feminino , Fluoxetina/uso terapêutico , Granuloma/diagnóstico por imagem , Granuloma/terapia , Humanos , Pessoa de Meia-Idade , Fenitoína/uso terapêutico , Fumarato de Quetiapina/uso terapêutico , Esquizofrenia Paranoide/psicologia , Esquizofrenia Paranoide/terapia , Convulsões/complicações , Convulsões/tratamento farmacológico
14.
Interact Cardiovasc Thorac Surg ; 28(3): 394-398, 2019 03 01.
Artigo em Inglês | MEDLINE | ID: mdl-30165660

RESUMO

OBJECTIVES: A small pulmonary granuloma (SPG) is often misdiagnosed as lung cancer in diabetic patients by positron emission tomography/computed tomography (PET/CT). The present study was conducted to investigate whether diabetes is the influencing factor and to determine other related factors that have an impact on the diagnostic results following PET/CT examination. METHODS: All clinical, imaging and pathological data of patients diagnosed with pulmonary nodules by PET/CT from January 2004 to December 2017 in our department were collected. Patients with an SPG who were wrongly diagnosed with lung cancer by PET/CT were enrolled (n = 79). The propensity score matching method was used to create a comparable control adenocarcinoma group (n = 395). Maximum standard uptake values, diabetes and fasting blood-glucose (FBG) were determined and analysed. RESULTS: The average maximum standard uptake values in the 2 groups were comparable (P = 0.801). Maximum standard uptake values in 5 subsections were not significantly different between the 2 groups (P = 0.135). The odds ratio (OR) of 3.326 [95% confidence interval (CI) 1.671-6.623] for diabetes favoured misdiagnosis and was statistically significant (P < 0.001). Furthermore, in patients with high FBG levels (≥7.0 mmol/l), the risk of misdiagnosis of SPG increased significantly compared with normal FBG level (OR 2.601, 95% CI 1.174-5.761; P = 0.015). CONCLUSIONS: Diabetes and high FBG level were the influencing factors in the false-positive results of lung cancer by PET/CT examination.


Assuntos
Diabetes Mellitus/diagnóstico , Erros de Diagnóstico , Granuloma/diagnóstico , Neoplasias Pulmonares/diagnóstico , Nódulos Pulmonares Múltiplos/diagnóstico , Tomografia Computadorizada com Tomografia por Emissão de Pósitrons/métodos , Feminino , Granuloma/complicações , Humanos , Masculino , Pessoa de Meia-Idade , Tomografia por Emissão de Pósitrons/métodos , Estudos Retrospectivos
16.
Med Hypotheses ; 122: 48-55, 2019 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-30593421

RESUMO

Nodular rheumatoid arthritis (RA) patients have raised rheumatoid factor (RF) and anti-citrullinated protein antibody (ACPA) levels, and are more likely to smoke than RA patients without nodules. Subcutaneous and pulmonary rheumatoid nodules (granulomas) frequently co-exist. Pulmonary rheumatoid nodules develop prior to RA development and have the immunological machinery to generate RF and ACPAs. Pulmonary granulomas have been observed in animal models exposed to cadmium (Cd) inhalation. Cigarette smoke increases pulmonary Cd exposure. It has been suggested that dust and cigarette smoke co-exposure increases localised pulmonary Cd adsorption. We hypothesise that subcutaneous nodular RA represents a distinct disease subtype induced by pulmonary rheumatoid nodule formation and the generation of high levels of RA associated autoantibodies initiated by Cd inhalation via cigarette smoke. Cohorts of RA patients attending rheumatology clinics in Cornwall, UK (total n = 504) were studied to determine the prevalence of nodular RA, with matched analysis (age, gender and social class) to compare urinary Cd, RF and ACPA levels stratifying for nodular disease and smoking. In cohort 1 45/303 (14.9%) of the RA patients under regular follow up had nodular disease. Of the RA smokers, 30/155 (19%) were nodular and of the RA non-smokers 15/148 (10%) were nodular. Smoking was significantly associated with nodular RA, odds ratio (OR) = 2.48 95% confidence interval (CI) 1.26-4.88, p = 0.008. Raised urinary Cd levels were significantly associated with nodular RA in non-dust exposed individuals, OR 2.26 (95% CI 1.08-4.73), p = 0.03 compared to dust exposed individuals, OR 0.78 (95% CI 0.35-1.76), p = 0.557, despite fewer pack years (py) at diagnosis (16 vs 20 py). Nodular RA smokers had significantly raised RF levels compared to RA smokers without nodular disease (median RF 171.5 (interquartile range (IQR) 48-394) vs median RF 31.7 (IQR 10.3-170.3), p < 0.00001). RF positivity was significantly more prevalent in nodular RA smokers compared to RA smokers without nodular disease (84/89 (94%) vs. 141/199 (71%), OR = 6.9 (95% CI 2.66-17.91), p < 0.00001). ACPA levels were also significantly raised in nodular smokers compared to non-nodular smokers (median ACPA 250 (IQR 145-426) vs 116 (1-257.5), p < 0.00001), as were ACPA positivity rates (83/89 (93%) vs 123/191 (64%), OR = 7.65 (95% CI 3.17-18.4), p < 0.0001). These pilot results support the hypothesis that nodular RA represents a distinct disease subtype initiated by cadmium inhalation, which we suggest induces pulmonary rheumatoid nodule formation and generation of RA-associated autoantibodies.


Assuntos
Artrite Reumatoide/diagnóstico , Autoanticorpos/química , Nódulo Pulmonar Solitário/diagnóstico , Adulto , Artrite Reumatoide/induzido quimicamente , Artrite Reumatoide/complicações , Cádmio/efeitos adversos , Estudos de Coortes , Feminino , Fibronectinas/química , Granuloma/induzido quimicamente , Granuloma/complicações , Humanos , Sistema Imunitário , Masculino , Pessoa de Meia-Idade , Modelos Estatísticos , Projetos Piloto , Fator Reumatoide/metabolismo , Reumatologia , Fatores de Risco , Fatores Sexuais , Fumar/efeitos adversos , Classe Social , Nódulo Pulmonar Solitário/complicações , Produtos do Tabaco/efeitos adversos
17.
Tuberk Toraks ; 66(3): 212-216, 2018 Sep.
Artigo em Turco | MEDLINE | ID: mdl-30479228

RESUMO

Introduction: Granulomatous lung disease (GLD) is caused by a wide range of conditions and it is challenge for pulmonologist. A detailed history of exposures is fundamental in GDL and has been found pivotal to reach a precise diagnosis. Materials and Methods: Between September 2014 and December 2016, the distribution of patients diagnosed with granulomatous lymphadenitis in the mediastinal/hilar lymph nodes by endobronchial ultrasound (EBUS) or mediastinoscopy was analyzed. To be listed as 'confident', a diagnosis of sarcoidosis required compatible histological, radiological and clinical findings in conjunction with negative cultures. Infectious entities listed as 'confident' had either microorganisms in tissue section, positive culture, positive serology or positive antigen detection in a consistent clinical pathological setting. Result: Granulomatous lymphadenitis was detected in 110 patients. The included 110 cases consisted of 70.9% women and median age of 53 (range 44-61) years. The final diagnosis of the patients was accepted to be sarcoidosis in 79 (71.8%), sarcoid like granulomas in 7 (6.4%), tuberculosis in 4 (3.6%), silicosis in 4 (3.6%), drug-associated granuloma in 2 (1.8%), hypersensitivity pneumonitis in 1 (0.9%), Chron disease in 1 (0.9%), unspecified in 12 (10.9%). Three patients were classified as tuberculosis based on culture. Conclusions: In this study, we found that the most common cause of granulomatous lymphadenitis was sarcoidosis. Contrary to expectations, the number of patients diagnosed with tuberculosis was very low.


Assuntos
Granuloma/patologia , Linfadenite/patologia , Mediastino/patologia , Adulto , Feminino , Granuloma/complicações , Granuloma/diagnóstico por imagem , Humanos , Linfonodos/patologia , Linfadenite/complicações , Linfadenite/diagnóstico por imagem , Masculino , Mediastino/diagnóstico por imagem , Pessoa de Meia-Idade , Ultrassonografia
18.
J Coll Physicians Surg Pak ; 28(11): 885-887, 2018 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-30369386

RESUMO

Acute granulomatous interstitial nephritis (GIN) is a rare cause of acute kidney injury (AKI) but treatable. It is present in 0.5 to 0.9% of native renal biopsies. Treatment with moderate dosage of steroids is associated with good prognosis. We herein review a case of a 48-year lady having AKI following drug-induced damage [non-steroidal anti-inflammatory drugs (NSAIDs) and antibiotics]. Her renal functions did not improve after one month of drug withdrawal, so renal biopsy was done that showed acute GIN. She was treated with intravenous then oral steroids that completely resolved AKI.


Assuntos
Lesão Renal Aguda/patologia , Granuloma/complicações , Rim/patologia , Nefrite Intersticial/complicações , Lesão Renal Aguda/etiologia , Biópsia , Creatinina/sangue , Feminino , Humanos , Rim/diagnóstico por imagem , Pessoa de Meia-Idade , Náusea/etiologia , Nefrite Intersticial/patologia , Ultrassonografia , Vômito/etiologia
20.
J Med Case Rep ; 12(1): 299, 2018 Oct 15.
Artigo em Inglês | MEDLINE | ID: mdl-30318016

RESUMO

BACKGROUND: Lymphadenopathy is not an uncommon presentation of adult onset Still's disease: it is present in up to two thirds of patients with adult onset Still's disease. The characteristic appearance of lymphadenopathy is described as intense, paracortical immunoblastic hyperplasia. Changes in light microscopy may resemble lymphoma, but immunohistochemistry reveals a benign polyclonal B cell hyperplasia. CASE PRESENTATION: We describe a 67-year-old Sri Lankan woman who manifested relapsing prolonged fever, raised inflammatory markers, arthralgia, myalgia, transient skin rash, and cervical lymphadenopathy histologically characterized by noncaseating granulomatous adenitis with central suppuration. Due to the fact of high prevalence of tuberculosis in the region, an extensive diagnostic evaluation was done to exclude the possibility of extrapulmonary tuberculosis; unsuccessful therapeutic trials of complete antituberculosis regime reliably excluded the possibility of tuberculosis and strengthened the diagnostic validity. Disease flares were characterized by systemic inflammatory response syndrome with immediate clinical and laboratory response to corticosteroids. After systematic diagnostic workup which ruled out possible malignant, rheumatic, or autoimmune diseases and infections previously described as causes of granulomatous adenitis, our patient was diagnosed as having adult onset Still's disease based on Yamaguchi criteria. She required a trial of indomethacin followed by methylprednisolone pulse therapy and long-term maintenance steroid therapy without steroid-sparing immunosuppressive agents or biological disease-modifying antirheumatic drugs. She achieved full disease remission in 3 months. Reevaluation after 6 months and 1 year did not reveal residual disease activity. CONCLUSIONS: To the best of our knowledge this is the first report of suppurative noncaseating granulomatous lymphadenitis attributed to adult onset Still's disease among Asian or South Asian ethnicities and it is also rarely reported among Europeans and North Americans. It is an extremely challenging situation to diagnose Still's disease with granulomatous lymphadenitis where tuberculosis is highly prevalent. This case highlights the importance of consideration of adult onset Still's disease as a potential diagnosis in a compatible clinical context in the presence of noncaseating granulomatous adenitis and indicates that one should not be misled into a diagnosis of tuberculosis by the fact of the high prevalence of tuberculosis, however, the exclusion of other diagnoses is a prerequisite.


Assuntos
Granuloma/complicações , Granuloma/patologia , Linfadenite/complicações , Linfadenite/patologia , Doença de Still de Início Tardio/complicações , Tuberculose , Idoso , Anti-Inflamatórios/uso terapêutico , Anti-Inflamatórios não Esteroides/uso terapêutico , Diagnóstico Diferencial , Feminino , Granuloma/tratamento farmacológico , Humanos , Indometacina/uso terapêutico , Linfadenite/tratamento farmacológico , Metilprednisolona/uso terapêutico , Supuração
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