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1.
Ned Tijdschr Geneeskd ; 1642021 03 02.
Artigo em Holandês | MEDLINE | ID: mdl-33651490

RESUMO

BACKGROUND: The levels of carbohydrate antigen 19-9 (CA19-9) are often tested to stage pancreatobiliary tumours or to monitor the effects of treatment of such tumours. Benign diseases can also be associated with elevated CA 19-9 levels, but extreme elevation is rare in such cases. CASE DESCRIPTION: An 84-year-old man was diagnosed with cholecystitis at the emergency department and received conservative treatment with antibiotics for this. After 5 weeks, the patient came to the outpatient clinic with unexplained icterus. Additional imaging revealed indications of malignancy and the CA19-9 concentration was 4240 kU/l. We suspected pancreatobiliary malignancy. However, ERCP revealed obstructive choledocholithiasis and we subsequently performed laparoscopic cholecystectomy. Based on histopathology examination of the gallbladder, a diagnosis of xanthogranulomatous cholecystitis was made. After surgery, CA19-9 levels decreased and the patient swiftly recovered. CONCLUSION: Significantly elevated CA19-9 levels are not proof of pancreatobiliary malignancy. Our case underscores that CA19-9 is not an ideal tumour marker and that the results of CA19-9 tests should therefore be interpreted with some caution.


Assuntos
Antígeno CA-19-9/sangue , Colecistite/diagnóstico , Xantomatose/diagnóstico , Idoso de 80 Anos ou mais , Neoplasias do Sistema Biliar/diagnóstico , Colecistectomia Laparoscópica , Colecistite/cirurgia , Diagnóstico Diferencial , Humanos , Masculino , Neoplasias Pancreáticas/diagnóstico , Xantomatose/cirurgia
3.
World Neurosurg ; 140: 56-59, 2020 08.
Artigo em Inglês | MEDLINE | ID: mdl-32437994

RESUMO

BACKGROUND: Xanthomas are benign lipomatous deposits that can be found systemically in various tissues including bones. Their presence in the skull remains a rare entity. Despite their benign characteristics, imaging modalities are often unable to distinguish them from malignant lesions. This leads to a diagnostic dilemma in patients with underlying malignancy. This case report highlights such a case where clinical history of prostate cancer and image findings were concordant with that of metastatic deposit in the parietal skull region. CASE DESCRIPTION: This 65-year-old gentleman was diagnosed with prostatic adenocarcinoma. During systemic workup for his tumor, he was found to have a right parietal skull lesion. Magnetic resonance imaging of the brain, as well as a bone scan, were consistent with that of a metastatic deposit. As treatment would be drastically affected by the diagnosis, an excision biopsy was performed. The histology was consistent with that of a bone xanthoma. CONCLUSIONS: Xanthomas are benign lesions that can be seen deposited in appendicular and axial skeleton. Skull lesions are rare with most case descriptions focusing on their presence in the frontoorbital regions and mandible and temporal bone. They usually have a benign course but may present with symptoms due to localized mass effect. Surgical intervention and histologic diagnosis may still be required in these lesions due to their lack of imaging characteristics that confirm their diagnosis through noninvasive methods.


Assuntos
Crânio/patologia , Xantomatose/patologia , Idoso , Neoplasias Ósseas/diagnóstico , Neoplasias Ósseas/patologia , Diagnóstico Diferencial , Humanos , Masculino , Metástase Neoplásica/diagnóstico , Metástase Neoplásica/patologia , Neoplasias da Próstata/patologia , Xantomatose/diagnóstico
4.
World Neurosurg ; 138: 27-34, 2020 06.
Artigo em Inglês | MEDLINE | ID: mdl-32081821

RESUMO

BACKGROUND: Xanthomatous hypophysitis (XH) is the rarest histologic type of primary hypophysitis. It is nonlymphocytic and characterized by an infiltration of the pituitary gland by lipid-laden histiocytes and macrophages. The clinical and radiologic features overlap heavily with pituitary adenomas and are prone to misdiagnosis. We describe a rare case of XH recurrence at 1 year, treated surgically. Moreover, we provide an updated review of the literature to further elucidate useful management and detection strategies. CASE DESCRIPTION: A 45-year-old woman presented with a history of menstrual irregularity for 9 months, amenorrhea, galactorrhea, and headache for 2 months duration. Preoperative endocrinologic studies showed increased prolactin levels. Magnetic resonance imaging of the sella showed a cystic lesion with suprasellar extension suggestive of a pituitary adenoma. The patient underwent transsphenoidal resection, showing a thick yellowish colloidal material. Histopathology showed necrotic tissue with no definitive diagnosis and no identified microorganisms. At 2 months after surgery, prolactin levels and menstrual cycle normalized. At 1 year after surgery, her menstrual cycle again became irregular. Repeat magnetic resonance imaging showed a recurrent mass and a second transsphenoidal resection was undertaken. Repeat histopathology was consistent with XH. The patient is 9 years postoperative and is doing well without evidence of recurrence. CONCLUSIONS: XH presents similarly to nonfunctional adenomas and diagnosis remains difficult without surgical pathology, requiring meticulous immunohistochemistry to prevent misdiagnosis. Thus, XH should be considered as a rare cause in the differential of disease of the sellar region. Management can parallel that of pituitary neoplasm, with a focus on radiosurgery, re-resection, and high-dose steroids.


Assuntos
Adenoma/diagnóstico , Hipofisite/diagnóstico , Hipofisite/patologia , Neoplasias Hipofisárias/diagnóstico , Xantomatose/diagnóstico , Diagnóstico Diferencial , Feminino , Humanos , Hipofisite/cirurgia , Pessoa de Meia-Idade , Recidiva , Xantomatose/patologia , Xantomatose/cirurgia
7.
World Neurosurg ; 133: e241-e251, 2020 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-31505289

RESUMO

BACKGROUND: Cystic sellar masses (CSMs) pose diagnostic and therapeutic challenges associated with subtotal cyst wall resection, cerebrospinal fluid (CSF) leak repair, and disease recurrence. Current magnetic resonance imaging (MRI) interpretation often cannot reliably differentiate CSMs, mandating adaptable intraoperative strategies. We reviewed our diagnostic and therapeutic experience after endoscopic endonasal approaches (EEAs) for CSMs. METHODS: A retrospective record review of patients with CSM managed via EEA at the University of Southern California from 2011 to 2018 was conducted. Patient demographics, preoperative characteristics, surgical details, pathologic findings, and postoperative outcomes were assessed. RESULTS: Analysis included 47 patients (mean age, 43.2 years); of these, 78.7% were women. Preoperative symptoms included headache (76.6%) and vision loss (42.6%). Histologically verified sellar pathology included 27 Rathke cleft cysts (RCCs) (57.4%), 17 cystic pituitary adenomas (CPAs) (36.2%), 2 arachnoid cysts (4.3%), and 1 xanthogranuloma (2.1%). Twelve patients (70.6%) with CPAs underwent complete resection and 5 (29.4%) underwent subtotal resection. All 27 patients with RCC and 2 patients with arachnoid cyst underwent complete fenestration and drainage. One xanthogranuloma was completely resected. There were 14 intraoperative (29.8%) and 4 postoperative CSF leaks (8.5%). Headaches, vision, and endocrinopathy improved in 69.2%, 80.0%, and 33.3% of patients with CPA and 73.9%, 71.4%, and 40.9% of patients with RCC, respectively. There were 2 RCC recurrences and 1 CPA recurrence over the follow-up period. CONCLUSIONS: Surgeons must prepare for versatile management strategies of CSMs based on pretest probability associated with MRI and intraoperative findings. Outcomes after EEA for CSMs show low complication profiles and excellent rates of headache and visual improvement, albeit lower rates of endocrine normalization.


Assuntos
Cistos do Sistema Nervoso Central/cirurgia , Cirurgia Endoscópica por Orifício Natural/métodos , Neuroimagem/métodos , Sela Túrcica , Adenoma/complicações , Adenoma/diagnóstico , Adenoma/diagnóstico por imagem , Adenoma/cirurgia , Adulto , Cistos do Sistema Nervoso Central/complicações , Cistos do Sistema Nervoso Central/diagnóstico , Cistos do Sistema Nervoso Central/diagnóstico por imagem , Vazamento de Líquido Cefalorraquidiano/etiologia , Drenagem , Fadiga/etiologia , Feminino , Cefaleia/etiologia , Humanos , Hipopituitarismo/etiologia , Complicações Intraoperatórias/etiologia , Masculino , Pessoa de Meia-Idade , Cavidade Nasal , Neoplasias Hipofisárias/complicações , Neoplasias Hipofisárias/diagnóstico , Neoplasias Hipofisárias/diagnóstico por imagem , Neoplasias Hipofisárias/cirurgia , Complicações Pós-Operatórias/etiologia , Estudos Retrospectivos , Sela Túrcica/diagnóstico por imagem , Sela Túrcica/cirurgia , Transtornos da Visão/etiologia , Xantomatose/complicações , Xantomatose/diagnóstico , Xantomatose/diagnóstico por imagem , Xantomatose/cirurgia
8.
Indian J Dermatol Venereol Leprol ; 86(2): 158-161, 2020.
Artigo em Inglês | MEDLINE | ID: mdl-30333358

RESUMO

Rosai-Dorfman disease or sinus histiocytosis with massive lymphadenopathy is a benign lympho-histiocytic proliferative disorder initially described with bilateral painless lymphadenopathy (90 %), fever, leukocytosis, elevated ESR, anemia, and polyclonal hypergammaglobulinemia (90 %). Extranodal forms occur in 43% of cases, the skin being the most common site. Around 10% of patients have skin lesions and in 3%, the disease is limited exclusively to the skin. Here, we report a male patient who presented with pure cutaneous lesions which mimic eruptive xanthoma clinically. However, the diagnosis was established histo pathologically. So, high level of clinical suspension is critical to avoid missing such cases.


Assuntos
Histiocitose Sinusal/diagnóstico , Dermatopatias/diagnóstico , Xantomatose/diagnóstico , Diagnóstico Diferencial , Histiocitose Sinusal/complicações , Humanos , Masculino , Pessoa de Meia-Idade , Dermatopatias/complicações , Xantomatose/complicações
10.
Rev. chil. ortop. traumatol ; 61(3): 112-115, 2020. ilus
Artigo em Espanhol | LILACS | ID: biblio-1177827

RESUMO

OBJETIVO Presentación de un inusual caso de xantomatosis cerebrotendinosa en un paciente de edad cuya primera manifestación fueron xantomas bilaterales del tendón de Aquiles. MATERIAL Y MÉTODOS Mujer de 62 años, que presenta tumoraciones, que presenta tumoraciones sólidas y polilobuladas, en la cara posterior de ambos tendones de Aquiles de 8 años de evolución. El diagnóstico se realizó mediante el hallazgo de hiperlipidemia y estudio genético. Se realió la exóresis quirúrgica parcial de las tumoraciones. RESULTADOS A los 5 años de la cirugía del pie izquierdo y 4 años del pie derecho la paciente estaba asintomática. Presentaba una fuerza para la flexión plantar bilateral de 5/5, pudiendo caminar y subir escaleras sin molestias. Presentaba una escala AOFAS de 85 y 90 puntos en el pie izquierdo y derecho, respectivamente. No hubo recidivas. DISCUSIÓN Los xantomas son depósitos de colesterol en el tejido conectivo de la piel, tendones o fascia, como resultado de una hiperlipoproteinemia. La importancia del caso radica en su sospecha diagnóstica, ya que la xantomatosis cerebrotendinosa suele manifestarse en pacientes de menos de 30 años de edad y en los que se ha recomendado la resección radical de las tumoraciones, e incluso del tendón, debido a las frecuentes recidivas. CONCLUSIÓN En pacientes de mayor edad con lesiones que infiltran el tendón, se puede optar por un tratamiento menos agresivo con un buen resultado clínico.


OBJETIVE Presentation of an unusual case of cerebrotendinous xanthomatosis in an elderly patient whose first manifestation was bilateral Achilles tendon xanthomas. MATERIAL AND METHODS 62-year-old woman presenting solid and polylobed tumors on the posterior aspect of both Achilles tendons for eight years. The diagnosis was made by means of hyperlipidemia and a genetic study. Surgical partial excision of the tumors was performed. RESULTS Five years after surgery on the left foot and four years after the right foot, the patient was asymptomatic. Bilateral plantar flexion force was 5/5, The patient was able to walk and climb stairs without discomfort. AOFAS score was 85 and 90 on the left and right feet, respectively. There were no recurrences. DISCUSSION Xanthomatosis is a genetic alteration with deposits of cholesterol in connective tissue of the skin, tendons or fascia, because of hyperlipoproteinemia. The importance of the present case lies in its diagnostic suspicion, since cerebrotendinous xanthomatosis manifests usually in patients under 30 years of age and in whom radical resection of tumors, and even of the tendon, has been recommended due to frequent recurrences . CONCLUSION In older patients with tumors that infiltrate the tendon, a less aggressive treatment can be chosen with a good clinical result.


Assuntos
Humanos , Feminino , Pessoa de Meia-Idade , Tendão do Calcâneo , Xantomatose/cirurgia , Xantomatose/diagnóstico , Xantomatose Cerebrotendinosa/complicações , Tendinopatia/cirurgia , Tendinopatia/diagnóstico , Imageamento por Ressonância Magnética , Xantomatose/etiologia , Xantomatose/diagnóstico por imagem , Tendinopatia/etiologia , Tendinopatia/diagnóstico por imagem , Hiperlipidemias
11.
Arch Pediatr ; 26(7): 411-414, 2019 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-31630906

RESUMO

Xanthomas are rare bone tumors that occur more often in patients with hyperlipidemia or metabolic disorders. We report the case of a 9-year-old child presenting with right shoulder pain after a trauma. A benign osteolytic lesion was found in the proximal humeral metaphysis. Curettage of the lesion was performed. Histologic findings confirmed intraosseous xanthoma. Although there was no evidence of dyslipidemia or metabolic disease, the patient suffered local recurrence at the 8-month follow-up, requiring a second surgery with bone grafting. This difficult and rare diagnosis must be kept in mind with osteolytic lesions.


Assuntos
Doenças Ósseas/diagnóstico , Úmero , Xantomatose/diagnóstico , Doenças Ósseas/patologia , Doenças Ósseas/cirurgia , Criança , Humanos , Úmero/patologia , Úmero/cirurgia , Hiperlipidemias , Masculino , Recidiva , Xantomatose/patologia , Xantomatose/cirurgia
13.
J Oral Pathol Med ; 48(10): 935-942, 2019 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-31355943

RESUMO

BACKGROUND: The aim of the present study was to report the clinicopathologic, radiographic and immunohistochemical features of five South American cases of intraosseous xanthomas of the mandible and to compare them to those detected in a literature review. METHODS: Clinical data were collected from the records of three Oral and Maxillofacial Pathology services in South America and compared with those compiled from a literature review based on a search of three electronic databases (PubMed, Web of Science and Scopus). All cases were evaluated by haematoxylin and eosin staining and immunohistochemistry for CD68 and S-100. RESULTS: The series comprised four females (80%) and one male (20%) with a mean age of 23.3 ± 10.9 years (range: 13-45 years). In four cases, there was involvement of the posterior region of the mandible (80%). The lesions presented radiographically as unilocular (60%) radiolucencies with punched-out margin (80%). All cases predominantly consisted of CD68-positive and S-100-negative xanthomatous cells. No recurrences were observed after curettage, with a median follow-up of 27 months. CONCLUSION: Intraosseous xanthoma of the jaws is a rare benign disorder. We report here five additional cases affecting the mandible, for a total of 36 cases of the jaws reported in the literature. Overall, this lesion has predilection for posterior sites of the mandible of asymptomatic young adults.


Assuntos
Neoplasias Ósseas/diagnóstico , Mandíbula/patologia , Xantomatose/diagnóstico , Adolescente , Adulto , Feminino , Humanos , Imuno-Histoquímica , Masculino , Pessoa de Meia-Idade , Adulto Jovem
15.
BMJ Case Rep ; 12(7)2019 Jul 22.
Artigo em Inglês | MEDLINE | ID: mdl-31337625

RESUMO

We present the case of a 34-year-old woman who was diagnosed to have adenocarcinoma of the caecum based on the clinical, radiological, histopathological and intraoperative findings. However, postoperative histopathology showed only features of xanthogranulomatous inflammation without any evidence of malignancy. This benign chronic inflammatory condition could present as a histological surprise. It is important for both surgeons and pathologists alike to be aware of this.


Assuntos
Adenocarcinoma/patologia , Colo Ascendente/patologia , Doenças do Colo/patologia , Neoplasias do Colo/patologia , Erros de Diagnóstico , Granuloma/patologia , Inflamação/patologia , Xantomatose/patologia , Adenocarcinoma/diagnóstico , Adenocarcinoma/cirurgia , Adulto , Biópsia , Colectomia , Doenças do Colo/diagnóstico , Neoplasias do Colo/diagnóstico , Neoplasias do Colo/cirurgia , Feminino , Granuloma/diagnóstico , Humanos , Inflamação/diagnóstico , Xantomatose/diagnóstico
16.
BMJ Case Rep ; 12(7)2019 Jul 01.
Artigo em Inglês | MEDLINE | ID: mdl-31266759

RESUMO

A case of multiple xanthogranulomas located bilaterally on arms, armpits, legs, trunk, abdomen, scalp, neck and plantar surfaces in a 25-year-old man is reported. The patient was evaluated by polarised light dermoscopy and reflectance confocal microscopy (RCM) that showed peculiar aspects. In particular, dermoscopy showed a homogeneous orange-yellowish hue that is related to the histopathological presence of foamy histiocytes and Touton giant cells; the last typically appeared at RCM as peculiar giant cells surrounded by highly refractive ring due to the cytoplasm rich of lipids. Laboratory investigations, including routine haematological examination, liver and renal function tests, serum lipid and sugar levels were normal. The patient started isotretinoin 20 mg once daily. After 6 months, some lesions flattened, leading to yellowish or hyperpigmented macules, but new lesions raised up.


Assuntos
Dermoscopia/métodos , Neoplasias Cutâneas/diagnóstico , Xantomatose/diagnóstico , Adulto , Fármacos Dermatológicos/uso terapêutico , Diagnóstico Diferencial , Humanos , Isotretinoína/uso terapêutico , Masculino , Microscopia Confocal , Neoplasias Cutâneas/tratamento farmacológico , Xantomatose/tratamento farmacológico
18.
Diagn Cytopathol ; 47(9): 939-942, 2019 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-31169968

RESUMO

Xanthomas are papulonodular skin lesions present in lipoprotein metabolism disorders, which result in cholesterol deposits in subcutaneous tissue, tendons, ligaments, periosteum, etc. A 11-year-old male presented with multiple soft tissue swellings, prominently over joints. Fine-needle aspiration (FNA) from multiple sites had similar appearance with foamy histocytes and giant cells. Oil Red O and polarized microscopy were also positive for fat. We describe an unusual case of tendinous and tuberous xanthoma diagnosed by cytology. Acquaintance with fine-needle aspiration cytology findings in xanthomas can help to avoid the need of surgical biopsy, as xanthomas can regress on medical therapy alone.


Assuntos
Neoplasias Cutâneas , Xantomatose , Biópsia por Agulha Fina , Criança , Humanos , Masculino , Neoplasias Cutâneas/diagnóstico , Neoplasias Cutâneas/metabolismo , Neoplasias Cutâneas/patologia , Xantomatose/diagnóstico , Xantomatose/metabolismo , Xantomatose/patologia
19.
Hautarzt ; 70(6): 438-442, 2019 Jun.
Artigo em Alemão | MEDLINE | ID: mdl-31076815

RESUMO

Cutaneous xanthomas may develop in patients with lipid abnormalities, chronic inflammatory diseases or cancer. Especially myeloproliferative diseases may be accompanied by dystrophic xanthomas. We report on a normolipemic patient with cutaneous xanthomas who was subsequently diagnosed with mycosis fungoides. In addition to illustrative clinical and histopathological images, we present an overview of reports on dystrophic xanthomas in mycosis fungoides, the most frequent cutaneous T­cell lymphoma.


Assuntos
Linfoma Cutâneo de Células T , Micose Fungoide/diagnóstico , Xantomatose/diagnóstico , Humanos , Neoplasias Cutâneas
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