Juvenile xanthogranuloma: Case report and review of the literature

Cypel, TKS; Rm, Zuker

doi:10.4172/plastic-surgery.1000572

Cited by 12 publications

(20 citation statements)

References 8 publications

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“…Los pacientes con mayor riesgo de manifestar XJ extracutáneos son los menores de dos años con XJ múltiples. 1 El cuadro de XJ diseminado, aunque es poco frecuente, es muy grave y puede poner en riesgo la vida del paciente, ya que presenta un compromiso cutáneo y visceral. Involucra diferentes órganos: tejidos blandos, músculos, ojos, pulmones, hígado, bazo, sistema nervioso central y nasofaringe.…”

Section: Discussionunclassified

Xantogranuloma juvenil múltiple: Variante inusual en un lactante

Pineda

Le-Bert

Aspée

2017

Rev. chil. dermatol

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ResumenEl Xantogranuloma Juvenil (XJ) es un tumor benigno de la infancia, constituido por histiocitos progresivamente lipidizados, en ausencia de anomalías metabólicas.Se reporta un caso de XJ múltiple en un lactante.Paciente masculino de 1 año y 11 meses de edad, sin antecedentes mórbidos. Consultó por lesiones de 15 meses de evolución en cuero cabelludo. Al examen físico destacaban dos pápulas amarillentas y tres máculas anaranjadas en cuero cabelludo. A la dermatoscopía se apreciaba un color amarillonaranja homogéneo. La biopsia incisional demostró infiltrado de histiocitos en dermis y células de Touton. A la inmunohistoquímica, los histiocitos fueron CD68 positivo, S100 negativo y CD1a negativo. Los exámenes de laboratorio y la evaluación por oftalmología no evidenciaron anormalidades.El XJ es un tumor benigno, siendo la forma más común de Histiocitosis no Langerhans. El 90% de las veces ocurre en etapas tempranas de la vida. Se caracteriza por una pápula o nódulo asintomático, amarillo-anaranjado, que compromete cabeza, cuello y/o tronco superior. Hasta el 82% de los casos se manifiesta de manera única. El ojo es el sitio extracutáneo más frecuentemente comprometido. La dermatoscopía describe un patrón de "puesta de sol". Su pronóstico es bueno y tiende a involucionar a los 3-6 años desde su aparición.Se presenta este caso dado que el XJ múltiple es menos frecuente de observar que su forma única y para recordar que su asociación con neurofibromatosis tipo 1 predispone a un mayor riesgo de desarrollar una leucemia mieloide crónica infantil. A case of multiple JX is reported in an infant.Male patient of 1 year and 11 months, without morbid history. Checking injuries in scalp of 15 months of evolution. At physical examination highlights two yellow papules and three orange macules in the scalp. Dermatoscopy shown a homogeneous yellow-orange color. The incisional biopsy showed infiltration of histiocytes in the dermis and Touton cells. At immunohistochemistry, histiocytes were CD68 positive, S100 negative and CD1a negative. Laboratory tests and evaluation by Ophthalmology showed no abnormalities.The JX is a benign tumor, been the most common form nonLangerhans histiocytosis. This taking place 90% of the time in early life, and characterized by yellow-orange asymptomatic papule or nodule, which undertake head, neck and / or upper body. Up to 82% of cases appears uniquely. The eye is the most frequently committed extracutaneous site. The dermoscopy describes a pattern of "setting sun". His prognosis is good and tends to return to the 3-6 years since their appearance.We present this case because the multiple JX is less common to observe that this singular form, and to remember that their association with neurofibromatosis type 1 predisposes to increased risk of developing childhood chronic myeloid leukemia.

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Section: Discussionunclassified

Xantogranuloma juvenil múltiple: Variante inusual en un lactante

Pineda

Le-Bert

Aspée

2017

Rev. chil. dermatol

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“…Histopathologically, xanthogranulomas show a dense infiltrate of histiocytes [3]. The lesions also have a variable number of Touton giant cells and principally perivascular and perilesional inflammatory cells [3,4]. Immuno histochemically, the lesion is positive for CD68, factor XIIIa, fascin, and alpha-antitrypsin and negative for S-100, CD1a, beta-actin, and desmin [1].…”

Section: Introductionmentioning

confidence: 96%

“…One other very rare diagnostic possibility that should be considered is a periapical granuloma with Touton-like giant cells. When present in the oral cavity, xanthogranulomas can appear similar to several more common entities [2][3][4][5][6]. Therefore, this case demonstrates the importance of biopsy in definitive diagnosis of oral lesions and lesions found at the apices of previously endodontically treated teeth.…”

Section: Introductionmentioning

confidence: 99%

“…Immuno histochemically, the lesion is positive for CD68, factor XIIIa, fascin, and alpha-antitrypsin and negative for S-100, CD1a, beta-actin, and desmin [1]. Most cases of cutaneous xanthogranuloma do not require treatment and have a favorable prognosis because the lesions spontaneously regress [1][2][3][4][5]. However, spontaneous regression has only been reported in the case of one oral lesion [2].…”

Section: Introductionmentioning

confidence: 99%

“…While the etiology is unknown, this entity is thought to be reactive [2]. Histopathologically, xanthogranulomas show a dense infiltrate of histiocytes [3]. The lesions also have a variable number of Touton giant cells and principally perivascular and perilesional inflammatory cells [3,4].…”

Section: Introductionmentioning

confidence: 99%

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A Nonhealing Periapical Radiolucency

Vargo

Bilodeau

Jaradat

et al. 2016

JDHODT

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Background: This case discusses a radiolucent lesion in the anterior mandible, which initially was thought to be due to endodontic failure. However, the final diagnosis was xanthogranuloma. Case description:We have reported a case of xanthogranuloma in the mandible found at the apices of two endodontically treated teeth. When present in the oral cavity, it can appear similar to several more common entities. While the radiolucent periapical lesion was thought to be the result of non-healing root canal therapy, the excisional biopsy yielded the diagnosis of xanthogranuloma. Removal of the lesion has resulted in excellent healing, as the surgeon expected. Practical implications:This case demonstrates the importance of biopsy in definitive diagnosis of oral lesions and lesions found at the apices of previously endodontically treated teeth.

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