Juvenile and adult xanthogranuloma: A 30-year single-center experience and review of the disorder and its relationship to other histiocytoses

Salari, Behzad; Dehner, Louis P.

doi:10.1016/j.anndiagpath.2022.151940

Cited by 13 publications

(5 citation statements)

References 120 publications

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“…10 Both disease entities follow a chronic course and may also present with systemic involvement of bone, internal organs, and soft tissue. 11 Occasionally, the distinction between LCH and JXG can be challenging, 10,12 and biopsies for H&E and immunohistochemistry remain necessary to distinguish these 2 entities. 9,13,14 Although these represent 2 distinct entities, occasional coexistence is possible, but rare.…”

Section: Discussionmentioning

confidence: 99%

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Langerhans Cell Histiocytosis Evolving into Juvenile Xanthogranuloma: Two Linked Entities

Shaker

Sangüeza

2023

The American Journal of Dermatopathology

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Background:Langerhans cell histiocytosis (LCH) represents a myeloid clonal proliferation that involves the skin and other organs. Occasionally, cases of LCH evolve into juvenile xanthogranuloma (JXG).Case Presentation:A 7-month-old boy presented with an itchy, flaky rash resembling seborrheic dermatitis affecting the scalp and eyebrows. The lesions started at 2 months old. On physical examination, there were reddish/brown lesions on the trunk, denuded areas on the groin and neck, and a large lesion behind his bottom teeth. In addition, there were thick white plaques in his mouth and thick whitish material in both ears. A skin biopsy showed features of LCH. Radiologic examination demonstrated several osteolytic lesions. Chemotherapy produced marked improvement. A few months later, the patient developed lesions with clinical and histologic features of XG.Conclusion:A possible association between LCH and XG is explained by lineage maturation development. Chemotherapy may play a role in modifying the production of cytokines that enhance the transformation or ‘maturation’ of Langerhans cells into multinucleated macrophages (Touton cells) characteristic of a more favorable proliferative inflammatory condition.

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Section: Discussionmentioning

confidence: 99%

“…Occasionally, the distinction between LCH and JXG can be challenging, 10,12 and biopsies for H&E and immunohistochemistry remain necessary to distinguish these 2 entities. 9,13,14 Although these represent 2 distinct entities, occasional coexistence is possible, but rare.…”

Section: Discussionmentioning

confidence: 99%

Langerhans Cell Histiocytosis Evolving into Juvenile Xanthogranuloma: Two Linked Entities

Shaker

Sangüeza

2023

The American Journal of Dermatopathology

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“…The nuclei of juvenile xanthogranuloma are round to oval and are negative for S100 and CD1a [17] . A combination of CD11c, CD4, CD1a, and CD163 or CD68 staining provides a more specific diagnostic outcome to distinguish JXG from other histiocytic diseases [18] . In RDD, enlarged cervical lymph nodes are a classic finding.…”

Section: Discussionmentioning

confidence: 99%

ALK-positive histiocytosis involving the cavernous sinus: A deceptive radiologic mimic of meningioma

Alizadeh¹,

Ravindran²,

Chkheidze³

et al. 2023

Radiology Case Reports

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“…These histiocytes are positive for CD68, CD163, CD4, CD11c, and factor XIIIa, but are generally negative for CD1a and S100, although S100 expression was reportedly observed in up to 32% of JXGs. 48 In cases of typical JXG, diagnosis of ALK-positive histiocytosis is recommended if ALK1 immunostaining or gene rearrangement is detected. 18 The morphology, immunophenotype, genetics, and clinical presentations of JXG may also overlap with ECD.…”

Section: Jxgmentioning

confidence: 99%

Pediatric Histiocytic Disorders: Morphology, Immunophenotype and Genetics

Cheng,

Zhu,

Pan

et al. 2023

J Clin Transl Pathol

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Histiocytic disorders are rare in childhood and often present with a wide spectrum of histological and clinical symptoms, making their diagnosis challenging. The pathological classification of histiocytic disorders has been evolving during the last few decades, and new diagnostic criteria and classifications have been recently updated. Herein, we review pediatric histiocytic disorders, focusing on the pathological features of morphology, immunophenotype, and newly discovered molecular data. These insights shed light on the pathogenesis of these disorders and may become therapeutic biomarkers.

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Juvenile and adult xanthogranuloma: A 30-year single-center experience and review of the disorder and its relationship to other histiocytoses

Cited by 13 publications

References 120 publications

Langerhans Cell Histiocytosis Evolving into Juvenile Xanthogranuloma: Two Linked Entities

Langerhans Cell Histiocytosis Evolving into Juvenile Xanthogranuloma: Two Linked Entities

ALK-positive histiocytosis involving the cavernous sinus: A deceptive radiologic mimic of meningioma

Pediatric Histiocytic Disorders: Morphology, Immunophenotype and Genetics

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