Xanthoma disseminatum with intracranial involvement: case report and literature review

Oka, Masahiro; Oniki, Shuntaro; Komatsu, Maiko; Ikeda, Tetsuya; Matsuo, Masafumi; Miyamoto, Yoshitomo; Akita, Hozuka; Kondo, Takeshi; Nishigori, Chikako

doi:10.1111/j.1365-4632.2009.04208.x

Cited by 17 publications

(33 citation statements)

References 25 publications

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“…In fact, the ECD [6,11,12], xanthogranuloma disseminatum [1,2,3,4,5,13] and juvenile xanthogranulomas [14] may have similar distributions in their CNS lesions. Finally, the XD diagnosis in the present patient is supported by the pattern of skin lesions and the absence of other symptoms/involvement as in the ECD.…”

Section: Discussionmentioning

confidence: 99%

“…Three clinical patterns of XD have been described [3]: a persistent form, a less common progressive form and a rare spontaneously regressive form [12]. CNS involvement with diabetes insipidus has been associated with the progressive clinical pattern, and the involvement of other areas occurs in less than 5% of XD [3].…”

Section: Discussionmentioning

confidence: 99%

“…CNS involvement with diabetes insipidus has been associated with the progressive clinical pattern, and the involvement of other areas occurs in less than 5% of XD [3]. …”

Section: Discussionmentioning

confidence: 99%

“…The effectiveness of radiation therapy is unclear due to the limited number of cases; even though radiation therapy often exhibited none or a small palliating stabilizing effect on XD as seen in our case, two reports experienced successful outcome in this context [15,16]. Considering systemic management, ECD treatment options included interferon-α, anakinra and Braf targeting drugs [3,17]. But unlike in ECD, the BRAF mutation has not yet been found in other NLH including multicentric reticulohistiocytosis (MCRH) and XD in the literature and in our case [18,19].…”

Section: Discussionmentioning

confidence: 99%

“…Among the areas of the central nervous system (CNS) involved in all the subtypes of NLH, infiltration of the pituitary gland is a frequent feature often expressed as diabetes insipidus. Nonetheless, lesions may affect any part of the brain [3,4]. The NLH prognosis is unpredictable, and mortality rates may reach close to 60% at 32 months [5].…”

Section: Introductionmentioning

confidence: 99%

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Cerebral and Cutaneous Involvements of Xanthoma Disseminatum Successfully Treated with an Interleukin-1 Receptor Antagonist: A Case Report and Minireview

Campero¹,

Campero

Guerrero

et al. 2016

Dermatology

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A young male presented with panhypopituitarism (including diabetes insipidus) and temporal lobe epilepsy. A histology specimen of cutaneous papules was diagnostic of non-Langerhans histiocytosis. The diagnosis of xanthoma granulomata was considered based on the clinical and brain MRI findings. Brain lesions significantly worsened over time despite radiotherapy until anakinra induced a complete clinical and radiological remission of all active lesions. Although a single case, the outcome of this patient with xanthoma disseminatum treated with an interleukin-1 receptor antagonist opens and strengthens new and recent physiopathogenic and treatment perspectives for the otherwise difficult-to-treat non-Langerhans cell histiocytosis. Similar results with anakinra have been observed in patients with Erdheim-Chester disease and in multicentric reticulohistiocytosis.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

“…CNS involvement with diabetes insipidus has been associated with the progressive clinical pattern, and the involvement of other areas occurs in less than 5% of XD [3]. …”

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Cerebral and Cutaneous Involvements of Xanthoma Disseminatum Successfully Treated with an Interleukin-1 Receptor Antagonist: A Case Report and Minireview

Campero¹,

Campero

Guerrero

et al. 2016

Dermatology

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Eruption of Yellow-Brown and Red-Brown Papules in a Young Adult With Polydipsia and Polyuria

2017

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A woman in her 20s presented with numerous grouped yellow-brown and red-brown papules on the upper eyelids, infraorbital rims, bilateral axillae, inframammary creases, lower back, buttocks, hips, inguinal folds, and proximal thighs (Figure , A and B). The lesions first appeared in her right axilla 6 months prior and were asymptomatic. The patient also noted a 3-month history of polyuria, polydipsia, and nocturia. Review of symptoms was otherwise negative. She had no notable medical or family history. On physical examination there were no other sites of involvement including mucosal surfaces. Shave biopsies were obtained from the right (Figure , C) and left axillae (Figure , D) and sent for histopathological examination. Yellow-brown papulesA Red-brown papules B Hematoxylin-eosin C CD68 stain D Figure. A, Clinical images show punctate yellow-brown papules on the upper eyelids and infraorbital rims; B, numerous red-brown papules in the right axilla. C, Histopathology of a shave biopsy sample from the right axilla (original magnification ×200). D, Histopathology of a shave biopsy sample taken from the left axilla (original magnification ×200). WHAT IS YOUR DIAGNOSIS? A. Xanthogranuloma B. Erdheim-Chester disease C. Xanthoma disseminatum D. Generalized eruptive histiocytoma Clinical Review & Education

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Hematopoietic Proliferations

Phung¹,

Wright²,

Pourciau³

et al. 2017

Pediatric Dermatopathology

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Xanthoma disseminatum with intracranial involvement: case report and literature review

Cited by 17 publications

References 25 publications

Cerebral and Cutaneous Involvements of Xanthoma Disseminatum Successfully Treated with an Interleukin-1 Receptor Antagonist: A Case Report and Minireview

Cerebral and Cutaneous Involvements of Xanthoma Disseminatum Successfully Treated with an Interleukin-1 Receptor Antagonist: A Case Report and Minireview

Eruption of Yellow-Brown and Red-Brown Papules in a Young Adult With Polydipsia and Polyuria

Hematopoietic Proliferations

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