Disseminated juvenile xanthogranulomatosis in a newborn resulting in liver transplantation

Haughton, Adrienne; Horii, Kimberly A.; Shao, Lei; Daniel, J.; Nopper, Amy J.

doi:10.1016/j.jaad.2007.09.012

Cited by 36 publications

(26 citation statements)

References 28 publications

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“…Interestingly, he had also had JXG on the arms, and it tended to spontaneously resolve. Disseminated JXG, which has multiple cutaneous lesions, is sometimes associated with visceral JXG in infancy (5,6). Both the present SGFP-JXG case and the patient in the literature showed no involvement of internal organs.…”

Section: Discussionmentioning

confidence: 46%

Symmetrical Giant Facial Plaque-type Juvenile Xanthogranuloma Persisting Beyond 10 years of Age

Sugiura¹,

Hasegawa²,

Shimoyama³

et al. 2014

Acta Derm Venerol

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Section: Discussionmentioning

confidence: 46%

Symmetrical Giant Facial Plaque-type Juvenile Xanthogranuloma Persisting Beyond 10 years of Age

Sugiura¹,

Hasegawa²,

Shimoyama³

et al. 2014

Acta Derm Venerol

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“…JXG has been discussed as a granulomatous response to an unknown stimulus, but the exact aetiology is unknown. JXG has been associated with juvenile chronic myelogenous leukaemia, neurofibromatosis type I, insulin‐dependent diabetes mellitus, aquagenic pruritus, urticaria pigmentosa and – as in our case – cytomegalovirus infection . Cutaneous JXG manifests in 40–70% of patients during the first year of life, with a male predominance …”

Section: Discussionmentioning

confidence: 55%

Successful treatment of systemic juvenile xanthogranulomatosis with cytarabine and 2‐chlorodeoxyadenosine: case report and review of the literature

et al. 2016

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The non-Langerhans cell histiocytosis (LCH) juvenile xanthogranulomatosis (JXG) is usually a benign disease limited to the skin. Only a few cases of systemic disease with at least two affected organs and lethal outcomes have been reported to date. Treatment is controversial and no standard protocol is available. We report the rare case of a 22-month-old boy presenting multiple erythematous brownish papules of the head, trunk and legs, which had developed starting from his 6th month of life. Additional symptoms were delayed psychomotor development, hydrocephalus and hepatosplenomegaly. Further diagnostics revealed a systemic JXG with involvement of the skin, central nervous system, liver and spleen. The patient did not respond to initial therapy with prednisone and vinblastine according to protocol III for LCH. However, further therapy with cytarabine and 2-chlorodeoxyadenosine followed by a consolidation phase with 2-chlorodeoxyadenosine alone was successful and the patient is in his 4th year of remission. We provide a comprehensive review of the reported cases of systemic JXG to date.

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“…3,4,12,13 Involvement of the central nervous system and liver was reported to be related to death. 3,4,14,15 Pulmonary involvement usually manifests as bilateral, diffuse, asymptomatic micro-or macronodules. 11,16,17 Interstitial lung pattern and mediastinal adenopathy have been reported.…”

Section: Discussionmentioning

confidence: 99%

Baby with neonatal systemic juvenile xanthogranuloma born within a cross-cousin marriage

Nacaroğlu

Can

Karkıner

et al. 2015

Dermatologica Sinica

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Disseminated juvenile xanthogranulomatosis in a newborn resulting in liver transplantation

Cited by 36 publications

References 28 publications

Symmetrical Giant Facial Plaque-type Juvenile Xanthogranuloma Persisting Beyond 10 years of Age

Symmetrical Giant Facial Plaque-type Juvenile Xanthogranuloma Persisting Beyond 10 years of Age

Successful treatment of systemic juvenile xanthogranulomatosis with cytarabine and 2‐chlorodeoxyadenosine: case report and review of the literature

Baby with neonatal systemic juvenile xanthogranuloma born within a cross-cousin marriage

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