Langerhans cell histiocytosis with prominent nail involvement

Bender, Robert W.; Seline, Alison E.; Siegel, Dawn H.; Sokumbi, Olayemi

doi:10.1111/cup.13352

Cited by 7 publications

(12 citation statements)

References 22 publications

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“…4 The third patient was treated with vinblastine followed by cytarabine but still progressed. 7 The nail changes improved after adding NB-UVB to the treatment regimen. However, disease recurred following tapering of phototherapy and replacement of vinblastine with methotrexate.…”

Section: Discussionmentioning

confidence: 96%

Onycholysis and purpuric nail striae in a child

Yang

Wang

Liu

2023

Pediatric Dermatology

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A 2-year-old boy presented with a 2-month history of recurrent fevers, abdominal distension, an eroded skin lesion in the right axilla, and dystrophic nails. Physical examination revealed hyperkeratosis, onycholysis, longitudinal grooving, and purpuric striae on his fingernails and toenails (Figures 1 and 2). A 2 Â 1 cm fusiform eroded plaque was present on his right axillae (Figure 3). Hepatosplenomegaly was noted on palpation. A punch biopsy was taken from the axillary skin lesion (Figure 4).

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Section: Discussionmentioning

confidence: 96%

Onycholysis and purpuric nail striae in a child

Yang

Wang

Liu

2023

Pediatric Dermatology

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“…Early‐onset (under the age of two years), extensive disease, and the presence of organ failure are the leading negative prognostic indicators . Nail involvement in LCH is an uncommon manifestation and rarely reported . It is thought to be associated with multisystem involvement and therefore a poor prognosis .…”

Section: Introductionmentioning

confidence: 99%

“…3 Nail involvement in LCH is an uncommon manifestation and rarely reported. 4,5 It is thought to be associated with multisystem involvement and therefore a poor prognosis. 1,6 Mataix 6 showed that it could be the initial manifestation in LCH.…”

Section: Introductionmentioning

confidence: 99%

Purpuric nail striae as the initial manifestation in multisystem Langerhans cell histiocytosis

Prayogo

Rahmayunita

Agustin

et al. 2019

Pediatric Dermatology

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Nail involvement in Langerhans cell histiocytosis (LCH) is very uncommon. We describe a 9‐month‐old boy with longitudinal purpuric bands, onychodystrophy, and paronychias on his fingernails and toenails, which antedated manifestations of recurrent bilateral spontaneous pneumothorax and elevated transaminases. The diagnosis of nail LCH was then confirmed by immunohistochemistry demonstrating positive S100 and CD1a staining in the epithelium of nail bed tissue. This case report affirms that nail involvement may be the initial manifestation of multisystem LCH and could portend an unfavorable prognosis.

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“…Nail involvement has also been considered an unfavorable prognostic sign and seems to be more frequent in multisystem-Langerhans cell histiocytosis. [2][3][4] A 41-year-old previously healthy Caucasian man, presented in October 2019, with multiple cutaneous erythematous papules over the scalp, persistent chronic paronychia with onychodystrophy and nail discoloration involving only the fifth finger of the right hand [Figure 1]. Differential diagnoses included onychomycosis, chronic candidiasis, Darier disease and Langerhans cell histiocytosis.…”

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confidence: 99%

“…Two papers investigated the BRAF mutational status (including present case) and both were wild-type. 4 Most patients were treated with polychemotherapy including prednisone, vinca alkaloids, 6-mercaptopurine, methotrexate, cyclophosphamide, etoposide, and/or cytosine arabinoside. 5/31 (16.1%) patients with follow-up data, achieved complete remission, while 9/31 (29.0%) died of disease after a median of 15.5 months.…”

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confidence: 99%