Cutaneous Oxalosis Due to Primary Hyperoxaluria.
Journal
The American Journal of dermatopathology
ISSN: 1533-0311
Titre abrégé: Am J Dermatopathol
Pays: United States
ID NLM: 7911005
Informations de publication
Date de publication:
01 Dec 2022
01 Dec 2022
Historique:
entrez:
17
11
2022
pubmed:
18
11
2022
medline:
22
11
2022
Statut:
ppublish
Résumé
A 19-year-old girl presented to the emergency department with a progressively painful purpuric lesion on the left dorsal foot, which had initially appeared 2 days prior. Three months earlier, she had been diagnosed with end-stage renal disease. Her medical history also included recurrent urolithiasis for the past 5 years and liver failure. Biopsy revealed oxalate crystals occluding vessels with secondary epidermal and dermal ischemia. Oxalate crystals were also visualized in the vessel walls and free in the subcutis. Genetic testing confirmed the diagnosis of primary hyperoxaluria type 1. She was treated with sodium thiosulfate, apixaban, pentoxifylline, wound care, and palliative care. At 4-month follow-up, the cutaneous manifestations of oxalosis were confined to only her feet, and she was undergoing evaluation for combined liver and kidney transplant. Cutaneous oxalosis because of primary hyperoxaluria should be considered in young patients presenting with purpuric lesions, recurrent urolithiasis, and early-onset renal failure.
Identifiants
pubmed: 36395453
doi: 10.1097/DAD.0000000000002307
pii: 00000372-202212000-00025
doi:
Substances chimiques
Oxalates
0
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
981-983Informations de copyright
Copyright © 2022 Wolters Kluwer Health, Inc. All rights reserved.
Déclaration de conflit d'intérêts
The authors declare no conflicts of interest.
Références
Cochat P. Primary hyperoxaluria type 1. Kidney Int. 1999;55:2533–2547.
Bogle MA, Teller CF, Tschen JA, et al. Primary hyperoxaluria in a 27-year-old woman. J Am Acad Dermatol. 2003;49:725–728.
van Woerden CS, Groothoff JW, Wanders RJA, et al. Primary hyperoxaluria type 1 in The Netherlands: prevalence and outcome. Nephrol Dial Transpl. 2003;18:273–279.
Shreberk-Hassidim R, Zaguri R, Maly A, et al. Skin manifestations of primary hyperoxaluria: a case report. Int J Dermatol. 2015;54:e478–e479.
Blackmon JA, Jeffy BG, Malone JC, Knable AL Jr. Oxalosis involving the skin: case report and literature review. Arch Dermatol. 2011;147:1302–1305.
Online Mendelian Inheritance in Man, OMIM® [Database Online]. Baltimore, MD: Johns Hopkins University; 1966. Updated June 4, 2022.
Georgesen C, Fox LP, Harp J. Retiform purpura: a diagnostic approach. J Am Acad Dermatol. 2020;82:783–796.
Somach SC, Davis BR, Paras FA, et al. Fatal cutaneous necrosis mimicking calciphylaxis in a patient with type 1 primary hyperoxaluria. Arch Dermatol. 1995;131:821–823.
Bridges AG. A challenging case of leg ulcers associated with retiform purpura: cutaneous oxalosis in the setting of primary type 1 hyperoxaluria. Mayo Clin Proc. 2020;95:1825–1826.
Vedvyas C, Winterfield LS, Vleugels RA. Calciphylaxis: a systematic review of existing and emerging therapies. J Am Acad Dermatol. 2012;67:e253–e260.
King BJ, El-Azhary RA, McEvoy MT, et al. Direct oral anticoagulant medications in calciphylaxis. Int J Dermatol. 2017;56:1065–1070.
Harambat J, Fargue S, Bacchetta J, et al. Primary hyperoxaluria. Int J Nephrol. 2011;2011:1–11.