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Published in: Journal of Medical Case Reports 1/2012

Open Access 01-12-2012 | Case report

Acute compartment syndrome of the forearm as a rare complication of toxic epidermal necrolysis: a case report

Authors: Tamer Kamal, Sherif Elnikety, Heba Mashaly, James Casha

Published in: Journal of Medical Case Reports | Issue 1/2012

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Abstract

Introduction

Toxic epidermal necrolysis lies within the spectrum of severe cutaneous adverse reactions induced by drugs, affecting skin and mucous membranes. Toxic epidermal necrolysis is considered a medical emergency as it is considered to be potentially fatal and carries a high mortality rate. To the best of our knowledge the association of toxic epidermal necrolysis and compartment syndrome has been rarely mentioned in the literature. In this case we treated the compartment syndrome promptly despite the poor general condition and skin status of our patient. Despite the poor skin condition, wound healing was uneventful with no complications.

Case presentation

A 62-year-old Caucasian man with a generalized macular-vesicular rash involving 90% of his body surface area and mucous membranes, as well as impaired renal and hepatic functions following ingestion of allopurinol for treatment of gout, was admitted to our hospital. Skin biopsies were taken and he was started on a steroid infusion. Within hours of admission, he developed acute compartment syndrome of the dominant forearm and hand.

Conclusions

Despite its rare incidence, toxic epidermal necrolysis is a condition with a high incidence of complications and mortality. Patients with severe conditions affecting a large degree of the skin surface area should be treated as promptly and effectively as patients with burns, with close monitoring and the anticipation that rare musculoskeletal complications might arise. The association of compartment syndrome and toxic epidermal necrolysis might lead to a rapid deterioration and fatal systemic involvement and multiple organ failures.
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Literature
1.
go back to reference Mockenhaupt M: Severe drug-induced skin reactions: clinical pattern, diagnostics and therapy. J Dtsch Dermatol Ges. 2009, 7: 142-160.PubMed Mockenhaupt M: Severe drug-induced skin reactions: clinical pattern, diagnostics and therapy. J Dtsch Dermatol Ges. 2009, 7: 142-160.PubMed
2.
go back to reference Mockenhaupt M: The current understanding of Stevens-Johnson syndrome and toxic epidermal necrolysis. Exp Rev Clin Immunol. 2011, 7: 803-813. 10.1586/eci.11.66.CrossRef Mockenhaupt M: The current understanding of Stevens-Johnson syndrome and toxic epidermal necrolysis. Exp Rev Clin Immunol. 2011, 7: 803-813. 10.1586/eci.11.66.CrossRef
3.
go back to reference Torres MJ, Mayorga C, Blanca M: Nonimmediate allergic reactions induced by drugs: pathogenesis and diagnostic tests. J Investig Allergol Clin Immunol. 2009, 19: 80-90.PubMed Torres MJ, Mayorga C, Blanca M: Nonimmediate allergic reactions induced by drugs: pathogenesis and diagnostic tests. J Investig Allergol Clin Immunol. 2009, 19: 80-90.PubMed
4.
go back to reference Roujeau JC, Kelly JP, Naldi L, Rzany B, Stern RS, Anderson T, Auquier A, Bastuji-Garin S, Correia O, Locati F, Mockenhaupt M, Paoletti C, Shapiro S, Shear N, Schöpf E, Kaufman DW: Medication use and the risk of Stevens-Johnson syndrome or toxic epidermal necrolysis. N Engl J Med. 1995, 333: 1600-1607. 10.1056/NEJM199512143332404.CrossRefPubMed Roujeau JC, Kelly JP, Naldi L, Rzany B, Stern RS, Anderson T, Auquier A, Bastuji-Garin S, Correia O, Locati F, Mockenhaupt M, Paoletti C, Shapiro S, Shear N, Schöpf E, Kaufman DW: Medication use and the risk of Stevens-Johnson syndrome or toxic epidermal necrolysis. N Engl J Med. 1995, 333: 1600-1607. 10.1056/NEJM199512143332404.CrossRefPubMed
5.
go back to reference Mockenhaupt M, Viboud C, Dunant A, Naldi L, Halevy S, Bouwes Bavinck JN, Sidoroff A, Schneck J, Roujeau JC, Flahault A: Stevens-Johnson syndrome and toxic epidermal necrolysis: assessment of medication risks with emphasis on recently marketed drugs. The EuroSCAR-study. J Invest Dermatol. 2008, 128: 35-44. 10.1038/sj.jid.5701033.CrossRefPubMed Mockenhaupt M, Viboud C, Dunant A, Naldi L, Halevy S, Bouwes Bavinck JN, Sidoroff A, Schneck J, Roujeau JC, Flahault A: Stevens-Johnson syndrome and toxic epidermal necrolysis: assessment of medication risks with emphasis on recently marketed drugs. The EuroSCAR-study. J Invest Dermatol. 2008, 128: 35-44. 10.1038/sj.jid.5701033.CrossRefPubMed
6.
go back to reference Halevy S, Ghislain PD, Mockenhaupt M, Fagot JP, Bouwes Bavinck JN, Sidoroff A, Naldi L, Dunant A, Viboud C, Roujeau JC, EuroSCAR Study Group: Allopurinol is the most common cause of Stevens-Johnson syndrome and toxic epidermal necrolysis in Europe and Israel. J Am Acad Dermatol. 2008, 58: 25-32. 10.1016/j.jaad.2007.08.036.CrossRefPubMed Halevy S, Ghislain PD, Mockenhaupt M, Fagot JP, Bouwes Bavinck JN, Sidoroff A, Naldi L, Dunant A, Viboud C, Roujeau JC, EuroSCAR Study Group: Allopurinol is the most common cause of Stevens-Johnson syndrome and toxic epidermal necrolysis in Europe and Israel. J Am Acad Dermatol. 2008, 58: 25-32. 10.1016/j.jaad.2007.08.036.CrossRefPubMed
7.
go back to reference Halevy S: Stevens-Johnson syndrome and toxic epidermal necrolysis--updates and innovations. Harefuah. 2010, 149: 186-190.PubMed Halevy S: Stevens-Johnson syndrome and toxic epidermal necrolysis--updates and innovations. Harefuah. 2010, 149: 186-190.PubMed
8.
go back to reference Laurisch S, Jaedtke M, Demir R, Sorrentino SA, Kielstein JT, Rennekampff HO, Vogt PM, Meyer GP, Fuchs M, Klein G, Drexler H, Schieffer B, Napp LC: Allopurinol-induced hypersensitivity syndrome resulting in death. Med Klin (Munich). 2010, 105: 262-266. 10.1007/s00063-010-1037-3.CrossRef Laurisch S, Jaedtke M, Demir R, Sorrentino SA, Kielstein JT, Rennekampff HO, Vogt PM, Meyer GP, Fuchs M, Klein G, Drexler H, Schieffer B, Napp LC: Allopurinol-induced hypersensitivity syndrome resulting in death. Med Klin (Munich). 2010, 105: 262-266. 10.1007/s00063-010-1037-3.CrossRef
9.
go back to reference Ventura F, Fracasso T, Leoncini A, Gentile R, de Stefano F: Death caused by toxic epidermal necrolysis (Lyell syndrome). J Forensic Sci. 2010, 55: 839-841. 10.1111/j.1556-4029.2010.01338.x.CrossRefPubMed Ventura F, Fracasso T, Leoncini A, Gentile R, de Stefano F: Death caused by toxic epidermal necrolysis (Lyell syndrome). J Forensic Sci. 2010, 55: 839-841. 10.1111/j.1556-4029.2010.01338.x.CrossRefPubMed
10.
go back to reference Bastuji-Garin S, Rzany B, Stern RS, Shear NH, Naldi L, Roujeau JC: Clinical classification of cases of toxic epidermal necrolysis, Stevens-Johnson syndrome, and erythema multiforme. Arch Dermatol. 1993, 129: 92-96. 10.1001/archderm.1993.01680220104023.CrossRefPubMed Bastuji-Garin S, Rzany B, Stern RS, Shear NH, Naldi L, Roujeau JC: Clinical classification of cases of toxic epidermal necrolysis, Stevens-Johnson syndrome, and erythema multiforme. Arch Dermatol. 1993, 129: 92-96. 10.1001/archderm.1993.01680220104023.CrossRefPubMed
11.
go back to reference Bastuji-Garin S, Fouchard N, Bertocchi M, Roujeau JC, Revuz J, Wolkenstein PJ: SCORTEN: a severity-of-illness score for toxic epidermal necrolysis. Invest Dermatol. 2000, 115: 149-153. 10.1046/j.1523-1747.2000.00061.x.CrossRef Bastuji-Garin S, Fouchard N, Bertocchi M, Roujeau JC, Revuz J, Wolkenstein PJ: SCORTEN: a severity-of-illness score for toxic epidermal necrolysis. Invest Dermatol. 2000, 115: 149-153. 10.1046/j.1523-1747.2000.00061.x.CrossRef
12.
go back to reference Nassif A, Bensussan A, Boumsell L, Deniaud A, Moslehi H, Wolkenstein P, Bagot M, Roujeau JC: Toxic epidermal necrolysis: effector cells are drug-specific cytotoxic T cells. J Allergy Clin Immunol. 2004, 114: 1209-1215. 10.1016/j.jaci.2004.07.047.CrossRefPubMed Nassif A, Bensussan A, Boumsell L, Deniaud A, Moslehi H, Wolkenstein P, Bagot M, Roujeau JC: Toxic epidermal necrolysis: effector cells are drug-specific cytotoxic T cells. J Allergy Clin Immunol. 2004, 114: 1209-1215. 10.1016/j.jaci.2004.07.047.CrossRefPubMed
13.
go back to reference Chung WH, Hung SI, Yang JY, Su SC, Huang SP, Wei CY, Chin SW, Chiou CC, Chu SC, Ho HC, Yang CH, Lu CF, Wu JY, Liao YD, Chen YT: Granulysin is a key mediator for disseminated keratinocyte death in Stevens-Johnson syndrome and toxic epidermal necrolysis. Nat Med. 2008, 14: 1343-1350. 10.1038/nm.1884.CrossRefPubMed Chung WH, Hung SI, Yang JY, Su SC, Huang SP, Wei CY, Chin SW, Chiou CC, Chu SC, Ho HC, Yang CH, Lu CF, Wu JY, Liao YD, Chen YT: Granulysin is a key mediator for disseminated keratinocyte death in Stevens-Johnson syndrome and toxic epidermal necrolysis. Nat Med. 2008, 14: 1343-1350. 10.1038/nm.1884.CrossRefPubMed
14.
go back to reference Morel E, Escamochero S, Cabañas R, Díaz R, Fiandor A, Bellón T: CD94/NKG2C is a killer effector molecule in patients with Stevens-Johnson syndrome and toxic epidermal necrolysis. J Allergy Clin Immunol. 2010, 125: 703-710. 10.1016/j.jaci.2009.10.030.CrossRefPubMed Morel E, Escamochero S, Cabañas R, Díaz R, Fiandor A, Bellón T: CD94/NKG2C is a killer effector molecule in patients with Stevens-Johnson syndrome and toxic epidermal necrolysis. J Allergy Clin Immunol. 2010, 125: 703-710. 10.1016/j.jaci.2009.10.030.CrossRefPubMed
15.
go back to reference Li X, Liang D, Liu X: Compartment syndrome in burn patients. A report of five cases. Burns. 2002, 28: 787-789. 10.1016/S0305-4179(02)00190-0.CrossRefPubMed Li X, Liang D, Liu X: Compartment syndrome in burn patients. A report of five cases. Burns. 2002, 28: 787-789. 10.1016/S0305-4179(02)00190-0.CrossRefPubMed
Metadata
Title
Acute compartment syndrome of the forearm as a rare complication of toxic epidermal necrolysis: a case report
Authors
Tamer Kamal
Sherif Elnikety
Heba Mashaly
James Casha
Publication date
01-12-2012
Publisher
BioMed Central
Published in
Journal of Medical Case Reports / Issue 1/2012
Electronic ISSN: 1752-1947
DOI
https://doi.org/10.1186/1752-1947-6-84

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