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Bronchiolitis obliterans as a long-term sequela of Stevens–Johnson syndrome and toxic epidermal necrolysis in children

Bronchiolitis obliterans as a long-term sequela of Stevens–Johnson syndrome and toxic epidermal necrolysis in children
Bronchiolitis obliterans as a long-term sequela of Stevens–Johnson syndrome and toxic epidermal necrolysis in children
Toxic epidermal necrolysis (TEN) and Stevens–Johnson syndrome (SJS) are characterized by widespread skin and mucosal blistering and necrosis. The triggers and long-term sequelae in children may differ from those reported for adults. Bronchiolitis obliterans (BO) is an uncommon complication, with only 15 previously reported cases, but can lead to significant long-term morbidity, requiring lung transplantation in some cases. We report three children with nondrug-related SJS (n = 1) TEN (n = 2) who developed BO. Two were treated with intravenous immunoglobulin therapy (2–2.4 g/kg) and all three survived. We highlight salient learning points from our cases and potential pitfalls in diagnosis of BO, including delayed onset, and we also review the literature.
0307-6938
Seccombe, E.L.
6f34388d-fec9-41ab-873a-37ed48ac507f
Ardern-Jones, M.
7ac43c24-94ab-4d19-ba69-afaa546bec90
Walker, W.
58aae223-5b0e-4f34-9ee7-58bb68278c3a
Austin, S.
57d051a5-86f6-47bf-a580-633d4def827b
Taibjee, S.
f105b15f-c60f-4919-9708-581c566c7fda
Williams, S.
762dd529-2acb-40b8-acf3-e3488eec8506
Hossain, Parwez
563de5fc-84ad-4539-9228-bde0237eaf51
Shenoy, D.
f39eaa8d-a430-4533-8a2a-cdaac530459d
Fityan, A.
76dc1f06-faf9-4676-abf4-c81de5eb3b4c
Seccombe, E.L.
6f34388d-fec9-41ab-873a-37ed48ac507f
Ardern-Jones, M.
7ac43c24-94ab-4d19-ba69-afaa546bec90
Walker, W.
58aae223-5b0e-4f34-9ee7-58bb68278c3a
Austin, S.
57d051a5-86f6-47bf-a580-633d4def827b
Taibjee, S.
f105b15f-c60f-4919-9708-581c566c7fda
Williams, S.
762dd529-2acb-40b8-acf3-e3488eec8506
Hossain, Parwez
563de5fc-84ad-4539-9228-bde0237eaf51
Shenoy, D.
f39eaa8d-a430-4533-8a2a-cdaac530459d
Fityan, A.
76dc1f06-faf9-4676-abf4-c81de5eb3b4c

Seccombe, E.L., Ardern-Jones, M., Walker, W., Austin, S., Taibjee, S., Williams, S., Hossain, Parwez, Shenoy, D. and Fityan, A. (2019) Bronchiolitis obliterans as a long-term sequela of Stevens–Johnson syndrome and toxic epidermal necrolysis in children. Clinical and Experimental Dermatology. (doi:10.1111/ced.13969).

Record type: Article

Abstract

Toxic epidermal necrolysis (TEN) and Stevens–Johnson syndrome (SJS) are characterized by widespread skin and mucosal blistering and necrosis. The triggers and long-term sequelae in children may differ from those reported for adults. Bronchiolitis obliterans (BO) is an uncommon complication, with only 15 previously reported cases, but can lead to significant long-term morbidity, requiring lung transplantation in some cases. We report three children with nondrug-related SJS (n = 1) TEN (n = 2) who developed BO. Two were treated with intravenous immunoglobulin therapy (2–2.4 g/kg) and all three survived. We highlight salient learning points from our cases and potential pitfalls in diagnosis of BO, including delayed onset, and we also review the literature.

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CED-2019-0073Edited-V2.ES - Accepted Manuscript
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Accepted/In Press date: 20 March 2019
e-pub ahead of print date: 25 March 2019

Identifiers

Local EPrints ID: 432980
URI: http://eprints.soton.ac.uk/id/eprint/432980
ISSN: 0307-6938
PURE UUID: d4a28b62-be4a-4710-bb4a-f77f4c99aad4
ORCID for Parwez Hossain: ORCID iD orcid.org/0000-0002-3131-2395

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Date deposited: 05 Aug 2019 16:30
Last modified: 15 Sep 2021 05:12

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