Ocular Manifestations of Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis in Children

Catt, Caroline; Hamilton, Gavin M.; Fish, Joel; Mireskandari, Kamiar; Ali, Asim

doi:10.1016/j.ajo.2016.03.020

Cited by 61 publications

(38 citation statements)

References 20 publications

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“…Ophthalmic complications were found in SJS/TEN with the incidence ranging from 20.0–81.0% [4, 6, 35–37]. In this study, short-term complications were seen in 66.7% of cases and long-term complications were seen in 36.1% of cases.…”

Section: Discussionmentioning

confidence: 49%

Clinical Features and Treatment Outcomes among Children with Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis: A 20-Year Study in a Tertiary Referral Hospital

Chatproedprai

Wutticharoenwong

Tempark

et al. 2018

Dermatology Research and Practice

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Aim To determine the probable causative factors, clinical features, and treatment outcomes of Stevens-Johnson syndrome (SJS), toxic epidermal necrolysis (TEN), and SJS-TEN overlap in children. Methods A 20-year database review of all children diagnosed with SJS/TEN/SJS-TEN overlap at the King Chulalongkorn Memorial Hospital, Thailand. Results 36 patients (M : F, 16 : 20) with the mean age of 9.2 ± 4.0 years were identified. There were 20 cases of SJS, 4 cases of SJS-TEN overlap, and 12 cases of TEN. Drugs were the leading cause for the diseases (72.3%); antiepileptics were the most common culprits (36.1%). Cutaneous morphology at presentation was morbilliform rash (83.3%), blister (38.9%), targetoid lesions (25.0%), and purpuric macules (2.8%). Oral mucosa (97.2%) and eye (83.3%) were the 2 most common mucosal involvements. Majority of the cases (77.8%) were treated with systemic corticosteroids, intravenous immunoglobulin, or both. Treatment outcomes between those who received systemic therapy and those who received only supportive care were comparable. Skin and eye were the principal sites of short-term and long-term complications. Conclusions SJS/TEN are not common but are serious diseases which lead to significant morbidities in children. Early withdrawal of suspicious causes and meticulous supportive care are very important. This study found that the systemic therapy was not superior to supportive care because the treatment outcomes for both groups were comparable.

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Section: Discussionmentioning

confidence: 49%

Clinical Features and Treatment Outcomes among Children with Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis: A 20-Year Study in a Tertiary Referral Hospital

Chatproedprai

Wutticharoenwong

Tempark

et al. 2018

Dermatology Research and Practice

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“…Most complications are ocular surface abnormalities and not vision threatening as most children (90% of cases) maintain good vision long-term. Blindness as a complication is not common (5–9% of cases) [ 19 ].…”

Section: Discussionmentioning

confidence: 99%

“…About 27–80% of hospitalized patients with SJS/TEN develop acute ocular complications [ 20 ], with conjunctivitis (78%) being the most common clinical sign in the pediatric population [ 19 ]. Conjunctivitis together with conjunctival membrane and sub conjunctival hemorrhage constitutes one of the earliest signs.…”

Section: Discussionmentioning

confidence: 99%

Severe eye complications from toxic epidermal necrolysis following initiation of Nevirapine based HAART regimen in a child with HIV infection: a case from Cameroon

et al. 2018

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BackgroundToxic epidermal necrolysis (TEN) is a rare life threatening dermatological disorder characterized by extensive epidermal detachment and erosion of mucous membranes. It is typically a side effect of some medications. Nevirapine, a nonnucleoside reverse transcriptase inhibitor (NNRTI) is one of the frequently used components of highly active antiretroviral therapy (HAART). Skin rash is its common adverse reaction, usually mild and rarely progressing to TEN. Ophthalmic involvement is common as well but rarely progresses to blindness especially in the pediatric population.Case presentationWe report the case of a 3 year 5 month old child diagnosed with HIV who developed TEN 8 days after starting a Nevirapine based HAART regimen. Drug withdrawal and supportive treatment alone were the modalities employed to achieve complete re-epithelization of lesions. Patient was lost to follow-up 6 months after being in care and was only seen 3 years later with total loss of vision.ConclusionBlindness, though rare, can be a long-term complication of TEN in children especially with HIV infection. Physicians and patient caregivers should closely monitor these patients, especially during their early stages of treatment amongst others for development of adverse drug reactions. Long-term retention in care is pivotal for identification and prompt management of ocular and other chronic complications, albeit recognizing management challenges in low resourced settings.

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“…Early and aggressive treatment of acute ocular manifestations is essential to limit long‐term sequelae and vision impairment in these patients, with evidence supporting the use of AMT. Ocular manifestations may also present late, necessitating follow‐up in all patients …”

Section: Reportmentioning

confidence: 99%

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

et al. 2019

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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) and 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.Stevens-Johnson syndrome (SJS) and toxic epidermal necrolysis (TEN) are rare, potentially life-threatening conditions, characterized by widespread skin and mucosal necrosis. SJS is defined as < 10% body surface area (BSA) detachment, SJS-TEN overlap as 10-30% BSA detachment and TEN as > 30% detachment. 1 The reported incidence is lower in children than adults: 5.3 vs. 9.3, 0.8 vs. 1.9 and 0.4 vs. 1.6 cases per million per year for SJS, SJS-TEN and TEN, respectively, in the USA. 2,3 Medications are usually culpable, although infections may be implicated, particularly in children. 4 Mortality in childhood is reportedly 0% (SJS), 4% (SJS-TEN) and 16% (TEN), 2 with deaths most frequently due to sepsis and organ failure. 2 However, similarly to adults, increased mortality is noted with higher degrees of epidermal detachment: 60-70% BSA involvement was associated with 37.5% mortality in one study. 5 Intravenous immunoglobulin (IVIG), ciclosporin and/or systemic corticosteroids have been used despite the relative lack of evidence, as summarized in recent guidelines. 6 Bronchiolitis obliterans (BO) is a rarely reported complication, but when present, is associated with significant long-term morbidity. We report three children with nondrug-related SJS/TEN who developed BO. ReportPatient 1 was a previously well 5-year-old boy who presented with a 2-day history of an erythematous maculopapular rash in association with fever and coryza. Overall, the skin changes involved 95% BSA, with 40% epidermal detachment. There were additional bilateral conjunctival defects with confluent superficial punctate epithelial erosions of the cornea. He had severe oral and upper airway ulceration with sloughing. Subsequent development of stridor necessitated intubation.Histological examination of a skin biopsy confirmed TEN, with negative direct immunofluorescence. Chest radiography was unremarkable. Bronchoscopy showed ulceration at the base of insertion of the endotracheal

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Ocular Manifestations of Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis in Children

Cited by 61 publications

References 20 publications

Clinical Features and Treatment Outcomes among Children with Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis: A 20-Year Study in a Tertiary Referral Hospital

Clinical Features and Treatment Outcomes among Children with Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis: A 20-Year Study in a Tertiary Referral Hospital

Severe eye complications from toxic epidermal necrolysis following initiation of Nevirapine based HAART regimen in a child with HIV infection: a case from Cameroon

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

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