Ocular involvement in children with localised scleroderma: a multi-centre study

Zannin, Maria Elisabetta; Martini, Giorgia; Athreya, Balu H.; Russo, Ricardo; Higgins, Gloria C.; Vittadello, Fabio; Alpigiani, M. G.; Alessio, Maria; Paradisi, Mauro; Woo, Patricia; Zulian, Francesco

doi:10.1136/bjo.2007.116038

Cited by 123 publications

(96 citation statements)

References 13 publications

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“…4,7,9,11,[13][14][15][16] These have been due to either direct muscle infiltration or peripheral nerve disease. In scleroderma, increased collagen deposition occurs, resulting in dermal thickening.…”

Section: Discussionmentioning

confidence: 99%

“…Several published articles have looked at the ocular impact of this disease [2][3][4][5][6][7][8][9][10][11][12][13][14][15][16] but no reviewed papers have documented the natural progression of LS and 'en coup de sabre' for as long as in this case.…”

Section: Discussionmentioning

confidence: 99%

“…3 In 2007 a large study of 750 patients with localised juvenile scleroderma was reported. 4 It identified 24 patients with significant ocular involvement, including eyelid and eyelash abnormalities, ectropion, anterior uveitis, episcleritis, refractive error, pupil mydriasis and enophthalmos. One patient had a paralytic strabismus due to a 6th nerve palsy.…”

Section: Discussionmentioning

confidence: 99%

“…'En coup de sabre' is an uncommon and seldom reported dermatological disorder; it is usually unilateral, but rare bilateral cases have been recorded. 7 It is often reported with ptosis or lid abnormalities [4][5][6][7][8][9][11][12][13][14][15][16] but concurrent oculomotor disorders are rare. 4,7,9,11,[13][14][15][16] Case report…”

Section: Introductionmentioning

confidence: 99%

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Linear scleroderma ‘en coup de sabre’ with ptosis and oculomotility disorders: case report and review of the literature

Taylor¹,

Riley²,

Bowyer

2010

British and Irish Orthoptic Journal

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Aim: To present a case of linear scleroderma (LS) involving the extraocular muscles and periorbita with follow-up over an 18-year period. Method: Initial presentation was of a female aged 9 years with progressive left unilateral ptosis which ultimately underwent surgical correction. Results: The main features of 'en coup de sabre' subsequently manifested on the left side of the face and head. An unusual feature was the characteristic subcutaneous cleft which also appeared on the opposite side above the right eyebrow. The left eye went on to develop a corneal ulcer with resulting poor visual acuity. An ipsilateral oculomotility disorder was then noted in addition to poor eyelid closure. Conclusion: This case highlights the need to exclude 'en coup de sabre' as a potential differential diagnosis in any case of progressive or recurrent ptosis, especially in childhood. As oculomotility may also be affected ultimately, it could lead to corneal compromise following ptosis correction.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Linear scleroderma ‘en coup de sabre’ with ptosis and oculomotility disorders: case report and review of the literature

Taylor¹,

Riley²,

Bowyer

2010

British and Irish Orthoptic Journal

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“…A radiograph of the skull may be helpful if bone is involved. Magnetic resonance imaging of the brain and skull may reveal abnormalities such as dilated ventricle, atrophy of the cortex and subcortical calcification [6]. A 10-25 MHz ultrasound scan can help to measure the skin thickness and assess disease activity [7].…”

mentioning

confidence: 99%

Concomitant en coup de sabre and plaque type morphea in the same patient: a rare occurrence

Arif¹,

Adil²,

Amin³

et al. 2017

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Morphoea (Localized Scleroderma)

Orteu

2016

Rook's Textbook of Dermatology, Ninth Edition

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Morphoea encompasses a group of related conditions characterized by varying degrees of sclerosis, fibrosis and atrophy in the skin and subcutaneous tissues, sometimes extending deeply into the muscle, bone, eye and brain. Extracutaneous manifestations occur in up to 25% of cases but in contrast to systemic sclerosis (SSc), no internal organ fibrosis or vascular changes occur. Antinuclear antibody positivity is common but the specific autoantibodies seen in SSc are rarely present. There is no increased mortality, but substantial morbidity may occur as a result of joint contractures, facial and limb asymmetry, extracutaneous manifestations and the psychological impact of the condition. Although previously considered self‐limiting, there is now emerging evidence that a protracted, relapsing–remitting course may be common. The key to successful treatment is the involvement of a multidisciplinary team and the initiation of therapy during the active inflammatory stage, before significant damage has occurred. Topical and phototherapies are appropriate in superficial forms of morphoea, whilst a majority of linear, generalized and deep forms require systemic therapy.

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Ocular involvement in children with localised scleroderma: a multi-centre study

Cited by 123 publications

References 13 publications

Linear scleroderma ‘en coup de sabre’ with ptosis and oculomotility disorders: case report and review of the literature

Linear scleroderma ‘en coup de sabre’ with ptosis and oculomotility disorders: case report and review of the literature

Concomitant en coup de sabre and plaque type morphea in the same patient: a rare occurrence

Morphoea (Localized Scleroderma)

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