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• W2021199113 abstract "We report the ocular findings in a 2.5-year-old girl with a history of congenital erosive and vesicular dermatosis at birth. We highlight the complexity of the associated nasolacrimal duct obstruction with canalicular scarring and review the ocular manifestations of this rare disease. We report the ocular findings in a 2.5-year-old girl with a history of congenital erosive and vesicular dermatosis at birth. We highlight the complexity of the associated nasolacrimal duct obstruction with canalicular scarring and review the ocular manifestations of this rare disease. Congenital erosive and vesicular dermatosis (CEVD) healing with reticulate scarring is a rare disease, with only 18 cases reported in the literature. It presents at birth, usually in premature infants, with crusted erosions and vesicles that heal relatively rapidly, forming unique reticulated scars over the trunk and limbs, tongue,1Cohen B.A. Esterly N.B. Nelson P.F. Congenital erosive and vesicular dermatosis healing with reticulated supple scarring.Arch Dermatol. 1985; 121: 361-367Crossref PubMed Scopus (44) Google Scholar, 2Gupta A.K. Rasmussen J.E. Headington J.T. Extensive congenital erosions and vesicles healing with reticulate scarring.J Am Acad Dermatol. 1987; 17: 369-376Abstract Full Text PDF PubMed Scopus (22) Google Scholar, 3Plantin P. Delaire P. Guillois B. et al.Congenital erosive dermatosis with reticulated supple scarring: first neonatal report.Arch Dermatol. 1990; 126: 544-546Crossref PubMed Scopus (21) Google Scholar and scalp,1Cohen B.A. Esterly N.B. Nelson P.F. Congenital erosive and vesicular dermatosis healing with reticulated supple scarring.Arch Dermatol. 1985; 121: 361-367Crossref PubMed Scopus (44) Google Scholar, 3Plantin P. Delaire P. Guillois B. et al.Congenital erosive dermatosis with reticulated supple scarring: first neonatal report.Arch Dermatol. 1990; 126: 544-546Crossref PubMed Scopus (21) Google Scholar, 4Sidhu-Malik N.K. Resnick S.D. Wilson B.B. Congenital erosive and vesicular dermatosis healing with reticulated supple scarring: report of three new cases and review of the literature.Pediatr Dermatol. 1998; 15: 214-218Crossref PubMed Scopus (17) Google Scholar, 5Mashiah J. Wallach D. Leclerc-Mercier S. Bodemer C. Hadj-Rabia S. Congenital erosive and vesicular dermatosis: a new case and review of the literature.Pediatr Dermatol. 2012; 29: 756-758Crossref PubMed Scopus (12) Google Scholar with relative sparing of the face and volar surfaces. Only 4 cases have been reported with ocular involvement,2Gupta A.K. Rasmussen J.E. Headington J.T. Extensive congenital erosions and vesicles healing with reticulate scarring.J Am Acad Dermatol. 1987; 17: 369-376Abstract Full Text PDF PubMed Scopus (22) Google Scholar, 4Sidhu-Malik N.K. Resnick S.D. Wilson B.B. Congenital erosive and vesicular dermatosis healing with reticulated supple scarring: report of three new cases and review of the literature.Pediatr Dermatol. 1998; 15: 214-218Crossref PubMed Scopus (17) Google Scholar, 5Mashiah J. Wallach D. Leclerc-Mercier S. Bodemer C. Hadj-Rabia S. Congenital erosive and vesicular dermatosis: a new case and review of the literature.Pediatr Dermatol. 2012; 29: 756-758Crossref PubMed Scopus (12) Google Scholar, 6Stein S. Stone S. Paller A.S. Ongoing blistering in a boy with congenital erosive and vesicular dermatosis healing with reticulated supple scarring.J Am Acad Dermatol. 2001; 45: 946-948Abstract Full Text Full Text PDF PubMed Scopus (19) Google Scholar including nasolacrimal duct obstruction, corneal scarring, cicatricial alopecia of the eyelashes, recurrent conjunctivitis, blepharitis, and macular scarring. A 2.5-year-old white-Asian girl with a diagnosis of CEVD was referred for bilateral epiphora that began soon after birth. She was born at 27 weeks gestation, weighing 1,180 g. At birth, she was noted to have a red blistering eruption of her trunk, scalp, and extremities and was diagnosed with congenital erosive and vesicular dermatosis. Differential diagnosis included ectodermal dysplasia, such as Rapp-Hodgkin syndrome. Testing failed to identify any mutation in the TP63 gene. She remained hospitalized for the first 10 weeks of life. General examination showed reticulated scarring of her skin over the trunk, extremities, and scalp (Figure 1A); the palm, soles, and face were spared. Her teeth and hair were normal, and her tongue showed no scarring. Cognitive and motor development was appropriate. On ophthalmological examination, her visual acuity was 20/40 in both eyes. Bilateral upper and lower eyelid erythema was noted at the margins, along with partial absence of the eyelashes of the upper and lower lids. An increased tear lake was observed in both eyes (Figure 1B). Her corneas were clear centrally, with superficial inferior peripheral vascular pannus measuring 2 mm bilaterally. The anterior segment and pupil examinations were otherwise normal. The posterior segment was normal, with an unremarkable optic disk and macula in both eyes. The clinical diagnosis of bilateral nasolacrimal duct obstruction secondary to CEVD was made. Probing under general anesthesia was attempted for both eyes. On the right side the superior canaliculus was scarred and nonpatent and stenosis was present in the inferior and common canaliculus that required monocanalicular intubation. On the left side both superior and inferior canalicular and nasolacrimal duct stenosis was present and was successfully treated with bicanalicular intubation. The silicone tube became loose in the right eye and required removal 9 days postoperatively. The tube in the left eye was removed 5 weeks postoperatively. The tearing improved, but residual epiphora persisted bilaterally. Congenital erosive and vesicular dermatosis with reticulated scarring is a rare disorder of unknown origin. Only 18 cases have been published in the literature. The condition typically presents at birth in a premature neonate with superficial erosions, vesicles, and bullae that affect up to 75% of the body surface area.4Sidhu-Malik N.K. Resnick S.D. Wilson B.B. Congenital erosive and vesicular dermatosis healing with reticulated supple scarring: report of three new cases and review of the literature.Pediatr Dermatol. 1998; 15: 214-218Crossref PubMed Scopus (17) Google Scholar The erosions heal relatively rapidly, leaving characteristic supple, reticulated scarring.6Stein S. Stone S. Paller A.S. Ongoing blistering in a boy with congenital erosive and vesicular dermatosis healing with reticulated supple scarring.J Am Acad Dermatol. 2001; 45: 946-948Abstract Full Text Full Text PDF PubMed Scopus (19) Google Scholar The trunk and extremities are most severely involved, with relative sparing of the face and volar surfaces.6Stein S. Stone S. Paller A.S. Ongoing blistering in a boy with congenital erosive and vesicular dermatosis healing with reticulated supple scarring.J Am Acad Dermatol. 2001; 45: 946-948Abstract Full Text Full Text PDF PubMed Scopus (19) Google Scholar Of the 18 reported cases, 4 with eye involvement have been reported, 2 with corneal scarring,4Sidhu-Malik N.K. Resnick S.D. Wilson B.B. Congenital erosive and vesicular dermatosis healing with reticulated supple scarring: report of three new cases and review of the literature.Pediatr Dermatol. 1998; 15: 214-218Crossref PubMed Scopus (17) Google Scholar, 5Mashiah J. Wallach D. Leclerc-Mercier S. Bodemer C. Hadj-Rabia S. Congenital erosive and vesicular dermatosis: a new case and review of the literature.Pediatr Dermatol. 2012; 29: 756-758Crossref PubMed Scopus (12) Google Scholar 1 with nasolacrimal duct obstruction with recurrent conjunctivitis and blepharitis along with partial absence of eyelashes,6Stein S. Stone S. Paller A.S. Ongoing blistering in a boy with congenital erosive and vesicular dermatosis healing with reticulated supple scarring.J Am Acad Dermatol. 2001; 45: 946-948Abstract Full Text Full Text PDF PubMed Scopus (19) Google Scholar and 1 with macular scarring.2Gupta A.K. Rasmussen J.E. Headington J.T. Extensive congenital erosions and vesicles healing with reticulate scarring.J Am Acad Dermatol. 1987; 17: 369-376Abstract Full Text PDF PubMed Scopus (22) Google Scholar The literature review of ocular manifestations in CEVD is summarized in Table 1. CEVD associated with nasolacrimal obstruction was first reported by Stein and colleagues6Stein S. Stone S. Paller A.S. Ongoing blistering in a boy with congenital erosive and vesicular dermatosis healing with reticulated supple scarring.J Am Acad Dermatol. 2001; 45: 946-948Abstract Full Text Full Text PDF PubMed Scopus (19) Google Scholar in 2001, but details of the blockage were not documented. Our case affirms the association of CEVD and this type of nasolacrimal duct obstruction. Our probing findings suggested stenosis bilaterally in the canaliculi and nasolacrimal ducts. We believe that the stenosis and obstruction were attributable to scarring due to mucosal involvement along the lacrimal duct during the acute insult of the disease in the newborn. The reported mucosal involvement of the tongue in this disease in 3 cases supports our hypothesis.1Cohen B.A. Esterly N.B. Nelson P.F. Congenital erosive and vesicular dermatosis healing with reticulated supple scarring.Arch Dermatol. 1985; 121: 361-367Crossref PubMed Scopus (44) Google Scholar, 2Gupta A.K. Rasmussen J.E. Headington J.T. Extensive congenital erosions and vesicles healing with reticulate scarring.J Am Acad Dermatol. 1987; 17: 369-376Abstract Full Text PDF PubMed Scopus (22) Google Scholar, 3Plantin P. Delaire P. Guillois B. et al.Congenital erosive dermatosis with reticulated supple scarring: first neonatal report.Arch Dermatol. 1990; 126: 544-546Crossref PubMed Scopus (21) Google Scholar, 7De Lange A. Bayet B. Debauche C. et al.Congenital erosive and vesicular dermatosis with reticulated scarring in a newborn: an innovative treatment using a silicone dressing.Pediatr Dermatol. 2009; 26: 735-738Crossref PubMed Scopus (7) Google ScholarTable 1Literature review of ocular manifestations in congenital erosive and vesicular dermatosisReferenceReported cases with eye examinations (n = 6)Ocular featuresSidhu-Malik et al,4Sidhu-Malik N.K. Resnick S.D. Wilson B.B. Congenital erosive and vesicular dermatosis healing with reticulated supple scarring: report of three new cases and review of the literature.Pediatr Dermatol. 1998; 15: 214-218Crossref PubMed Scopus (17) Google Scholar Mashiah et al,5Mashiah J. Wallach D. Leclerc-Mercier S. Bodemer C. Hadj-Rabia S. Congenital erosive and vesicular dermatosis: a new case and review of the literature.Pediatr Dermatol. 2012; 29: 756-758Crossref PubMed Scopus (12) Google Scholar present study3 (50%)Corneal scar/neovascularizationStein et al,6Stein S. Stone S. Paller A.S. Ongoing blistering in a boy with congenital erosive and vesicular dermatosis healing with reticulated supple scarring.J Am Acad Dermatol. 2001; 45: 946-948Abstract Full Text Full Text PDF PubMed Scopus (19) Google Scholar present study2 (33%)Nasolacrimal obstructionStein et al,6Stein S. Stone S. Paller A.S. Ongoing blistering in a boy with congenital erosive and vesicular dermatosis healing with reticulated supple scarring.J Am Acad Dermatol. 2001; 45: 946-948Abstract Full Text Full Text PDF PubMed Scopus (19) Google Scholar present study2 (33%)Partial absence of eye lashesStein et al,6Stein S. Stone S. Paller A.S. Ongoing blistering in a boy with congenital erosive and vesicular dermatosis healing with reticulated supple scarring.J Am Acad Dermatol. 2001; 45: 946-948Abstract Full Text Full Text PDF PubMed Scopus (19) Google Scholar present study2 (33%)BlepharitisStein et al6Stein S. Stone S. Paller A.S. Ongoing blistering in a boy with congenital erosive and vesicular dermatosis healing with reticulated supple scarring.J Am Acad Dermatol. 2001; 45: 946-948Abstract Full Text Full Text PDF PubMed Scopus (19) Google Scholar1 (17%)Recurrent conjunctivitisGupta et al2Gupta A.K. Rasmussen J.E. Headington J.T. Extensive congenital erosions and vesicles healing with reticulate scarring.J Am Acad Dermatol. 1987; 17: 369-376Abstract Full Text PDF PubMed Scopus (22) Google Scholar1 (17%)Macular scarDe Lange7De Lange A. Bayet B. Debauche C. et al.Congenital erosive and vesicular dermatosis with reticulated scarring in a newborn: an innovative treatment using a silicone dressing.Pediatr Dermatol. 2009; 26: 735-738Crossref PubMed Scopus (7) Google Scholar1 (17%)Normal eye examination Open table in a new tab Our case highlights difficulties in managing the bilateral canalicular and nasolacrimal duct obstruction associated with CEVD. Simple probing is unlikely to be effective in these cases. Silicone intubation during the early stages of the disease to prevent closure of the nasolacrimal ducts from scarring may be indicated. In cases where scarring recurs, further balloon catheter dilatation may be required. Other ocular manifestations of CEVD include corneal scarring, alopecia of eyelashes, recurrent conjunctivitis, blepharitis, and macular scarring. Corneal scarring due to corneal neovascularization is the most common manifestation of CEVD. Mashiah and colleauges5Mashiah J. Wallach D. Leclerc-Mercier S. Bodemer C. Hadj-Rabia S. Congenital erosive and vesicular dermatosis: a new case and review of the literature.Pediatr Dermatol. 2012; 29: 756-758Crossref PubMed Scopus (12) Google Scholar reported corneal neovascularization affecting visual acuity and requiring cyclosporine treatment. Details of management of corneal scarring in another case were not reported.4Sidhu-Malik N.K. Resnick S.D. Wilson B.B. Congenital erosive and vesicular dermatosis healing with reticulated supple scarring: report of three new cases and review of the literature.Pediatr Dermatol. 1998; 15: 214-218Crossref PubMed Scopus (17) Google Scholar Corneal neovascularization in our patient was peripheral and did not affect visual acuity; treatment was not required. Corneal neovascularization and scarring in these cases may possibly be due to previous keratoconjunctivitis occurring during the acute phase of the disease. Associated recurrent conjunctivitis and blepharitis in another case6Stein S. Stone S. Paller A.S. Ongoing blistering in a boy with congenital erosive and vesicular dermatosis healing with reticulated supple scarring.J Am Acad Dermatol. 2001; 45: 946-948Abstract Full Text Full Text PDF PubMed Scopus (19) Google Scholar are likely secondary to significant nasolacrimal duct obstruction. The authors conducted a MEDLINE (PubMed) search for the period1966 to 2012 using the following terms: congenital erosive AND vesicular dermatosis, ocular diseases, nasolacrimal obstruction. Only articles published in English language were considered." @default.
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• W2021199113 title "Bilateral canalicular and nasolacrimal duct obstruction in congenital erosive and vesicular dermatosis: a case report and review of the literature" @default.
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