Aplasia cutis congenita as a result of interstitial laser therapy for fetal reduction in monochorionic twins: Conservative approach and outcome

Samedi, Veronica Mugarab; Lodha, Abhay; Elsharkawy, Adel; Awad, Essa Al

doi:10.1016/j.ijscr.2017.10.007

Cited by 6 publications

(5 citation statements)

References 10 publications

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“…This differs from published experience that death before 14 weeks of gestation results in ACC usually on the trunk, while after 14 weeks it is mostly on the extremities 7 . The same pattern of symmetrical, extensive ACC occurs after selective foetal reduction, and in both situations can be attributed to placental damage secondary to twin death, without implicating laser burns 8 …”

Section: Resultscontrasting

confidence: 77%

“…7 The same pattern of symmetrical, extensive ACC occurs after selective foetal reduction, and in both situations can be attributed to placental damage secondary to twin death, without implicating laser burns. 8 Type 6: aplasia cutis congenita associated with epidermolysis bullosa (Bart syndrome)…”

Section: Type 1: Scalp Aplasia Cutis Congenita Without Multiple Anomaliesmentioning

confidence: 99%

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Classification of aplasia cutis congenita: a 25‐year review of cases presenting to a tertiary paediatric dermatology department

Sathishkumar

Ogboli

Moss

2020

Clin. Exp. Dermatol.

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Background Aplasia cutis congenita (ACC) is a rare, congenital disorder characterized by localized or widespread absence of skin at birth with heterogeneous clinical presentation. The classification proposed by Frieden in 1986 is widely used. Aim To establish whether, 34 years on, the Frieden classification still meets the needs of dermatologists. Methods We conducted a retrospective chart review of all patients with a diagnosis of ACC presenting over a 25‐year period to a tertiary paediatric dermatology department. We compiled demographic data, clinical characteristics (e.g. number, location and morphology of the lesions), imaging and genetic results where available, and other associated abnormalities, and grouped them according to the Frieden classification. For Type 6 ACC (Bart syndrome) we reviewed neonatal photographs of all babies born with epidermolysis bullosa (EB) over 5 years. Results Excluding Type 6, there were 56 children with ACC. The scalp was involved in 82.1%, and Type 1 was the commonest type. Over 5 years, 13 of 108 neonates (12%) with EB were born with the appearance of Type 6 ACC. Two children did not fit Frieden’s original classification and one had a previously undescribed association of ACC with cleft lip/palate‐ectodermal dysplasia 1 syndrome. Conclusion We conclude that the Frieden classification remains valid with some modifications. Type 3 ACC probably represents a mosaic RASopathy syndrome, while Type 7 could cover nongenetic ACC attributable to trauma. Type 8 should be subdivided into two subgroups: teratogenic and infective. Type 9 covers at least four subgroups. The classification will continue to evolve as new genes and pathomechanisms emerge.

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Section: Resultscontrasting

confidence: 77%

Section: Type 1: Scalp Aplasia Cutis Congenita Without Multiple Anomaliesmentioning

confidence: 99%

Classification of aplasia cutis congenita: a 25‐year review of cases presenting to a tertiary paediatric dermatology department

Sathishkumar

Ogboli

Moss

2020

Clin. Exp. Dermatol.

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“…One case series reported ACC incidence of 8% (2 of 26 newborns after laser ablation at 13 and 14 weeks) after MFPR. 10 One similar case report suggested that the mechanism of ACC was laser burn 11 ; however, a baby born after spontaneous cotwin loss during a MCDA pregnancy presented similar truncal and thigh lesions characteristic of ACC type 5, 12 thereby disproving laser burn as the plausible cause.…”

Section: The Incidence Of Multiple Gestation In Southmentioning

confidence: 99%

A case of aplasia cutis congenita following fetal reduction of triplet pregnancy conceived through in vitro fertilization

Kim¹,

Kim²,

Min³

et al. 2022

Turk J Pediatr

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Background. Aplasia cutis congenita (ACC) is a rare congenital localized skin defect that is mostly diagnosed in the newborn or infant period. ACC type 5 often involves the trunk or extremities accompanied by fetus papyraceous (FP) or placental infarcts. The etiology and pathogenesis of this rare type of ACC are not well known. In this case, we report an ACC type 5 with a definite etiology.Case. We report a preterm infant with ACC type 5, with diffuse bilateral leg lesions found at birth. He was the first baby of dichorionic twin after reduction from a dichorionic triplet pregnancy conceived through in vitro fertilization. A fetus papyraceous was found in juxtaposition with the affected baby's placenta. After 37 days of hospitalization, his leg lesions were successfully epithelized with supportive care. He is regularly visiting the Dermatology clinic for scar care and shows normal development without motor limitation.Conclusions. Herein, we present a preterm infant with ACC type 5 and the placental pathology with fetus papyraceous of the artificially reduced monochorionic co-twin of the affected infant. We suggest a precautious decision in multifetal pregnancy reduction (MFPR) in dichorionic triplets, presenting ACC type 5 as an adverse outcome of MFPR.

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“…The exact etiology of ACC is not completely understood. Several factors have been proposed as possible causes, e.g., vascular disruption, thrombotic events, trauma, amniotic defects, chromosomal abnormalities or gene mutations, ectodermal dysplasia, defective closure of the neural tube, and teratogenic events such as intrauterine infections, or drugs and medications during pregnancy [4][5][6].…”

Section: Introductionmentioning

confidence: 99%

“…The most common lesion location of ACC is the scalp in 70% [ 7 , 8 ]; however, any skin site can be affected [ 7 ]. The estimated incidence of ACC is approximately 0.5–3 in 10,000 newborns [ 2 , 5 ], regardless of gender or ethnicity [ 5 ]. The clinical appearance of ACC is heterogeneous and it can be associated with various systemic congenital anomalies.…”

Section: Introductionmentioning

confidence: 99%

Membranous aplasia cutis congenita in trisomy 18

Cammarata‐Scalisi

Diociaiuti

Guerrero³

et al. 2020

Ital J Pediatr

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Background: Aplasia cutis congenita (ACC) is a rare congenital condition characterized by the absence of skin layers and sometimes other underlying structures, in a localized or widespread area. The exact etiopathogenesis is not yet completely understood. Membranous ACC (MACC) also described as bullous or cystic ACC is a clinical subtype of ACC, covered with a membranous or glistening surface, and appears as a flat scar. There are less than 20 cases reported in the literature. It has been proposed an abortive form of a defective closure of the neural tube. On the other hand, the trisomy 18 is a chromosomal abnormality characterized by a broad clinical spectrum and the presence of defective closure of the neural tube. Case presentation: We report on an 18-months-old Venezuelan boy, who presented on the parietal scalp a distinctive localized MACC appearing as an oval lesion covered with a membranous surface, characterized by the absence of hairs and the presence of a sharp hair collar. The karyotype in peripheral blood was 47,XY,+ 18. Conclusions: This is the second case report of ACC in trisomy 18 and reinforces the interpretation of a nonfortuitous association as well as of a defective closure of the neural tube as pathogenetic mechanism. The case highlights the importance of examining for dermatological alterations such as ACC in cases of chromosomopathy.

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Aplasia cutis congenita as a result of interstitial laser therapy for fetal reduction in monochorionic twins: Conservative approach and outcome

Cited by 6 publications

References 10 publications

Classification of aplasia cutis congenita: a 25‐year review of cases presenting to a tertiary paediatric dermatology department

Classification of aplasia cutis congenita: a 25‐year review of cases presenting to a tertiary paediatric dermatology department

A case of aplasia cutis congenita following fetal reduction of triplet pregnancy conceived through in vitro fertilization

Membranous aplasia cutis congenita in trisomy 18

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