An Exploratory Clinical Trial of a Novel Treatment for Giant Congenital Melanocytic Nevi Combining Inactivated Autologous Nevus Tissue by High Hydrostatic Pressure and a Cultured Epidermal Autograft: Study Protocol

Morimoto, Naoki; Jinno, Chizuru; Sakamoto, Michiharu; Kakudo, Natsuko; Yamaoka, Tetsuji; Kusumoto, Kenji

doi:10.2196/resprot.6195

Cited by 13 publications

(14 citation statements)

References 18 publications

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“…As for the cutoff point of 50%, this is based on the consensus that a wound size reduction of 20% to 40% in 2 and 4 weeks is a reliable predictor of healing at 12 weeks in the treatment of EB. Also, in the clinical trial of CEA for postoperative wounds of giant congenital melanocytic nevi, engraftment of 60% or more is reported to be a proper cutoff point (Morimoto et al, 2016). Taking these findings into consideration, we set the cutoff point at 50% or more in this clinical trial.…”

Section: Primary Endpointmentioning

confidence: 99%

Cultured Epidermal Autografts from Clinically Revertant Skin as a Potential Wound Treatment for Recessive Dystrophic Epidermolysis Bullosa

Matsumura

Fujita

Shinkuma

et al. 2019

Journal of Investigative Dermatology

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Inherited skin disorders have been reported recently to have sporadic normal-looking areas, where a portion of the keratinocytes have recovered from causative gene mutations (revertant mosaicism). We observed a case of recessive dystrophic epidermolysis bullosa treated with cultured epidermal autografts (CEAs), whose CEAgrafted site remained epithelized for 16 years. We proved that the CEA product and the grafted area included cells with revertant mosaicism. Based on these findings, we conducted an investigator-initiated clinical trial of CEAs from clinically revertant skin for recessive dystrophic epidermolysis bullosa. The donor sites were analyzed by genetic analysis, immunofluorescence, electron microscopy, and quantification of the reverted mRNA with deep sequencing. The primary endpoint was the ulcer epithelization rate per patient at 4 weeks after the last CEA application. Three patients with recessive dystrophic epidermolysis bullosa with 8 ulcers were enrolled, and the epithelization rate for each patient at the primary endpoint was 87.7%, 100%, and 57.0%, respectively. The clinical effects were found to persist for at least 76 weeks after CEA transplantation. One of the three patients had apparent revertant mosaicism in the donor skin and in the post-transplanted area. CEAs from clinically normal skin are a potentially well-tolerated treatment for recessive dystrophic epidermolysis bullosa.

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Section: Primary Endpointmentioning

confidence: 99%

Cultured Epidermal Autografts from Clinically Revertant Skin as a Potential Wound Treatment for Recessive Dystrophic Epidermolysis Bullosa

Matsumura

Fujita

Shinkuma

et al. 2019

Journal of Investigative Dermatology

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“…A novel surgical technique showing promising results in GCMN is full-thickness excision of the nevus tissue, followed by its inactivation by high hydrostatic pressure (200 MPa for 10 min) and replanting it to its original site [ 9 , 10 ]. However, more clinical trials with a larger sample size and longer follow-up periods are needed to determine the success rate of this technique.…”

Section: Discussionmentioning

confidence: 99%

Giant Congenital Melanocytic Nevi: An Update and Emerging Therapies

2018

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Giant congenital melanocytic nevi (GCMN) are a rare occurrence. Gain-of-function mutation in the NRAS gene is found to be associated with GCMN, causing abnormal proliferation of embryonic melanoblasts. The two major complications associated with GCMN are malignant melanoma and neurocutaneous melanosis. Treatment of GCMN has conventionally been surgical. However, the role of NRAS inhibitors and inactivation of nevus tissue by high hydrostatic pressure are being explored. We present a case of a 1-day-old neonate born with GCMN, along with a review of the literature.

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“…Traditional surgery techniques including tissue expansion are mature, but its limits have inhibited its applying in GCMN treatment. Morimoto et al [ 9 ] had treated the GCMN combining inactivated autologous nevus tissue by high hydrostatic pressure and a cultured epidermal auto graft and achieved great results. And this method not only diminishes the damage of the normal skin, but also acquires good appearance.…”

Section: Discussionmentioning

confidence: 99%

An alternative strategy treated giant congenital melanocytic nevi with epidermis and superficial dermis of the lesions

Wang²,

Luo³

et al. 2018

Medicine

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Giant congenital melanocytic nevi (GCMN) are defined as rare pigmented lesions that are believed to form between weeks 9 and 20 of gestation. It is difficult to reconstruct large defects after the removal of the lesions and it has posed a great challenge to the plastic surgeon and dermatologist.Given all those difficulty reconstructing the defects, we try to explore an alternative way to resurfacing the defect after removal of GCMN.Patients with GCMN received single-stage excision. Following the subcutaneous tissue and deep dermis were discarded, epidermis and superficial dermis were harvested as graft substitutes to reconstruct the defects in situ.All of the grafted tissue survived well and skin color in the surgical area gradually became lighter. During the periodicity of follow-up, neither hypertrophic scars nor recurrence were observed. Furthermore, histopathology examination demonstrated that there are no distinct melanocytes gathered in the postoperation lesions.For those GCMN which is difficult to reconstruct with traditional methods, resection of the lesion followed by reconstruction with epidermis skin and superficial dermis from the lesions in situ may be a feasible and alternative therapy method.

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An Exploratory Clinical Trial of a Novel Treatment for Giant Congenital Melanocytic Nevi Combining Inactivated Autologous Nevus Tissue by High Hydrostatic Pressure and a Cultured Epidermal Autograft: Study Protocol

Cited by 13 publications

References 18 publications

Cultured Epidermal Autografts from Clinically Revertant Skin as a Potential Wound Treatment for Recessive Dystrophic Epidermolysis Bullosa

Cultured Epidermal Autografts from Clinically Revertant Skin as a Potential Wound Treatment for Recessive Dystrophic Epidermolysis Bullosa

Giant Congenital Melanocytic Nevi: An Update and Emerging Therapies

An alternative strategy treated giant congenital melanocytic nevi with epidermis and superficial dermis of the lesions

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