Treatment of complex periorbital venolymphatic malformation in a neonate with a combination therapy of sirolimus and prednisolone

Dermatologic Therapy
David KimJoyce Teng

Abstract

Venolymphatic malformations (VLMs) are vascular anomalies consisting of both veins and lymph vessels. A 2-week-old newborn presented with large VLMs on the left forehead, temple, preauricular area, and orbit. Patient was at imminent risk for permanent vision loss due to a localized mass effect. Surgical excision or debulking was contraindicated due to its complexity and proximity to the left eye, and the patient failed to respond to the sildenafil treatment and sclerotherapy. Patient was subsequently started on oral sirolimus 0.8 mg/m(2) twice daily in combination with prednisolone 2 mg/kg daily. The patient had an excellent therapeutic outcome for 7 months with complete preservation of vision before treatment was discontinued. However, 2 months after the medical treatments were discontinued, her VLM rebounded. She responded to the combination therapy again after a failed treatment with the mTOR inhibitor alone. This case demonstrates that the sirolimus and prednisolone combination therapy could be beneficial for treatment of complex VLM intractable to other treatments.

References

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Citations

Jun 5, 2015·European Journal of Pediatrics·Herwig LacknerChristian E Urban
Nov 8, 2016·Journal of Pediatric Hematology/oncology·Dov C GoldenbergRolf Gemperli
Jun 21, 2018·Lymphatic Research and Biology·Susanne WiegandAndreas Dietz
Aug 29, 2018·Pediatric Dermatology·Michael A Cardis, Cynthia M C DeKlotz
Jan 24, 2019·Pediatric Blood & Cancer·Kiersten W RicciDenise M Adams
Feb 7, 2018·Orbit·Wolf A LagrèzeJochen Rössler
Jun 25, 2019·Current Opinion in Ophthalmology·Stacey C Lam, Hunter K L Yuen
Dec 7, 2018·Current Treatment Options in Pediatrics·Reema PadiaJonathan Perkins
Jan 29, 2020·Ophthalmic Plastic and Reconstructive Surgery·Marissa K ShojiSuzanne K Freitag
Sep 26, 2020·Clinics in Dermatology·Shayan CheraghlouKeith A Choate
Mar 20, 2021·Journal of Vascular Surgery. Venous and Lymphatic Disorders·Marlies Geeurickx, Veerle Labarque
Jun 4, 2021·Journal of Vascular Surgery·Yi JiYongbo Zhang
Mar 30, 2019·Journal of Vascular Surgery·Arash FereydooniNaiem Nassiri
Jan 20, 2016·Pediatrics·Denise M AdamsRichard G Azizkhan

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