ArticleMedicine2017
The use of sirolimus in the treatment of giant cystic lymphangioma: Four case reports and update of medical therapy.
Article in Medicine, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers, 1 of them a synthesis that pooled it.
What it found
Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.
The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
18 citing papers in PubMed, 1 synthesis or guideline pooled it, 45 citations in OpenAlex.
- Sirolimus for vascular anomalies in the first year of life: a systematic review.Journal of perinatology : official journal of the California Perinatal Association · 2024Pooled it
- Sirolimus Therapy and Dyslipidemia in Neonates and Infants: A Retrospective Cohort and Pharmacovigilance Analysis.Paediatric drugs · 2026Article
- Intrauterine Bleomycin Administration for Fetal Lymphatic Malformation: A Novel Therapeutic Approach-Case Report and Literature Review.Journal of clinical medicine · 2026Review
- Prenatal ultrasound diagnosis, intrauterine monitoring and postnatal management of a giant fetal abdominopelvic lymphangioma: a case report and scoping review.Frontiers in pediatrics · 2026Article
- Laparoscopic management of giant abdominal cystic lymphangiomas: experience from the queen Fabiola children's university hospital.BMC surgery · 2025Article
- A refractory right cervical mediastinal lymphangioma with medication and insertion of a Denver shunt.Journal of surgical case reports · 2024Article
- A multi-step approach to overcome challenges in the management of head and neck lymphatic malformations, and response to treatment.Orphanet journal of rare diseases · 2024Article
- Successful Management of Retropharyngeal Lymphangioma with Stridor Using Sirolimus: A Case Report.Indian journal of otolaryngology and head and neck surgery : official publication of the Association of Otolaryngologists of India · 2024Article
- Outcome of Sclerotherapy in a Preterm Infant with a Giant Fetal Neck Lymphatic Malformation: A Case Report.International journal of women's health · 2023Article
- Antiproliferative therapy with sirolimus and propranolol for congenital vascular anomalies in newborns (Case reports).Experimental and therapeutic medicine · 2021Article
- Lymphangioma of the fetal neck within the PIK3CA-related-overgrowth spectrum (PROS): A case report.Clinical case reports · 2021Article
- Optimization of Initial Dose Regimen for Sirolimus in Pediatric Patients With Lymphangioma.Frontiers in pharmacology · 2021Article
- Operative Management of Neonatal Lymphatic Malformations: Lesson Learned From 57 Consecutive Cases.Frontiers in pediatrics · 2021Article
- Positron emission tomography/computed tomography findings of multiple cystic lymphangiomas in an adult: A case report.World journal of clinical cases · 2020Article
- Early Adjuvant Medication With the mTOR Inhibitor Sirolimus in a Preterm Neonate With Compressive Cystic Lymphatic Malformation.Frontiers in pediatrics · 2020Article
- Successful treatment of diffuse pulmonary lymphangiomatosis with sirolimus.Respiratory medicine case reports · 2020Article
- Article
- Novel clinical phenotype of generalised lymphatic dysplasia in a neonate: a missed diagnosis.BMJ case reports · 2019Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
11 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
rationaleLymphatic malformations (LMs) are rare and benign anomalies resulting from the defective embryological development of the primordial lymphatic structures. Due to their permeative growth throughout all tissue layers, treatment is often challenging. Small asymptomatic lesions can be conservatively managed, while symptomatic lesions require active management. Surgery has been historically considered the treatment of choice, but today less invasive therapeutic options are preferred (sclerotherapy, laser therapy, oral medications). However, there are not uniform therapeutic protocols. Sirolimus is an oral medication that has been reported to be effective in the recent literature. Here we present the case of 4 newborns with giant multicystic lymphangioma treated with oral sirolimus after surgical resection had failed. PATIENT CONCERNS: At birth the LMs were clinically appreciated as giant masses involving different organs and structures. DIAGNOSES: All patients had a prenatal diagnosis of giant multicystic lymphangioma confirmed at histological and cytological analysis.
interventionsPatients were treated with oral sirolimus after unsuccessful surgical resection. OUTCOMES: In all patients, sirolimus determined an overall reduction of the mass and a global involution from the macro- to the microcystic composition. Sirolimus was safe and poor disadvantages had been observed. The main and isolated adverse effect at laboratory analysis was progressive dyslipidemia, with increasing levels of total cholesterol and triglycerides. LESSONS: To date, our experience with sirolimus in the management of LMs is favorable. We recommend the use of sirolimus after unsuccessful surgical excision have been tried or when the surgical approach is not feasible. A multidisciplinary follow-up is needed to monitor disease evolution.
Indexed as
Identifiers
What OpenQuestion holds
Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.