Evidence map›Paper›PMID 39044220›Full record

ArticleOrphanet journal of rare diseases2024

A multi-step approach to overcome challenges in the management of head and neck lymphatic malformations, and response to treatment.

Valentina Trevisan, Eugenio De Corso, Germana Viscogliosi, Roberta Onesimo, Alessandro Cina, Marco Panfili, Lucrezia Perri, Cristiana Agazzi, Valentina Giorgio, Donato Rigante and 11 more

Registry-linked trialAbstract read
In one paragraph

Article in Orphanet journal of rare diseases, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT06892964 (Prospective and Retrospective Clinical Study of Lymphatic Malformations), which is not on this map. Cited by 2 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed, 1 pooled it
–field-weighted citation impact
1 · What the graph read from 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.

2 · The registry

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.

NCT06892964 narecruitingnot on this map

Prospective and Retrospective Clinical Study of Lymphatic Malformations: Institution of an Italian REGistry of Lymphatic Malformations and of a Biobank for Biological Sample Collection

TypeinterventionalSponsorFondazione Policlinico Universitario Agostino Gemelli IRCCSRan2024 to 2044Enrolled70ConditionsLymphatic Malformation, Lymphatic Abnormality, Lymphangioma, Lymphatic AbnormalitiesArmsGenetic profiling of all eligible patients
3 · Its place in the literature

Who cites it

2 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
  2. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

21 authors.

Valentina TrevisanCenter for Rare Diseases and Birth Defects, Department of Woman and Child Health and Public Health, Fondazione Policlinico Universitario "A. Gemelli" IRCCS, Largo A. Gemelli 8, Rome, 00168, RM, Italy.
Eugenio De CorsoUnit of Otorhinolaryngology, Fondazione Policlinico Universitario "A. Gemelli" IRCCS, Rome, RM, Italy.
Germana ViscogliosiUOC Radiodiagnostica e Neuroradiologia, Dipartimento di Diagnostica per Immagini, Fondazione Policlinico Universitario A. Gemelli IRCCS, Radioterapia, Rome, Italy.
Roberta OnesimoCenter for Rare Diseases and Birth Defects, Department of Woman and Child Health and Public Health, Fondazione Policlinico Universitario "A. Gemelli" IRCCS, Largo A. Gemelli 8, Rome, 00168, RM, Italy.
Alessandro CinaUOC Radiodiagnostica e Neuroradiologia, Dipartimento di Diagnostica per Immagini, Fondazione Policlinico Universitario A. Gemelli IRCCS, Radioterapia, Rome, Italy.
Marco PanfiliUOC Radiodiagnostica e Neuroradiologia, Dipartimento di Diagnostica per Immagini, Fondazione Policlinico Universitario A. Gemelli IRCCS, Radioterapia, Rome, Italy.
Lucrezia PerriCenter for Rare Diseases and Birth Defects, Department of Woman and Child Health and Public Health, Fondazione Policlinico Universitario "A. Gemelli" IRCCS, Largo A. Gemelli 8, Rome, 00168, RM, Italy.
Cristiana AgazziCenter for Rare Diseases and Birth Defects, Department of Woman and Child Health and Public Health, Fondazione Policlinico Universitario "A. Gemelli" IRCCS, Largo A. Gemelli 8, Rome, 00168, RM, Italy.
Valentina GiorgioCenter for Rare Diseases and Birth Defects, Department of Woman and Child Health and Public Health, Fondazione Policlinico Universitario "A. Gemelli" IRCCS, Largo A. Gemelli 8, Rome, 00168, RM, Italy.
Donato RiganteCenter for Rare Diseases and Birth Defects, Department of Woman and Child Health and Public Health, Fondazione Policlinico Universitario "A. Gemelli" IRCCS, Largo A. Gemelli 8, Rome, 00168, RM, Italy.
Giovanni VentoUniversità Cattolica del Sacro Cuore, Rome, RM, Italy.
Patrizia PapacciNeonatology Unit, Department of Woman and Child Health and Public Health, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Rome, 00168, Italy.
Filomena Valentina ParadisoScuola di Specializzazione in Chirurgia Pediatrica, Università Cattolica Sacro Cuore, Roma, Italy.
Sara SilvaroliScuola di Specializzazione in Chirurgia Pediatrica, Università Cattolica Sacro Cuore, Roma, Italy.
Lorenzo NanniUniversità Cattolica del Sacro Cuore, Rome, RM, Italy.
Nicoletta RestaDepartment of Biomedical Sciences and Human Oncology (DIMO), Medical Genetics, University of Bari "Aldo Moro", Bari, Italy.
Marco CastoriDivision of Medical Genetics, Fondazione IRCCS-Casa Sollievo della Sofferenza, Foggia, San Giovanni Rotondo, Italy.
Jacopo GalliUnit of Otorhinolaryngology, Fondazione Policlinico Universitario "A. Gemelli" IRCCS, Rome, RM, Italy.
Gaetano PaludettiUniversità Cattolica del Sacro Cuore, Rome, RM, Italy.
Giuseppe ZampinoCenter for Rare Diseases and Birth Defects, Department of Woman and Child Health and Public Health, Fondazione Policlinico Universitario "A. Gemelli" IRCCS, Largo A. Gemelli 8, Rome, 00168, RM, Italy.
Chiara LeoniCenter for Rare Diseases and Birth Defects, Department of Woman and Child Health and Public Health, Fondazione Policlinico Universitario "A. Gemelli" IRCCS, Largo A. Gemelli 8, Rome, 00168, RM, Italy. chiara.leoni@policlinicogemelli.it.ORCID 0000-0002-4089-637X

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundLymphatic malformations are vascular developmental anomalies varying from local superficial masses to diffuse infiltrating lesions, resulting in disfigurement. Patients' outcomes range from spontaneous regression to severe sequelae notwithstanding appropriate treatment. The current classification guides, in part, clinicians through the decision-making process, prognosis prediction and choice of therapeutic strategies. Even though the understanding of molecular basis of the disease has been recently improved, a standardized management algorithm has not been reached yet.

resultsHere, we report our experience on five children with different lymphatic anomalies of the head and neck region treated by applying a multidisciplinary approach reaching a consensus among specialists on problem-solving and setting priorities.

conclusionsAlthough restitutio ad integrum was rarely achieved and the burden of care is challenging for patients, caregivers and healthcare providers, this study demonstrates how the referral to expert centres can significantly improve outcomes by alleviating parental stress and ameliorating patients' quality of life. A flow-chart is proposed to guide the multidisciplinary care of children with LMs and to encourage multidisciplinary collaborative initiatives to implement dedicated patients' pathways.

Indexed as

Lymphatic AbnormalitiesChildChild, PreschoolFemaleHeadHumansInfantMaleNeckQuality of LifeAlpelisibLymphangiomaLymphatic malformationsmTORPersonalized medicineSirolimus

Identifiers

PMID39044220
PMCPMC11265367

What Socratic holds

Textmetadata
LicenceCC BY
Read underepoch 390

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 Socratic graph.