Evidence map›Paper›PMID 33738294›Full record

ArticleFrontiers in medicine2021

Challenges in Transition From Childhood to Adulthood Care in Rare Metabolic Diseases: Results From the First Multi-Center European Survey.

Karolina M Stepien, Beata Kieć-Wilk, Christina Lampe, Trine Tangeraas, Graziella Cefalo, Nadia Belmatoug, Rita Francisco, Mireia Del Toro, Leona Wagner, Anne-Grethe Lauridsen and 9 more

Open access · goldAbstract read
In one paragraph

Article in Frontiers in medicine, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 42 papers.

0numbers the graph read from it
0cells of the map it votes in
42citing papers in PubMed
16.3field-weighted citation impact, top 1% of its field
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.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

42 citing papers in PubMed, 69 citations in OpenAlex.

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  12. Bridging the knowledge gap in rare bone disorders: insights from the APCO Asia-Pacific Rare Bone Disorders Engagement (ASPIRE) survey.Osteoporosis international : a journal established as result of cooperation between the European Foundation for Osteoporosis and the National Osteoporosis Foundation of the USA · 2025
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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

19 authors at 10 institutions in 7 countries.

Karolina M StepienAdult Inherited Metabolic Diseases, Salford Royal NHS Foundation Trust, Salford, United Kingdom.
Beata Kieć-WilkDepartment of Metabolic Diseases and Diabetes, Krakow University Hospital, Krakow, Poland.
Christina LampeDepartment of Child Neurology, Center for Rare Diseases Giessen (ZSEGI), Justus-Liebig University, Giessen, Germany.
Trine TangeraasNorwegian National Unit for Newborn Screening, Division of Paediatric and Adolescent Medicine, Oslo University Hospital, Oslo, Norway.
Graziella CefaloDepartment of Maternal and Child Health, San Paolo Hospital, University of Milan, ASST Santi Paolo e Carlo, Milan, Italy.
Nadia BelmatougReferral Center for Lysosomal Diseases, AP-HP Nord, Beaujon Hospital, Paris University, Clichy, France.
Rita FranciscoPortuguese Association for Congenital Disorders of Glycosylation and other Rare Metabolic Diseases, Lisbon, Portugal.
Mireia Del ToroPediatric Neurology Department, University Hospital Vall d'Hebron, Universitat Autónoma de Barcelona, Barcelona, Spain.
Leona WagnerGerman-Speaking Self-Help Group for Alkaptonuria (DSAKU) e.V., Stuttgart, Germany.
Anne-Grethe LauridsenInternational Gaucher Alliance, Dursley, United Kingdom.
Sylvia SestiniItalian Association of Patients With Alkaptonuria (aimAKU), Siena, Italy.
Nathalie WeinholdMetabolic Unit, Interdisciplinary Centre for Metabolism: Endocrinology, Diabetes and Metabolism (UP) and Children's Hospital, Charité University Hospital Berlin, Berlin, Germany.
Andreas HahnDepartment of Child Neurology, Justus-Liebig University, Giessen, Germany.
Chiara MontanariDepartment of Maternal and Child Health, San Paolo Hospital, University of Milan, ASST Santi Paolo e Carlo, Milan, Italy.
Valentina RovelliDepartment of Maternal and Child Health, San Paolo Hospital, University of Milan, ASST Santi Paolo e Carlo, Milan, Italy.
Cinzia M BellettatoMetabERN, Regional Coordinating Center for Rare Diseases, Udine University Hospital, Udine, Italy.
Laura PaneghettiMetabERN, Regional Coordinating Center for Rare Diseases, Udine University Hospital, Udine, Italy.
Corine van LingenMetabERN, Regional Coordinating Center for Rare Diseases, Udine University Hospital, Udine, Italy.
Maurizio ScarpaMetabERN, Regional Coordinating Center for Rare Diseases, Udine University Hospital, Udine, Italy.
University of Udine · ITOspedale San Paolo · ITUniversity of Giessen · DEAssistance Publique – Hôpitaux de Paris · FRCharité - Universitätsmedizin Berlin · DEKrakow Cardiovascular Research Institute · PLOslo University Hospital · NOSalford Royal NHS Foundation Trust · GBUniversitat Autònoma de Barcelona · ESUniversity of Milan · IT

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Inherited Metabolic Diseases (IMDs) are rare diseases caused by genetic defects in biochemical pathways. Earlier diagnosis and advances in treatment have improved the life expectancy of IMD patients over the last decades, with the majority of patients now surviving beyond the age of 20. This has created a new challenge: as they grow up, the care of IMD patients' needs to be transferred from metabolic pediatricians to metabolic physicians specialized in treating adults, through a process called "transition." The purpose of this study was to assess how this transition is managed in Europe: a survey was sent to all 77 centers of the European Reference Network for Hereditary Metabolic Disorders (MetabERN) to collect information and to identify unmet needs regarding the transition process. Data was collected from 63/77 (81%) healthcare providers (HCPs) from 20 EU countries. Responders were mostly metabolic pediatricians; of these, only ~40% have received appropriate training in health issues of adolescent metabolic patients. In most centers (~67%) there is no designated transition coordinator. About 50% of centers provide a written individualized transition protocol, which is standardized in just ~20% of cases. In 77% of centers, pediatricians share a medical summary, transition letter and emergency plan with the adult team and the patient. According to our responders, 11% of patients remain under pediatric care throughout their life. The main challenges identified by HCPs in managing transition are lack of time and shortage of adult metabolic physician positions, while the implementations that are most required for a successful transition include: medical staff dedicated to transition, a transition coordinator, and specific metabolic training for adult physicians. Our study shows that the transition process of IMD patients in Europe is far from standardized and in most cases is inadequate or non-existent. A transition coordinator to facilitate collaboration between the pediatric and adult healthcare teams should be central to any transition program. Standardized operating procedures, together with adequate financial resources and specific training for adult physicians focused on IMDs are the key aspects that must be improved in the rare metabolic field to establish successful transition processes in Europe.

Indexed as

adulthood (18 years and older)adult metabolic patientchallengecontinuity of careinherited metabolic disease(s)rare diseasetransition process

Identifiers

PMID33738294
PMCPMC7962750
OpenAlexW3129998749

What OpenQuestion holds

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Registered trials

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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.