MRI for Response Assessment of Extensive Lymphatic Malformations in Children Treated With Sirolimus

Durand, Rachelle; Reid, Janet R.; Belasco, Jean B.; Shellikeri, Sphoorti; Calle‐Toro, Juan S.; Cahill, Anne Marie; Srinivasan, Abhay

doi:10.2214/ajr.20.24378

Cited by 6 publications

(10 citation statements)

References 41 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…One of these studies, Ozeki et al, 17 included adults and children but only the pediatric data was extracted for this review. Ten retrospective case series [19][20][21][22][23][24][25][26][27][28] and five individual case reports [29][30][31][32][33] of sirolimus use in cervicofacial LMs were also included; these are summarized in Table II. Assessment of risk of bias for all included studies is shown in Table III.…”

Section: Search Resultsmentioning

confidence: 99%

“…Those studies that reported their surveillance regimen checked full blood count, renal profile, bone profile, hepatic profile, and sirolimus levels monthly. 16,28…”

Section: Sirolimus Dosing and Surveillancementioning

confidence: 99%

“…Out of the included studies, three 17,19,28 gave all patients trimethoprim-sulfamethoxazole prophylaxis against Pneumocystis (PJP). Out of the total of 33 patients in these studies, 4 patients had grade 3-4 events during treatment.…”

Section: Antibiotic Prophylaxismentioning

confidence: 99%

See 2 more Smart Citations

Sirolimus for Pediatric Cervicofacial Lymphatic Malformation: A Systematic Review and Meta‐Analysis

Kamhieh,

Mitra,

Burnett

et al. 2023

The Laryngoscope

View full text Add to dashboard Cite

ObjectiveThis study is a systematic review and meta‐analysis of the efficacy and safety of sirolimus in the management of pediatric cervicofacial lymphatic malformations (LMs).Data SourcesEMBASE, Medline, Scopus, and Cochrane databases were searched, along with the reference list of all included articles.Review MethodsThe study protocol was registered with PROSPERO and a systematic literature search strategy was designed and conducted with the aid of a medical librarian. All studies including case reports were included, with pooled analysis of raw data. A meta‐analysis was conducted of magnetic resonance imaging (MRI), clinical, and airway outcomes.ResultsThirteen case series and five individual case reports were included. Meta‐analysis showed 78% (95% CI 57%–94%) of 62 patients had a reduction in LM volume, on MRI criteria, by 20% or more, and 32% (95% CI 11%–57%) had a reduction of 50% or more. Further meta‐analysis showed 97% (95% CI 88%–100%) of 78 patients reported some clinical improvement on sirolimus. Sirolimus may be of particular value in management of airway LMs; out of 27 tracheostomy‐dependent patients, meta‐analysis showed 33% (95% CI 1%–78%) were decannulated after starting sirolimus. Individual patient meta‐analysis on 24 individuals showed a statistically significant better response to sirolimus when initiated under the age of 2 years.ConclusionThis review and meta‐analysis support the efficacy of sirolimus in pediatric LMs of the head, neck, and airway. A large multi‐center trial is needed to further explore its role and limitations. Laryngoscope, 2023

show abstract

Section: Search Resultsmentioning

confidence: 99%

“…Those studies that reported their surveillance regimen checked full blood count, renal profile, bone profile, hepatic profile, and sirolimus levels monthly. 16,28…”

Section: Sirolimus Dosing and Surveillancementioning

confidence: 99%

See 1 more Smart Citation

Sirolimus for Pediatric Cervicofacial Lymphatic Malformation: A Systematic Review and Meta‐Analysis

Kamhieh,

Mitra,

Burnett

et al. 2023

The Laryngoscope

View full text Add to dashboard Cite

show abstract

“…MRI also plays an important role in post-treatment follow-up, particularly in cases of large or complex LMs [28,29]. MR lymphangiography may help to direct therapeutic interventions in certain cases, such as when thoracic duct embolization is considered for a lymphatic leak, and can evaluate central lymphatic ducts after surgical or endovascular intervention [30,31].…”

Section: Accepted Manuscriptmentioning

confidence: 99%

“…However, no guidelines currently exist to define the most appropriate timing of repeat imaging, which instead is typically decided on a case-by-case basis. Development of imaging treatment response criteria represents an active area of research [28,29].…”

Section: Accepted Manuscriptmentioning

confidence: 99%

Lymphatic Anomalies in Children: Update on Imaging Diagnosis, Genetics, and Treatment

Snyder

Sarma

Borst

et al. 2022

American Journal of Roentgenology

View full text Add to dashboard Cite

The publication of this Accepted Manuscript is provided to give early visibility to the contents of the article, which will undergo additional copyediting, typesetting, and review before it is published in its final form. During the production process, errors may be discovered that could affect the content of the Accepted Manuscript. All legal disclaimers that apply to the journal pertain. The reader is cautioned to consult the definitive version of record before relying on the contents of this document.

show abstract

Bowel perforation following percutaneous sclerotherapy of an intra-abdominal lymphatic malformation

et al. 2022

View full text Add to dashboard Cite

MRI for Response Assessment of Extensive Lymphatic Malformations in Children Treated With Sirolimus

Cited by 6 publications

References 41 publications

Sirolimus for Pediatric Cervicofacial Lymphatic Malformation: A Systematic Review and Meta‐Analysis

Sirolimus for Pediatric Cervicofacial Lymphatic Malformation: A Systematic Review and Meta‐Analysis

Lymphatic Anomalies in Children: Update on Imaging Diagnosis, Genetics, and Treatment

Bowel perforation following percutaneous sclerotherapy of an intra-abdominal lymphatic malformation

Contact Info

Product

Resources

About