Oral manifestations and dental care management of a young patient with lymphangioma of the tongue: A case report

Chouchène, Farah; Masmoudi, Fatma; Baaziz, Ahlem; Maatouk, Fethi; Ghédira, Hichem

doi:10.1002/ccr3.4537

Cited by 4 publications

(6 citation statements)

References 26 publications

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“…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%

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

Kamhieh,

Mitra,

Burnett

et al. 2023

The Laryngoscope

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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

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Section: Search Resultsmentioning

confidence: 99%

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

Kamhieh,

Mitra,

Burnett

et al. 2023

The Laryngoscope

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“…Zhang reported to use a reduced starting dose in neonates (0.5 mg/m 2 ). Other starting doses were 1–2 mg per day [ 31 ], 0.1 mg/kg per day [ 35 ], 0.05 mg/kg twice daily [ 16 ], 0.6 mg/m 2 per twice daily dose [ 33 ], 0.07 mg/kg twice daily [ 18 ], 0.4–0.8 mg/m 2 twice daily [ 20 ] or in two fractions [ 27 ], and 1.5 mg/kg/day administered in two divided daily doses [ 37 ]. In five publications, the initial dose of sirolimus was not reported [ 12 , 14 , 15 , 22 , 38 ].…”

Section: Resultsmentioning

confidence: 99%

“…In one study each, 3.5–6 ng/mL [ 30 ], 4–8 ng/mL [ 21 ], 4–10 ng/mL [ 38 ], 4–13 ng/mL [ 36 ], 5–12 ng/mL [ 33 ], 10–12 ng/mL [ 27 ], 10–13 ng/mL [ 19 ] and 12–20 ng/mL [ 35 ] were the target concentrations. Eight studies did not report the target blood level of sirolimus [ 12 , 13 , 15 , 18 , 22 , 29 , 32 , 37 ]. Not in all studies the planned target trough level for sirolimus was achieved [ 14 , 20 ].…”

Section: Resultsmentioning

confidence: 99%

“…Twenty-three studies commented on possible adverse events associated with and probably being a result of sirolimus therapy [ 15 – 17 , 19 – 31 , 33 – 39 ]. In five studies no patients experienced side effects [ 15 , 22 , 29 , 30 , 37 ]. In the other 18 studies, different adverse effects were reported [ 16 , 17 , 19 – 21 , 23 – 28 , 31 , 33 – 36 , 38 , 39 ], the most common were hyperlipidemia, neutropenia and infections such as cellulitis.…”

Section: Resultsmentioning

confidence: 99%

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Efficacy of sirolimus in children with lymphatic malformations of the head and neck

Wiegand

Dietz

Wichmann

2022

Eur Arch Otorhinolaryngol

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Purpose Children with extensive lymphatic malformations of the head and neck often suffer from functional impairment and aesthetic deformity which significantly affect the quality of life and may be life-threatening. Treatment with sirolimus has the potential to improve symptoms and downsize lymphatic malformations. This systematic review summarizes the current information about sirolimus treatment of lymphatic malformations of the head and neck in children, its efficacy and side effects. Methods A systematic search of the literature regarding studies on sirolimus treatment of children with lymphatic malformations of the head and neck was performed in PubMed, Embase, and Google Scholar up to July 2021 with the search terms “lymphatic malformation”, “lymphangioma”, “cystic hygroma”, “low-flow malformation”, “sirolimus”, “rapamycin”, “mTOR inhibitor” and “children”. Results In all, 28 studies including 105 children from newborn to 17 years treated with sirolimus for lymphatic malformations of the head and neck were analyzed. The most frequent initial dose was 0.8 mg/m2 per dose, twice daily at 12-h interval. The target blood level differed between studies, 10–15 ng/mL and 5–15 ng/mL were most often used. More than 91% of the children responded to sirolimus treatment which lasts from 6 months to 4 years. Typical side effects were hyperlipidemia, neutropenia and infections. Methods Sirolimus could be an effective treatment for children with large complicated lymphatic malformations of the head and neck. As not all patients will benefit from treatment, the decision to treat sirolimus should be made by a multidisciplinary team.

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“…The lymphatic endothelium reveals positive staining for marker D2-40. Small blood vessels positively stained for CD31 are also present in the lamina propria [9,14,15]. Prenatal chromosomal analysis including 13, 18, 21, X, and Y may reveal the increased possibility for lymphangioma formation.…”

Section: Discussionmentioning

confidence: 99%

Diagnostic and Therapeutic Challenges of a Large Tongue Lymphangioma in a Child: A Case Report

Andreadis,

Ndreu,

Tsekos

et al. 2023

Cureus

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Lymphangiomas are rare, painless, benign tumors in infancy or early childhood resulting from a congenital malformation of the lymphatic vessels with variable clinical appearance. We report the case of a six-year-old male child who presented with a micronodular surface of the tongue, a burning sensation, difficulties during swallowing and mastication, as well as speech disturbances. Histological examination of the excised tongue specimen showed an angiomatous lesion of the lamina propria that comprised many wide thin-walled spaces. MRI examination revealed an area of disparate T2 signal maximum diameter of 2-3 cm with cystic texture in the middle and frontal left part of the tongue. The lesion was diagnosed as a lymphangioma and was managed through pre-operative embolization and surgery. Tongue lymphangiomas may lead to aesthetic problems, functional issues (like dysphagia, airway obstruction, and speech difficulties), psychological disturbances, poor oral hygiene, and occasional bleeding associated with oral trauma. A major fraction of patients also experience infections, often leading to a significant increase in lesion size. A combination of preoperative embolization and surgical excision could be chosen considering its large size and the age of the patient, and to further eliminate the possibility of recurrence. Early diagnosis and radical treatment are critical in its management.

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Oral manifestations and dental care management of a young patient with lymphangioma of the tongue: A case report

Abstract: This is an open access article under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs License, which permits use and distribution in any medium, provided the original work is properly cited, the use is non-commercial and no modifications or adaptations are made.

Cited by 4 publications

References 26 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

Efficacy of sirolimus in children with lymphatic malformations of the head and neck

Diagnostic and Therapeutic Challenges of a Large Tongue Lymphangioma in a Child: A Case Report

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