Oral lymphatic malformations: A multicenter study of 208 cases and literature review

Meirelles, Daniela Pereira; Couto, Aline Maria do; Silva, Leni V. O.; Abreu, Lucas Guimarães; Sousa, Suzana Cantanhede Orsini Machado de; Custódio, Marcos; Tarquínio, Sandra Beatriz Chaves; Batista, Aline Carvalho; Mendonça, Elismauro Francisco; Libório‐Kimura, Tatiana Nayara; Louredo, Brendo Vinícius Rodrigues; Romañach, Mário José; Andrade, Bruno Augusto Benevenuto De; Nonaka, Cassiano Francisco Weege; Alves, Pollianna Muniz; Sena, Luana Samara Balduino de; Mesquita, Ricardo Alves; Aguiar, Maria Cássia Ferreira de

doi:10.1002/hed.26854

Cited by 9 publications

(16 citation statements)

References 48 publications

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“…At present, surgical treatment is the first choice for oral and maxillofacial lymphatic malformation. At the same time, sclerosing agent injection, interventional embolization, freezing, laser, oral sirolimus, and oral celecoxib can be used for those who cannot completely remove the focus 8–11 …”

Section: Discussionmentioning

confidence: 99%

“…At the same time, sclerosing agent injection, interventional embolization, freezing, laser, oral sirolimus, and oral celecoxib can be used for those who cannot completely remove the focus. [8][9][10][11] Sirolimus is a mTOR receptor inhibitor, which can inhibit cell proliferation and angiogenesis and effectively treat various types of vascular malformations. 12,13 Sirolimus has been widely used in various refractory vascular malformations.…”

Section: Discussionmentioning

confidence: 99%

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Occurrence of Rhabdomyosarcoma After Surgery Combined with oral Sirolimus for Mixed Vascular Malformation of the Tongue

Lin

Han

Cui

et al. 2023

Journal of Craniofacial Surgery

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Vascular malformation is the general term of a kind of lesions originated from lymphatic vessels and vascular tissues, which contains a variety of components called mixed vascular malformation. Rhabdomyosarcoma (RMS) is a kind of soft tissue sarcoma, originating from striated muscle cells or mesenchymal cells. RMS and vascular malformation mostly occur in children, and common in the head and neck, but their simultaneous occurrence is rare. A 9-year-old boy who was hospitalized for a second attack of combined vascular malformation: hemolymphangioma. The child experienced severe upper airway obstruction and tongue bleeding. Postoperative pathology demonstrated hemolymphangioma combined with RMS. Subsequently, he was transferred to the oncology department for chemotherapy and lately died of RMS with lung metastasis. The secondary RMS may be related to the usage of sirolimus. Because of its uncertain border, vascular malformation in the oral and maxillofacial region is difficult to completely remove by surgical resection, and local recurrence could be often observed. Due to its rapid progress and continuous bleeding, the possibility of malignant tumor should be considered and multidisciplinary comprehensive treatment should be actively taken. Besides, family history of related malignant tumors and immune function should be investigated in detail before choosing the application of oral sirolimus.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Occurrence of Rhabdomyosarcoma After Surgery Combined with oral Sirolimus for Mixed Vascular Malformation of the Tongue

Lin

Han

Cui

et al. 2023

Journal of Craniofacial Surgery

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“…In a multicenter study of oral lymphatic malformations, disagreement between clinical and histopathological diagnoses was encountered in 58.2% of the lesions. 22 Considering that vascular lesions within the mouth have a broad etiopathogenesis, the final diagnosis of such entities may be challenging. 23 To improve the diagnostic process and establish a definite diagnosis of an uncommon lesion, this study described the immunohistochemical profile of an oral hemangiolymphangioma.…”

Section: Discussionmentioning

confidence: 99%

Unveiling an oral hemangiolymphangioma

Ferreira-Santos¹,

Santos²,

Scherma³

et al. 2023

Autops Case Rep

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Hemangiolymphangioma is a very rare vascular malformation that develops as a combination of dilated venous and lymphatic vessels. We describe an unusual case of hemangiolymphangioma of the tongue affecting an adult man who complained of an uncomfortable, slowly progressing exophytic irregular dark red-violaceous nodular mass on his tongue that impaired speech and swallowing for two weeks. The clinical differential diagnoses were Kaposi’s sarcoma and a COVID-19-related lesion. A complete blood count and serology for HIV-1 and 2 and RT-PCR for COVID-19 were requested and results were negative. An incisional biopsy was performed. Microscopically, the lesion exhibited several dilated vessels lined by normal-appearing endothelial cells, some filled with prominent intravascular erythrocytes and others containing proteinaceous eosinophilic material resembling lymphatic vessels, in close association with hyperkeratosis, papillomatosis, and acanthosis. From immunohistochemical analysis, most vessels were found to be CD34 positive, some highlighted by α-SMA, whereas D2-40 was focal. Positive staining for some lymphatic and blood vessel markers, i.e. , D2-40 and CD34, respectively, indicates a mixed derivation of the lesion. HHV-8 was negative. Clinical features, the congested blood vessels with ectasia in intimate association with hyperplastic epithelium, and the immunohistochemical profile supported the final diagnosis of oral hemangiolymphangioma. The patient underwent minimally invasive surgical excision with no intercurrences. After 18 months of follow-up, there were no signs of relapse.

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“…From a structural point of view, lymphatic malformations can be classified as microcystic, macrocystic or mixed ( 3 , 5 ). At intraoral level, most lesions are microcystic and mainly affect the tongue, more specifically the dorsum of the tongue.…”

Section: Introductionmentioning

confidence: 99%

“…At intraoral level, most lesions are microcystic and mainly affect the tongue, more specifically the dorsum of the tongue. Other locations that may be affected are the lips, buccal mucosa, or palate ( 5 ).…”

Section: Introductionmentioning

confidence: 99%

Gingival lymphatic malformation. An atypical case report

Alberdi-Navarro,

Ruiz-Ochoa,

De Juan-Galindez

et al. 2023

J Clin Exp Dent

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Lymphatic malformations are a rare pathology that presents a highly variable clinical expression. Intraorally, it mainly affects the dorsum of the tongue. The objective of this work is to present a case of lymphatic malformation in an atypical location. A 20-year-old male who attended the clinic for multiple vesicular lesion in attached gingiva, asymptomatic and of unknown evolution. Removal of the lesion and subsequent histological analysis were performed, which showed a microcystic lymphatic vascular lesion. Immunohistochemistry for D2-40 was performed, which corroborated the lymphatic origin of the lesion. At 6 months, no recurrence of the lesion was recognized. Clinicians should include lymphatic malformations in the differential diagnosis of multiple vesicular lesions. Knowing the oral manifestations of this entity is essential for its proper diagnosis and clinical management. Key words: Gingiva, oral lymphatic malformation, diagnosis.

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Oral lymphatic malformations: A multicenter study of 208 cases and literature review

Cited by 9 publications

References 48 publications

Occurrence of Rhabdomyosarcoma After Surgery Combined with oral Sirolimus for Mixed Vascular Malformation of the Tongue

Occurrence of Rhabdomyosarcoma After Surgery Combined with oral Sirolimus for Mixed Vascular Malformation of the Tongue

Unveiling an oral hemangiolymphangioma

Gingival lymphatic malformation. An atypical case report

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