gms | German Medical Science

23. Wissenschaftliche Jahrestagung der Deutschen Gesellschaft für Phoniatrie und Pädaudiologie e. V.

Deutsche Gesellschaft für Phoniatrie und Pädaudiologie e. V.

15. - 17.09.2006, Heidelberg

An unusual case of vascular malformation requiring additional orthodontic, swallowing and voice therapy after intralesional Bleomycin injection

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  • corresponding author presenting/speaker Daniela Tatulescu - Medical Center for Diagnostic and Treatment of the Ministry of Defence "Academician Stefan Milcu", Bucharest, Romania
  • author Ionut Secheli - University Children's Hospital „M.S.Curie”, Bucharest, Romania
  • author Marian Secheli - University Children's Hospital „M.S.Curie”, Bucharest, Romania

Deutsche Gesellschaft für Phoniatrie und Pädaudiologie. 23. Wissenschaftliche Jahrestagung der Deutschen Gesellschaft für Phoniatrie und Pädaudiologie. Heidelberg, 15.-17.09.2006. Düsseldorf, Köln: German Medical Science; 2006. Doc06dgppV27

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Veröffentlicht: 5. September 2006

© 2006 Tatulescu et al.
Dieser Artikel ist ein Open Access-Artikel und steht unter den Creative Commons Lizenzbedingungen (http://creativecommons.org/licenses/by-nc-nd/3.0/deed.de). Er darf vervielf&aauml;ltigt, verbreitet und &oauml;ffentlich zug&aauml;nglich gemacht werden, vorausgesetzt dass Autor und Quelle genannt werden.


Abstract

Vascular malformations are errors of the vascular morphogenesis. Depending on the dominating vessel of the lesion, we can describe several types of malformations: capillary, venous, arterial, lymphatic and mixed. We report the case of a 12 year old child who presented at birth a mixed venous and lymphatic malformation at several levels, includind tongue, oral cavity, vestibular cavity and chin.

As the child did not respond to the medical treatment, he was proposed an embolization procedure after having undergone a previous venography in a different medical center. Due to the high costs of such a procedure, the beginning of the treatment with intralesional Bleomycin injection has been decided. Bleomycin, a polypeptidic antineoplasic antibiotic, was recently introduced in the treatment of vascular anomalies as sclerosant agent if injected intralesionally.

After 5 injections under general anesthesia, the lesions disappeared; the lesions didn't reappear and even more, the tongue returned to its normal proportions.

The vascular malformation which determined macroglossia since birth, affected during its development, the entire oral cavity. Nowadays, the child whose tongue returned to its normal proportion after 12 years cannot achieve a frontal occlusion which creates discomfort during mastication; the child must still undergo several orthodontical and surgical correcting procedures and a speech training.


Text

Hemangiomas or benign vascular tumors which represent a benign neoplasic proliferation of endothelial cells. The dynamic self-limited proliferative vascular lesion dislocates and pushes away the tissue from the place where it grows without invading it, which is important for the surgeon who excises the llesion and must restore the integrity of the remaining tissue.

Vascular malformations are errors or the vascular morphogenesis. These lesions are steady and appear as areas with vascular or lymphatic-like shape. They have irregular disorganized and abnormal shape and invade the surrounding tissue.

Depending on the dominating vessel of the lesion, we can describe several types of malformations: capillary, venous, arterial, lymphatic and mixed.

Obviously, depending on the existing blood vessels, the blood flow is also different, which allows us to describe: vascular malformations with low flow and vascular malformations with high flow (these descriptions are allowed by Echo Doppler and especially by the Magnetic Resonance with contrast substance or Arteryography).

Syndromes associated with vascular anomalies. The most representative syndromes are : Kasabach Merritt Syndrome, Klippel Trenaunay Weber Syndrome – Angioostehypertrophy, Sturge Weber Dimitri Syndrome, Rendu Osler Weber Syndrome, Von Hippel Lindau Syndrome, Maffucci Syndrome.

Vasculophathie more common in adults, with lesions which appear in cases of hepatic cirrhosis (vascular stars), hyperestrogenism, puberty, menopause, pregnancy, or senile.

From anatomopathological point of view: Hemangiomas appear as a hyperplasia of endothelial cells with a basic multilamelar membrane which dislocate the surrounding tissue and have, in proliferative phase, a big number of mast cells. During the involutive period, with low mitotic activity, the number of the mast cells is normal.

Vascular malformations represents collections of disorganized abnormal blood vessels with normal endothelium with a basic unilamelar membrane and a normal number of mast cells; they invade the neighboring tissue and often present intravascular thrombosis. Depending on the dominating vessel of the lesion, we can describe several types of malformation: capillary, venous, arterial – which realize arteriovenouse fistulae and have an important blood flow, sometimes influencing the developing area.

It is well known the fact that, unlike vascular malformations that grow invading into the neighboring tissue and don’t involute and cannot be completely eradicated, hemangiomas involute spontaneously leaving residual scarring. This classification does not include the malignant lesions whose starting point is a component of the vascular system.

Material and method

We present a new treatment method through a particular procedure: sclerotherapy with intralesional Bleomycin injections. The technique is not new, but the injected substance seems to have real advantages.

The study concerning the sclerotherapy with intralesional injections does not apply to vascular lesions associated with syndromes or related to chronic liver disorders, drug administration or hormonal states.

Bleomycin, a polypeptidic antineoplasic antibiotic, was recently introduced in the treatment of vascular anomalies as sclerosant agent if injected intralesionally. Excellent results allowed the usages of Bleomycin on a large scale in the local treatment of vascular anomalies located especially on the child’s face and neck.

We report the case of a 12 year old child who presented at birth a mixed venous and lymphatic malformations at several levels, includind tongue, oral cavity, vestibular cavity and chin.

As the child did not respond to the medical tratment, he was proposed an embolization procedure after having undergone a previous venography in a different medical center. Due to the high costs of such a procedure, the beginning of the treatment with intralesional Bleomycin injection has been decided.

Results and conclusions

After 5 injections under general anesthesia, the lesions disappeared; the lesions didn't reappear and more, the tongue returned to its normal proportions.

The vascular malformation which determined macroglossia since birth, affected during its development, the entire oral cavity. Nowadays, the child whose tongue returned to its normal proportion after 12 years cannot achieve a frontal occlusion which creates discomfort during mastication; the child must still undergo several orthodontical and surgical correcting procedure and a speech training.

Now the child has a normal life. He can speak and eat normally.

The procedure was not traumatizing with no adverse reaction. We have successfully paved the way for subsequent correcting surgeries.


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