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Case Reports
. 2009 Oct;29(5):274-8.

Arteriovenous malformation of the base of tongue in pregnancy: case report

F Martines  1 , V Immordino
Affiliations
Case Reports

Arteriovenous malformation of the base of tongue in pregnancy: case report

F Martines et al. Acta Otorhinolaryngol Ital. 2009 Oct.

Abstract
in English, Italian

Arteriovenous malformation of the head and neck is a rare vascular anomaly but when present is persistent and progressive in nature and can represent a lethal benign disease. An unusual case is presented of an arteriovenous malformation of the base of tongue in a 32-year-old primigravida at 23.2 weeks of gestation with a history of haemoptysis. The patient was admitted to hospital with 10.7 g/dl of haemoglobin and 32.1% of haematocrit but due to recurrent massive haemoptysis, in the next few days, dropped to 6.7 g/dl of haemoglobin and 20.2% of haematocrit which required immediate blood transfusions. To maintain the upper airways patent the patient underwent tracheostomy; during angiography, showing an arteriovenous malformation with its feeding arteries (lingual artery, internal maxillary artery, and maxillary artery) embolization was made without a significant blood flow reduction. After surgical ligation of the external carotid artery, on the right side, the patient was readmitted for further angiographic evaluation, which confirmed complete occlusion of the carotid artery but, at the same time, revealed the integrity of the arteriovenous malformation perfusion on account of a new feeding artery (left lingual artery). A new superselective catheterization of the lingual artery was performed but due to the effect of progesterone, which causes smooth muscle relaxation and leads to arteriovenous malformation dilatation and rupture, the primigravida again presented haemoptysis. In agreement with the gynaecologists, the patient was given betamethasone to induce foetal lung maturation, and induction of labour was planned at 26 weeks, and a healthy baby was delivered naturally. Over the following days, the patient had no further haemoptysis and so far clinical examination showed no evidence of the original mass (slight haemorrhagic suffusion of the right anterior amygdala region).

Le malformazioni arterovenose del distretto testa-collo sono delle forme cliniche patologiche rare, lentamente progressive che a seconda della sede e della dimensione possono risultare letali. Gli Autori presentano un raro caso di malformazione arterovenosa della base della lingua in una donna di 32 anni primipara alla 23,2 settimana di gestazione con una storia clinica di ripetuti episodi di emottisi nelle settimane antecedenti al ricovero. Pervenuta in regime di urgenza con valori di 10,7 g/dl di emoglobina e 32,1% di ematocrito, a seguito di nuovi episodi di emottisi giungeva a valori di emoglobina di 6,7 g/dl e di ematocrito del 20,2% e veniva pertanto sottoposta a trasfusione. Si eseguiva inoltre una tracheostomia per mantenere la pervietà delle vie aeree ed una angiografia, che mostrava una malformazione arterovenosa alimentata dalle arterie del distretto della carotide esterna di destra. A seguito di un tentativo non riuscito di embolizzazione della malformazione si decideva per una legatura chirurgica della carotide esterna di destra. La gestante veniva successivamente sottoposta a una nuova seduta di angiografia che confermava l’occlusione completa della carotide esterna destra ma evidenziava nel contempo l’integrità della malformazione arterovenosa la cui perfusione veniva assicurata dalla arteria linguale controlaterale ed induceva ad un nuovo tentativo di embolizzazione della linguale sinistra. Per l’incremento dei valori di progesterone che favoriscono la dilatazione e la rottura delle malformazioni arterovenose ed a seguito di nuovi episodi di emottisi, consultati i ginecologi alla paziente veniva somministrato betametasone a dosi tali da favorire la maturazione fetale e indurre il parto alla 26 settimana. Nei giorni seguenti il parto la paziente non ha più presentato episodi di emottisi e la malformazione arterovenosa si è lentamente ridotta fino a scomparire, residuando attualmente soltanto una lieve soffusione emorragica della regione tonsillare anteriore destra.

Keywords: Arteriovenous malformations; Pregnancy; Tongue; Vascular malformation.

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Figures

Fig. 1
Fig. 1
Pharynx/hypopharynx endoscopic examination.
Fig. 2
Fig. 2
Angio CT showing original AVM.
Fig. 3
Fig. 3
Angio CT after ligation of external carotid artery confirmed integrity of AVM.
Fig. 4
Fig. 4
Oral cavity examination six months after labour.
See this image and copyright information in PMC

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