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. 2019 Dec 27;9(1):19916.
doi: 10.1038/s41598-019-56217-0.

Rectosigmoidal manifestations of venous malformations: MR imaging findings and interdisciplinary therapeutic modalities

Richard Brill  1 , Eva Brill  2 , Wibke Uller  3 , Veronika Teusch  3 , Hubert Gufler  4 , Simone Hammer  3 , Claudia Fellner  3 , Katja Evert  5 , Constantin Goldann  4 , Maximilian Helm  4 , Jonas Rosendahl  6 , Walter A Wohlgemuth  4
Affiliations

Rectosigmoidal manifestations of venous malformations: MR imaging findings and interdisciplinary therapeutic modalities

Richard Brill et al. Sci Rep. .

Erratum in

Abstract

The aim of this study was to identify the frequency of rectosigmoidal involvement in patients with venous malformations (VM) of the lower extremities and to demonstrate multidisciplinary therapeutic options. The medical records and magnetic resonance images (MRI) of patients with VM of the lower extremities, over a six-year period, were reviewed retrospectively in order to determine the occurrence of rectosigmoidal involvement. Vascular interventions, surgical treatments, percutaneous and hybrid (endoscopy-guided angiography) sclerotherapy and procedural complications (according to Clavien-Dindo classification) were also noted. Of the 378 patients with vascular malformation of the lower limbs, 19 patients (5%) had documented venous rectosigmoidal malformation. All of these 19 patients reported episodes of rectal bleeding, while seven patients (36.8%) also had anemia. All patients underwent endoscopy. By endoscopy, seven patients (36.8%) showed discreet changes, and 12 patients (63.2%) showed pronounced signs of submucosal VM with active (47.3%) or previous (15.7%) bleeding. Treatment was performed in all patients with pronounced findings. Six patients underwent endoscopy-guided hybrid sclerotherapy, one patient underwent endoscopic tissue removal, one patient received percutaneous sclerotherapy and one patient received a combination of transvenous embolization and hybrid sclerotherapy. Three patients required open surgery. No complications occurred after conservative treatments; however, one complication was reported after open surgery. None of the treated patients reported further bleeding and anemia at the end of the follow-up period. In this cohort, rectosigmoidal VM occurred in 5% of patients presenting with a high incidence of rectal bleeding. Percutaneous or endoscopy-guided hybrid sclerotherapy appeared to be a safe and effective alternative to surgery.

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Conflict of interest statement

The authors declare no competing interests.

Figures

Figure 1
Figure 1
Therapeutic algorithm for the treatment of bleeding from a VM.
Figure 2
Figure 2
MRI of a 35-year old patient with expanded VM, both rectosigmoidal and in lower limbs. (*) Rectosigmoidal lumen; (I) rectosigmoidal intramural VMs associated with massively dilated rectosigmoidal wall.
Figure 3
Figure 3
TWIST MRI of a 10-year old girl with expanded sigmoidal VM. Circle: Massively dilated vessels with venous flow characteristics.
Figure 4
Figure 4
Angiography of a 10-year old girl with VM: Endoscopy-guided puncture of an intramural sigmoidal VM and venous angiography during intervention; (*) coils (transvenous angiographic embolization).
Figure 5
Figure 5
Histologic picture of a 35-year old patient with large VM: HE staining shows dilatated blood vessels (marked with a triangle) with thin walls in the submucosa (original magnification 40×).
Figure 6
Figure 6
Macroscopic picture of a 35-year old patient with large VM (white rectangle): Gross view of the surgical specimen of a rectum and sigmoid colon with prominent blood vessels, particularly in the surrounding soft tissue.
Figure 7
Figure 7
(a) Angiography of a 35-year old patient with VM and postoperative bleeding (arrows) out of the inferior rectal artery. (b) No bleeding after embolization.
See this image and copyright information in PMC

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