Polymethylmethacrylate bone cement utilization for hemostasis in an intraosseous arteriovenous malformation
Mehmet Dadaci, Munur Selcuk Kendir, Zikrullah Baycar, Bilsev Ince
Department of Plastic and Reconstructive and Aesthetic Surgery, Meram Faculty of Medicine, Necmettin Erbakan University, Konya, Turkey
Correspondence Address:
Dr. Munur Selcuk Kendir
Department of Plastic and Reconstructive and Aesthetic Surgery, Meram Faculty of Medicine, Necmettin Erbakan University, Meram, Konya 42060
Turkey
Source of Support: None, Conflict of Interest: None
DOI: 10.4103/tjps.tjps_19_22
A 15-year-old girl applied to the outpatient clinic with an arteriovenous malformation, measuring approximately 20 cm × 10 cm, on the anterior aspect of the right knee. She was admitted to operation because of the failure of the previous embolization attempts. Intraoperatively, there was uncontrolled bleeding from the nidus which is located at the medulla of the tibia. After the failure of initial efforts, hemostasis could be achieved using polymethylmethacrylate cement. Arteriovenous malformations (AVMs) may cause life-threatening bleeding. In uncontrolled intraosseous bleeding, bone cement could be used for hemostasis. In this study, we present an AVM case and our experience about its management.
Keywords: Arteriovenous malformation, hemostasis, polymethylmethacrylate
Errors in fetal vascular development are believed to be the basis for vascular malformations.[1] Vascular malformations often have stable endothelium. As a result of their steady nature, they never regress and are usually present from the beginning of life. The lesions enlarge proportionately with the growth of the child in general.[2],[3] Even though arteriovenous malformations (AVMs) most commonly arise within the central nervous system, the lesion might be seldom seen in extremities or head-and-neck region.[1] A pan-vascular study investigating concomitant lesions is not indicated in these patients.[4],[5]
The existing lesion can range from high-flow arteriovenous connections to low-flow lesions that may be venous, lymphatic, or a combination of both.[6] In high-flow AVMs, the shunt lacks the normal resistance of the capillary system and may cause a significant level of arterial steal phenomenon, which leads to distal ischemia.[7] The clinical presentation may be completely asymptomatic or, in contrast, may present with a swollen mass, pain, bleeding, growth retardation, venous hypertension, ischemia, and rarely high-output heart failure.
Polymethylmethacrylate (PMMA) is the most preferred synthetic material for bone substitution.[8] It is an excellent choice due to the high biocompatibility, low cost, easy handling characteristics, sufficient strength, and moldability in situ.[9] The cement, which is enshaped outside the body or in the defect area, freezes within 10–20 min and becomes usable to replace the bony tissue.[10] Aseptic loosening, infection of the prosthesis, and thermal necrosis to surrounding tissue are the most common complications with PMMA in orthopedic applications.[11]
In this study, we aimed to present our experience of using PMMA cement in the pursuit of hemostasis and bone reconstruction in the management of a case of intramedullary extended high-flow vascular malformation located on the anterior aspect of the right tibia.
Case ReportA 15-year-old girl applied to the outpatient clinic with a vascular-looking mass, present since birth, measuring approximately 20 cm × 10 cm, located on the anterior aspect of the right knee. The main complaints of the patient were severe pain and burning sensation. In the physical examination, a dark red-colored lesion with bullous structures starting from the lower end of the right femur and covering the upper one-third of the tibia with irregular borders was observed [Figure 1]. Sensorial and motor examinations of the extremity were normal. Arterial pulses could be taken from the distal extremities. It was learned that AVM embolization was tried by interventional radiology twice before, but embolization could not be accomplished due to the high flow.
Figure 1: Preoperative appearance of the lesion located on the knee. (a) Anterior view. (b) Medial view. (c) Lateral viewIn the angiographic examination, superficially localized arteriovenous malformation fed from the femoral artery and popliteal artery branches with multiple venous drainage was observed in the popliteal region. In the magnetic resonance imaging examination of the right leg, it was reported that the density of vascularity increased in the intraosseous part of the tibia and the structure of the bone was seriously affected [Figure 2].
Figure 2: The radiographic appearance of the lesion. (a) X-ray imaging. (b) MR-angiography. MR: Magnetic resonancePossible outcomes were discussed with family and patient, and operation decision was taken. It was started under a tourniquet. After a superficial incision around the lesion, the connections of the vascular malformation with the tibia and surrounding soft tissue were ligated up to the proximal part of the tibia, which was the part of the bone that nidus was thought to be lobated. We observed that the mass extended into the medulla of the tibia through two pieces of 0.5-cm diameter foramina located on the proximal one-third of the tibia. The mass was scraped off, and the foramina were obliterated with bone wax. After the tourniquet was deflated, very high flow bleeding, which was thought to be nidus bleeding, started from the medulla. The bone wax plug did not provide enough durability to stop the bleeding, and it popped out under the influence of the high-pressure bleeding. The tourniquet was then inflated again. This time, for obliteration, PMMA bone cement was prepared and placed in two foramina on the tibia. The tourniquet was deflated again, waiting for the cement to set, and although there was no bleeding from the two foramina of the tibia, high-flow bleeding occurred from almost every region of the shaft of the tibia. As the bleeding could not be controlled, the tourniquet was inflated again. Then, a strip of bone was elevated after vertical osteotomy, which included the holes on the tibia, and the tibial medulla was exposed and the nidus located on the tibia medulla was curetted [Figure 3]. Then, PMMA bone cement was placed in the cavity formed in the bone to fulfill the bone defect and provide hemostasis. At the end of the cement freezing period, it was observed that the patient's bleeding control was achieved. In the intraoperative period, 4 units of Erythrocyte Suspension (ES) and 2 units of fresh frozen plasma transfusions were applied to the patient. The reconstruction was postponed to the second session, and the operation was terminated [Figure 4].
Figure 3: (a) Schematic view of the foramina on the tibia. (b) The appearance of the bone defect after osteotomyReconstruction stage was planned for the 4 days after. There were soft-tissue defects in the upper one-third of the tibia, and the bone cement was also exposed. Gastrocnemius muscle-skin flap was performed for soft-tissue reconstruction of the tibia and for the coverage of the cement. To repair skin defects that could not be covered by the flap, a partial thickness skin graft was applied to the defect area and the reconstruction was completed [Figure 5].
Figure 5: The appearance of the right knee at the postoperative 20th day. (a) Lateral view. (b) Anterior view. (c) Medial view. (d) The flexor capacity of the kneePathological examination of the resected lesion was reported as arteriovenous malformation. No significant AVM filling was observed in the arteriographic examination performed at the postoperative 20th day. The patient was discharged on the postoperative 25th day. Postoperative rehabilitation was completed in the late postoperative period [Figure 6].
Figure 6: The angiographic studies at the postoperative 20th day. (a) Early phase of angiography. (b) Late phase of angiography. (c) Arterial mapping of the lower limbIn the postoperative 2nd year, pink-purple color changes were observed in the skin around the operated area during a routine follow-up, which was suggesting recurrence. The patient was consulted with interventional radiology, and selective embolization was performed. The patient did not have any additional complaints in the subsequent follow-ups.
DiscussionVascular malformations can be divided into low-flow and high-flow malformations according to their hemodynamic characteristics.[12] Extremities are extremely rare locations for AVMs. Intraosseous vascular anomalies account for only 0.5%–1% of all intraosseous vascular lesions.[13] For all vascular lesions, it would be beneficial to conduct a radiological study that will reveal the dimensions and characteristics of the lesion before discussing treatment options.[14]
The recommended treatment for small, isolated, and regional AVMs is the resection and reconstruction of the lesion.[3] The main goal in the treatment of high-flow arteriovenous vascular malformations is to eradicate the nidus. This is best accomplished with a liquid embolic agent that will penetrate into the nidus.[15] This is followed by surgery resection 24–48 h later. This procedure is intended to reduce bleeding during surgery and does not reduce resection margins. Large AVMs usually require embolization in stages.[12],[16] Surgical resection is another option for high-flow AVMs that cannot be controlled by embolization.[17]
Surgical resection is the best option in the management of AVMs, but complete resection of the lesion is often impossible.[18] Incomplete resections and embolization can induce aggressive growth of the remaining nidus, and the risk of recurrence is up to 50% within the first 5 years.[19],[20] For this reason, the best results in the treatment of complex and large lesions are obtained with a combination of treatment options.[12]
In this case, although it was tried to embolize two times before the patient's application to our clinic, the attemptions were unsuccessful. Therefore, there was no other treatment option other than a complex surgical intervention. Due to the patient's severe symptoms, the presence of a lesion that made the patient's suffering and the patient and her family's severe desire for surgery; the operation was planned after the possible risks and complications were explained to the patient and her family in detail. In such cases, it should be kept in mind that intraoperative uncontrolled bleeding may be encountered in patients with a history of multiple failed embolization attempts.
In the event of acute bleeding within the bone that cannot be stopped by acceptable methods, other conventional methods are ligation of the feeding vessels of the bone or removal of the organ in question.[21] In our case, if it had not been possible to control the bleeding with PMMA molding, ligation of the branches of the popliteal artery to the tibia would have been performed. However, because of ischemia, avascular necrosis of the bone will occur with a high probability and a dysfunctional extremity can be expected in the chronic period. As another method, limb amputation, is catastrophic for the patient and family, it should be preferred if only the bleeding is at a life-threatening level and cannot be controlled by any other method.
PMMA bone cement was used as an embolizing agent for treating intraosseous arteriovenous lesions in a previous study.[22] What makes our case unique is that an intraoperative hemorrhage with terrible possible consequences could be controlled thanks to the compression caused by the mass effect of PMMA and the exothermic reaction of the cement, while it sets. Simultaneously, bone reconstruction with PMMA was also completed. It has been shown in this study that the usage of PMMA bone cement for bleeding control provides effective hemostasis.
Recurrence in our patient a few years after the operation is an expected situation due to the nature of the lesion. We recommend that the patient should be informed that vascular malformations may recur and that patients should be followed closely in the postoperative period.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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