Acta Neurochirurgica https://doi.org/10.1007/s00701-017-3440-5 CASE REPORT - BRAIN TUMORS Surgical treatment of a gauzoma with associated obliterative arteriopathy and review of the literature Eric S. Nussbaum 1 & Kevin M. Kallmes 2 1 & Jodi Lowary & Leslie A. Nussbaum 1 Received: 23 November 2017 / Accepted: 14 December 2017 # Springer-Verlag GmbH Austria, part of Springer Nature 2017 Abstract We report a case of a 50-year-old woman whose 0.5 mm middle cerebral artery (MCA) aneurysm was treated with gauze wrapping at an outside facility. She returned 9 months later with seizures and an inflammatory process in the region of the prior aneurysm. Surgical re-exploration at that time was aborted. Two years later, she presented with a gauzoma associated with local inflammatory response and severe narrowing of the MCA. A common carotid artery to MCA bypass was performed, followed by surgical removal of the gauze and inflammatory material. Over a 3-month period, she recovered with significant improvement in her preoperative neurological deficits. Keywords Intracranial aneurysm . Microsurgical wrapping . Gauzoma . Extracranial-intracranial bypass Abbreviations MCA middle cerebral artery PCommA left posterior communicating artery MRI magnetic resonance imaging STA superior temporal artery CCA common carotid artery CT computed tomography FLAIR fluid-attenuated inversion recovery ICA internal carotid artery Aneurysms that are not amenable to endovascular treatment or surgical clipping because of their broad bases can be treated by microsurgical muslin- or gauze wrapping or combined clip-wrapping as an alternative to parent artery sacrifice or aneurysmectomy with relatively good outcomes [15, 10]. However, muslin- or gauze wrapping can induce a local inflammatory response, which, in severe cases, may result in reactive changes in adjacent tissues with clinical manifestations [4, 5]. We report a case of a gauzoma with secondary * Kevin M. Kallmes kall0156@umn.edu 1 Gillette Hospital Department of Neurosurgery, National Brain Aneurysm Center, St Paul, MN, USA 2 Duke University Law School, 201 Science Drive, Durham, NC 27708, USA edema and severe narrowing of the MCA that was successfully managed with extracranial-intracranial bypass and foreign body removal. A literature review of cases of adverse reactions to gauze wrapping was completed as well. Case report In 2011, a 48-year-old woman presented to an outside facility with a 6-mm aneurysm on her left posterior communicating artery (PCommA) and a 0.5-mm aneurysm at the bifurcation of her left middle cerebral artery (MCA; see Fig. 1), both unruptured. Both aneurysms were repaired surgically; the PCommA aneurysm was clipped, while the MCA aneurysm was wrapped with surgical gauze. Although the patient recovered from the surgery without immediate sequelae, 9 months following surgery, she began experiencing complex partial seizures. Magnetic resonance imaging (MRI) showed inflammation in the left temporal lobe, while magnetic resonance angiography showed decreased flow through the left MCA. The original surgeon performed surgical re-exploration, during which he found an encapsulated and scarred mass. Because of the extent of the scarring, he did not explore further within the scarred tissue and made no major intervention before closing. In 2014, approximately 2 years following the second craniotomy, the patient presented to the National Brain Aneurysm Center with progressive neurological deficits, Acta Neurochir Fig. 1 Pre-operative 3D angiography of 0.5 mm left MCA bifurcation aneurysm (arrow) including right side hemiparesis, speech trouble, and seizures. Repeat MRI demonstrated edema associated with a ringenhancing mass that we identified as a gauzoma (Fig. 2). Catheter angiographic examination revealed near-complete occlusion of the M1 segment of the left MCA with multiple Fig. 2 a Axial T2-weighted MRI demonstrates heterogeneous signal in the region of the left MCA. There is apparent abrupt termination of the proximal M1 segment. There is apparent hemosiderin staining and a possibility of hypertrophy of small vessels in expected area of MCA trifurcation. b Axial Fluidattenuated inversion recovery (FLAIR) MRI showing diffuse weight matter hyperintensity (arrow) compatible with vasogenic edema with some mass effect. The enhancing region is isointense to gray matter. c T1weighted MRI with contrast showing evidence of prior craniotomy in left temple region with pronounced focal area of enhancement (arrow) in inferior frontal lobe. d Inferior FLAIR MRI at the level of the insula showing vasogenic edema (arrow) in the anterior temporal white matter with some mass effect collateral channels, that had developed to partially reconstitute distal flow, were visible, consistent with Moyamoya disease (Figs. 2b and 3) [26]. Collateral flow from the anterior cerebral artery had also developed to partially reconstitute flow. The patient was started on dexamethasone at a dose of 4 mg every 6 h, which was tapered over 2 weeks to 2 mg every 8 h. This was then switched to a maintenance dose of prednisone for 6 weeks. This resulted in a reduction in inflammation on MRI, but the patient reported severe water retention and weight gain and began experiencing steroid-psychosis. Prednisone treatment was therefore discontinued, and a trial of azathioprine was undertaken. However, after 2 weeks, the patient reported that side effects continued at an intolerable level. At this point, based on persistent symptoms of hemiparesis, dysphasia, and headache, and given the progressive inflammation and restriction of the MCA, we performed a long saphenous vein graft and exploratory surgery. The previous surgeon had sacrificed the patient’s ipsilateral superior temporal artery (STA); therefore, we performed a long saphenous vein graft from the common carotid artery (CCA) to the MCA to ensure adequate distal blood flow before exploring the inflammatory tissue. We were concerned that during exploration and removal of the granuloma, we might interrupt the fine Acta Neurochir Fig. 4 Intraoperative image of gauzoma (white circle) resection with adjacent saphenous vein graft between distal portion of common carotid and MCA (white arrows) gauzoma (Fig. 5a), and patency of the saphenous vein graft (Fig. 5c). Fig. 3 Pre-operative left internal carotid artery (ICA) injection angiogram showing abnormality in M1 segment of MCA (arrow) with impaired flow and Bneovascularization^ collaterals at the level of gauzoma collateral channels that had developed to reconstitute the distal MCA potentially resulting in a severe ischemic injury. Using an orbitocranial approach, we were able to complete the bypass and then expose and completely remove a mass of dense, fibrous tissue surrounding a cluster of gauze fragments (Fig. 4). Postoperatively, the patient was managed with a rapid dexamethasone taper. She made significant improvements over her preoperative deficits, and at 3-month follow-up, her hemiparesis, dysphasia, and headache syndrome had resolved. She reported mild residual Bclouding of her thinking,^ but this represented a significant improvement over her preoperative status. MR imaging after 3 months demonstrated dramatic improvement in the local edema, complete resection of the Discussion In this case report, we highlight the presentation and treatment options for patients suffering from gauzoma formation following intracranial aneurysm surgery. This report is notable because symptoms related to adjacent inflammation and scarring, specifically partial complex seizures, occurred relatively early after surgery. Progressive symptoms, delayed for 2 more years, resulted from not only the parenchymal inflammation but also from a profound arterio-obliterative process that resulted in near-complete occlusion of the M1 segment. We also demonstrate the potential utility of saphenous vein graft bypass for gauzoma-caused parent artery narrowing accompanying surgical removal of the gauze wrappings. This was determined to be necessary based on rapid progression of edema and deteriorating neurological condition secondary to ischemia. Fig. 5 a Three-month follow-up T2 MRI showing complete resection of gauzoma. b Three-month follow-up FLAIR MRI showing complete resection of gauzoma. c Three-month follow-up MRA demonstrating patency of MCA bypass Acta Neurochir Table 1 Literature Review of Gauzoma/Muslinoma Cases Author, date Age/ Initial treatment sex Second presentation symptomsa Second presentation treatment Outcome Carney et al. 1983 [7] Repka et al. 1984 [24] Tomsak 1985 [30] Marcus et al. 1986 [19] 44/F Headache, blurred vision No treatment pursued Partial temporal field defect Optic neuropathy Exploratory surgery, removal of abscess Decadron, exploratory surgery and scar removal Surgical removal of lesions, dipyridamole, dexamethasone, and cyclophosphamide Clipping of unruptured MCA aneurysm, surgical removal of shredded gauze wrapping Asymptomatic Clipping, gauze wrapping 32/M Clipping, muslin wrapping 62/F Clipping, muslin wrapping 24/F Clipping, muslin wrapping Vision loss, pupillary defect, optic atrophy Blurred vision, diplopia, bitemporal hemianopia Chambi et al. 63/F 1990 [8] Gauze wrapping Headache, nausea, vomiting Case 2 39/F Case 3 71/F Case 4 31/F Clipped and gauze Headache, low-grade fever, wrapped vision loss Clipped and gauze Headache, fever, right painless wrapped ptosis, loss of consciousness Clipped and gauze Epileptic events, olfactory wrapped hallucinations Case 5 65/F Gauze wrapping Case 6 35/F Gauze wrapping Lethargy, confusion, fever, fatigue, headache, photophobia, blurred vision Olfactory hallucinations, visual defect, visual hallucinations Haisa et al. 56/F Muslin wrapping Visual field defect 1990 [14] McFadzean 53/M Clipping, gauze Vision loss in left eye, blurring et al. 1991 wrapping in right eye [20] Onoue et al. 44/F Failed muscle Diplopia 1992 [21] wrapping, gauze wrapping Felsberg 42/F Muslin wrapping Headache, decreasing et al. 1993 visual acuity [11] Prabhu et al. 53/F Pterional Blindness, optic atrophy 1994 [23] craniotomy, gauze-wrapping Case 2 47/F Clipping, Contraction of visual field wrapping Kirollos et al. 47/F Muslin wrapping Headache, vision loss, visual 1997 [16] field defect Case 2 59/F Clipping, Deteriorating vision, right wrapping homonymous hemianopia Lee et al. 61/F Clipping, gauze Recurrent visual field loss 1997 [18] wrapping Visual field defect, right eye Bhatti et al. 33/F Frontal pain 2000 [4] craniotomy, muslin wrapping Case 2 63/F Muslin wrapping Vision loss Berger et al. 2003 [3] 69/M Supraorbital access, muslin wrapping Vision loss 64/M Vision loss, visual field defect Vision stabilized Asymptomatic Recurrent blindness, hydrocephalus diagnosis; death after cardiac arrest No treatment pursued Partial visual field deficit Diphenylhydantoin sodium treatment Persistent third nerve palsy Intravenous cloxacillin and cefotaxime, Asymptomatic later diphenylhydantoin sodium and carbamazepine Treated with intravenous ceftazidime, Asymptomatic vancomycin, and tobramycin Diphenylhydantoin sodium Mass removed during craniotomy Treatment for oligomenorrhea supervened, visual status stable Visual field defect and acuity unchanged Dexamethasone Improvement in vision, left visual field deficit remained Mass removed and aneurysmal neck barely clipped Asymptomatic Surgical removal of muslin, abscess drained Right visual acuity significantly improved Dexamethasone No improvement No treatment pursued No improvement Exploratory surgery, bacterial species found in abscess cavity; flucloxacillin Treated with hydrocortisone, surgical removal of abscess Two surgical decompressions of optic nerve, removal mass No treatment pursued Visual field deficit Deteriorating visual field deficit Partial recovery of visual function Small inferonasal defect in the right visual field Oral corticosteroids Significant improvement in left and right visual acuity Methyl-prednisolone Slight visual improvement in the left eye Acta Neurochir Table 1 (continued) Second presentation treatment Outcome Corticosteroids, eventual surgery with lysis of adhesions & dissection Stable, no improvement to vision Right temporalgia, decreased visual acuity Steroid pulse therapy, surgical removal of mass Residual left hemianopsia in the right eye Endovascularly Headache, right eye pain, coiled, Ray-Tec vision loss wrapped Left homonymous Unsuccessful hemianopia, right optic endovascular, neuropathy clipping, gauze wrapping Taravati et al. 52/M Clipping, gauze Blurred vision 2006 [29] wrapping Surgical decompression of optic nerve Patient’s vision had deteriorated to no light perception Slight visual improvement Author, date Age/ Initial treatment sex Brochert et al. 2003 [6] Fujimura 50/F et al. 2003 [12] Goldsberry 43/F et al. 2004 [13] Subramanian 61/F et al. 2005 [28] Second presentation symptomsa Clipping, muslin wrapping Clipping, gauze wrapping Andres et al. 2007 [1] 67/F Yoon et al. 2010 [31] 51/M Pterional Visual field defect craniotomy, gauze-wrapping Headache 53/M Pterional craniotomy, gauze-wrapping Right side hemiparesis, 64/M Pterional headache craniotomy, gauze-wrapping 52/F Pterional Decreased visual acuity craniotomy, gauze-wrapping 58/F Pterional Headache craniotomy, gauze-wrapping 53/F Muslin wrapping Asymptomatic Case 2 Case 3 Case 4 Case 5 Slater et al. 2014 [27] Clipping, gauze wrapping Left side hemiparesis, gait disturbance Dexamethasone Oral prednisone Episodic bitemporal visual field loss Ceftriaxone, ornidazole, vancomycin, exploratory surgery, mass removal No treatment pursued Asymptomatic Asymptomatic Conservative steroid therapy Asymptomatic Dexamethasone, clopidogrel, and heparin Partially improved right side hemiparesis, headaches Conservative steroid therapy Persistent optic neuropathy Conservative steroid therapy Asymptomatic No treatment pursued Asymptomatic; abnormal peri-aneurysmal T2 signal, vascular narrowing, ring-enhancing mass Asymptomatic; abnormal peri-aneurysmal T2 signal, ring-enhancing mass Significant improvement of all symptoms Case 2 70/F Muslin wrapping Asymptomatic No treatment pursued Present case 51/F Gauze wrapping Right side hemiparesis, dysphasia, seizures Decadron, CCA-MCA bypass and surgical removal of gauze a All secondary conditions listed here were progressive at presentation. CCA-MCA common carotid artery-middle carotid artery Case series that did not provide individual patient symptoms, treatments, and outcomes were not included in this review The inflammation and mass formation caused by gauzomas have been associated with reduced cerebral blood volume in past cases [17], though none has reported complete occlusion of the parent artery. Combined with the risk to the parent artery in microsurgical removal of the gauze wrapping, this makes bypass a useful method of addressing parent artery narrowing and prophylactically ensuring distal flow. Bypass, especially STA-MCA bypass, is also a recommended treatment for Moyamoya disease regardless of its etiology [9]. The patency of our patient’s CCA-MCA bypass at follow-up indicates that, even after the gauzoma was removed, the MCA was inadequately able to deliver the requisite blood volume. Dense, localized scar tissue formation has been considered a beneficial reaction to contact with cotton or gauze-wrapping surgeries, in that it strengthens the walls of the aneurysm [4, 25]. However, both the scar tissue and inflammation can cause complications or parent artery narrowing in 3.5–5% of patients and granuloma formation in 1.5% of patients treated with gauze wrapping [22, 2], and in our case, the scar tissue made surgical removal of the shredded gauze more difficult. Acta Neurochir Table 2 Treatments based on patient presentation Treatment Symptoms No treatment Steroids Anti-seizurea Antibioticsb Surgical removal Surgical decompression Headache 2 (28.6%) 3 (30.0%) 1 (33.3%) 2 (66.7%) 3 (23.1%) 1 (50.0%) Vision loss/visual field deficit 5 (71.4%) 7 (70.0%) 0 (0.0%) 1 (33.3%) 11 (84.6%) 2 (100.0%) Hallucinations Seizure/palsy 0 (0.0%) 0 (0.0%) 0 (0.0%) 0 (0.0%) 2 (66.7%) 2 (66.7%) 1 (33.3%) 1 (33.3%) 0 (0.0%) 1 (7.7%) 0 (0.0%) 0 (0.0%) Hemiparesis Fever 0 (0.0%) 1 (14.3%) 1 (10.0%) 0 (0.0%) 0 (0.0%) 0 (0.0%) 0 (0.0%) 1 (33.3%) 2 (15.4%) 0 (0.0%) 0 (0.0%) 0 (0.0%) Loss of consciousness/fatigue 0 (0.0%) 0 (0.0%) 1 (33.3%) 1 (33.3%) 0 (0.0%) 0 (0.0%) Asymptomatic Total patients (n = 36) 2 (28.6%) 7 0 (0.0%) 10 0 (0.0%) 3 0 (0.0%) 3 0 (0.0%) 13 0 (0.0%) 2 Patients with multiple symptoms were counted in all relevant categories. Percentages are given based on treatment category a One patient was treated with antibiotics and then anti-seizure medication and was counted in both categories b One patient was treated surgically and then given antibiotics to treat an infection found during surgery and was counted in both categories These potential drawbacks, as well as the risk of both infection and ischemia [2], should be considered before aneurysm wrapping. In our literature review of muslinoma and gauzoma patients (Table 1), presentation of patients with muslinomas or gauzomas was relatively consistent, meaning that knowledge of the patient’s surgical history can combine with their symptoms to provide consistent diagnostic criteria. The majority of patients presented with vision loss, visual field deficits, and/or optochiasmatic arachnoiditis (26/36, 72.2%). Other common symptoms included headache (12/36, 33.3%), visual (1/36, 2.8%) and olfactory hallucinations (2/36, 5.6%), seizures (4/36, 11.1%), and hemiparesis (3/36, 8.3%). For symptoms based on treatment chosen, see Table 2. The imaging used to demonstrate the gauzoma was mostly computed tomography (CT) (18/36, 50%) or MRI (17/36, 47.2%). MRI was recommended by Bhatti et al. based on its capacity to enable easier mass identification [4]. Table 3 Given that most patients in our review were stable upon presentation with gauzoma, treatment with steroids was pursued in ten cases (27.8%). In the case of steroid treatment, outcomes included diminished symptoms (6/10, 60.0%), persistent neurological defect (2/10, 20.0%), and a total lack of symptoms (2/10, 20.0%). Surgical decompression was used to lower intracranial pressure in two cases (5.6%), resulting in subsided symptoms for one patient (50.0%). In seven of the 36 cases (19.4%), no treatment was pursued. Exploratory surgery and removal attempts were undertaken in the plurality of cases (13/36, 36.1%), usually because of rapid worsening of symptoms, although gauzoma location also influenced treatment decisions. At follow-up, five of the 13 patients (38.5%) who had undergone surgical removal had their symptoms subside, four (30.8%) were asymptomatic, three (23.1%) experienced a persistent neurological defect, and one (7.7%) died of cardiac arrest. Otherwise, if the patient showed active signs of infection in the affected area, antibiotics were prescribed. For full data concerning patient outcomes, see Table 3. Outcomes based on treatment methods Treatment Outcome Asymptomatic Symptoms subsided Persistent neurological deficit Death Total patients (n = 36) No treatment Steroids Anti-seizurea Antibioticsb Surgical removal Surgical decompression 3 (42.9%) 2 (28.6%) 2 (28.6%) 0 (0.0%) 7 2 (20.0%) 6 (60.0%) 2 (20.0%) 0 (0.0%) 10 1 (33.3%) 2 (66.7%) 0 (0.0%) 0 (0.0%) 3 2 (66.7%) 1 (33.3%) 0 (0.0%) 0 (0.0%) 3 4 (30.8%) 5 (38.5%) 3 (23.1%) 1 (7.7%) 13 0 (0.0%) 1 (50.0%) 1 (50.0%) 0 (0.0%) 2 a One patient was treated with antibiotics and then anti-seizure medication and was counted in both categories b One patient was treated surgically and then given antibiotics to treat an infection found during surgery and was counted in both categories Acta Neurochir Our patient had also had previous exploratory surgery, which should be taken into account as a cause of further scarring that may not be seen in other cases. However, the parent artery narrowing we observed is a common consequence of gauze-wrapping. Therefore, cerebral revascularization should be considered as a potential treatment if inflammation limits parent artery blood flow. Lastly, despite a limited pool of cases, surgical excision has a good prognosis for patients with progressive vision loss and inflammation in the case that steroids do not resolve the patient’s symptoms. 3. 4. 5. 6. 7. Conclusion The symptoms of gauzomas are relatively consistent, and our case demonstrates that disabling edema and parent artery narrowing can be addressed with cerebral revascularization and removal of scar tissue with good neurological outcome. This demonstrates the necessity of considering surgical options when steroids do not resolve the symptoms of inflammation. 8. 9. 10. Funding The United Hospital Foundation provided financial support in the form of grant funding. The sponsor had no role in the design of conduct of this research. Compliance with ethical standards Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper. Conflict of interest The authors declare that they have no conflict of interest. Ethical approval All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. For this type of study, formal consent is not required. 11. 12. 13. 14. 15. Informed consent Informed consent was obtained from all individual participants included in the study. 16. References 1. 2. Andres RH, Guzman R, Weis J, Schroth G, Barth A (2007) Granuloma formation and occlusion of an unruptured aneurysm after wrapping. 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J Neuroophthalmol 5:71 Yoon MA, Kim E, Kwon BJ, Kim JE, Kang HS, Park JH, Sohn CH, Kim JH, Lee DH (2010) Muslinoma and muslin-induced foreign body inflammatory reactions after surgical clipping and wrapping for intracranial aneurysms: imaging findings and clinical features. J Neurosurg 112:640–647. https://doi.org/10.3171/2009.7.jns081625 Comments This is an instructive case report, well-documented and presented, of a gauze ball that developed and became highly symptomatic around a miniscule unruptured MCA aneurysm. There are several educational points that we can take away. 1. The placement of extravascular foreign material is not always benign. In this case, a 6 mm PCoA UIA was clipped electively (I would agree with this), and a minuscule (0.5 mm) MCA blip was layered with gauze at the same operation. I freely admit that I have done this too, many years past. In light of current data, however, we must question whether anything at all should have been done at the MCA location, especially in consideration of the consequences to this patient, who developed seizures and hemiparesis and had three major craniotomies, all when she never had any SAH or clinical problem at all. Foreign material is just that we usually get away with it, but the strategy of prophylactic aneurysm wrapping is not benign, as we can see here. 2. The patient had an aggressive revascularization strategy based on vessel imaging. I understand that she had hemiparesis and that her STA was surgically absent. For my part, however, the clinical workup and the justification for saphenous vein bypass would have been strengthened by the acquisition of perfusion data, which is not presented here. This would improve the value of this case report as well. It is, in sum, an interesting case and illustrates well that no good deed goes unpunished. Christopher M. Loftus Philadelphia, PA, USA