Vascular stenting of a malignant arterial blowout as a bridge to effective systemic and regional therapy.

Locally advanced cutaneous squamous cell carcinoma can erode into blood vessels, leading to vascular blowout, requiring emergent surgical intervention. We describe a first case of this disease complication which was effectively managed with endovascular stenting as a bridge to effective systemic and regional therapy. We discuss the efficacy of this staged approach which is novel and timely in a clinical environment of increasingly effective systemic therapies.

Feasibility and Intermediate Results of Transcarotid Revascularization with a Prosthetic Conduit.

BACKGROUND: Transcarotid artery revascularization (TCAR) is a hybrid technique with excellent initial outcomes. The technical success and safety of TCAR is heavily dependent on an anatomically suitable common carotid artery (CCA). Many patients do not meet anatomic criteria and therefore are not eligible for this therapy. We sought to extend the eligibility of TCAR to patients with unfavorable CCA anatomy via the adoption of a prosthetic arterial conduit. METHODS: A single-center retrospective study of patients with critical carotid artery stenosis who underwent TCAR via a prosthetic conduit between June 2019 and October 2021 was performed. All patients in the study were considered high-risk for carotid endarterectomy based on anatomic features, such as restenosis post-carotid endarterectomy and neck radiation. Unfavorable CCA anatomy was defined as a clavicle to carotid bifurcation distance <5 cm, a CCA diameter <6 mm, and/or significant atherosclerotic disease at the intended arterial access site. The primary outcome of interest was technical success. Secondary outcomes included perioperative complications, intermediate and long-term patency, intermediate and long-term stroke and/or mortality and in-hospital length of stay. Follow-up ranged from 1 to 29 months. RESULTS: Eight patients underwent 10 TCAR procedures via a prosthetic conduit. A total of 2 procedures (20%) were performed on female patients and 8 procedures (75%) were performed on male patients. The mean age was 65 years old (standard deviation 11 years). Technical success was 100%. The 30-day ipsilateral stroke rate was 0%. The 30-day patency was 90%. There was no re-exploration for hemorrhage and 30 day mortality was 0%. CONCLUSIONS: TCAR is an excellent option for carotid artery revascularization. Unfavorable CCA anatomy has limited its applicability. TCAR via a prosthetic conduit has the potential to expand eligibility for this promising therapy.

Restoring Venous Patency with the ClotTriever Following Deep Vein Thrombosis.

BACKGROUND: Although randomized data remain inconclusive, invasive endovenous therapy is increasingly favored in patients with extensive iliocaval or iliofemoral deep vein thrombosis (DVT) to reduce the rates of postthrombotic syndrome. Previously, pharmacomechanical thrombectomy was the therapy of choice, but the Inari ClotTriever device is an appealing, purely mechanical, alternative. It may reduce bleeding risk, intensive care unit admission, and the need for multiple procedures when compared with traditional thrombolysis. We present a series of 18 patients treated with the ClotTriever for extensive iliocaval or iliofemoral DVT. METHODS: The Inari ClotTriever is a percutaneous mechanical thrombectomy system consisting of an expandable nitinol collection bag that is dragged along the vein wall, separating and capturing thrombus for collection into the retrieval sheath. We retrospectively reviewed all patients undergoing ClotTriever thrombectomy since the device became available at our quaternary referral center in June 2019. The review of these patients' records was determined to be exempt by our institutional review board. RESULTS: Eighteen patients underwent ClotTriever thrombectomy between June 2019 and November 2021. Most patients (N = 16, 89%) presented within 2 weeks of symptom onset, and identifiable provoking factors were present in all patients. The most common provoking factor was anatomy, with May-Thurner syndrome present in 8 patients. All patients had restoration of unimpeded venous flow in the treated segments, although 3 had some residual nonflow limiting thrombus. There were no bleeding events or repeat venous procedures. The median postprocedure length of stay of 2 days. Postoperative venous imaging was performed in 15 patients and showed patency of the treated segment in 14 patients. Revised Clinical Venous Severity Scores were available in 14 patients during the course of follow-up. Of these, 9 patients' highest scores were 0, 2 patients' highest scores were 2, 2 patients' highest scores were 4, and 1 patient had a high score of 8. CONCLUSIONS: Venous flow was re-established in all 18 patients treated with the ClotTriever in this series, with no bleeding complications, and median postprocedure length of stay of 2 days. All patients with available follow-up, except 1, retained patency of the treated venous segments, and most had mild postthrombotic syndrome or none at all. These findings suggest that the ClotTriever is a safe and effective way to treat extensive iliocaval/femoral DVT.

Cerebral monitoring during transcarotid artery revascularization with flow reversal via transcranial doppler ultrasound examination.

OBJECTIVE: Transcarotid artery revascularization (TCAR) is a hybrid technique for carotid artery revascularization that relies on proximal carotid occlusion with flow reversal for distal embolic protection. The hemodynamic response of the intracranial circulation to flow reversal is unknown. In addition, the rate and pattern of cerebral embolization during flow reversal has yet to be investigated. The aim of this study was to characterize cerebral hemodynamic and embolization patterns during TCAR. METHODS: A single-institution retrospective study of patients with carotid artery stenosis undergoing TCAR with intraoperative transcranial Doppler (TCD) monitoring of the middle cerebral artery (MCA) was performed. Primary outcomes included changes in MCA velocity and MCA embolic signals observed throughout TCAR. RESULTS: Eleven patients underwent TCAR with TCD monitoring of the ipsilateral MCA. The average MCA velocity at baseline was 50.6 ± 16.4 cm/s. MCA flow decreased significantly upon initiation of flow reversal (50.5 ± 16.4 cm/s vs 19.1 ± 18.4 cm/s; P = .02). The reinitiation of antegrade flow resulted in a significant increase in the number of embolic events compared with baseline (P = .003), and embolic events were observed in two patients during flow reversal. CONCLUSIONS: TCD monitoring of patients undergoing TCAR revealed that the initiation of flow reversal results in a decrement in ipsilateral MCA velocity. Furthermore, embolic events can occur during flow reversal and are significantly associated with the reinitiation of antegrade flow in the internal carotid artery. However, both of these hemodynamic events were well-tolerated in our cohort. These findings suggest that TCAR remains a safe neuroprotective strategy for carotid revascularization.

Retrograde Transamputation Revascularization: A Case Report.

Below-knee amputation remains the most common level of amputation in patients with lower extremity gangrene and critical limb ischemia. Failure to heal, requiring additional operative debridement or conversion to an above-knee amputation remains a significant cause of patient morbidity. There remains no definitive diagnostic test that can accurately predict healing of the amputation site. We report a case utilizing a hybrid technique of retrograde transamputation revascularization via balloon angioplasty. This proximal, retrograde approach allows for relatively easy crossing and treatment of the infrainguinal chronic total occlusions (CTOs), improving arterial inflow for optimal wound healing.

Negative pressure wound therapy reduces surgical site infections.

OBJECTIVE: Surgical site infection (SSI) with lower extremity incisions represents a modifiable source of major morbidity. Our institutional bundled care protocol to decrease SSI includes optimization of perioperative risk factors, dedicated wound closure tray, and voluntary use of a closed surface negative pressure wound therapy (cNPWT) device applied over closed incisions in the operating room. This study examined the individual effect of cNPWT on SSI reduction and other perioperative outcomes. METHODS: All patients with lower extremity or infrainguinal incisions between January 2016 and December 2017 were prospectively identified and tracked for infectious complications. All patients were treated with the same perioperative care bundle to reduce SSI. cNPWT was applied over closed incisions at the discretion of the surgeon. The 90-day outcomes regarding SSI, return to operating room, death, and readmission were tracked. Univariate and multivariate analysis using binary logistic regression for factors associated with SSI was performed for patients with and without cNPWT devices, with P < .05 determined to be significant. RESULTS: There were 504 patients included, 225 with cNPWT and 279 with standard dressings. Between the groups, there were no major differences in mean age, mean body mass index, perioperative transfusions, use of prosthetic, reoperative field, dialysis status, and presence of diabetes. There were significantly more women (39.6% vs 27.2% female; P < .01) and active smokers (47.1% vs 30.2%; P < .01) in the cNPWT group along with increased mean operative times (238.3 vs 189.0 minutes; P < .01). Univariate analysis revealed significantly fewer SSIs with cNPWT (9.8% vs 19.0% in standard dressings; P < .01) along with decreased perioperative mortality (5.8% vs 11.2%; P = .04). There were no differences in return to operating room (27.6% cNPWT vs 27.7% standard; P = .97) or readmissions (29.8% cNPWT vs 26.5%; P = .43), but more returns to the operating room were for wound-related problems in the standard dressings group (48.3% vs 26.2%; P < .01). Binary logistic regression using an SSI end point demonstrated that female sex increases SSI (odds ratio, 2.43; confidence interval, 1.37-4.30; P < .01), whereas cNPWT reduces SSI (odds ratio, 0.32; confidence interval, 0.17-0.63; P < .01). CONCLUSIONS: The use of negative pressure wound therapy devices decreases the incidence of infrainguinal wound infections. This occurs as an independent factor as part of a patient care bundle targeting modifiable variables in perioperative care.

Jejunal arterial access for retrograde mesenteric stenting.

Endovascular approaches have replaced open surgical revascularization in most patients with mesenteric ischemia; however, flush ostial occlusions may not be amenable to traditional antegrade access. Retrograde mesenteric stenting has been previously described, but this technique requires a formal laparotomy and dissection of the proximal superior mesenteric artery. We present here a modification of this technique that requires only a "mini-laparotomy" and no open vascular repair of the superior mesenteric artery as well as a review of our initial institutional experience with this procedure. Our approach differs from previously described work by minimizing mesenteric dissection, avoiding the need for repair of an arteriotomy, and limiting the size of the laparotomy incision in this population of profoundly comorbid patients.

Laparotomy during endovascular repair of ruptured abdominal aortic aneurysms increases mortality.

OBJECTIVE: Subset analyses from small case series suggest patients requiring laparotomy during endovascular repair of ruptured abdominal aortic aneurysms (REVAR) have worse survival than those undergoing REVAR without laparotomy. Most concomitant laparotomies are performed for abdominal compartment syndrome. This study used data from the American College of Surgeons National Surgical Quality Improvement Program to determine whether the need for laparotomy during REVAR is associated with increased mortality. METHODS: Data were obtained from the 2005 to 2013 National Surgical Quality Improvement Program participant user files based on Current Procedural Terminology (American Medical Association, Chicago, Ill) and International Classification of Diseases-9 Edition coding. Patient and procedure-related characteristics and 30-day postoperative outcomes were compared using Pearson χ2 tests for categoric variables and Wilcoxon rank sum tests for continuous variables. A backward-stepwise multivariable logistic regression model was used to identify patient- and procedure-related factors associated with increased death after REVAR. RESULTS: We identified 1241 patients who underwent REVAR, and 91 (7.3%) required concomitant laparotomy. The 30-day mortality was 60% in the laparotomy group and 21% in the standard REVAR group (P < .001). The major complication rate was also higher in the laparotomy group (88% vs 63%; P < .001). Multivariable analysis showed laparotomy was strongly associated with 30-day mortality (odds ratio, 5.91; 95% confidence interval, 3.62-9.62; P < .001). CONCLUSIONS: Laparotomy during REVAR is a commonly used technique for the management of elevated intra-abdominal pressure and abdominal compartment syndrome development. The results of this study strongly confirm findings from smaller studies that the need for laparotomy during REVAR is associated with significantly worse 30-day survival.

The impact of intraoperative shunting on early neurologic outcomes after carotid endarterectomy.

BACKGROUND: Although the need for intraoperative shunting during carotid endarterectomy (CEA) is intensely debated, relatively few studies have compared the neurologic outcomes of patients undergoing CEA with or without shunts. The objective of our analysis was to determine the impact of intraoperative shunting during CEA on the incidence of postoperative stroke. METHODS: The 2012 CEA-targeted American College of Surgeons National Surgical Quality Improvement Program database was used for this analysis. The preoperative and operative characteristics of patients undergoing CEA with or without intraoperative shunting were compared. From this overall sample, propensity score techniques were then used to match patients with or without intraoperative shunting for a number of variables, including age, degree of ipsilateral and contralateral carotid stenosis, presence of several anatomic or physiologic risk factors, anesthesia modality, and use of patch angioplasty vs primary arteriotomy closure. The 30-day postoperative mortality and combined stroke/transient ischemic attack (TIA) rates of this matched cohort were then compared. A similar analysis was also performed on a subgroup of patients with severe stenosis or occlusion of the contralateral carotid artery. RESULTS: A total of 3153 patients were included for initial analysis (2023 "no-shunt" patients vs 1130 "shunt" patients). From this overall sample, propensity score matching yielded a cohort of 1072 patients with or without intraoperative shunt placement who were well matched for all known patient- and procedure-related factors. There was no significant difference in the incidence of postoperative stroke/TIA between the two groups of this matched cohort (3.4% in the no-shunt group vs 3.7% in the shunt group; P = .64). Analysis of a similarly well matched subgroup of patients with severe stenosis or occlusion of the contralateral carotid artery demonstrated a statistically nonsignificant increase in the incidence of postoperative stroke/TIA with the use of intraoperative shunting (4.9% in the no-shunt group vs 9.8% in the shunt group; P = .08). CONCLUSIONS: There is no clinical benefit to intraoperative shunting during CEA, even in patients who may be at high risk for intraoperative cerebral hypoperfusion due to severe stenosis or occlusion of the contralateral carotid artery.

Mycotic Saccular Abdominal Aortic Aneurysm in an Infant after Cardiac Catheterization: A Case Report.

Abdominal aortic aneurysms (AAAs) are a rare entity in the pediatric population. Children with mycotic (infectious) AAA in particular are at risk of life-threatening rupture due to their rapid expansion coupled with aortic wall thinning and deterioration. Here, we present the case of a 10-month-old infant with prior 2-staged repair for hypoplastic left heart syndrome that was incidentally discovered to have a mycotic AAA on abdominal ultrasound (US) for evaluation of renovascular hypertension. Before the time of evaluation with US, the infant had developed methicillin-resistant Staphylococcus aureus bacteremia 3 days after cardiac catheterization with percutaneous thoracic aortic balloon angioplasty. She had normal aortic contours on contrasted computed tomography scan of the abdomen approximately 2 weeks before the aforementioned US evaluation. This infant subsequently underwent open aneurysmorrhaphy with cryopreserved vein patch angioplasty with resolution of her aneurysmal segment.

Spliced arm vein grafts are a durable conduit for lower extremity bypass.

BACKGROUND: Many patients with peripheral vascular disease (PAD) requiring revascularization do not have adequate ipsilateral great saphenous vein (GSV) for constructing a bypass because of intrinsic vein disease or prior harvesting for limb or coronary bypass. Prosthetic conduits have poor long-term patency, especially for distal bypass. With advancing endovascular sophistication, tibial angioplasty may be a good revascularization option, but we hypothesize that using spliced arm vein for distal lower extremity bypass is still a well-tolerated and more durable solution. METHODS: A retrospective chart review was conducted of all PAD patients undergoing lower extremity bypass or tibial angioplasty for lifestyle-limiting claudication or critical limb ischemia at a single institution over a 7-year period. Statistical analysis was conducted by Kaplan-Meier survival analysis and Cox proportional hazards model. Statistical significance was set at P = 0.05. RESULTS: From 2005 to 2012, there were 120 patients who underwent infrageniculate revascularization with conduit other than GSV. Over half of the patients (66 patients, 71.2% male, mean age 62 years) underwent bypass operations using arm vein conduit, and 88% of those bypasses were to tibial vessels. Patency was 100% at 1 year and 85% at 2 years. There was no impact on patency or amputation rate based on the source of vein or the number of splices. Forty-three patients underwent tibial angioplasty and patency was 70% at 1 year and 50% at 2 years. CONCLUSIONS: When GSV is not available, spliced arm vein grafts provide durable lower extremity revascularization with favorable patency and limb preservation rates. Spliced arm vein grafts should be considered over prosthetic grafts and angioplasty alone in patients with distal occlusive disease.

Outcomes of surgical revascularization for lower extremity arterial thromboembolism in patients with advanced malignancy.

OBJECTIVE: The objective of this study was to describe the outcomes of surgical revascularization for lower extremity arterial thromboembolism in patients with advanced malignancy. METHODS: The 2005-2011 American College of Surgeons National Surgical Quality Improvement Program database was used for this study. Preoperative characteristics and postoperative outcomes of patients with known advanced malignancy who underwent surgical revascularization for lower extremity arterial thromboembolic disease were assessed and compared with those of patients without known advanced malignancy. Parsimonious multivariate logistic regression analysis was used to determine the independent association between advanced malignancy and 30-day postoperative mortality and morbidity after adjustment for demographic characteristics, acute and chronic comorbid disease burden, history of peripheral arterial disease, functional and nutritional status, acuity of presentation, and procedure type. RESULTS: The study included 136 patients with advanced malignancy who underwent surgical revascularization for lower extremity arterial thromboembolism for analysis (65% thromboembolectomy, 26% thromboendarterectomy, and 22% bypass grafting). The 30-day mortality and morbidity rates in these patients were 30.2% and 38.2%, respectively. Compared with patients without advanced malignancy, patients with advanced malignancy had a significantly greater risk of postoperative death (adjusted odds ratio, 5.92; 95% confidence interval, 3.69-9.52; P < .001) but not morbidity (adjusted odds ratio, 1.28; 95% confidence interval, 0.87-1.87; P = .21). CONCLUSIONS: Our study is the largest to date to describe the outcomes of patients with advanced malignancy who undergo surgical revascularization for arterial thromboembolism of the lower extremities. Such patients suffer high rates of early postoperative mortality and morbidity, especially when emergency operation is required. Early involvement of palliative care specialists is warranted in these patients to ensure that the decision to pursue surgical revascularization is aligned with their goals of care.

Atypical aortic thrombus: should nonoperative management be first line?

BACKGROUND: Aortic thrombus in the absence of atherosclerotic plaque or aneurysm is rare, and its optimal management remains unclear. Although atypical aortic thrombus (AAT) has been historically managed operatively, successful nonoperative strategies have been recently reported. Here, we report our experience in treating patients with AAT that has evolved from a primarily operative approach to a first-line, nonoperative strategy. METHODS: Records of patients treated for AAT between 2008 and 2011 at our institution were reviewed. RESULTS: Ten female and three male patients with ages ranging from 27 to 69 were identified. Seven were treated operatively and 6 nonoperatively. Initial presentation was variable and included limb thromboembolic events (n = 6), visceral ischemia (n = 5), and stroke (n = 1). Associated risk factors included hypercoagulability (76%; n = 10) and hyperlipidemia (38%, n = 5). In the nonoperative group, complete thrombus resolution was obtained via anticoagulation (n = 5) or systemic thrombolysis (n = 1). Complete thrombus extraction was achieved in all operative patients. There were 11 significant complications in 5 of the 7 patients (71%) in the operative group, including intraoperative lower extremity embolism, pericardial effusion, stroke, and 1 death. There was 1 complication in the patients treated nonoperatively. The median hospital length of stay was 9 days (range 3-49) for those treated nonoperatively and 30 days (range 4-115) for those undergoing operative thrombectomy. CONCLUSIONS: Although AAT has traditionally been treated operatively, nonoperative management of AAT with anticoagulation or thrombolysis is feasible in selected patients and may lessen morbidity and length of hospitalization in those patients for whom it is appropriate.

Endovascular Treatment of a Multi-visceral Aortic Conduit Blowout With Parallel Stent Grafts and Coils.

Multi-visceral transplantation (MVT) is a complex surgical procedure involving the transplantation of multiple abdominal organs as a single unit, typically used as bailout treatment of patients with devastating abdominal pathologies. Due to the complexity of the procedure, major and even life-threatening complications can happen. Vascular complications, including anastomotic breakdowns or pseudoaneurysms due to infections, can be universally lethal. Open surgical repair is often not an option due to the hostile operative field. We report a case of endovascular salvage of multi-visceral aortic conduit blowout utilizing parallel stent grafts and coils without sacrifice of the transplanted viscera. This combination can successfully control bleeding and maintain graft perfusion in this rare but devastating complication.

Endovascular Management of Aortic Stump Blowout by Parallel Grafting and Coil Embolization of Visceral Aorta.

Aortic graft and endograft infections remain a significant source of morbidity and mortality after abdominal aortic aneurysm repair. With graft excision and extra-anatomic bypass, an infrarenal aortic stump remains which can have suture line dehiscence and catastrophic stump blowout. Treatment of this is extremely challenging, especially for severely co-morbid patients who cannot undergo major surgery, or in patients with a hostile abdomen. We present a case study of a 74-year-old male found to have an aortoenteric fistula (AEF). This case broadens operative options for this type of patient population by demonstrating an endovascular technique for addressing aortic stump blowout by parallel grafting and coil embolization of the visceral aorta.

D-dimer as a Rule-Out for Aortic Dissection.

Introduction Acute aortic dissection (AAD) represents a significant diagnostic challenge with a high mortality rate if not treated promptly. This challenge arises from the diverse clinical presentations of AAD, and its symptom overlap with other medical conditions. Although both helical CT and transesophageal echocardiography are reliable diagnostic tools for AAD, they are not feasible for every suspected case. Furthermore, limited research on D-dimer's utility in ruling out AAD has been conducted due to the condition's rarity. Methods This study utilizes the TriNetX database (https://trinetx.com/), encompassing data from 54 healthcare organizations across the United States over the past two decades from 85 million patients. The objective is to evaluate the sensitivity of an elevated D-dimer level in diagnosing AAD across a much larger patient cohort than previously studied. Results Retrospectively analyzing this dataset, there were 1,319 patients identified with a confirmed AAD who had undergone D-dimer testing within a day of diagnosis. Of these, 1,252 patients exhibited D-dimer levels exceeding 400 ng/ml while 1,227 had levels surpassing 500 ng/ml. Notably, a D-dimer cutoff of 400 ng/ml demonstrated a sensitivity of 0.949 while a 500 ng/ml cutoff yielded a sensitivity of 0.930. Conclusion This large retrospective cohort study demonstrates that a blood D-dimer level is highly sensitive in assaying for AAD. The D-dimer levels analyzed showed a remarkable sensitivity in ruling out AAD, avoiding the need for more invasive testing in low-risk patients.

Surgical trainee participation during infrainguinal bypass grafting procedures is associated with increased early postoperative graft failure.

OBJECTIVE: This study was conducted to determine the potential effect of surgical trainee participation during infrainguinal bypass procedures on postoperative graft patency rates. METHODS: Data from the National Surgical Quality Improvement Program (NSQIP) Participant User Files from 2005 through 2009 were retrospectively reviewed, using propensity score matching, to identify all patients undergoing infrainguinal bypass grafting procedures, excluding those who had prior operation ≤30 days of the index procedure. A separate analysis was performed on a subset of procedures from the entire NSQIP sample that was matched on propensity for intraoperative surgical trainee participation. The primary predictor variable was intraoperative surgical trainee participation. The main outcome measure was the 30-day postoperative graft failure rate. RESULTS: For the entire sample of 14,723 NSQIP patients undergoing infrainguinal bypass grafting, 30-day graft failure rates were significantly higher when a surgical trainee participated (5.8%) vs without participation (3.9%; P < .0001). For the cohort of 9234 patients matched on their propensity for intraoperative trainee participation, this difference in graft failure rate remained significant (5.0% with participation vs 4.0% without participation; P = .02). CONCLUSIONS: Surgical trainee participation is an independent risk factor for technical failure after infrainguinal bypass grafting. Prospective evaluation is needed to determine the cause of this increase in graft failure rates for procedures that involve surgical trainees.

Vertebral artery transection with pseudoaneurysm and arteriovenous fistula requiring antegrade and retrograde embolization.

Traumatic vertebral artery injury is a rare, life-threatening injury that has been increasingly managed with endovascular intervention. However, an antegrade endovascular approach alone can fail to occlude traumatic pseudoaneurysms (PSAs) and arteriovenous fistulas (AVFs), requiring high-risk surgical reoperation. We have presented the case of a 27-year-old man with traumatic right vertebral artery PSA and AVF. Despite successful ipsilateral coil embolization, the PSA and AVF persisted via retrograde filling from the contralateral vertebral artery. Distal coil embolization was achieved through the contralateral vertebral artery in a novel "up and over" approach through the basilar artery. The findings from our case report have broadened the endovascular options for complicated traumatic injuries.

Venous thoracic outlet syndrome secondary to first rib osteochondroma in a pediatric patient.

Osteochondromas account for 30% to 50% of benign osseous tumors and 10% of all bone tumors. Most of these lesions are found incidentally on imaging studies obtained for other reasons. Vascular compromise due to osteochondroma is a rare but well-recognized phenomenon and typically occurs in the lower extremity as a result of a tumor mass projecting into the popliteal fossa. We present the very rare case of a pediatric patient with venous thoracic outlet syndrome due to an osteochondroma of the first rib, and to our knowledge, this report is only the second such occurrence in the medical literature.