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1.
Colorectal Dis ; 2024 Sep 30.
Artigo em Inglês | MEDLINE | ID: mdl-39350340

RESUMO

AIM: The aim of this work was to investigate the incidence of enterocutaneous fistula (ECF), including both small bowel and colonic fistulas, in a defined population of 1.04 million during a 10-year period and to describe aetiology, treatments, care consumption and outcome. METHOD: A comprehensive search algorithm including diagnostic and procedural codes, enterostomal therapy nurse notes and in-hospital care for >60 days yielded 1970 search hits. After reviewing medical records, 187 patients with ECF were identified. RESULTS: The annual incidence of ECF was 2.3 per 100 000, the incidence of ECF with intestinal failure type II was 0.9 per 100 000. Spontaneous closure of the fistula occurred in 16.0% of patients, while closure was seen in 97.3% of patients who underwent reconstructive surgery with recurrences in 6.7% and 8.3%, respectively. Cumulative ECF-related in-hospital care until closure or end of follow-up was 4 (range 0-61) weeks. Eighty-eight patients (47%) received home-based healthcare including parenteral feeding and/or fistula wound care. The estimated overall mortality at 1, 3 and 5 years was 33.7%, 42.1% and 47.6% respectively. Mortality was mainly in patients without spontaneous closure or reconstructive surgery, and the risk of ECF-related death was 30.2%. CONCLUSION: This study defines the population-based incidence of ECF and reports a high overall mortality rate. Initial survivors were characterized by either spontaneous closure or eligibility for later reconstructive surgery, but with an eventual mortality rate of approximately 20%. ECF patients are high consumers of care: 55.1% needed ≥4 weeks in hospital and many received home-based healthcare.

2.
J Dig Dis ; 2024 Oct 01.
Artigo em Inglês | MEDLINE | ID: mdl-39350692

RESUMO

OBJECTIVES: Histopathological characteristics of granulomas in perianal fistula of patients with Crohn's disease (CD) remain unexplored. We aimed to assess the histopathological features of granulomas in perianal fistula in CD. METHODS: A retrospective analysis was conducted by reviewing the medical and pathological records of 4430 cases who underwent perianal fistulectomy at our hospital between June 2015 and June 2023. The patients were divided into the CD group, tuberculosis (TB), and non-CD group, respectively, based on their final diangosis. The detection rate of granulomas and differential histopathological features were investigated. RESULTS: Among the 4430 patients, granulomas were identified in 41 cases, including 25 had CD, 2 had pulmonary TB, and 14 only exhibiting perianal lesions with no other comorbidities. Additionally, there were altogether 93 CD cases, resulting in a detection rate of granuloma of 26.9%, which was considerably higher than that in the non-CD group (26.9% vs 0.3%, p < 0.001). The majority (85.7%) of the perianal fistula tissues in the non-CD group contained foreign body giant cells, while this was observed in only 1 (4.0%) out of the 25 cases with CD. We proposed that granulomas in the perianal fistula in the non-CD group were mostly foreign body granulomas. Moreover, granulomas in the non-CD group were larger than that of the CD group (1135 µm vs 519 µm, p < 0.001). CONCLUSION: Most CD cases have less granulomas (≤3) and no foreign body giant cells. Ribbon-like granulomas can be seen only in CD cases.

3.
World J Gastrointest Endosc ; 16(9): 533-539, 2024 Sep 16.
Artigo em Inglês | MEDLINE | ID: mdl-39351174

RESUMO

BACKGROUND: Esophagopericardial fistula (EPF) is a rare, life-threatening condition with limited scientific literature and no established management guidelines. This case report highlights a successful multidisciplinary approach and the innovative use of endoscopic vacuum assisted closure (endoVAC) therapy in treating this complex condition. CASE SUMMARY: A 16-year-old male with a history of esophageal atresia and colon interposition presented with progressive chest pain, fever, and dyspnea. Imaging revealed an EPF with associated pleural and pericardial effusions. Initial management with an esophageal stent failed, prompting the use of an endoVAC system. The patient underwent multiple endoVAC device changes and received broad-spectrum antibiotics and nutritional support. The fistula successfully closed, and the patient recovered, demonstrating no new symptoms at a 6-month follow-up. CONCLUSION: EndoVAC therapy can effectively manage EPF, providing a minimally invasive treatment option.

4.
Heart Rhythm ; 2024 Sep 27.
Artigo em Inglês | MEDLINE | ID: mdl-39353499

RESUMO

BACKGROUND: The Manufacturer and User Facility Device Experience (MAUDE) database houses medical device reports of adverse events involving medical devices marketed in the United States submitted to the U.S. Food and Drug Administration (FDA) by mandatory and voluntary reporters. The MAUDE database is frequently used in clinical studies to report on device-related complications. Data about its efficacy are scarce. OBJECTIVE: To compare the mandatory MAUDE database (MAUDE group) with the invitation-based POTTER-AF Study (POTTER-AF 1 group) regarding data quality, procedural characteristics, diagnosis, treatment, and survival. METHODS: The reports of esophageal fistula esophageal fistula following atrial fibrillation (AF) ablation in the MAUDE database were compared to those in the POTTER-AF Study between 01/08/2009 and 31/08/2019. RESULTS: Esophageal fistula was reported in 47 patients in the MAUDE group and in 81 in the POTTER-AF 1 group. Procedures were performed with radiofrequency, cryoenergy or laser energy in 66.0%, 31.9% and 2.1% (MAUDE group) and in 96.3%, 2.5% and 1.2% (POTTER-AF 1 group). The median time to symptoms was 21 (14, 32.5) days (MAUDE group) and 18.0 (6.8, 22.3) days (POTTER-AF 1 group; p=0.031). The diagnostic method was reported in 38.3% of patients in the MAUDE group and in 98.8% in the POTTER-AF 1 group, the treatment in 57.4% and 100% and the outcome in all patients. In the MAUDE group, treatment was surgical (51.9%), endoscopic (37.0%), combined (3.7%) or conservative (7.4%), compared to 43.2%, 19.8%, 7.4% and 29.6% in the POTTER-AF 1 group. Overall mortality was 76.6% in the MAUDE group and 61.7% in the POTTER-AF 1 group (p=0.118). CONCLUSION: In the mandatory MAUDE database, less esophageal fistula cases were reported as compared to an invitation-based study. The data quality in the MAUDE database was significantly poorer.

5.
Cureus ; 16(8): e68348, 2024 Aug.
Artigo em Inglês | MEDLINE | ID: mdl-39355074

RESUMO

Iatrogenic coronary artery fistula (CAF) can occur following acute myocardial infarction, percutaneous coronary intervention (PCI) procedures, and heart surgery. Iatrogenic CAF linked to PCI has a low incidence rate. Early diagnosis and treatment are crucial when an iatrogenic fistula develops, as it may lead to cardiac tamponade, myocardial infarction, or death. In this report, we present a case of iatrogenic CAF secondary to coronary perforation caused by guidewire-induced trauma, followed by stent implantation and balloon inflation in the context of acute coronary syndrome (ACS). It was successfully managed through prolonged balloon inflation within the previously implanted stent just prior to the rupture zone.

8.
J Oral Implantol ; 2024 Oct 03.
Artigo em Inglês | MEDLINE | ID: mdl-39360465

RESUMO

An oroantral communication (OAC) may form in the upper molar region after tooth extraction. The patient is a 59-year-old female, who is a non-smoker. At the initial visit, teeth #14, #15, and #17 were missing. After tooth #16 was extracted due to apical periodontitis, a bone defect with a diameter of approximately 4 mm was observed, leading to the formation of an oroantral fistula (OAF). Another window was created in the lateral wall adjacent to the superior part of the bone defect at the fistula site to achieve closure of the OAF through bone formation and simultaneously perform sinus floor elevation (lateral approach) for implant placement. Through this lateral window, instruments were inserted into the maxillary sinus towards the bone defect at the fistula site. During this process, the remaining bone between the lateral window and the bone defect at the fistula site was carefully removed with instruments, connecting the two bone defects to facilitate the manipulation of the instruments. The Schneiderian membrane was elevated without enlarging the tear. Six months after these surgeries, a CBCT scan confirmed the closure of the fistula with hard tissue and the elevation of the sinus floor. Subsequently, three implants were placed, and prosthetic treatment was completed. Follow-up data is provided, including periapical X-ray and CBCT images taken two years and three months after surgery (one year and three months after the placement of the final prosthetic structure). The progress so far has been favorable.

9.
World Neurosurg X ; 24: 100401, 2024 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-39380788

RESUMO

Pediatric type IVc perimedullary arteriovenous fistulae (PAVF) comprise a rare subcategory of spinal vascular malformations in which multiple spinal arteries directly connect with draining veins resulting in high-flow arteriovenous shunting and large intradural venous varicosities. Complete disconnection of the fistula is necessary to prevent hemorrhage or spinal compression. A surgical, rather than endovascular, approach proves favorable under specific circumstances. Due to the rarity of these pediatric fistulae, no large studies exist to enumerate these circumstances. This case report fills this void by detailing several considerations which favored surgery for a type IVc PAVF in a 17-year-old female patient.

10.
Radiol Case Rep ; 19(12): 6465-6469, 2024 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-39380824

RESUMO

Coronary artery fistulas (CAF) are rare abnormalities involving a connection between a coronary artery and an adjacent vessel or heart chamber. Here we discuss the case of a 47-year-old male patient who had multiple gunshot wounds (GSWs) to the chest and abdomen, suffering a through and through bullet wound to the heart from the left ventricle (LV) through the left anterior descending (LAD) coronary artery and exiting from the right ventricle (RV). At the time of his hospitalization, he underwent a non-ECG gated trauma CT scan and subsequent cardiac catheterization that showed patient has a CAF between the LAD and RVOT. Roughly 3 years after his injury, the patient had an ECG-gated coronary CT scan showing the CAF is still present. The patient is now experiencing symptoms of heart failure with suspected worsening of shunt flow from the fistula. This case sheds light on CAFs, their presentation and potential complications to raise awareness for clinicians and radiologists.

11.
AME Case Rep ; 8: 86, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-39380853

RESUMO

Background: Intraductal papillary mucinous neoplasm of the bile duct (IPMN-B) is a neoplastic disease of the bile duct with papillary hyperplasia and mucus secretion, which originates from the duct epithelium and rarely complicates with fistula formation. Case Description: The patient was admitted to the hospital due to abdominal pain and yellow skin. Laboratory results showed alanine aminotransferase 299 U/L, total bilirubin 350 µmol/L, computerized tomography showed severe dilatation of common bile duct and extrahepatic bile duct. Magnetic resonance cholangiopancreatography showed the intra- and extrahepatic bile ducts were markedly dilated, and the signal of the bile ducts was uneven. Endoscope identified a large amount of mucus above the papilla that flowing out from the fistula. Further cholangiography through the fistula showed significant dilatation of the extrahepatic bile duct. SpyGlass examination showed a large amount of gelatinous mucus in the bile duct lumen and "fish-scaly or coral" changes in the mucosa of the right anterior branch bile duct, hepatic hilum as well as lower common bile duct. IPMN-B with choledochoduodenal fistula was diagnosed. The patient was then discharged with nasal biliary drainage and biliary stenting, oral antipyretic and hepatoprotective drugs. The patient's biliary obstruction and symptoms of infection improved with medical treatment but recured. Unfortunately, the patient died 10 months after his first visit. Conclusions: SpyGlass has advantages in identifying the nature and extent of lesions, providing important references for diagnosis and treatment. Endoscopic intervention relieves biliary obstruction to some extent in patients with high operative risk or reluctance to undergo surgery.

12.
AME Case Rep ; 8: 87, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-39380863

RESUMO

Background: Medullary thyroid carcinoma (MTC) is a rare and aggressive type of thyroid cancer. Patients with this condition typically manifest palpable neck swelling and compressive symptoms. Biochemical evaluation and neck ultrasound play vital roles in diagnosis. The management options differ based on the extent of the disease. Case Description: This paper describes a 47-year-old male patient diagnosed with MTC invading the trachea and larynx. He presented with a 5 cm × 5 cm hard thyromegaly on the right side with right-sided level IV lymphadenopathy, measuring approximately 2 cm. He underwent total thyroidectomy, total laryngectomy, and bilateral neck dissection. Postoperatively, the patient developed a neck abscess and pharyngocutaneous fistula (PCF), which was managed surgically and had a satisfactory outcome. After 128 days of inpatient care, he was discharged in a stable condition with resolution of complications and had no evidence of local recurrence during the 6-month follow-up. Conclusions: MTC is a rare type of thyroid neoplasia that can manifest with various symptoms resulting from either the primary lesion or secondary invasion. Surgery remains the mainstay of treatment, however, there are limited options and no approved adjuvant therapies for patients with disseminated MTC. Complications that arise after total thyroidectomy and laryngectomy can be noteworthy and demand careful surveillance and immediate treatment to prevent further deterioration.

13.
AME Case Rep ; 8: 90, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-39380871

RESUMO

Background: This study highlights an unusual and previously unreported adverse event (AE) following the minimally invasive treatment of pancreatic walled-off necrosis (WON). The standard treatment for WON currently involves primary drainage via an ultrasound-guided endoscopic, typically transgastric, approach. This method is associated with lower mortality and morbidity rates compared to traditional surgery. However, emerging AEs from these procedures may necessitate the involvement of a multidisciplinary team. Our case highlights the potential for gastrovesical fistula development as a rare AE following endoscopic drainage. Treatment for our patient prioritized individualized and non-surgical strategy, although surgical revision was also considered. Case Description: A 42-year-old male presented with a large symptomatic pancreatic WON refractory to conservative management, necessitating transgastric drainage. Despite the gradual evacuation of the WON contents, treatment was complicated by stent-related issues, including inadvertent bladder penetration. Rather than surgical correction, a collaborative approach among urology, gastroenterology, and surgery teams was employed, focusing on conservative treatment strategies. This approach successfully resolved the fistula, leading to the patient's full recovery. Conclusions: Given the increasing use of endoscopic transluminal drainage in (peri)pancreatic collections, it is crucial to be aware of all potential AEs. To our knowledge, this is the first documented case of gastrovesical fistula following drainage of WON. Early recognition and a multidisciplinary approach are vital to manage this event.

14.
Artigo em Inglês | MEDLINE | ID: mdl-39384502

RESUMO

Sialocele is a common complication of parotid tumour surgery. Aspiration and pressure dressing are the most common conservative treatments for sialoceles. However, they occasionally exhibit a refractory nature. In this case report, a minocycline injection was administered for refractory sialocele following a condylar fracture treated using the retromandibular anterior transparotid approach. Aspiration of the sialocele and the same amount of minocycline injection using three-way stopcocks were performed four weeks after surgery. After the injection, the buccal swelling completely disappeared without complications, including facial palsy. Percutaneous injection of minocycline might be the first choice for postoperative refractory sialocele following condylar fracture.

15.
Surg Endosc ; 2024 Oct 09.
Artigo em Inglês | MEDLINE | ID: mdl-39384654

RESUMO

BACKGROUND AND AIMS: Post-surgical biliary leaks (PSBL) are one of the most prevalent and significant adverse events emerging after liver or biliary tract surgeries. Endoscopic retrograde cholangiopancreatography (ERCP) alone or combined with another approach (Rendez Vous) as treatment of PSBL obtains optimal outcomes due to the possibility of modifying the resistances in the biliary tree. METHODS: A retrospective double-center study was conducted in two tertiary centers. Consecutive patients who underwent at least one attempt of PSBL correction by ERCP or Rendez Vous procedure between January 2018 and August 2023 were included. The primary outcome was overall endoscopic clinical success. In contrast, the secondary outcomes were hospital stay exceeding five days and endoscopic clinical success with the first endoscopic procedure at the tertiary center. Both univariate and multivariate analyses were used to assess outcomes. RESULTS: 65 patients were included. Patients with one or multiple) leaks had more possibility to achieve the endoscopic clinical success compared to those affected by the association of leaks and stricture (96% vs 67%, p value 0.005). Leaks occurring in the main biliary duct had less probability (67%) to achieve the overall endoscopic clinical success compared to those in the end-to-end anastomosis (90%), in the resection plane or biliary stump (96%) or first or secondary order biliary branches (100%, p value 0.038). A leak-bridging stent positioning had more probability of achieving the endoscopic clinical success than a not leak-bridging stent (91% vs 53%, p value 0.005). CONCLUSIONS: ERCP and Rendez Vous procedures are safe and effective for treating PSBL, regardless of the type of preceding surgery, even if technical or clinical success was not achieved on the first attempt. A stent should be placed, if feasible, leak-bridging to enhance treatment efficacy.

16.
Cureus ; 16(9): e68591, 2024 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-39371760

RESUMO

Anterior cervical discectomy and fusion (ACDF) is a commonly performed procedure. One of its most feared complications is esophageal injury (EI). There is no standard approach on how to treat these injuries. TachoSil® (Corza Medical GmbH, Düsseldorf, Germany) is a fibrinogen-containing patch that has been used in various surgical areas. Here, we present a 68-year-old male patient who was diagnosed with an EI with secondary surgical site infection following a three-level ACDF (C4/5, C5/6, and C6/7). Initially, the patient underwent incision and drainage (I&D) of the surgical site abscess, primary repair of the esophageal tear, and esophageal catheter placement to create a controlled esophagocutaneous fistula. Postoperatively, he was diagnosed with a leak and underwent a second I&D procedure. The primary repair of the EI was augmented with a TachoSil patch, and the patient was started on glycopyrrolate. The site of EI was well-sealed with no re-leaks, and the patient was discharged after he had completed a course of intravenous (IV) antibiotics and had been on parenteral nutrition for a total of 40 days. This case shows that the use of TachoSil to augment the repair of ACDF-associated EI, and glycopyrrolate to decrease salivation could decrease the risk of leak and enhance the healing process. This is an observation that needs to be scrutinized in future studies.

17.
Can J Kidney Health Dis ; 11: 20543581241284749, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-39371934

RESUMO

Rationale: Pyoderma gangrenosum (PG) is a rare neutrophilic dermatosis which gives rise to painful ulcers. Pyoderma gangrenosum can be triggered by trauma, a phenomenon called pathergy. Here, we report the first case of PG arising from pathergy due to needle insertion overlying an arteriovenous fistula (AVF). This case report seeks to inform nephrologists about PG, this yet unreported presentation, and management in the context of hemodialysis. Presenting concerns: A 69-year-old woman presented to dermatology clinic for erythemato-violaceous plaques with central ulceration at the site of needle insertion overlying her AVF. The patient was known for chronic renal insufficiency secondary to C3 glomerulonephritis, for which she received hemodialysis. After an accidental burn which lead to appearance of a painful ulcer, following each needle insertion for hemodialysis, she would develop an erythematous papule that progressed to a painful ulcer with erythematous-violaceous borders. Diagnosis: Pyoderma gangrenosum was clinically diagnosed and both clinical and paraclinical evaluation did not reveal any secondary cause of PG. Intervention: Dialysis via AVF was suspended due to the risk of triggering more PG and was temporarily pursued by central venous catheter. The patient was initially treated with prednisone and topical corticosteroids. Furthermore, owing to the high recurrence rate of PG, colchicine was initiated in prevention to avoid resorting to immunosuppressive or long-term corticotherapy. Outcomes: The patient's lesions improved on prednisone, which was then tapered over 1 month. Following prednisone taper and continuing improvement of PG on colchicine and topical corticosteroids alone, the decision was taken to recommence dialysis via AVF after performing a negative pathergy test. Topical corticosteroids were ceased due to the risk of cutaneous atrophy and were replaced by pimecrolimus ointment. The patient has continued dialysis via AVF ever since, without recurrence. Novel Finding: This is the first case reported of PG arising from pathergy due to needle insertion overlying an AVF. Colchicine may be a safe and effective therapy for long-term treatment of PG in the context of hemodialysis.


Contexte: Le Pyoderma gangrenosum (PG) est une dermatose neutrophilique rare qui provoque des ulcères douloureux. Le PG peut être déclenché par un traumatisme, un phénomène appelé pathergie. Nous rapportons ici le premier cas de PG résultant d'une pathergie au site d'insertion de l'aiguille recouvrant une fistule artérioveineuse. Ce rapport de cas vise à informer les néphrologues sur le PG, sur cette présentation non encore rapportée et sur sa prise en charge dans le contexte de l'hémodialyse. Présentation du cas: Une femme âgée de 69 ans s'étant présentée à une clinique de dermatologie pour soigner des plaques érythémateuses violacées avec ulcération centrale au site d'insertion de l'aiguille recouvrant sa fistule artérioveineuse (FAV). La patiente était connue pour une insuffisance rénale chronique secondaire à une glomérulonéphrite à C3, pour laquelle elle recevait des traitements d'hémodialyse. À la suite d'une brûlure accidentelle ayant entraîné l'apparition d'un ulcère douloureux, la patiente développait une papule érythémateuse qui évoluait vers un ulcère douloureux aux bords érythémateux violacés après chaque insertion d'aiguille pour l'hémodialyse. Diagnostic: Le Pyoderma gangrenosum a été diagnostiqué cliniquement et l'évaluation, tant clinique que paraclinique, n'a révélé aucune cause secondaire de PG. Intervention: En raison du risque accru de déclenchement de PG, la dialyse par la FAV a été suspendue et remplacée temporairement par la dialyse par cathéter veineux central. La patiente a d'abord reçu un traitement de prednisone et de corticostéroïdes topiques. En raison du taux élevé de récidive de PG, un traitement préventif à la colchicine a été initié pour éviter le recours à une corticothérapie immunosuppressive ou à long terme. Résultats: Les lésions de la patiente se sont améliorées avec le traitement à la prednisone, celui-ci a ensuite été réduit progressivement sur une période d'un mois. Après la réduction progressive de la prednisone et en raison de l'amélioration continue du PG avec le traitement par colchicine et corticostéroïdes topiques seulement, la décision a été prise de recommencer la dialyse par la FAV après un test de pathergie négatif. Un risque d'atrophie cutanée a justifié l'arrêt des corticostéroïdes topiques qui ont été remplacés par une pommade de pimecrolimus. Depuis, la patiente a poursuivi sa dialyse par FAV, sans récidive. Principales observations: Il s'agit du premier cas signalé de PG résultant d'une pathergie au site d'insertion de l'aiguille recouvrant une fistule artérioveineuse. La colchicine peut s'avérer un traitement sûr et efficace pour le traitement à long terme du PG dans le contexte de l'hémodialyse.

18.
Eur J Case Rep Intern Med ; 11(10): 004806, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-39372152

RESUMO

Aorto-atrial fistula (AAF) and aorto-ventricular fistula (AVF) are a rare, but potentially life-threatening condition, where an anomalous connection forms between the aortic structures and the cardiac atria or ventricles. The exact incidence of this condition is unknown, as are the major precipitating factors and best treatment options. It may be congenital, secondary to conditions such as aortic dissection, infective endocarditis, or valve replacement, or iatrogenic in nature. In the case reported here a 72-year-old male who was found to have an infective endocarditis complicated by right AAF and AVF. This case illustrates the important role of transesophageal echocardiography in the evaluation of a patient with subacute infective endocarditis with a very rare complication. LEARNING POINTS: Aortic fistulas are a rare, potentially fatal, complications of subacute infective endocarditis.Early recognition is greatly assisted by transesophageal echocardiography.

19.
J Surg Case Rep ; 2024(10): rjae617, 2024 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-39372394

RESUMO

Cholecystocutaneous fistula is an exceedingly rare type of external biliary fistula, where an abnormal connection forms between the gallbladder and the skin. Cholecystocutaneous fistula commonly develops in the setting of chronic calculus cholecystitis or following a previous surgical intervention involving the biliary tract. Patients with cholecystocutaneous fistula often present with systemic symptoms, such as fever, nausea, and vomiting, as well as localized symptoms in the right upper quadrant of the abdomen, where the external opening of the fistula is typically found. Ultrasonography, computed tomography, magnetic resonance imaging, magnetic resonance cholangiopancreatography (MRCP), and fistulograms (computed tomography or X-ray) are commonly used. Computed tomography has proven to be more effective than ultrasonography in delineating the fistulous tract and the associated fluid collections. Open cholecystectomy with excision of the fistulous tract is considered the gold standard and is curative in the majority of cases. However, a laparoscopic approach has become a viable alternative, especially in the hands of experienced surgeons.

20.
Eur Heart J Case Rep ; 8(10): ytae507, 2024 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-39372649

RESUMO

Background: Vieussens' arterial ring (VAR) is an embryonic conotruncal ring remnant that represents a rare vascular anomaly involving a connection between the right coronary artery (RCA) and the left anterior descending artery (LAD). We describe a unique case of VAR associated with large aneurysmal dilatation, which presents as the formation of a fistula between the coronary artery and pulmonary artery. Case summary: An 80-year-old Japanese woman presented with an asymptomatic mediastinal mass that was incidentally detected on computed tomography. Subsequent imaging over 2 years revealed the progression of the two masses connecting the RCA to the LAD measuring 8 × 7 mm and 28 × 21 mm in diameter. Transthoracic echocardiography identified a cystic lesion anterior to the right ventricular outflow tract, and colour Doppler imaging confirmed flow into the pulmonary artery. Furthermore, coronary angiography revealed a large aneurysm arising from the LAD, with an efferent vessel communicating with the pulmonary artery. Surgical intervention involved resection of the aneurysms and closure of the coronary-pulmonary artery fistula, which yielded favourable postoperative outcomes. Discussion: Vieussens' arterial ring is a rare but clinically significant anomaly with varied presentations; the incidence of VAR remains uncertain. In this case, surgical resection and closure of the fistula were performed to mitigate the risk of rupture and to address the potential haemodynamic consequences. Understanding and documenting such cases will contribute to refining treatment approaches and improving patient care in cardiovascular medicine.

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