Abstract Background Single coronary artery (SCA) is a rare congenital anomaly that may have serious clinical consequences in different clinical settings despite often being asymptomatic. Case Summary We report a case of a 75-year-old patient with a SCA originating from the right sinus of Valsalva presenting for pre-operative assessment for mitral valve (MV) stenosis. The patient developed cardiogenic shock post MV replacement surgery on grounds of iatrogenic occlusion of the ectopic left main coronary artery which was treated with a percutaneous revascularization procedure. Discussion SCA represents a major anatomical variant that can significantly impact clinical outcomes, particularly during cardiac surgery. An abnormal coronary anatomy poses technical challenges, due to potential inadvertent injury or compression of the anomalous artery relative to the extent of the myocardium at risk.

This video tutorial explains how to treat an aortic aneurysm affecting the arch, descending, and thoracoabdominal segments, in the presence of an anatomical aortic arch variation. The procedure involves replacing the entire aortic arch and ascending aorta using the classic elephant trunk technique, creating a platform for a planned staged endovascular treatment. Special emphasis is placed on cerebral protection strategies, including preservation of an anomalous left vertebral artery and special reinforcement of a distal arch anastomosis. The presence of an anomalous artery arising directly from the aortic arch increased the technical difficulty, requiring customized revascularization to ensure appropriate left posterior cerebral circulation. This case reinforces the idea that open techniques can be safe for selected elderly patients with complex anatomies, highlighting the importance of tailored strategies and careful surgical execution.

Trigeminocerebellar artery (TCA) is known as a causable artery of trigeminal neuralgia (TN) because this anomalous artery has unfavorable anatomic features to perform the microvascular decompression. We aimed to clarify the long-term outcomes of TN caused by TCA and demonstrate surgical concepts with various techniques for its treatment. Patients who underwent surgeries for TN by TCA were retrospectively reviewed with intraoperative findings, surgical techniques, and long-term outcomes were investigated. Surgical treatments of TN were performed on 146 patients, and 13 patients (9%) had symptoms owing to TCA. Patients in TCA group (average age: 59.1 years; sex [male:female = 3:10]; affected side [right:left = 10:3], affected area [V2: 8; V3: 1; V1 + 2: 3; and V2 + 3: 1]) had TCAs with the following anatomic characteristics: penetration of the trigeminal nerve (8 patients; 62%) and presence of several perforators (all patients) (median: 3 branches). Various surgical techniques (transposition: 5 patients [38.5%]; arterial reshaping: 1 patient [12.5%]; arterial reshaping + interposition: 2 patients [25.0%]; and partial sensory rhizotomy + transposition: 5 patients [62.5%]) were applied as required. Although 1 patient required postoperative oral medication, the other 12 patients (92%) experienced complete symptom resolution immediately after the surgery, and no recurrence was observed during the median follow-up period of 72 months. This study among 146 patients with TN observed 13 patients (9%) caused by TCA and confirmed the favorable long-term outcomes in 12 patients (92%) with complete resolution of symptom without recurrence. As TCA has disadvantageous anatomic structures to be separated from trigeminal nerve, to treat TN caused by TCA, contemporary standard techniques are insufficient at times and various case-by-case surgical techniques must be required.

The anomalous origin of the right pulmonary artery from the ascending aorta is a rare congenital anomaly usually diagnosed in infancy. When diagnosed later during adolescence, it presents diagnostic and treatment challenges, especially in determining whether pulmonary vascular disease can be reversed. We report the case of an 11-year-old girl who experienced increasing fatigue and was diagnosed with anomalous origin of the right pulmonary artery from the ascending aorta through echocardiography, CT angiography, and cardiac catheterisation. Segmental pulmonary hypertension was noted, but the operability was uncertain. A new non-invasive test combining 2D phase-contrast cardiac MRI with inhaled iloprost was performed. Flow measurements revealed a 42% rise in right pulmonary artery flow, indicating preserved vasoreactivity. Lung biopsy confirmed pulmonary vascular changes consistent with Heath-Edwards Stage II-III. Based on these findings, surgical reimplantation of the anomalous artery into the main pulmonary artery was performed. The postoperative recovery after surgery was uneventful, and follow-up catheterisation demonstrated normalised haemodynamics without residual stenosis. This case highlights the potential value of cardiac MRI-based vasoreactivity testing with inhaled iloprost as an additional tool alongside standard diagnostics for evaluating operability in late-presenting anomalous origin of the right pulmonary artery from the ascending aorta. Combining imaging, histopathology, and haemodynamic data allowed a personalised and safe surgical approach.

Y-incision aortic annular enlargement (Y-AAE) is increasingly utilized to facilitate valve upsizing. Its application in patients with an anomalous left circumflex artery (LCx) introduces additional complexity. We present a case of a Y-AAE with aortic valve replacement in a patient with an anomalous LCx originating from the right coronary sinus. The annulus was enlarged from 25 mm to accommodate a size 29 prosthesis. The anomalous LCx was carefully dissected from the aortic root and preserved, with no postoperative myocardial ischaemia. We reviewed 4 institutional cases of Y-AAE with upsizing by 3 valve sizes or to the largest size (size 29). One patient required primary repair of an LCx injury. During follow-up of up to 3 years, no patient developed myocardial ischaemia. These findings suggest that Y-AAE can be safely performed in the presence of an anomalous LCx, enabling optimal valve upsizing.

ABSTRACTAnomalous systemic arterial supply to the left basal segment is a rare congenital vascular anomaly, particularly when complicated by dissection of the feeding artery. This case report aims to present a unique instance of this condition and to review relevant literature. We present a young patient who was hospitalized with sudden chest pain, leading to the discovery of an anomalous artery originating from the thoracic aorta that supplied blood to the basal segment of the left inferior lobe of the lung, accompanied by dissection of the feeding artery. The patient underwent successful surgical resection of the anomalous vessel via thoracotomy, and postoperative recovery was unremarkable during follow‐up evaluations. This case highlights the importance of recognizing anomalous systemic arterial supply as a potential cause of acute chest pain and underscores the need for timely surgical intervention.

Coronary artery anomalies are rare congenital conditions that are usually detected incidentally during coronary angiography or post-mortem examinations. Although many variants are clinically asymptomatic, they can be fatal in some cases. In this article, we present a 35-year-old male patient who presented with acute inferior myocardial infarction causing complete atrio-ventricular block. Angiography revealed a rare coronary anomaly, the left circumflex artery originating from the right sinus of Valsalva. The lesion, caused by severe stenosis in the proximal segment of this anomalous vessel, was successfully treated with primary percutaneous coronary intervention. This case highlights the diagnostic and therapeutic challenges of such anatomic variations in the setting of acute coronary syndrome and reviews the current literature on interventional strategies.

A COVID-19-positive baby with acute respiratory failure and a working diagnosis of bronchiolitis deteriorated rapidly despite usual resuscitative measures. Respiratory acidosis and oxygenation improved with invasive ventilation; however, the patient became progressively hypotensive and showed a poor response to vasopressors. Bedside ultrasound showed right heart dilatation, troponin was raised, and the ECG had ischaemic changes. Echocardiography diagnosed anomalous aortic origin of the right pulmonary artery (RPA), with continuous left-to-right shunt and holodiastolic flow reversal in the abdominal aorta. This raised suspicion of a coronary steal phenomenon, explaining deterioration in response to typical resuscitation. An oxygen saturation (SpO2) target of 80-88% and permissive hypercapnia helped restore balanced circulations and improve coronary perfusion. He ultimately underwent a complete surgical repair with good recovery. We discuss opportunities for earlier recognition of cardiac differential diagnoses in a neonate with respiratory failure, including the use of bedside ultrasound, which is of interest to paediatric generalists, intensivists and emergency practitioners.

An anomalous origin of the right pulmonary artery is a rare phenomenon in neonates with transposition of the great arteries. We report a case of dextro-transposition of the great arteries with an anomalous right pulmonary artery originating from the innominate artery, diagnosed by cardiac CT angiography and the cinematic rendering technique. Although surgical correction was possible, it was deferred due to complex anatomy and parental preference. This case highlights the diagnostic value and role of advanced imaging in complex congenital heart disease.

Commando procedure and coronary artery bypass grafting for severe mitral anular calcification and anomalous left circumflex artery from the right coronary artery

Rationale:The posterior circulation represented by the vertebrobasilar artery system is responsible for 20% of brain perfusion. Posterior circulation ischemia can cause a variety of symptoms due to the broad area supplied by the vertebrobasilar artery. In addition, stroke is the second leading cause of death globally, highlighting the importance of studying any potential contributing factors.Patient concerns:A 68-year-old nonsmoker and nonalcoholic with a history of diabetes type 2 and a previous stroke, presented with 2 days of visual disturbance with a burning sensation associated with occipital headache, fever, and polyuria.Diagnosis:He was admitted to our hospital due to his symptoms and during his evaluation, a computed tomography scan was performed and showed a bilateral calcification on the vertebral arteries. Magnetic resonance angiography showed a variant vessel connected between the vertebral arteries at the level of medulla.Intervention:The patient was admitted to the intensive care unit after being diagnosed with stroke complicated with aspiration pneumonia and diabetic ketoacidosis. He was treated with IV fluids and insulin pump without using thrombolysis or vascular intervention due to the time of symptoms.Outcomes:The patient’s condition and laboratory tests improved and was discharged after 2 weeks.Lessons:This study investigates an extremely rare variant of the Circle of Willis and the potential consequences associated with such a variant, including stroke, which is considered one of the most lethal outcomes. The study highlights the importance of investigating the association between this variant and the incidence and frequency of stroke. The patient in the study developed stroke twice and atherosclerosis due to this variant.

Abstract Vascular anomalies during pancreaticoduodenectomy are critical considerations to prevent surgical complications. This case report presents a unique confluence of vascular anomalies encountered during a pylorus-preserving pancreaticoduodenectomy (PPPD), highlighting the importance of thorough preoperative imaging and intraoperative vigilance. A 35-year-old female underwent PPPD for the second part of the duodenum lesion. Intraoperatively, we identified an anomalous right hepatic artery arising from the superior mesenteric artery (SMA), in addition to the usual right and left hepatic arteries from the proper hepatic artery. Furthermore, the superior mesenteric vein (SMV), splenic vein, and inferior mesenteric vein (IMV) converged separately to form the portal vein. Despite these anatomical variations, the PPPD was completed successfully with the preservation of all hepatic arterial branches. The coexistence of an anomalous additional right hepatic artery from the SMA and separate convergence of SMV, IMV, and splenic vein represents a unique vascular pattern. This case contributes to the growing body of literature on vascular variations in hepatopancreatic biliary surgery and emphasizes the need for flexible, informed, and cautious approaches to ensure optimal patient outcomes.

Identifying the blood supply from a systemic artery is crucial for diagnosing extralobar pulmonary sequestration. Contrast-enhanced computed tomography (CT) can accurately delineate the origin and course of anomalous systemic arteries and venous drainage. However, it remains challenging to make an accurate diagnosis in cases with atypical CT manifestations. To improve our understanding of the diversity of extralobar pulmonary sequestration and facilitate a more accurate diagnosis, especially for cases with atypical CT manifestations. A retrospective review was conducted on 58 patients who had confirmed extralobar pulmonary sequestration by surgery between November 2019 and December 2023. Patient demographics, clinical manifestations, and CT findings were reviewed. Among the 58 patients, 38 (65.5%) were male, and the left-to-right ratio was 47 to 11. It typically appeared triangular or polygonal, with the sharp angular sign observed in 55 patients (94.8%). The low-density branch sign was identified in 45 patients (77.6%) via contrast-enhanced CT. Lesions were located as follows: above the diaphragm in 45 cases (77.6%), within the diaphragm in seven cases (12.1%), and below the diaphragm in two cases (3.4%), presenting as solid masses; two instances (3.4%) involved anterior mediastinum lesions that presented as cystic-solid masses. In two cases (3.4%), the sequestered lung communicated with the lower esophagus, presenting as multiple air-filled cystic lesions. Abnormal arterial blood supply was detected in 54 patients (93.1%), whereas venous drainage was identified in 52 patients (89.7%). The presence of the sharp angular sign in a solid mass with clear boundaries suggests a diagnosis of extralobar pulmonary sequestration on routine CT, regardless of its location above, within, or below the diaphragm. CT angiography is a reliable imaging modality for documenting feeding arteries and draining veins, which confirms the diagnosis of extralobar pulmonary sequestration. The low-density branch sign on contrast-enhanced CT indicates that the lesion comprises lung tissue and helps establish a diagnosis of pulmonary sequestration.

Structural and Functional Intersection: A Case of ACS with Anomalous Left Circumflex Artery

Abstract Anomalous origin of the pulmonary artery (AOPA) is a rare congenital condition with significant clinical implications. We present a unique case of a 5-day-old neonate exhibiting respiratory distress, diagnosed with AOPA from the left innominate trifurcation confirmed by computed tomography pulmonary angiography (CTPA). This specific variant has not been previously reported. The patient had a large sub-aortic ventricular septal defect (VSD) and an overriding aorta. Early diagnosis and surgical intervention are critical for improving outcomes, underscoring the need for heightened clinical awareness and prompt management of this condition.

Congenital anomalies of the cervical spine are rare, often asymptomatic conditions that may only be detected incidentally. However, they can have significant clinical implications, particularly when associated with vascular anomalies. This case highlights a unique combination of congenital hypoplasia of the C6 pedicle and superior articulating facet, resulting in an anomalous course of the vertebral artery (VA). A 28-year-old female presented to the emergency department after a minor trauma without neurological deficits. Imaging revealed congenital hypoplasia of the right C6 pedicle and superior articulating facet, absence of the C6 transverse foramen, and anomalous entry of the right VA at the C5 transverse foramen. The patient was managed conservatively with a cervical collar and remained neurologically intact at follow-up. This case underscores the importance of thorough radiological evaluation in identifying rare cervical spine malformations and their potential clinical significance. Recognition of such anomalies is critical for surgical planning and risk mitigation, especially during anterior cervical spine procedures. https://thejns.org/doi/10.3171/CASE25161.

ANOMALOUS CIRCUMFLEX CORONARY ARTERY AND HEREDITARY HEMORRHAGIC TELANGIECTASIA: THE PERFECT INGREDIENTS FOR A STENT THROMBOSIS

Anomalous Celiac Artery in Pulmonary Sequestration

Pulmonary sequestration was first described by Pryce in 1946. It is a developmental abnormality of the lung, in which a portion of lung tissue is isolated from the normal lung and receives blood supply from an anomalous artery branching off the body's circulatory system. In recent years, advances in diagnostic imaging, particularly computed tomography (CT), have made the preoperative diagnosis of pulmonary sequestration more straightforward. Thoracoscopic surgery has increasingly been used for treatment;however, there is still no definitive evidence regarding the extent of resection or the use of automatic suturing devices for the dissection of abnormal arteries. In this report, we summarize the cases of pulmonary sequestration treated surgically at our hospital and discuss the clinical presentation and surgical techniques.

The superficial ulnar artery is a rare anatomical variant that can be misidentified as the radial artery during radial forearm free flap harvest, potentially leading to ischemic complications. Awareness of this anomaly is crucial for reconstructive surgeons to prevent inadvertent vascular injury. We present a case where intraoperative identification techniques helped distinguish this variant, ensuring a successful surgical outcome. A 54-year-old male with squamous cell carcinoma involving the upper lip, buccal mucosa, left commissure, and lateral third of the lower lip underwent wide local excision and modified radical neck dissection. Reconstruction was planned using a radial forearm free flap. Preoperative Allen's test confirmed adequate ulnar artery perfusion. During flap harvest, an unexpected arterial structure was encountered superficial to the flexor muscles, initially mimicking the radial artery. Further dissection revealed the true radial artery between the flexor carpi radialis and brachioradialis muscles, confirming the superficial vessel as an anomalous ulnar artery. The superficial ulnar artery was preserved and buried within the flexor muscle bundle to avoid trauma. Postoperatively, the flap showed satisfactory perfusion, and the patient's hand function remained intact. This case highlights the importance of recognizing vascular anomalies during forearm flap harvest to prevent iatrogenic complications. Simple intraoperative techniques, such as careful proximal tracing of vascular bundles and the "tug test," can aid in distinguishing the true radial artery from aberrant vessels. Preoperative vascular imaging may be beneficial in selected cases where anatomical variations are suspected.