Odontogenic Maxillary Sinusopathies: a Radiological Classification.

Purpose: Before implant surgery, a preoperative radiological evaluation is recommended for recognizing maxillary inflammatory conditions. In order to avoid a failure of the dental procedure and prevent medical-legal consequences, it is necessary to treat patients suffering of maxillary sinus pathologies. The classification proposed in our study aims to standardize the reference values for mucosal thickening and to verify their association with the odontogenic or disventilatory causes of sinus pathology. Methods: The maximum mucosal thickness was measured at the level of the maxillary sinus floor: mucosal thickness was present when greater than 1 mm and was classified according to its extension within the sinus cavity. Results: Imaging data of 270 adult patients were included, performed for dental diagnosis and treatment planning, and they were divided into four main classes: Class I (85 pt.), mucosal thickness lesser than 2 mm, not pathological. Class II A (52 pt.), mucosal thickness between 2 and 5 mm, localized to the maxillary sinus floor: it is still considered non-pathological, and a "wait-and-see" approach is recommended. Class II B (46 pt.), mucosal thickness greater than 5 mm but localized at sinus floor: pathological mucosa with odontogenic etiology, requiring dental treatment. Class III A (39 pt.), mucosa thicker than 5 mm and concentric, most likely due to sinus ventilation disfunction: it requires maxillary antrostomy. Class III B (30 pt.), sinonasal manifestations such as nasal polyposis, retention cysts, mucocele, dental foreign body: pathological mucosa to be treated with ESS. Class IV (12 pt.), oroantral fistula: nasal endoscopic or oral approach. Conclusions: Our classification intends to suggest the better therapeutic option, in case of sinus pathology, according to the entity and pathogenesis of the mucosal thickening, in order to reduce complication and failure rate of dental surgery.

Treatment of Sinusitis Following Dental Implantation: A Systematic Review and Meta-Analysis.

BACKGROUND: Implantological procedures aimed at rehabilitating upper jaw edentulous patients (dental implant placement and/or maxillary sinus grafting) can sporadically result in sinusitis. In these patients, endoscopic sinus surgery is the most commonly employed treatment, but clinical scenarios and comprehensive management strategies are extremely heterogeneous across studies. OBJECTIVE: We sought to systematically define treatment strategies and related success rates for sinusitis following dental implantation, detailing different current treatment choices and concepts. METHODS: Adopting a PRISMA-compliant review framework, systematic searches were performed in multiple databases using criteria designed to include all studies published until November 2020 focusing on the treatment of human sinusitis following dental implantation. We selected all original studies, excluding case reports, specifying treatment modalities with objective treatment success definitions. Following duplicate removal, abstract and full-text selection, and quality assessment, we reviewed eligible articles for treatment modalities and success rates, which were pooled in a random-effects meta-analysis. RESULTS: Among 581 unique citations, eight studies (181 patients) were selected. Seven studies were retrospective case series. All studies relied on endoscopic sinus surgery, often coupled with intraoral accesses, and assessed therapeutic success endoscopically. The pooled treatment success rate was 94.7% (95% confidence interval, 91.5%-98%). Failures were treated in seven of 15 cases with further antibiotic therapies and in another seven cases with surgical revision. A single patient was lost to follow-up. CONCLUSIONS: Endoscopic sinus surgery appears to be the most frequent treatment of choice for sinusitis following dental implantation, with excellent success rates. The protean clinical picture drawn from the selected studies calls for the standardization of diagnostics and definitions in this field to enable direct comparisons between the results of different studies. The role of postoperative antibiotic therapies, which have been employed unevenly across studies, should also be prospectively investigated.

Maternal multiple sclerosis is not a risk factor for neurodevelopmental disorders in offspring.

BACKGROUND: Childhood neurodevelopmental disorders (NDDs), including specific learning disorders (SLD), attention deficit/hyperactivity disorder (ADHD) and autism spectrum disorder (ASD), are pathogenically linked to familial autoimmunity and maternal immune-mediated diseases during pregnancy. OBJECTIVE: We studied maternal MS as a potential risk factor for NDDs occurrence in offspring. METHODS: MS and control mothers were subjected to questionnaires to ascertain NDD diagnosis in their progeny and the occurrence of both autoimmune and neurodevelopment disorders in their families. Suspected NDD cases were evaluated to confirm or rule out the diagnosis. RESULTS: Of the 322 MS women, 206 (64%) have 361 children; of these, 27 (7.5%) were diagnosed with NDD (11% ADHD; 22% ASD; 67% SLD). NDD-risk in offspring was associated to family history of autoimmunity and to NDDs both in MS and non-MS mother families (r = 0.75; p = 0.005) whereas it was not associated to maternal MS. CONCLUSIONS: For the first time, we demonstrate that maternal MS does not predispose children to higher risk for NDD. On a mechanistic view, we suggest that the intrinsic organ-specific nature of MS does not impair the mother-child cross-talk in decidua nor does it influence fetal neurodevelopment.

Global and local brain connectivity changes associated with sudden unilateral sensorineural hearing loss.

Recent studies suggest that even moderate sudden sensorineural hearing loss (SSNHL) causes reduction of gray matter volume in the primary auditory cortex, diminishing its ability to react to sound stimulation, as well as reorganization of functional brain networks. We employed resting-state functional MRI (rs-fMRI), in conjunction with graph-theoretical analysis and a newly developed functional "disruption index," to study whole-brain as well as local functional changes in patients with unilateral SSNHL. We also assessed the potential of graph-theoretical measures as biomarkers of disease, in terms of their relationship to clinically relevant audiological parameters. Eight patients with moderate or severe unilateral SSNHL and 15 healthy controls were included in this prospective pilot study. All patients underwent rs-fMRI to study potential changes in brain connectivity. From rs-fMRI data, global and local graph-theoretical measures, disruption index, and audiological examinations were estimated. Mann-Whitney U tests were used to study the differences between SSNHL patients and healthy controls. Associations between brain metrics and clinical variables were studied using multiple linear regressions, and the presence or absence of brain network hubs was assessed using Fisher's exact test. No statistically significant differences between SSNHL patients and healthy controls were found in global or local network measures. However, when analyzing brain networks through the disruption index, we found a brain-wide functional network reorganization (p < 0.001 as compared with controls), whose extent was associated with clinical impairment (p < 0.05). We also observed several functional hubs in SSNHL patients that were not present in healthy controls and vice versa. Our results demonstrate a brain involvement in SSNHL patients, not detectable using conventional graph-theoretical analysis, which may yield subtle disease clues and possibly aid in monitoring disease progression in clinical trials.

Anatomic Variations of Ethmoid Roof and Risk of Skull Base Injury in Endoscopic Sinus Surgery: Statistical Correlations.

BACKGROUND: Recent developments in endoscopic sinus surgery (ESS) have increased the need to investigate the complex anatomic variations in the ethmoid roof and skull base, to inform the surgeon about the risk of damaging these crucial areas during ESS. OBJECTIVE: To offer a detailed description of sinus anatomy focusing on the key surgical landmarks in ESS and frontal recess surgery to standardize a systematic approach during the preoperative sinuses imaging evaluation.Methodology: A total of 220 computed tomography (CT) scans were reviewed to obtain six sets of measurements: the depth of the cribriform plate (CP); the length of the lateral lamella of the cribriform plate (LLCP); the angle formed by the LLCP and the continuation of the horizontal plane passing through the CP; the position of the anterior ethmoidal artery (AEA) at the skull base; the extent of frontal sinus pneumatization (FSP); the type of superior attachment of the uncinate process (SAUP). RESULTS: The length of the LLCP was statistically significantly correlated with the different Keros classification types, the angle formed by the LLCP with the continuation of the horizontal plane passing through the CP, and with the AEA position at the skull base. The depth of the olfactory fossa was correlated with FSP. CONCLUSIONS: According to the Keros and Gera classifications, the data obtained from these evaluations allow the assessment of anatomic-radiological risk profiles and can help identify those patients who are high risk for ethmoid roof injury.

Treatment of Paranasal Sinus Fungus Ball: A Systematic Review and Meta-Analysis.

OBJECTIVES: Paranasal sinus fungus ball is a common non-invasive mycosis with excellent long-term surgical treatment results. The present systematic review and meta-analysis were undertaken to define current treatment concepts and success rates in paranasal sinus fungus ball treatment. METHODS: Systematic searches were performed in multiple databases with criteria designed to include all studies published until May 2020 focusing on paranasal sinuses fungus ball treatment in humans. We selected studies including at least 10 patients, specifying treatment modalities, providing a minimum 6-month follow-up, and objectivating treatment success. After duplicate removal, abstract and full-text selection, and quality assessment, we reviewed eligible articles for treatment modalities and success rates. Success rates were pooled in a random effect meta-analysis and compared according to the use of intraoperative sinus lavages and postoperative antibiotics. RESULTS: Among 740 unique citations, 14 studies were deemed eligible. Most (n = 11) were retrospective case series. All studies relied on endoscopic sinus surgery. Intraoperative lavages were proposed in 10 studies and postoperative antibiotics in 7 (for all patients in 5 studies and for selected patients in 2). No significant heterogeneity was observed between results (Cochran's Q P = .639, I2 test = 0). Treatment success rate was 98.4% (95% confidence interval 97.4%-99.3%). Intraoperative sinus toilette and postoperative antibiotics didn't significantly improve the success rate. CONCLUSION: Endoscopic sinus surgery shows excellent results in fungus ball treatment. Further prospective studies might help further reducing antibiotics prescriptions in these patients and improve their management.

Definition and management of invasive fungal rhinosinusitis: a single-centre retrospective study.

OBJECTIVES: The purpose of this study was to correlate acute invasive fungal rhinosinusitis (AIFRS) and chronic invasive fungal rhinosinusitis with underlying diseases, aetiological microorganisms, clinical symptoms, radiological findings, and surgical and medical treatment to determine the subset of patients who require more accurate diagnostic investigation and to prevent irreversible complications. METHODS: This retrospective monocentric study included 17 patients who underwent endoscopic sinus surgery evaluated by paranasal computed tomography and magnetic resonance imaging. Age, sex and symptoms, and location of the invasive fungal infection and the causative fungus were analysed. RESULTS: In total, 4 patients were affected by the AIFRS form, and 13 by the chronic form. Diabetes mellitus was reported in 41.17% of cases, and haematological diseases in 23.52%. The maxillary sinuses were involved in 47.05% of cases and sphenoidal sinuses in 52.94%; Aspergillus fumigatus was the fungus in 76.47% of cases, and Zygomycetes in 23.53%. CONCLUSIONS: An understanding of the different types of fungal sinusitis and knowledge of their features play a crucial role in reaching prompt diagnosis and initiation of appropriate therapy, which is essential to avoid a protracted or fatal outcome.

Third molar maturity index and legal age in different ethnic populations: Accuracy of Cameriere's method.

Molar development is widely accepted as a reliable indicator of chronological age in a forensic context. A quantitative method for age estimation has been proposed by Cameriere et al. based on the relationship between chronological age and the third molar maturity index (I3M), which is the ratio between the two apical pulp widths and the total tooth length. Cameriere's cut off value of 0.08 was found to be a reliable tool in assessing the threshold of 18 years of age. The aim of this study was to evaluate the accuracy of the I3M in different ethnic populations focusing on its specificity (subjects correctly classified as <18 years based on I3M >0.08) and sensitivity (subjects correctly classified as ≥18 years based on I3M <0.08). A review of 22 scientific articles was performed, representing data from panoramic radiographs of 12,286 individuals (5723 males and 6563 females) from all over the world, including some ethnical subgroups. The I3M showed an overall sensitivity for both sexes ranging from 51.8% to 93.4% and a specificity ranging from 86.2% to 100%. The proportion of correctly classified individuals ranged from 74% to 95%. These results can be useful to refer the age estimation with the margin of error of subjects correctly classified as minors (specificity) or adults (sensitivity), according to sex, ethnicity and geographical distribution. The I3M can be considered a suitable method for estimating adulthood in forensic settings, regardless of sex. However, ethnic group can affect the accuracy.

Diagnostic Criteria for Odontogenic Sinusitis: A Systematic Review.

BACKGROUND: Odontogenic sinusitis affects a significant proportion of patients with paranasal sinus infections. Nevertheless, no shared diagnostic criteria for this condition have yet been implemented and published studies differ in their definition of the disease. OBJECTIVE: The present systematic review of the literature was undertaken to characterize and analyze the different diagnostic criteria currently employed for odontogenic sinusitis. METHODS: Systematic searches for studies published between 2009 and 2019 were performed in Medline, Embase, Web of Science, Cochrane Library, and ClinicalTrials.gov databases. Search criteria were designed to identify all studies focusing, even partially, on odontogenic sinusitis. Human original studies except single case reports published in the English, French, German, Spanish, or Italian language were included. We removed duplicate abstracts and conducted full-text reads, data extraction, and quality assessment procedures (using the Oxford Centre for Evidence-based Medicine levels of evidence and National Heart Lung and Blood Institute Study Quality Assessment Tools). We reviewed articles for diagnostic criteria, both in terms of definition and etiology identification. RESULTS: Among 1,000 unique citations, 63 studies were deemed eligible. Most articles (n = 45) were retrospective case series; a single randomized clinical trial was available. Only 49 studies reported diagnostic criteria, yet relied marginally on published guidelines (n = 10 articles) for identifying sinusitis, often choosing instead to develop their own clinical (n = 15 articles), endoscopic (n = 12 articles), and/or radiologic (n = 30 articles) criteria. For odontogenic focus identification, 14 papers required a multidisciplinary evaluation, 11 papers required a time relationship between dental procedures and sinusitis, 24 papers required oroscopy and/or dental evaluation, and 53 papers required computed tomography. CONCLUSIONS: Current diagnostic criteria for odontogenic sinusitis are extremely heterogeneous. Establishing shared diagnostic criteria aimed at defining both sinusitis and related odontogenic foci would spur collaboration between investigators and support more comprehensive outcomes evaluations together with a better understanding of treatment options.

Global and local reorganization of brain network connectivity in sudden sensorineural hearing loss.

Recent reports suggested that even moderate sudden sensorineural hearing loss (SSNHL) can be partly responsible for a loss of gray matter volume in the primary auditory cortex, hence reducing the capacity of the auditory cortical areas to react to sound stimulation. There is also evidence for a plastic reorganization of brain functional networks visible as enhanced local functional connectivity. The aim of this study was to use rs-fMRI, in conjunction with graph- theoretical analysis and a newly developed functional "disruption index" to study whole-brain as well as local functional changes in patients with acute and unilateral sensorineural hearing loss. No statistically significant differences in global or local network measures we found between SSNHL patients and healthy controls. However, when analyzing local metrics through the disruption index k, we found negative values for k which were statistically different from zero both in single subject analysis. Additionally, we found several associations between graph-theoretical metrics and clinical parameters.

Sex estimation of skeletons in middle and late adulthood: reliability of pelvic morphological traits and long bone metrics on an Italian skeletal collection.

There are several metric and morphological methods available for sex estimation of skeletal remains, but their reliability and applicability depend on the sexual dimorphism of the remains as well as on the availability of preserved bones. Some studies showed that age-related changes on bones can cause misclassification of sex. The purpose of this study was to establish the reliability of pelvic morphological traits and metric methods of sex estimation on relatively old individuals from a modern Italian skeletal collection. The data for this study were obtained from 164 individuals of the Milano CAL skeletal collection and average age of the samples was 75 years. In the pelvic morphological method, the recalibrated regression formula of Klales and colleagues (2012), pre-auricular sulcus, and greater sciatic notch morphology were used for sex estimation. With regard to the metric method, 15 standard measurements from upper and lower limbs were analyzed for sexual dimorphism. The results showed that in pelvic morphological approach, the application of regression formula of the revised Klales and colleague formula (2017) resulted in 100% accuracy. Classification rates of metric methods vary from 75.19 to 90.73% with the maximum epiphyseal breadth of proximal tibia representing the most discriminant parameter. This study confirms that the effect of age on sex estimation methods is not substantial, and both metric and morphological methods of sex estimation can be reliably applied to individuals of Italian descent in middle and late adulthood.

HPV infection and clinical profiles in laryngeal diseases. A preliminary study.

PURPOSE: The study analysed the presence of HPV in samples tissue from laryngeal chronic hyperplastic inflammation, with and without pre-neoplastic potential, and from squamous cell carcinoma of the larynx. The aim of this analysis was to evaluate the presence/absence of different types of HPV and their relationship to the clinical profile of the patients studied (habit of smoking and drinking). METHODS: Sixty cases were randomly selected from patients undergoing surgical treatment of the larynx for inflammatory/ neoplastic lesions and of neck nodes. Patients underwent standard clinical workup, comprising medical history and physical examination, panendoscopy, whole-body CT scan (in cancer patients), diagnostic or therapeutic microlaryngoscopy with laryngeal biopsy, and HPV evaluation. RESULTS: The HPV analysis showed an increased risk for heavy smokers of HPV positivity, as well as precancer lesions and cancer. Type 6 and 16 seem to be prevalent in all types of laryngeal mucosa disease, but pre-neoplastic conditions versus cancer seem to show a wider variety of HPV infections while cancer patients are invariably affected by types 6 and 66. Heavy smoking is related to HPV infection likewise alcohol in association with smoking. Advanced T is more associated with HPV positivity. CONCLUSIONS: These data impose a closer follow-up of smokers and pre-neoplastic cases and the utility of the broadspectrum polymerase chain reaction assay in laryngeal dysplastic and cancer lesions. This study may allow to develop biomarkers for early detection or recurrence surveillance, to identify therapeutic targets, and to begin individualization of treatment based on the biology of these tumours. KEY WORDS: HPV infection, Larynx, Laryngeal chronic hyperplastic inflammation, Squamous cell carcinoma.

The synergy between radiographic and macroscopic observation of skeletal lesions on dry bone.

The diagnosis of bone lesions is a fundamental part of the study of skeletal remains, both in the archeological and forensic context. On the one side, the literature proved the relevance of radiography for the detection of bone lesions; on the other side, the careful macroscopic observation of the morphology of bone lesions is often underestimated. For this study, we examined and performed plain radiography on 14 skeletons of the CAL Milano Cemetery Skeletal Collection diagnosed with rheumatoid arthritis, diabetes, multiple myeloma, metastatic cancer, and osteomalacia to compare the macroscopic morphology and radiographic visualization of bone lesions. At least 200 osteolytic lesions and 65 areas of proliferative bone reaction (either spongiosclerotic or periosteal) were studied. We realized "comparative sets" of macroscopic pictures and radiographic imaging of the same skeletal elements to allow comparisons of detection and recognition of bone lesions. As a result, while trabecular lesions may be lost through naked eye observation, many lesions can also be unperceived on radiographs due to contrast, including periosteal reactions, osteolytic lesions, and spongiosclerosis. The aim of this research was to investigate the strengths and pitfalls of digital radiography and macroscopic analysis and to demonstrate the synergy of a complementary approach between the two methods for lesion analysis in dry bone.

The role of intraoperative stroke volume variation on bleeding during functional endoscopic sinus surgery.

BACKGROUND: Functional endoscopic sinus surgery (FESS) is a minimally-invasive surgical technique for patients with paranasal sinus pathology. Surgical bleeding reduces operative field visibility and increases the incidence of serious complications. Epinephrine injection into the nasal mucosa and controlled hypotension are used to minimize bleeding. Hypotension carries risks and sometimes does not reduce surgical bleeding. The goal of this study is to discover which hemodynamic parameter better correlates with surgical bleeding. METHODS: We enrolled 55 patients undergoing FESS. Inclusion criteria: male or female with chronic rhinosinusitis (CRS), older than 18 years, ASA I to III and primary surgery. Exclusion criteria: ASA>III, cerebrovascular and cardiac disorders, supraventricular tachycardia, renal or hepatic diseases, non-treated arterial hypertension, beta-blocking agent therapy, platelet-inhibiting agent or anticoagulant therapy, coagulopathy, pregnancy, clotting disorders, presence of neoplastic lesions and history of cranio-facial surgery. We used standard ASA plus ClearSight to assess hemodynamic parameters. Surgical procedures were performed by one surgeon and divided in ten surgical times (from T0 to T9). Intraoperative bleeding was assessed using the Fromme-Boezaart Scale. RESULTS: Analysis between all the hemodynamic parameters registered and the Fromme-Boezaart Score showed a negative correlation between surgical bleeding and stroke volume variation (SVV) only. When dichotomizing according to Fromme-Boezaart Score (above or below 2), SVV was the only parameter which showed significant differences between groups. A cut-off of 12.5% in SVV is optimal to distinguish the group with the better surgical visibility from the group with the worst one. CONCLUSIONS: Targeting SVV larger than 12% achieves a possible reduction of the intraoperative bleeding in patients undergoing FESS.

Long-term outcome of autistic spectrum disorder: a retrospective case study in a southern italian region.

BACKGROUND: Autism Spectrum Disorder (ASD) is a complex neurodevelopmental disorder, characterized by impaired social communication and restricted and repetitive behaviours, as well as associated features including intellectual disability and impaired sensorimotor function. Despite a growing interest in this devastating disorder for families and young parents, there are no certainties as regards its aetiology, although a significant genetic background is considered to be important. Since there is little information about the social adaptation and quality of life of patients with Autism Spectrum Disorder, we decided to study and evaluate the long-term outcome and quality of life in a sample of children, adolescent and young adults. METHODS: This is a case study of subjects diagnosed with ASD and followed by clinics and rehabilitation centers in Campania region, in the south of Italy. The study sample was composed by 110 patients (83 males, 27 females), aged between 8.1 and 28.0 years (mean 20.6; median 21.2; SD ± 4.85), recruited in 8 rehabilitation centers of Campania region. A follow-up interview was performed by means of a questionnaire administered to the parents/caregivers of patients at a mean age of their son/daughter of 20.6 years (median 21.2 years; range 8.1-28.0). RESULTS: Reports from parents or caregivers show an overall improvement with regard to social and adaptive abilities in a group of teen-agers and young adults with ASD. Major concerns on significant quality of life parameters such as independent living, work experiences, friendships and relationships, accommodation type, recreational activities and personal autonomy were persisting. CONCLUSIONS: The present study shows an overall improvement with regard to social and adaptive abilities in a large number of subjects. Considerable problems are related to autonomy, employment opportunities and social relationships of these patients. Parents need more recreational activities and continuous support with facilities for families.

Procoagulant, tissue factor-bearing microparticles in bronchoalveolar lavage of interstitial lung disease patients: an observational study.

Coagulation factor Xa appears involved in the pathogenesis of pulmonary fibrosis. Through its interaction with protease activated receptor-1, this protease signals myofibroblast differentiation in lung fibroblasts. Although fibrogenic stimuli induce factor X synthesis by alveolar cells, the mechanisms of local posttranslational factor X activation are not fully understood. Cell-derived microparticles are submicron vesicles involved in different physiological processes, including blood coagulation; they potentially activate factor X due to the exposure on their outer membrane of both phosphatidylserine and tissue factor. We postulated a role for procoagulant microparticles in the pathogenesis of interstitial lung diseases. Nineteen patients with interstitial lung diseases and 11 controls were studied. All subjects underwent bronchoalveolar lavage; interstitial lung disease patients also underwent pulmonary function tests and high resolution CT scan. Microparticles were enumerated in the bronchoalveolar lavage fluid with a solid-phase assay based on thrombin generation. Microparticles were also tested for tissue factor activity. In vitro shedding of microparticles upon incubation with H2O2 was assessed in the human alveolar cell line, A549 and in normal bronchial epithelial cells. Tissue factor synthesis was quantitated by real-time PCR. Total microparticle number and microparticle-associated tissue factor activity were increased in interstitial lung disease patients compared to controls (84±8 vs. 39±3 nM phosphatidylserine; 293±37 vs. 105±21 arbitrary units of tissue factor activity; mean±SEM; p<.05 for both comparisons). Microparticle-bound tissue factor activity was inversely correlated with lung function as assessed by both diffusion capacity and forced vital capacity (r²â€Š= .27 and .31, respectively; p<.05 for both correlations). Exposure of lung epithelial cells to H2O2 caused an increase in microparticle-bound tissue factor without affecting tissue factor mRNA. Procoagulant microparticles are increased in interstitial lung diseases and correlate with functional impairment. These structures might contribute to the activation of factor X and to the factor Xa-mediated fibrotic response in lung injury.

Brainstem arteriovenous malformation presenting with dyspraxic handwriting in a young girl.

We report the case of a 11-year-old girl who developed an isolated hand-writing disorder with dysgraphia at the beginning of the school year in the sixth grade. A brain magnetic resonance angiography showed a round arteriovenous malformation sited in the left side of the midbrain extending to the ipsilateral medio-basal thalamus. Child neurologists should never neglect a thorough neurological evaluation in case of isolated worsening of handwriting, to rule out possible underlying organic causes.

A previously undescribed complex coronary artery anomaly.

Coronary artery anomalies are uncommon disorders. According to the literature, ≈1% of the general population is affected by a coronary artery abnormality. Coronary artery anomalies are often not associated with clinical signs, symptoms, or complications; nevertheless, they can be associated with congenital heart diseases and lead to sudden death. However, these anomalies are more often discovered as incidental findings at the time of coronary angiography or autopsy. The clinical relevance of coronary artery anomalies is closely related to the functional ability to provide adequate blood supply to the myocardial tissue. We describe a complex left coronary artery anomaly, not previously reported in medical literature, involving origin, course, and distribution of this vessel.