Dejan Radakovic, Khaled Hamouda, Kiril Penov, Constanze Bening, Sameh Sayed, Carsten Gietzen, Rainer G Leyh, Ivan Aleksic
Different arterial cannulation strategies are feasible for veno-arterial extracorporeal membrane oxygenation (VA-ECMO) in postcardiotomy shock. We aimed to analyze potential benefits and safety of different arterial cannulation strategies. We identified 158 patients with postcardiotomy cardiogenic shock requiring VA-ECMO between 01/10 and 01/19. Eighty-eight patients were cannulated via axillary or femoral artery (group P), and 70 centrally via the ascending aorta directly or through an 8 mm vascular graft anastomosed to the ascending aorta (group C)...
January 1, 2021: ASAIO Journal: a Peer-reviewed Journal of the American Society for Artificial Internal Organs
Satyajeet Misra, Bikram Kishore Behera, Chappity Preetam, Satyapriya Mohanty, Rudra Pratap Mahapatra, Priyank Tapuria, Anirudh Elayat, Anindya Nayak, Kunal Kotkar, John S McNeil, Randal S Blank
Tracheal tumors or masses causing critical airway obstruction require resection for symptom relief. However, the location and extent of these tumors or masses often preclude conventional general anesthesia and tracheal intubation. Peripheral cardiopulmonary bypass often is required before anesthetizing these patients. Herein, two cases of patients with tracheal masses, in whom awake peripheral cardiopulmonary bypass was instituted, are reported. The first case was that of an obese male child weighing 102 kg, with tracheal rhinoscleroma, who developed Harlequin, or north-south, syndrome after institution of femorofemoral venoarterial partial cardiopulmonary bypass...
November 25, 2020: Journal of Cardiothoracic and Vascular Anesthesia
Kenji Miyata, Mitsuko Akaihata, Yasuto Shimomura, Toshinori Hori, Kenitiro Kaneko, Akihisa Okumura
A 1-year-old boy presented with a 4-month history of hypertension, ptosis of the right upper eyelid, left hemifacial sweating, and flushing. He was diagnosed with Harlequin syndrome associated with Horner syndrome. Computed tomography revealed a mass lesion in the right superior mediastinum. Therefore, the patient underwent total tumor resection. Histological examination demonstrated ganglioneuroblastoma. The MYCN oncogene was not amplified, and the mitosis-karyorrhexis index was low. Accordingly, radiation and chemotherapy were not performed...
October 30, 2020: Child's Nervous System: ChNS: Official Journal of the International Society for Pediatric Neurosurgery
Saud Alobaida, Joseph M Lam
Hereditary sensory and autonomic neuropathy (HSAN) type IV, also known as congenital insensitivity to pain with anhidrosis (OMIM 256800), is part of a family of neurodegenerative disorders that manifest with variable sensory and autonomic neuropathies. In this report, we present a unique dermatological finding in a patient with HSAN type IV: bilateral harlequin syndrome that occurred in association with unilateral Horner syndrome, traumatic alopecia and Riga-Fede disease.
September 28, 2020: Pediatric Dermatology
Lauren Schultz, Amelia Mackarey, Caleb Oh, Paul Kent
Our goal is to describe a case of Harlequin syndrome associated with microwave ablation in the treatment of a symptomatic paraspinal mass in a child, along with a summary of the literature. Our patient is the only known case of persistent Harlequin syndrome associated with microwave ablation treatment of a symptomatic paraspinal mass. Harlequin syndrome is a rare neurological condition characterised by unilateral sweating and flushing of the face, neck and/or upper chest. The specific mechanism is unclear, but the majority of cases are believed to be a result of contralateral lesions along the sympathetic chain...
August 18, 2020: BMJ Case Reports
W P J Van Oosterhout
BACKGROUND: The harlequin sign or syndrome is a rare cranial autonomic condition characterized by unilateral diminished flushing and sweating of the face (and sometimes arm), in response to heat or exercise. It results from autonomic, mainly sympathetic dysfunction. Although the idiopathic form is the most common, underlying structural abnormalities in the head, neck, and thorax need to be excluded. METHODS AND RESULTS: Here, we describe the first case of the combination of primary cluster headache and the harlequin syndrome in a 49-year-old female patient...
July 11, 2020: Headache
Carlo Contento, Andrea Battisti, Bruno Agrò, Marina De Marco, Alessandra Iaiza, Laura Pietraforte, Patrizia Pisani, Antonio Proietti, Emiliano Vitalini, Andrea Montalto, Francesco Musumeci
OBJECTIVES: The Harlequin syndrome is a complication observed in patients receiving peripheral venoarterial extracorporeal membrane oxygenation. This condition is defined as a critical variation in the oxygen saturation between the upper and the lower part of the body deriving from a poor lung function. METHODS: Between July 2018 and November 2019, a total of 60 patients (42 men and 18 women; mean age 57.4 ± 10.0 years; range = 28-71 years) underwent peripheral venoarterial extracorporeal membrane oxygenation in our center...
May 2020: Perfusion
Michelle S Rovner, Amanda T Redding, Bethany Jacobs Wolf, Jeffrey Alexander Wharton, Caitlyn J Risely, Cory M Furse
BACKGROUND: Harlequin syndrome presents as differences in facial coloring due to unilateral flushing. This is the result of the inability to flush on the affected side due to the disruption of vasomotor and sudomotor sympathetic activity. The neurologically intact side appears flushed. A 2°C temperature difference between the flushed and nonflushed sides of the face has been detected in patients presenting with Harlequin syndrome. This difference in temperature might be detectable even in the absence of unilateral flushing, and this subclinical manifestation of the syndrome may occur more often than realized...
May 2020: Paediatric Anaesthesia
Cheng Ni, Ru-Hong Cheng, Jia Zhang, Jian-Ying Liang, Ru-Qu Wei, Ming Li, Zhi-Rong Yao
BACKGROUND: Neu-Laxova syndrome (NLS) is a rare hereditary disorder featuring intrauterine growth retardation, remarkable oedema with skin restriction, limb contracture, ichthyosis, and craniofacial anomaly. NLS shares multiple overlapping characteristics with several other inheritable refractory diseases: for example, harlequin foetus and restrictive dermopathy. To date, many NLS patients have been described, although the number of NLS cases with clear genetic aetiology remains limited...
December 1, 2019: European Journal of Dermatology: EJD
U Heiler, T Pitzen, M Fetter, M Ruf
Harlequin syndrome is a rare combination of symptoms, characterized by unilateral facial anhidrosis and paleness on the affected side, becoming obvious by contralateral flushing mainly during sports activity. The syndrome is mostly idiopathic, however it is also described as a complication of thoracic surgery, i.e. superior lobectomy. Here, we report on two cases of Harlequin syndrome following scoliosis surgery at the cervicothoracic junction.
December 2019: Der Orthopäde
Charles St-Arnaud, Marie-Michèle Thériault, Michael Mayette
No abstract text is available yet for this article.
February 2020: Canadian Journal of Anaesthesia
Richard Wagner, Martin Lacher, Andreas Merkenschlager, Moritz Markel
Harlequin syndrome (HS) is a rare dysautonomia of the sympathetic nervous system leading to asymmetric facial flushing and sweating. In the literature, only a few cases of HS after thoracoscopic tracheoesophageal fistula (TEF) repair are reported. We report on a newborn with TEF who developed HS after thoracoscopic repair. On the first day of life, the girl (3,480 g, gestation age: 41 week) underwent thoracoscopic repair of a type C esophageal atresia (TEF; OR time 105 minute) without complications. The postoperative course was uneventful, the patient swallowed and thrived well and did not require esophageal dilatations...
January 2019: European Journal of Pediatric Surgery Reports
Khadija Elboukhari, Hanane Baybay, Sara Elloudi, Zakia Douhi, Fatima Zahra Mernissi
Harlequin's syndrome is a rare dysautonomic syndrome of the face characterized by sweating with flush of one side and anhidrosis of the contralateral side. Mostly idiopathic although several secondary cases have been reported in the literature, the purpose of the treatment is mainly aesthetic and functional. We report the case of a patient having harlequin syndrome in its idiopathic form with a literature review.
2019: Pan African Medical Journal
James D Triplett, Eduardo E Benarroch, Jeremy K Cutsforth-Gregory
Pure autonomic failure (PAF) is a progressive syndrome of neurogenic orthostatic hypotension, widespread anhidrosis, urinary retention, and constipation without other neurologic manifestations. It is generally considered a peripheral ganglionic synucleinopathy. Natural history studies have described risk factors for the conversion of PAF to Parkinson's disease, multiple system atrophy, or dementia with Lewy bodies, yet the early stages of PAF are not well characterized. We present a patient with unilateral anhidrosis, contralateral facial flushing and hyperhidrosis consistent with Harlequin syndrome that, over 6 years, progressed to PAF, suggesting that PAF may present with focal autonomic impairment prior to generalized autonomic failure...
September 2019: Autonomic Neuroscience: Basic & Clinical
Mary Clare McKenna, Poornima Menon, Shane Smyth, Sean Murphy
Harlequin syndrome is a disorder of the autonomic nervous system. It clinically presents as a distinct line of hemifacial sympathetic denervation. We describe a case of Harlequin syndrome with co-existing central first-order Horner syndrome in the setting of a large thalamic hemorrhage with intraventricular extension.
July 10, 2019: Journal of Stroke and Cerebrovascular Diseases: the Official Journal of National Stroke Association
Jaeyoon Chung, Mark Berguson, Andrew Mendelson, Rebecca Barnett, Eric S Schwenk, Nathaniel R Evans, Scott W Cowan, Jordan E Goldhammer
No abstract text is available yet for this article.
March 18, 2019: Journal of Cardiothoracic and Vascular Anesthesia
Andrea Bräutigam, Gabor Mark Somfai, Heinrich Gerding
No abstract text is available yet for this article.
April 2019: Klinische Monatsblätter Für Augenheilkunde
Navid Pourtaheri, Derek Z Wang, Robert P Lesko, Christopher M Bonfield, Peter Taub, Anand R Kumar
Background and Significance: Apert syndrome is a congenital disorder of patients who typically present with bilateral coronal craniosynostosis and varying degrees of complex syndactyly of the hands and feet, among other features. We describe a unique presentation of a rare Apert-like patient with unilateral coronal craniosynostosis and complex syndactyly of the hands and feet. Case Report: A 2-year-old male patient presented to the craniofacial clinic with his mother due to a concerning head shape...
February 2019: Plastic Surgery
Julio Federico Camperio Ciani, Jorge Guerrel, Eric Baitchman, Rigoberto Diaz, Matthew Evans, Roberto Ibáñez, Heidi Ross, Eric Klaphake, Bradley Nissen, Allan P Pessier, Michael L Power, Caitlin Arlotta, Donna Snellgrove, Brad Wilson, Brian Gratwicke
Spindly Leg Syndrome (SLS) is a persistent animal welfare issue associated with the rearing of amphibians in captivity. We conducted two experiments to investigate the effects of diet, water composition and overfeeding on prevalence of SLS in newly metamorphosed harlequin frogs (Atelopus spp.). In our first experiment, we offered 400 full-sibling tadpoles of Atelopus certus isocaloric diets in treatments of 31%, 37%, 42% and 48% crude protein respectively. Tadpoles fed higher protein diets metamorphosed faster, but the incidence of SLS exceeded 80% in all treatments leading to the conclusion that variation in dietary protein was not responsible for causing SLS...
2018: PloS One
Ambreen Ijaz, Musharraf Jelani, Anila Panezai, Tahmina Rabbani, Iftikhar A Rasool, Jamil Ahmad, Abdul Wali
No abstract text is available yet for this article.
September 30, 2018: Congenital Anomalies
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