Harlequin Syndrome Related to Perioperative Intercostal Blockade: A Case Report.

Harlequin syndrome is a rare syndrome characterized by hemifacial flushing and altered facial sweating, with only a few case reports related to intercostal blockades. We present a case of Harlequin syndrome in a 65-year-old woman after intercostal blockade for video-assisted thoracoscopic lobectomy. One hour postoperatively, the patient became nauseated and presented with flushing of the right half of the face with a clear line of demarcation. Within 3 hours, the flushing disappeared. In this case report, we discuss Harlequin syndrome in relation to intercostal blockade and encourage clinicians to consider this syndrome in the differential diagnosis when encountering similar symptoms.

Hypohidrosis as an immune-related adverse event of checkpoint inhibitor therapy.

Background: Immune checkpoint blockade therapies including cytotoxic-T-lymphocyte antigen 4 (CTLA-4) and programmed cell death protein-1 (PD-1) inhibitors have become indispensable tools for treating melanoma and other cancers. An increasing number of diverse cutaneous adverse reactions to immunotherapy have been documented in the literature and have been reported to affect up to 40% of patients treated with targeted therapies. Method & results: Herein, we report a case of a patient with metastatic melanoma treated with checkpoint inhibitor therapy who developed vitiligo, gastritis and hepatitis, all identified as adverse immune events and attributable to his immunotherapy regimen. He subsequently developed acquired idiopathic generalized hypohidrosis with biopsy of lesional skin demonstrating a peri-eccrine lymphocytic infiltrate. Conclusion: These findings suggest this acquired generalized hypohidrosis represents a lymphocyte-mediated adverse immune event related to this patient's checkpoint inhibitor therapy.

Botulinum toxin type D blocks autonomic cholinergic synapses in humans: discussion of a potential therapeutic use.

Based on epidemiological data it was believed that botulinumtoxin type D (BT-D) may not block human cholinergic synapses. We wanted to investigate BT-D's effect on the autonomic cholinergic synapse in humans. For this, we compared in four volunteers intraindividually the hypohidrotic effect of intradermal BT-D and BT-A in Minor's iodine starch sweat test. Altogether, we studied BT-D in doses of 4, 8, 16 and 32MU and BT-A in doses of 2, 4, 8 and 16MU at weekly intervals throughout a period of 13 weeks. All BT doses were diluted in 0.2 ml 0.9% NaCl/H2O. Overall 704 data points were collected. Combined over all four subjects and all four doses BT-D's hypohidrotic effect intensity was half of BT-A's. BT-D's effect peaked around 5 weeks, BT-A's around 7 weeks. BT-D's effect duration was around 12 weeks, of BT-A's was around 14 weeks. For both BT types the hypohidrotic effect was dose dependent. BT-D, when injected intradermally, can block autonomic cholinergic synapses in humans. Compared to BT-A, BT-D's effect intensity was half and its effect duration was some 2 weeks shorter. With its weaker and shorter effect BT-D does not seem to promise therapeutic effects superior to BT-A.

Unilateral anhidrosis: A rare complication of thoracic epidural analgesia.

Management of pain following thoracotomy is an important issue for the control of early morbidity. We herein present the case of a patient who was referred to our hospital after a fall from a height. Right-sided multiple rib fractures, hemopneumothorax, and diaphragmatic rupture were detected. Thoracic epidural catheterization was performed for pain management just before thoracotomy. The patient developed unilateral anhidrosis postoperatively. We discuss this rare complication of thoracic epidural analgesia with a review of relevant literature.

Diffusion of two botulinum toxins type A on the forehead: double-blinded, randomized, controlled study.

BACKGROUND: Different diffusion of different botulinum toxin type A (BoNTA) preparations may account for differences in outcomes in cosmetic clinical practice. OBJECTIVES: A double-blind, randomized, self-controlled study was performed to evaluate the diffusion characteristics of onabotulinumtoxinA and a Chinese type A botulinum toxin (CBTX-A). MATERIALS AND METHODS: Healthy volunteers (N = 20) were recruited to receive a 0.05-mL (2 U) injection of BoTNA at four forehead sites (medial forehead (subcutaneous (SC)) and temporal forehead (intradermal (ID))). On day 14, the Minor's iodine starch test was performed and photographs were taken for calculating the area and dimensions of anhydrotic area. RESULTS: When BoNTAs were different, the anhidrosis ID area was significantly greater with CBTX-A than onabotulinumtoxinA, the vertical dimension was significantly longer with CBTX-A ID than onabotulinumtoxinA ID and the horizontal dimension was significantly greater with CBTX-A ID than onabotulinumtoxinA ID. The area of anhidrosis SC was significantly greater with CBTX-A than onabotulinumtoxinA. When injection depths were different, the mean horizontal dimension was significantly greater with onabotulinumtoxinA SC than ID. Comparing the dimension of the same BoNTA and injection method, the vertical dimension was significantly greater than the horizontal dimension. CONCLUSION: OnabotulinumtoxinA diffuses less than CBTX-A. ID injection technique may result in less diffusion than SC.

Concomitant horner and harlequin syndromes after inferior alveolar nerve block anesthesia.

INTRODUCTION: Intraoral anesthesia is probably the most commonly used procedure in dentistry. METHODS: Although inferior alveolar nerve block (IANB) anesthesia is one of the safest procedures to anesthetize the mandibular teeth, side effects of IANB anesthesia can still give rise to potential risks for patients. Fortunately, most observed alterations are transient and self-limited. RESULTS: The complications of IANB anesthesia are varied in nature and could be specifically categorized into systemic, localized, and distant complications. When the complications occur around the orofacial structures including the temporomandibular joint, middle ear, facial skin, and the eye, which are away from the oral cavity, it can be defined as distant complications. However, to our best knowledge, the concomitant occurrence of neurologic phenomena such as Horner syndrome combined with cutaneous complications in a patient receiving IANB anesthesia has never been discussed. CONCLUSIONS: In this exceptional case, the unusual Horner syndrome manifestations related to unilateral ptosis, miosis, and anisocoria were simultaneously developed with skin ischemia, paresthesia, and asymmetric flushing after the administration of IANB anesthesia.

Hypohidrosis induced by topiramate in an adult patient.

Hypohidrosis is an uncommon and reversible side effect of topiramate treatment, reported mainly in children. This report presents an adult patient with complex partial seizures who was treated with topiramate and developed hypohidrosis coupled with hyperthermia, related to high environmental temperature and physical exercise. Reduced sweat response was confirmed using the Neuropad test. Signs and symptoms ceased after drug discontinuation. During topiramate treatment, it is important to recognise this side effect, although the exact causal mechanism has not yet been clarified.

Headache in three new cases of Harlequin syndrome with accompanying pharmacological comparison with migraine.

BACKGROUND: Harlequin syndrome (HS) is a rare autonomic disorder characterised by unilateral diminished sweating and flushing of the face in response to heat or exercise. Some patients with HS complain of headache. METHODS: We present three new cases to characterise their headache phenotype and pharmacology and review the literature of cases where headache was described. RESULTS Two out of the three patients presented with episodes of unilateral headache associated with exercise: in one case the headache had migrainous features and was contralateral to the side where the flushing occurred, whereas the second patient, who had had migraine attacks in the past, had a brief throbbing headache, with no associated symptoms, ipsilateral to the facial flushing. The third woman had migraine but the attacks were not associated with HS. Pharmacological characterisation suggested the HS and migraine were biologically distinct. HS was not triggered by nitroglycerin and was unaffected by sumatriptan, dihydroergotamine and ergotamine. HS and migraine did not occur together. In the literature, we found six patients with both HS and headache, five of whom had migraine. CONCLUSIONS: These data do not show any correlation between the phenotypic expression of migraine and HS suggesting the syndromes are pathogenetically independent.

Comparison of the spread of three botulinum toxin type A preparations.

Botulinum toxins are frequently used in esthetics to improve the appearance of facial wrinkles. In this setting, precise localization of the neurotoxin is required to produce the desired clinical effects. Unwanted effects can occur if the neurotoxin diffuses into untargeted muscle. Therefore, a neurotoxin with low and predictable spread would be preferable for esthetic applications. The aim of this study was to investigate the spread of three approved botulinum toxin type A preparations, with and without complexing proteins, by measuring and comparing the size of the anhidrotic halos they produced following injection of equivalent doses in an identical volume into the forehead of patients. The results showed that incobotulinumtoxinA and onabotulinumtoxinA displayed comparable spread at 6 weeks (maximal area of anhidrosis within 6 weeks) and area under the effect curve (AUEC) over 6 months. However, abobotulinumtoxinA, when assuming a 1:2.5 injection volume ratio, produced a statistically significantly greater maximal area of anhidrosis within 6 weeks and AUEC over 6 months compared with incobotulinumtoxinA. All preparations were well tolerated. The results of this study demonstrate that incobotulinumtoxinA and onabotulinumtoxinA have comparable spread, while abobotulinumtoxinA has significantly greater spread than incobotulinumtoxinA.

Hypohidrosis and hyperthermia during topiramate treatment in children.

Topiramate is one of the newer generation antiepileptic drugs with a beneficial clinical effect on various seizure types. In this study, we present the clinical findings of hypohidrosis and hyperthermia with topiramate in pediatric patients. The data were collected retrospectively on 173 patients diagnosed as epilepsy on topiramate treatment, and hypohidrosis-related symptoms induced by topiramate were found in 22 patients. Their mean age was 64.45 +/- 56.63 months. The mean duration of topiramate treatment was 7.09 +/- 2.46 months, and the mean dose was 5.37 +/- 1.75 mg/kg/day. All of the patients complained of hypohidrosis and hyperthermia. Six (27.2%) of them had facial flushing, 4 (18.1%) had heat sensation and only 1 (4.5%) had lethargy. Hypohidrosis-related symptoms resolved after discontinuation of the medication. In conclusion, children treated with topiramate should be cautioned regarding these potential adverse effects and advised to avoid its use during the hot summer season.

Effect of prolonged administration of clenbuterol on airway reactivity and sweating in horses with inflammatory airway disease.

OBJECTIVE: To determine whether prolonged administration of clenbuterol results in tachyphylaxis, specifically regarding its bronchoprotective properties and effect on sweating in horses. ANIMALS: 8 Thoroughbreds with inflammatory airway disease. PROCEDURES: In a crossover design, horses received clenbuterol (0.8 µg/kg, p.o., q 12 h) or placebo for 21 days, with a washout period of ≥ 30 days between the 2 treatments. Airway reactivity was evaluated by use of flowmetric plethysmography and histamine broncho-provocation before (day 0; baseline) and every 7 days after the start of treatment. Sweat function was evaluated via response to epinephrine administered ID before and every 10 days after the start of treatment. RESULTS: The concentration of histamine required to increase total airway obstruction by 35% (PC35) was significantly reduced during treatment with clenbuterol (mean change, 11.5 mg/mL), compared with during administration of the placebo (mean change, -1.56 mg/mL), with a peak effect at 14 days. Tachyphylaxis was evident by day 21, with 7 of 8 horses having a PC35 below the baseline value (mean change, -0.48 mg/mL), which returned to baseline values during the washout period. No effect of clenbuterol was seen in sweat response to epinephrine administration. CONCLUSIONS AND CLINICAL RELEVANCE: Clenbuterol initially reduced airway sensitivity to inhaled histamine, but tachyphylaxis that resulted in increased airway reactivity was evident by day 21. Although no effects on sweating were detected, the technique may not have been sensitive enough to identify subtle changes. Prolonged administration of clenbuterol likely results in a clinically important reduction in its bronchodilatory effects.

Hipertermia asociada a anhidrosis en el tratamiento del trastorno de control de impulsos con topiramato / Hyperthermia-associated anhydrosis in the treatment of impulse control disorder with topiramate

El topiramato es un fármaco antiepiléptico utilizado en el tratamiento de algunos trastornos psiquiátricos. Dentro de su perfil de efectos secundarios se encuentra la posibilidad de producir anhidrosis que en algunas ocasiones puede asociarse a cuadros de hipertermia. En este artículo se presenta el caso de un niño de 11 años diagnosticado de un trastorno del control de impulsos. Durante el tratamiento con topiramato presentó un cuadro de hipertermia asociado a anhidrosis coincidiendo con los meses más calurosos en la ciudad en la que residía, y que requirió la suspensión del fármaco para el control del cuadro sintomático

Topiramate is an anti-epileptic drug used to treat some psychiatric disorders. Among its possible side effects is anhidrosis, which in some cases is linked to cases of hyperthermia. This paper reports the case of a 11 year-old boy diagnosed with a impulse control disorder. During the course of treatment with topiramate the boy presented symptoms of hyperthermia associated with anhidrosis, coinciding with the hottest months of the year in his home city, requiring the suspension of the drug in order to bring the symptoms under control