Bier anemic spots, cyanosis, and urticaria-like eruption (BASCULE) syndrome in a 15-year-old male, responsive to high-dose fexofenadine.

We describe an unusual presentation of Bier anemic spots, cyanosis, and urticaria-like eruption (BASCULE) syndrome in a 15-year-old male, recalcitrant to low-dose anti-histamines, which subsequently responded to high-dose fexofenadine.

[A Case of Methemoglobinemia Caused by Toluidine Revelation with Dyspnea and Cyanosis].

Toluidine is a known cause of bladder cancer, but it is less widely recognized as a cause of methemoglobinemia because methemoglobinemia is rare. We herein report a case of methemoglobinemia caused by toluidine in a 50-year-old man. A solution of toluidine overflowed from its container during transportation and adhered to the man's clothes, but he drove to his workplace 100 km away without changing his clothes or undergoing decontamination. Before arriving at his workplace, he developed dyspnea and called emergency services, and he was then transported to a local hospital. He had significant cyanosis upon arrival, and arterial blood gas analysis revealed a high methemoglobin level of 44%. He was diagnosed with toluidine-induced methemoglobinemia and was transported to our hospital, where he was admitted to the intensive care unit. Treatment for methemoglobinemia was started immediately after hospitalization, and the patient's symptoms and methemoglobin level improved. Methemoglobinemia should be considered in workers who handle toluidine and develop cyanosis and dyspnea.

Use of terlipressin in critically ill children with liver disease.

BACKGROUND: Terlipressin, a long-acting synthetic analogue of vasopressin has been used in the adult population for various indications including hepatorenal syndrome (HRS-AKI), esophageal variceal hemorrhage (EVH) and shock, but its use in pediatrics is still limited to individualized cases and data on safety and efficacy is scant. METHODS: We reviewed the patient records of children with liver disease and Acute Kidney Injury requiring terlipressin admitted to the Paediatric Intensive Care Unit (PICU) of King's College Hospital, London from January 2010-December 2017, with special emphasis on its effect on renal parameters and adverse event profile. RESULTS: Twenty-one terlipressin administration records in a total of 16 patients (median) (IQR) 10 years (6.1-14.4) were included. The drug was initially given as a bolus dose in all cases, followed by either bolus or infusion with median dosage being 5.2 (3.8-6.7) mcg/kg/hour. After administration, a sustained increase of mean arterial pressure was observed. There was an improvement in serum creatinine (Cr) (at 24 h; p = 0.386) and increase in urine output (UO), especially in the hepatorenal syndrome subgroup (HRS-AKI). We found minimal evidence of gastrointestinal side effects including feeding intolerance and vasoconstrictive side effects including cyanosis / ischaemia of extremities. CONCLUSION: Terlipressin was found to be safe in critically sick children with liver disease with positive impact on renal parameters which might be taken as a surrogate marker of HRS reversal, though effects on outcomes are difficult to ascertain. It is important to be aware of all its side-effects and actively watch for them. Future prospective studies are warranted to validate these findings.

An Uncommon Complication With Use of Topical Local Anesthetic Agents: Methemoglobinemia.

Although the use of topical local anesthetics is generally safe, several potentially fatal complications have been reported. Methemoglobinemia is a rare but potentially fatal complication. Methemoglobin is a naturally occurring oxidized metabolite of hemoglobin, and physiologic levels (<1%) are normal. Methemoglobinemia can be congenital or acquired. Several drugs including topical anesthetic agents like benzocaine can induce this condition. Sudden appearance of cyanosis, with a disproportionately better oxygen saturation of 85% after use of local anesthetics can be a helpful for diagnosis.

Purple glove syndrome occurring after oral administration of phenytoin in therapeutic doses: mechanism still a dilemma.

Purple glove syndrome is a rare and poorly understood complication of phenytoin use, occurring almost always with its intravenous formulation. This syndrome comprises of pain, purple discoloration, and edema distal to the site of intravenous administration of phenytoin. We hereby report an unusual case, wherein purple glove syndrome was seen on oral formulation of phenytoin in its therapeutic dose.

Central cyanosis on a psychiatric unit treated at the Salford Royal Hospital.

We describe a case of acquired methaemoglobinaemia due to frequent use of the 'legal high' known as 'Pink Panthers'. This contains 5,6-Methylenedioxy-2-aminoindane and 2-Aminoindane, both amphetamine analogues with the potential to cause methaemoglobinaemia. Furthermore, the most common 'cutting agent' for legal highs in the UK is benzocaine, also known to cause methaemoglobinaemia. Given the increasing prevalence of legal highs, particularly those containing added benzocaine, such presentations may become more common. Furthermore, in one case series, benzocaine gel used for toothache was the second most common reason for hospitalisation due to acquired methaemoglobinaemia after dapsone use. Indeed, the Federal Drug Agency has issued as public warning as to the risk of these products. We therefore think that clinicians and the public should be made more aware of the risk associated with such agents.

Rapid-onset methemoglobinemia from traditional-medicine-induced potassium nitrate poisoning: successful treatment with methylene blue-a case report.

BACKGROUND: Potassium nitrate poisoning is a rare but potentially serious condition that can result in methemoglobinemia and subsequent cyanosis. This case report presents a unique instance of rapid-onset methemoglobinemia resulting from the ingestion of a traditional medicine preparation containing potassium nitrate, known as "kalmi shora." CASE PRESENTATION: A 14-year-old Pakistani boy reported to the emergency department with a history of sudden-onset headache, drowsiness agitation, irritability, and generalized cyanosis. Pulse oximetry showed a concerning oxygen saturation level of 58%, whereas arterial blood gas analysis revealed a normal partial pressure of oxygen (90 mmHg). The profile of abrupt onset of symptoms, generalized cyanosis, and the discrepancy between the partial pressure of oxygen and oxygen saturation readings necessitated a comprehensive assessment including inquiries into potential toxins. The peculiar appearance of the blood, resembling chocolate in color, further indicated the possibility of methemoglobinemia. The patient was successfully treated with methylene blue, leading to a prompt resolution of symptoms. CONCLUSION: This case highlights the significance of considering toxin exposures, such as traditional-medicine-induced poisoning, in emergency settings. The report contributes to the medical literature by highlighting the potential risks associated with traditional remedies and emphasizes the critical role of prompt diagnosis and intervention in optimizing patient outcomes. Recognition of the specific etiology of methemoglobinemia, in this case, traditional medicine ingestion, is essential for effective management in emergency medicine.

Management of isopropyl nitrate-induced methaemoglobinaemia in pregnancy.

A pregnant female in her early 30s presented with cyanosis and oxygen saturation of 78%. She ingested isopropyl nitrate mistaking it for cannabidiol. Her arterial blood gas showed a methaemoglobin of >30% (outside the measuring range). She was treated with 120 mg of methylthioninium chloride (2 mg/kg) and symptoms improved. Her pregnancy progressed but was induced at 36 weeks because her child was small for gestational age. Methaemoglobinaemia is a rare presentation in pregnancy. There have been no reported cases of isopropyl nitrate-induced methaemoglobinaemia in pregnancy. Historically, intra-amniotic methylthioninium chloride was used in amniocentesis but use stopped after links to fetal malformations and neonatal death were made. There is no evidence outlining the risks of isopropyl nitrate in pregnancy and limited data on fetal effects from maternal exposure to intravenous methylthioninium chloride. This case adds to the evidence that treating methaemoglobinaemia may outweigh the risks of maternal exposure to methylthioninium chloride.

Acquired methemoglobinemia induced by indoxacarb poisoning: a case report.

Indoxacarb, a large-spectrum non-organophosphorus oxadiazine insecticide, is broadly used in farming whose mechanism of action is the blockage of voltage-gated sodium channels of insects. There is restricted data on human poisoning. We report a case of an 18-year-old male patient without comorbidities presented with unconsciousness and cyanosis after the intentional ingestion of indoxacarb in a suicide attempt. Methemoglobinemia was clinically suspected and was successfully treated after methylene blue injection, associated with supportive and symptomatic management. This case emphasizes the importance of considering methemoglobinemia after indoxacarb ingestion in addition to its early recognition and timely injection of methylene blue which led to complete recovery without sequelae.

An unusual complication of cocaine toxicity.

A 25-year man presented to the Accident and Emergency Department complaining of dizziness and shortness of breath after taking 70 grams of cocaine over 10 hours. He said a friend had noticed that his skin had turned dark blue. On examination the patient was severely centrally and peripherally cyanosed. His pulse oximeter oxygen saturations were 88% on air. An arterial blood gas showed a methaemoglobin level of 45.6%. The patient was diagnosed with cocaine-induced methaemoglobinaemia and given methyl thioninium chloride (methylene blue). He made an uneventful recovery.

Syncope and Methemoglobinemia Preceded by Amyl Nitrite 'Popper' Inhalation.

INTRODUCTION: Methemoglobinemia represents an uncommon but potentially serious cause of presentation to the emergency department, resulting in hypoxemia and even death. The symptoms and clinical findings in this condition can be nonspecific and therefore methemoglobinemia can be easily missed if the clinician is not familiar with it. This report presents a case caused by recreational drug use which has rarely been documented previously. CASE REPORT: A 23-year-old male with a history of asthma presents to the emergency department for an episode of syncope after inhalation of amyl nitrite "poppers". He had normal vitals other than tachycardia but was found to have nailbed and perioral cyanosis, a classic but uncommon presentation that is demonstrated in the included clinical image. He was found to have methemoglobinemia caused by his use of amyl nitrite and received supportive care but did not require methylene blue. CONCLUSION: Emergency physicians should familiarize themselves with the classic physical exam findings in methemoglobinemia in order to identify and treat this condition promptly. While this patient had a good outcome with only supportive care and observation, his presentation and the etiology of his condition offer an important teaching point. The possibility of methemoglobinemia after recreational "popper" use should be considered when working up a patient who presents with cyanosis and hypoxemia.

Comparison of the repeated dose toxicity of isomers of dinitrotoluene.

Dinitrotoluene (DNT) is a nitroaromatic explosive used in propellant mixtures and in the production of plastics. Isomers of DNT were administered daily via oral gavage to male Sprague-Dawley rats for 14 days to determine the subacute toxicity of individual isomers of DNT. The 3,5-DNT isomer was the most toxic isomer, inducing weight loss and mortality within 3 days. Cyanosis and anemia were observed for all isomers. Exposure to 2,4-, 2,6-, and 3,5-DNT resulted in decreased testes mass and degenerative histopathological changes. Increased splenic mass was observed for 2,4-, 2,6-, and 2,5-DNT. Extramedullary hematopoiesis of the spleen was noted for all isomers, while lymphoid hyperplasia of the spleen was noted for all isomers except 2,5-DNT. Increased liver mass was observed for 2,3-DNT and 3,4-DNT. Hepatocellular lesions were observed for 2,6-DNT and 2,4-DNT. Neurotoxic effects were noted for 3,4-DNT, 2,4-DNT, and 3,5-DNT.

Two cases of fatal methemoglobinemia caused by self-poisoning with sodium nitrite: A case report.

RATIONALE: Sodium nitrite intoxication reportedly causes severe methemoglobinemia. Recent studies reported that most clinically significant cases resulted from intentional exposure in suicidal attempts. We describe 2 cases of severe methemoglobinemia secondary to intentional sodium nitrite intoxication in suicidal attempts. PATIENTS CONCERNS: A 26-year-old man and 20-year-old woman attempted suicide by taking sodium nitrite, and were brought to the emergency department. DIAGNOSIS: The male patient collapsed at the scene. He ingested approximately 18 g of sodium nitrate, and his methemoglobin level was 90.3%. The female patient was conscious, but was cyanotic. She ingested approximately 12.5 g of sodium nitrite, and her methemoglobin level was 54.6%. INTERVENTIONS: The male patient received advanced cardiac life support in the emergency department. Methylene blue was immediately administered for the female patient. OUTCOMES: The male patient died despite aggressive resuscitation. The female patient's cyanosis resolved, and her methemoglobin level decreased to 1.2% 3 hours later. LESSONS: The immediate administration of methylene blue in severe methemoglobinemia patients prevented fatal consequences. The public should be informed about the accessibility and toxicity of sodium nitrite.

Methemoglobinemia due to use of poppers: a case report.

BACKGROUND: Methemoglobinemia is an excess of oxidized hemoglobin in the blood, affecting oxygen transportation. It is characterized by central cyanosis that does not respond to oxygen therapy. Prognosis is excellent when treated adequately and rapidly. We present a case report of a 38-year-old Caucasian man suffering from methemoglobinemia due to the use of poppers. CASE PRESENTATION: A 38-year-old Caucasian man known as a smoker and addicted to cocaine was admitted to the emergency department with dyspnea, agitation, and central cyanosis that started approximately 3 hours before admission. The persistent hypoxia despite high-flow oxygen therapy and a history of poppers use helped to reveal a condition known as methemoglobinemia. CONCLUSIONS: Our case highlighted a typical clinical presentation of methemoglobinemia. This possible life-threatening condition can occur after ingestion or inhalation of poppers, commonly sold in sex shops for recreational purposes. This can be easily confirmed by the methemoglobin level of the blood gases, provided the emergency physician considers this diagnosis. Rapid treatment with intravenous methylene blue is effective and leads to a favorable prognosis.

[Unexplained cyanosis in two newborns: neonatal methemoglobinemia after maternal perineal infiltration of prilocaine]. / Onbegrepen cyanose bij twee neonaten.

BACKGROUND: Methemoglobinemia is a rare cause of neonatal cyanosis in the newborn. It is considered a medical emergency. Failure of recognition or appropriated treatment could result in serious disease and neonatal death. Neonatal methemoglobinemia can be caused by both hereditary and acquired factors. CASE DESCRIPTION: We present two cases of newborns who developed severe cyanosis a few hours after birth due to methemoglobinemia. This was thought to be related to the local maternal perineal infiltration of prilocaine during childbirth. Though rare, prilocaine is the most potent agent to induce methemoglobinemia compared to other local aneasthetics. After intravenous administration of methylene blue, both newborns fully recovered. CONCLUSION: Neonatal methemoglobinemia is a rare and potentially fatal complication of local anesthetics, particularly prilocaine, administered to the mother during childbirth. Midwives, obstetricians, gynecologists and pediatricians should be aware of this complication. The use of other local anesthetics, including lidocaine, should be considered.

A Case of Dapsone-induced Mild Methemoglobinemia with Dyspnea and Cyanosis.

Dear Editor, Dapsone is a dual-function drug with antimicrobial and antiprotozoal effects and anti-inflammatory features (1). In dermatology, it is a first choice for conditions such as leprosy, IgA pemphigus, dermatitis herpetiformis, and linear IgA bullous dermatosis, or an adjunctive treatment for, e.g. bullous pemphigoid (BP) and pemphigus vulgaris (1). However, dapsone is associated with some adverse effects, including methemoglobinemia (1). Methemoglobin (MetHb) concentrations of less than 15% usually cause no symptoms in patients with normal hemoglobin concentrations (2). Herein, we report the case of a patient with BP who developed dyspnea because of dapsone-induced methemoglobinemia that was as mild as 4.7%. A 93-year-old man was diagnosed with BP based on skin manifestations (Figure 1, a and b), histopathological findings (Figure 1, c and d), and anti-BP180 NC16A antibody titer determined by chemiluminescence enzyme immunoassay (279 U/mL) 3 years earlier. His comorbidities included diabetes mellitus, chronic heart failure, right pleural effusion, and brain infarction. The patient had been successfully treated with oral prednisolone, so the steroid was tapered to 4 mg/day. The blisters recurred, however, and new ones kept developing even though the prednisolone was increased to 25 mg/day. Dapsone (75 mg/day) was begun as adjunctive treatment, and new blister formation ceased. At one week from dapsone initiation, the patient developed dyspnea, and his oxygen saturation as measured by pulse oximetry decreased to 88% on room air. At presentation, his blood pressure was 118/78 mmHg, the heart rate was 95 beats/minute, and axillary temperature was 36.3 °C. Neurological examination and consciousness findings remained unchanged compared with findings before dyspnea onset. Chest examination showed normal breath and heart sounds, but lip and peripheral cyanosis was present. Blood tests revealed a white blood cell count of 12,920/µl; red blood cells, 370×104/µl; hemoglobin, 11.7 g/dl; and CMV antigenemia (or C7-HRP), negative. Chest CT and echocardiography indicated no remarkable change compared with imaging from one year earlier. Arterial blood gas analysis showed a pH of 7.454, PaO2 63.1 mmHg, PaCO2 35.4 mmHg, HCO3- 24.3 mmol/L, SaO2 92.4%, and MetHb of 4.7%. These findings indicated a saturation gap (difference between SpO2 and SaO2) induced by MetHb. Upon cessation of dapsone, MetHb levels and SpO2 returned to normal and the dyspnea resolved, implicating dapsone in the methemoglobinemia (Figure 1, e). Differential diagnoses were pulmonary disease, heart disease, neuromuscular disease, sepsis, and drug intoxication. These possibilities were ruled out by the physical examination, drug history, vital signs, blood tests, and chest CT and echocardiography. In normal individuals, MetHb levels are less than 1% (2). Healthy patients with normal hemoglobin concentrations develop cyanosis at MetHb level of 15-20%, dyspnea at 20-50%, and coma at 50-70%, and die at more than 70% (2). However, patients with hematologic disease, acidosis, or cardiopulmonary diseases, for example, present with symptoms even with MetHb levels less than 15% (2,3). We inferred that our patient presented with dyspnea even under mild methemoglobinemia because he had anemia, chronic heart failure, and right pleural effusion. The occurrence of dapsone-induced methemoglobinemia with obvious symptoms is rare (1,4). Clinicians should be aware that methemoglobinemia symptoms are influenced not only by MetHb concentrations but also by comorbidities.

[Methemoglobin intoxication by prilocaine in EMLA. Accidental intoxication of an infant with scald injuries]. / Methämoglobinintoxikation durch Prilocain in EMLA. Akzidentelle Intoxikation bei einem Kleinkind mit Verbrühungen.

The case of an infant who had received EMLA(R) for local pain therapy after scalding to 5% of the body surface with boiling water is reported. Due to the application of EMLA(R) on the injured skin and exceeding the recommended doses of prilocaine and lidocaine the child developed symptomatic methemoglobinemia. During surgical wound dressing the boy showed cyanosis, decreased peripheral oxygen saturation and potentially suffered a general seizure. With a symptomatic therapy including mechanical ventilation and anticonvulsive drugs the methemoglobinemia normalized within 9 h. The child recovered without any neurological impairment after wound treatment was completed.