Adult ; Dapsone ; adverse effects ; Drug Eruptions ; Humans ; Male ; Syndrome

Humans ; Dapsone/adverse effects* ; Drug Hypersensitivity/etiology* ; Hypersensitivity ; Syndrome

Adult ; Dapsone ; adverse effects ; Humans ; Infectious Mononucleosis ; chemically induced ; Male ; Pemphigus ; drug therapy ; Syndrome

Methemoglobin (MetHb) is an oxidation product of hemoglobin in which the sixth coordination position of ferric iron is bound to a water molecule or to a hydroxyl group. The most common cause of acquired MetHb-emia is accidental poisoning which usually is the result of ingestion of water containing nitrates or food containing nitrite, and sometimes the inhalation or ingestion of butyl or amyl nitrite used as an aphrodisiac. We herein report a case of MetHb-emia after ingestion of an aphrodisiac, later identified as dapsone by gas chromatograph/mass selective detector (GC/MSD). A 24-year old male was admitted due to cyanosis after ingestion of a drug purchased as an aphrodisiac. On arterial blood gas analysis, pH was 7.32, PaCO2 26.8 mmHg, PaO2 75.6 mmHg, and bicarbonate 13.9 mmol/L. Initial pulse oxymetry was 89%. With 3 liter of nasal oxygen supplement, oxygen saturation was increased to 90-92%, but cyanosis did not disappear. Despite continuous supplement of oxygen, cyanosis was not improved. On the fifth hospital day, MetHb was 24.9%. Methylene blue was administered (2 mg/kg intravenously) and the patient rapidly improved. We proved the composition of aphrodisiac as dapsone by the method of GC/MSD.
Administration, Oral ; Adult ; Antidotes/therapeutic use ; Aphrodisiacs/adverse effects* ; Case Report ; Cyanosis/drug therapy ; Cyanosis/chemically induced ; Cyanosis/blood ; Dapsone/adverse effects* ; Human ; Male ; Methemoglobinemia/drug therapy ; Methemoglobinemia/chemically induced* ; Methylene Blue/therapeutic use

Drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome is a life-threatening adverse drug reaction with systemic manifestations. Dapsone is known to be useful for treatment of leprosy and various dermatologic conditions. We report a patient with prurigo pigmentosa who developed DRESS syndrome after dapsone treatment. She presented with lymphadenopathy, fever, eosinophilia, skin rash, and elevated liver enzymes. Initial lymph node and skin biopsy was suggestive of peripheral T-cell lymphoma. Initially, she was treated with chemotherapy. A week later after complete remission of skin symptoms, new skin lesions recurred. TCR-gene rearrangement was examined to show negative results and she was diagnosed as dapsone induced DRESS syndrome. This case emphasizes the importance of differential diagnosis of lymphoma and DRESS syndrome.
Biopsy ; Dapsone ; Diagnosis, Differential ; Drug Hypersensitivity ; Drug Hypersensitivity Syndrome* ; Drug Therapy ; Drug-Related Side Effects and Adverse Reactions ; Eosinophilia ; Exanthema ; Fever ; Humans ; Leprosy ; Liver ; Lymph Nodes ; Lymphatic Diseases ; Lymphoma ; Lymphoma, T-Cell, Peripheral ; Prurigo ; Pseudolymphoma ; Skin

Adverse drug reactions can cause considerable discomfort. They can be life-threatening in severe cases, requiring or prolonging hospitalization, impeding proper treatment, and increasing treatment costs considerably. Although the incidence of severe cutaneous adverse reactions (SCARs) is low, they can be serious, have permanent sequelae, or lead to death. A recent pharmacogenomic study confirmed that genetic factors can predispose an individual to SCARs. Genetic markers enable not only elucidation of the pathogenesis of SCARs, but also screening of susceptible subjects. The human leukocyte antigen (HLA) genotypes associated with SCARs include HLA-B*57:01 for abacavir (Caucasians), HLA-B*58:01 for allopurinol (Asians), HLA-B*15:02 (Han Chinese) and HLA-A*31:01 (Europeans and Koreans) for carbamazepine, HLA-B*59:01 for methazolamide (Koreans and Japanese), and HLA-B*13:01 for dapsone (Asians). Therefore, prescreening genetic testing could prevent severe drug hypersensitivity reactions. Large-scale epidemiologic studies are required to demonstrate the usefulness and cost-effectiveness of screening tests because their efficacy is affected by the genetic differences among ethnicities.
Allopurinol ; Carbamazepine ; Cicatrix ; Dapsone ; Drug Hypersensitivity ; Drug Hypersensitivity Syndrome ; Drug-Related Side Effects and Adverse Reactions ; Epidemiologic Studies ; Genetic Markers* ; Genetic Testing ; Genotype ; Health Care Costs ; HLA Antigens ; Hospitalization ; Humans ; Incidence ; Leukocytes ; Mass Screening ; Methazolamide ; Pharmacogenetics ; Stevens-Johnson Syndrome

INTRODUCTIONA 22-year-old Malay soldier developed dapsone hypersensitivity syndrome 12 weeks after taking maloprim (dapsone 100 mg/pyrimethamine 12.5 mg) for anti-malarial prophylaxis.

CLINICAL PICTUREHe presented with fever, rash, lymphadenopathy and multiple-organ involvement including serositis, hepatitis and thyroiditis. Subsequently, he developed congestive heart failure with a reduction in ejection fraction on echocardiogram, and serum cardiac enzyme elevation consistent with a hypersensitivity myocarditis.

TREATMENTMaloprim was discontinued and he was treated with steroids, diuretics and an angiotensin-converting-enzyme inhibitor.

OUTCOMEHe has made a complete recovery with resolution of thyroiditis and a return to normal ejection fraction 10 months after admission.

CONCLUSIONIn summary, we report a case of dapsone hypersensitivity syndrome with classical symptoms of fever, rash and multi-organ involvement including a rare manifestation of myocarditis. To our knowledge, this is the first case of dapsone-related hypersensitivity myocarditis not diagnosed in a post-mortem setting. As maloprim is widely used for malaria prophylaxis, clinicians need to be aware of this unusual but potentially serious association.

Abdominal Pain ; drug therapy ; Adult ; Anti-Inflammatory Agents, Non-Steroidal ; adverse effects ; therapeutic use ; Biopsy ; Dapsone ; adverse effects ; therapeutic use ; Diagnosis, Differential ; Drug Hypersensitivity ; complications ; pathology ; Echocardiography ; Electrocardiography, Ambulatory ; Fever ; drug therapy ; Follow-Up Studies ; Humans ; Male ; Myocarditis ; diagnosis ; etiology ; Radiography, Thoracic ; Skin ; pathology ; Thyrotoxicosis ; diagnosis ; etiology