UNASSIGNED: Stevens-Johnson syndrome (SJS) is a rare and unusual hypersensitivity reaction to certain drugs like allopurinol, commonly used for treating gout. SJS is recognized by extensive necrosis and detachment of skin and mucus membranes. Pancytopenia, characterized by decreased levels of red blood cells, white blood cells and platelets, is an exceedingly rare occurrence in the rare disorder SJS.
UNASSIGNED: The authors present a 61-year-old male who exhibited symptoms of fever and rash for 5 days accompanied by pancytopenia and liver injury.
UNASSIGNED: The abdomen and bilateral lower extremities exhibited several well-defined dusky-colored hyperpigmented macular lesions. Initially, these lesions were small, tender, erythematous, and raised, later transitioning to a dark red. Multiple distinct ulcerations were present on the lips and buccal cavity. Additionally, there was denudation of the skin with bleeding observed between the toes of both legs. The causality was assessed as a definite adverse drug reaction according to the Naranjo and ALDEN algorithm. The patient received treatment consisting of intravenous steroid along with prophylactics antibiotics. The individual\'s pancytopenia was resolved without requiring any blood cells or plasma or platelet concentrate transfusion.
UNASSIGNED: The exact pathophysiology of SJS associated with pancytopenia has not yet been fully elucidated. The authors\' study hypothesized that the cause of pancytopenia in SJS could be either the direct cytotoxicity of drugs or immune-mediated damage to the bone marrow cells. Additional studies are necessary to establish the precise pathophysiology of the condition. Moreover, our study also indicates that pancytopenia can resolve in SJS without the need for blood cells or plasma or platelet concentrate transfusion. Once more, further studies are required to establish precise management strategies for managing SJS associated with pancytopenia.

Canine leishmaniosis is a vector-borne disease caused by Leishmania infantum, and clinical manifestations of infection range from absent or severe to fatal and result from immune-mediated mechanisms. In dogs, the most common clinical signs of leishmaniosis include skin lesions and lymphadenomegaly. However, the presence of other nontypical signs has been described, and diagnosing these cases can be challenging. The aim of the present short communication was to describe the impact of the formation of circulating immunocomplexes due to L. infantum in a dog with leishmaniosis affected by a massive venous thrombus of the caudal vena cava and external iliac veins. On admission, the dog presented bilateral cutaneous vasculopathy of the thigh and renal disease due to L. infantum infection. Two weeks after starting anti-Leishmania treatment based on meglumine antimoniate and allopurinol administration, the animal developed acute claudication of the hind limbs with the presence of a thrombus in the caudal vena cava and the external iliac veins and a high level of circulating immunocomplexes detected by enzyme-linked immunosorbent assay. Exacerbation of the humoral immune response, along with deposition of circulating immune complexes in the tissues and the concurrent presence of kidney and liver damage, might have contributed to an imbalance in haemostasis in this patient. Future studies should evaluate and analyse the pathological mechanisms contributing to thrombosis in dogs with leishmaniosis.

Allopurinol-induced drug reaction syndrome with eosinophilia and systemic symptoms (A-DRESS) is a well-described condition in adults, whereas it is uncommon among children. We describe a case of A-DRESS in a 16-year-old male with steroid-dependent nephrotic syndrome. He presented a life-threatening clinical course with persisting fever, skin rash, eosinophilia, lymphadenopathy, distributive shock, and herpesvirus 6 detection. The withdrawal of allopurinol and a combination of intravenous immunoglobulins (IVIGs) and systemic corticosteroids led to the patient\'s recovery without sequelae. Drug reaction with eosinophilia and systemic symptoms (DRESS) in pediatrics is rare and can present in a severe form. Early diagnosis and timely treatment are critical for prognostic purposes. This report suggests the potentially crucial role of IVIG in the treatment of patients with A-DRESS.

Drug-induced hypersensitivity syndrome (DIHS) is a severe type of cutaneous adverse event involving systemic organ failures. In some cases of DIHS, acute renal failure takes place, and it becomes necessary to perform hemodialysis. However, the clinical outcome of renal failure in the course of treatment of DIHS remains unclear. Herein, we report a case of DIHS complicated with acute renal failure, which requires hemodialysis. Furthermore, we also review the DIHS cases accompanied by acute renal failure with hemodialysis in the English case report literature.

Drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome is a drug reaction commonly related to eosinophilia, from uncertain epidemiology, and without consensus for diagnosis and treatment globally. It presents a great challenge in its management and is characterized by fever, lymphadenopathy, skin rash, and multisystemic involvement. An aggressive and difficult-to-manage clinical case is presented in a 50-year-old man with chronic kidney disease due to diabetes mellitus type 2 and systemic arterial hypertension, who developed an unusual variant similar to DRESS and Stevens-Johnson syndrome (SJS) overlap secondary to allopurinol, with skin manifestations without eosinophilia, but fulfilling clinical and laboratory criteria for DRESS and SJS syndrome.

A 9-year-old domestic cat, positive for antibodies to feline immunodeficiency virus (FIV), was brought to a veterinary clinic with alopecia, ulcerative skin lesions, and upper respiratory tract (URT) signs. This was after being treated for suspected allergic dermatitis, without clinical improvement, for 2 y. Biopsy of the skin and fine-needle aspirates of the spleen and of the lymph nodes were taken which detected the presence of Leishmania amastigotes. Leishmania infection was further confirmed by detection of a high titer of anti-Leishmania antibodies (≥ 3200) with an indirect fluorescent antibody technique (IFAT) serology. After the diagnosis of feline leishmaniosis (FeL) was made, allopurinol and meglumine antimoniate were started and led to quick and complete clinical improvement. After 7 mo, allopurinol administration was briefly interrupted but was resumed following relapse of the skin lesions. One month later, the cat was treated for suspected acute kidney injury, which prompted reduction of the total daily dose of allopurinol by 50%. The cat remained clinically well, with complete resolution of the cutaneous and URT signs, for nearly 24 mo after the diagnosis of FeL; at which point it was euthanized for worsening cardiac disease. To our knowledge, this represents a rare case of successful treatment of FeL with a suspected nephrotoxic effect associated with long-term use of allopurinol. Further studies are required to clarify the relationship, if any, between leishmaniosis and congestive heart failure in cats.
Suivi à long terme d’un cas de leishmaniose féline traité par une association d’allopurinol et d’antimoniate de méglumine. Un chat domestique de 9 ans, positif pour les anticorps contre le virus de l’immunodéficience féline (FIV), a été présenté dans une clinique vétérinaire avec une alopécie, des lésions cutanées ulcéreuses et des signes des voies respiratoires supérieures (URT). Ceci après avoir été traité pour une suspicion de dermatite allergique sans amélioration clinique, pendant 2 ans. Une biopsie de la peau et des ponctions à l’aiguille fine de la rate et des ganglions lymphatiques ont été réalisées et ont détecté la présence d’amastigotes de Leishmania. L’infection à Leishmania a été confirmée par la détection d’un titre élevé d’anticorps sériques anti-Leishmania (≥ 3200) par une technique d’immunofluorescence indirecte (IFAT). Après le diagnostic de leishmaniose féline (FeL), un traitement à l’allopurinol et l’antimoniate de méglumine a été instauré et a entraîné une amélioration clinique rapide et complète. Après 7 mois, l’administration d’allopurinol a été brièvement interrompue mais a été reprise après la rechute des lésions cutanées. Un mois plus tard, le chat a été traité pour une lésion rénale aiguë suspectée, ce qui a entraîné une réduction de 50 % de la dose quotidienne totale d’allopurinol. Le chat est resté cliniquement en bonne santé, avec une résolution complète des signes cutanés et urinaires, pendant près de 24 mois après le diagnostic de FeL; à quel point il a été euthanasié pour aggravation de la maladie cardiaque.À notre connaissance, ceci représente un cas rare de traitement réussi de FeL avec un effet néphrotoxique suspecté associé à une utilisation à long terme d’allopurinol. D’autres études sont nécessaires pour clarifier la relation, le cas échéant, entre la leishmaniose et l’insuffisance cardiaque congestive chez les chats.(Traduit par Dr Serge Messier).

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BACKGROUND: Drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome is an uncommon yet serious adverse drug hypersensitivity reaction with the presentations including rash, fever, lymphadenopathy, and internal organ involvement. Sarcoidosis is a systematic granulomatous disease with unknown etiology. We herein report a case of pulmonary sarcoidosis secondary to allopurinol-induced DRESS.
METHODS: A 37-year-old man with a history of hyperuricemia was treated with allopurinol for three weeks at a total dose of 7000 milligrams before developing symptoms including anorexia, fever, erythematous rash, and elevated transaminase. The patient was diagnosed with DRESS and was treated with prednisone for 6 mo until all the symptoms completely resolved. Three months later, the patient presented again because of a progressively worsening dry cough. His chest computed tomography images showed bilateral lung parenchyma involvement with lymph node enlargement, which was confirmed to be nonnecrotizing granuloma by pathological examination. Based on radiologic and pathological findings, he was diagnosed with sarcoidosis and was restarted on treatment with prednisone, which was continued for another 6 mo. Reexamination of chest imaging revealed complete resolution of parenchymal lung lesions and a significant reduction in the size of the mediastinal and hilar lymph nodes. Following a 6-month follow-up of completion of treatment, the patient\'s clinical condition remained stable with no clinical evidence of relapse.
CONCLUSIONS: This is the first case in which pulmonary sarcoidosis developed as a late complication of allopurinol-induced DRESS. The case indicated that the autoimmune reaction of DRESS may play an important role in the pathogenesis of sarcoidosis.

Chronic diarrhea is a clinical sign associated with canine leishmaniosis, varying from 3 % to 30 % of prevalence. However, its occurrence in dogs has been mostly associated with chronic kidney or liver disease. Leishmania organisms can cause inflammation of the digestive tract with chronic diarrhea as the only clinical manifestation, although it has been poorly documented in dogs. The aim of this retrospective observational study was to describe dogs with chronic diarrhea as the main clinical sign associated with leishmaniosis. All cases had a complete blood count, biochemistry, urinalyses, and diagnostic tests for leishmaniosis. Exclusion criteria included renal or hepatic disease and/or previous diagnosis of gastrointestinal disease. Twenty-three dogs were included. Small bowel diarrhea was present in 7/23 (30.4 %), large bowel diarrhea in 9/23 (39.2 %) and mixed diarrhea in 7/23 (30.4 %). Gastrointestinal biopsies were performed in 8/23 dogs and Leishmania amastigotes were found in all of them. In the others, leishmaniosis was diagnosed by serology in 10/15 dogs (66.7 %), serology plus blood PCR in 3/15 (20.0 %), lymph node cytology in 1/15 (6.7 %), and blood PCR in 1/15 (6.7 %). All dogs treated had a complete resolution of diarrhea with specific treatment for leishmaniosis alone, based on meglumine antimoniate (75-100 mg/kg SID SC for 1 month) plus allopurinol (10 mg/kg BID PO ≥ 6 months). This study suggests that leishmaniosis should be also included in the differential diagnosis of dogs from endemic areas presenting with the primary problem of large-bowel, small-bowel, or mixed-bowel chronic diarrhea.

UNASSIGNED: HLA-B*58:01 is a well-known risk factor for allopurinol-induced severe cutaneous adverse reactions (SCARs). However, only a minority of HLA-B*58:01 carriers suffer SCARs after taking allopurinol. The aim of this study was to investigate subsidiary genetic markers that could identify those at further increased risk of developing allopurinol-induced drug reaction with eosinophilia and systemic symptoms (DRESS) in subjects with HLA-B*58:01.
UNASSIGNED: Subjects with B*58:01 were enrolled (21 allopurinol-induced DRESS and 52 allopurinol-tolerant control). HLA-A, -B, -C and -DRB1 alleles were compared. Comparison of risk between HLAs and allopurinol-induced SCAR in separate populations was performed to support the results. Kruskal-Wallis test, Pearson\'s chi-square test, Fisher\'s exact test and binary logistic regression were used to analyze the risk of SCAR development.
UNASSIGNED: Frequencies of A*24:02 (71.4 vs. 17.3%, p < 0.001, odds ratio [OR] = 12.0; 95% confidence interval [CI], 3.6-39.2) were significantly higher in B*58:01 (+) DRESS than B*58:01 (+) tolerant controls. In addition, DRB1*13:02 further increased the risk of DRESS. The phenotype frequency of A*24:02/DRB1*13:02 was significantly higher in the B*58:01 (+) DRESS group than in the B*58:01 (+) tolerant controls (52.4% vs. 5.8%, p < 0.001, OR, 66.0; 95% CI, 6.1-716.2). In 2782 allopurinol user cohort, the overall prevalence of DRESS was 0.22%, which increased to 1.62% and 2.86% in the presence of B*58:01 and B*58:01/A*24:02, respectively.
UNASSIGNED: The additional secondary screening with A*24:02 and DRB1*13:02 alleles may identify those at further increased risk of allopurinol-induced DRESS in B*58:01 carriers.