Insulin autoimmune syndrome in an occidental woman: a case report and literature review

SUMMARY Insulin autoimmune syndrome (IAS, Hirata's disease) is a rare hypoglycemic disorder characterized by spontaneous hypoglycemia associated with extremely high circulating insulin levels and positive anti-insulin antibody results. Thus far, most cases have been reported in Asian countries, notably Japan, with few cases reported in western countries. As a possible cause, it is associated with the use of drugs containing sulfhydryl radicals, such as captopril. This report refers to a 63-year-old female Brazilian patient with a history of postprandial hypoglycemia. After extensive investigation and exclusion of other causes, her hyperinsulinemic hypoglycemia was considered to have likely been induced by captopril. Most cases of IAS are self-limiting. However, dietary management, corticosteroids, plasmapheresis, and rituximab have already been used to treat patients with IAS. In our case, after discontinuation of captopril, an initial decrease in insulin autoantibody levels was observed followed by improvement in episodes of hypoglycemia. Although it is a rare disease, IAS should be considered in the differential diagnosis of endogenous hyperinsulinemic hypoglycemia. Patients with suspected IAS must be screened for autoimmunity-related drugs for insulin. Initial clinical suspicion of IAS can avoid unnecessary costs associated with imaging examinations and/or invasive surgical procedures.

Perfil microbiológico e desfechos clínicos de úlceras em pés de diabéticos internados / Microbiological profile and clinical outcome of severe foot ulcers of diabetic inpatients

Descrever o perfil microbiológico e os desfechos clínicos de úlceras graves em pés diabéticos de pacientes internados em um hospital universitário de atenção terciária no estado do Ceara, Brasil. Métodos: Conduziu-se uma análise retrospectiva de dados obtidos nos prontuários médicos de todos os pacientes diabéticos internados entre janeiro de 2006 a junho de 2007, nas enfermarias do Serviço de Endocrinologia e Diabetes do Hospital Universitário Walter Cantídio (Universidade Federal do Ceará), por úlceras graves em pés diabéticos, com no mínimo grau 2 da classificação de Wagner, refratárias ao tratamento ambulatorial. Dados clínicos (sexo, idade, tempo de diabetes e co-morbidades) de cada paciente assim como as características microbiológicas do material colhido das suas ulceras em pés ou das suas peças cirúrgicas (amputações) foram obtidos. Resultados: Foram identificados no período 17 diabéticos, todos tipo 2, com idade de 58,11±10,8 anos e 12,4±8,4 anos de doença, 58,8% homens. Das úlceras, 41,1% eram grau 2, 35,2% grau 3, 11,7% grau 4 e 11,7% grau 5 de Wagner, 64,7% com menos de 3 meses de evolução. Realizaram limpeza cirúrgica 82,3% dos pacientes e amputações 47%, sendo identificada osteomielite em 47% dos casos. Antibioticoterapia empírica foi iniciada em todos os pacientes, sendo ciprofloxacina/metronidazol o esquema mais usado (76,5%). Houve cultura negativa em 12,5% das realizadas. Nas positivas, os germes mais freqüentes foram: S. aureus (57,1%); S. viridans (28,7%); P. aeruginosas (28,7%) e M. morganii (28,7%) A maioria (75%) dos S. aureus isolados eram meticilino-resistentes, mas sensíveis à vancomicina. Conclusão: Observouse a presença de flora polimicrobiana com grande número de patógenos multirresistentes e elevada prevalência de osteomielite e amputações em diabeticos portadores de úlceras graves, neuropatia e doença vascular periférica.

To describe the microbiological profile and clinical outcomes of diabetic foot ulcers of inpatients of a tertiary university hospital, at Ceara, Brazil. Methods: We conducted a retrospective analysis of medical charts data of all diabetic inpatients of the Endocrine and Diabetes Unit of Walter Cantídio University Hospital (Federal University of Ceará), admitted from January, 2006 to June, 2007 for severe foot ulcers (minimum of grade 2 of Wagner`s classification), which were refractory to ambulatory treatment. Clinical data from each patient were recorded (sex, age, diabetes duration, and comorbidities) as well as microbiological characteristics of foot ulcers and surgical (amputations) material. Results: We identified 17 diabetic patients, all type 2, aged 58.11 ± 10.8 years and 12.4 ± 8.4 years of disease, 58.8% male. Of ulcers, 41.1% were grade 2; 35.2% grade 3; 11.7% grade 4 and 11.7% grade 5 of Wagner; 64.7% with less than 3 months of evolution. Debridement was performed in 82.3% of patients and amputation in 47%; osteomyelitis was identified in 47% of cases. All patients started empiric antibiotic therapy, where ciprofloxacin/metronidazole was the most used scheme (76.5%). Cultures were negative in 12.5% of the patients. In the positive ones, the most prevalent bacterial pathogens detected in the culture materials were: S. aureus (57.1%); S. viridans (28.7%); P. aeruginosas (28.7%); M. morganii (28.7%). The majority (75%) of isolated S. aureus were methicillin-resistant, but were sensitive to vacomicin. Conclusion: We observed the presence of polymicrobial flora with a large number of multiresistant pathogens and high prevalence of osteomyelitis and amputations in diabetic patients with severe ulcers, neuropathy and peripheral vascular disease.

Tumores testiculares bilaterais por hiperplasia congênita de restos adrenais / Bilateral testicular tumors caused by congenital adrenal rest hyperplasia

OBJETIVOS: Tumores testiculares são uma rara condição associada à hiperplasia adrenal congênita (HAC) que decorrem da hiperplasia de restos adrenais intratesticulares (HRA), raramente ocorrendo associados a neoplasias malignas. Sua diferenciação histológica com tumores de células de Leydig é muito difícil, podendo levar a orquiectomias desnecessárias. O objetivo deste relato foi apresentar esse dilema diagnóstico em um paciente com HAC e tumores testiculares bilaterais. MÉTODOS: Relatou-se o caso de um paciente masculino, 16 anos, com diagnóstico de HAC desde os 3 anos de idade, que apresentava tumorações testiculares endurecidas, indolores e de crescimento lento, sendo encaminhado para orquiectomia bilateral. RESULTADOS: Foi decidido por tratamento conservador com prednisona, havendo significativa diminuição do volume testicular e normalização dos níveis de andrógenos. CONCLUSÃO: Este caso demonstra a importância de sempre se considerar a hipótese de HRA intratesticulares no diagnóstico diferencial dos tumores testiculares. A investigação e a conduta devem ser conduzidas de maneira cautelosa para se evitar orquiectomias desnecessárias.

OBJECTIVES: Testicular tumors are a rare condition associated with congenital adrenal hyperplasia (CAH), originated from intratesticular adrenal rest tumors, and they are rarely associated with malignant tumors. Their histological differentiation from Leydig-cell tumors is quite difficult, which would lead to inappropriate orchiectomies. Thus the objective of this report was to present this diagnostic dilemma. METHODS: Reported the case of 16-yr-old boy with previous diagnosis of CAH with bilateral testicular enlargement who was recommended to be submitted to a bilateral orchiectomy. RESULTS: Considering this findings, it was decided to treat conventionally with prednisone with significant reduction of testicular volume, and normalization of androgens levels. CONCLUSION: This case shows the importance of intratesticular adrenal rest tumors in the differential diagnosis of testicular tumors. Cautious approach during investigation and treatment are recommended to avoid inappropriate orchiectomies.