RESUMO
There are only six cases in literature that describe development of dystonia with Sjogren's syndrome (SS). We describe a case of a 43-year-old woman who presented with symptoms including movement disorder, sensory neurogenic bladder, sensory loss and neuropathic pain, migraine like headaches, musculoskeletal pain, Raynaud's phenomenon and dysautonomia. Symptoms started in 2000, with weakness that progressed to dystonia in 2003. Diagnostic work-up was inconclusive with negative inflammatory serologies, cerebrospinal fluid and MRI for many years. After patient developed sicca syndrome with dry eyes and mouth in 2009, her rheumatoid factor titre was elevated (550 IU/mL), erythrocyte sedimentation rate, anti-Sjogrens syndrome-related antigen A (anti-Ro/SSA) and anti-SSB/La: anti-Sjogrens syndrome-related antigen B (anti-La/SSB) became positive. Lip biopsy confirmed diagnosis of SS. She was diagnosed with primary SS with neurological involvement. Her symptoms responded well to intravenous methylprednisolone. Symptoms stabilised with trials of immune-suppressive therapy. This is a case that demonstrates the delay of diagnosing SS with preceding unique neurological association.
Assuntos
Distonia/diagnóstico , Síndrome de Sjogren/diagnóstico , Adulto , Anticorpos Antinucleares/imunologia , Distonia/etiologia , Distonia/imunologia , Feminino , Humanos , Imunossupressores/uso terapêutico , Transtornos de Enxaqueca/diagnóstico , Transtornos de Enxaqueca/etiologia , Transtornos de Enxaqueca/imunologia , Neuralgia/diagnóstico , Neuralgia/etiologia , Neuralgia/imunologia , Disautonomias Primárias/diagnóstico , Disautonomias Primárias/etiologia , Disautonomias Primárias/imunologia , Doença de Raynaud/diagnóstico , Doença de Raynaud/etiologia , Doença de Raynaud/imunologia , Glândulas Salivares Menores/patologia , Transtornos de Sensação/diagnóstico , Transtornos de Sensação/etiologia , Transtornos de Sensação/imunologia , Síndrome de Sjogren/complicações , Síndrome de Sjogren/tratamento farmacológico , Síndrome de Sjogren/imunologia , Síncope/diagnóstico , Síncope/etiologia , Síncope/imunologia , Bexiga Urinaria Neurogênica/diagnóstico , Bexiga Urinaria Neurogênica/etiologia , Bexiga Urinaria Neurogênica/imunologiaRESUMO
Natural rubber latex is the second most implicated agent in intraoperative anaphylactic reactions. This report describes a case of intraoperative anaphylaxis occurring in a non-atopic fourteen-year-old girl undergoing multiple surgical procedures, but without spina bifida, in which latex surgical gloves were the main culprit for the anaphylactic reactions. Clinical manifestations of an anaphylactic reaction were also experienced during the examination of the possible cause of intraoperative anaphylaxis by skin prick testing with a latex allergen extract. Skin tests with anesthetics were negative. Specific IgE to latex was positive at 92.9 kUA/L (class 5). The molecular basis for the reported intraoperative anaphylaxis was ascribed to three low-molecular mass latex allergens (10-15 kD) detected in the brand of latex surgical gloves used during the operation. Given the potential of a dramatic outcome, latex allergy testing as a regular preoperative measure may contribute to the reduction of anaphylactic reactions during surgical interventions.