Oculomasticatory myorhythmia in Whipple's disease: The Lance
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A 58-year-old woman presented with a 7-year history of chronic intermittent arthritis and weight loss. She had also developed alterations in her sleep–wake cycle, anxiety, and oculomotor abnormalities, followed by progressive cognitive impairment and intermittent agitation. She was hospitalised for impaired consciousness and respiratory failure requiring intubation.

Clinical examination revealed an oculomasticatory myorhythmia, leading to suspicion of Whipple's disease. Diagnosis of classical Whipple's disease was confirmed by a positive periodic-acid-Schiff stain on jejunal biopsy samples and PCR detection of Tropheryma whipplei in cerebrospinal fluid. After antibiotic treatment for aspiration pneumonia (a combination of 4g piperacillin and 500mg tazobactam, administered intravenously every 8 h for 5 days), she received a combination of sulfadiazine, doxycycline, and hydroxychloroquine for Whipple's disease. Cognitive impairment and myorhythmia persisted without any improvement.

Whipple's disease is a rare systemic infectious disease caused by T whipplei. The classic form of the disease associated with various manifestations including neurological involvement. Oculomasticatory myorhythmia is defined as pendular vergence oscillations of the eyes that are synchronous with masticatory myorhythmia. Although uncommon, masticatory myorhytmia is considered pathognomonic of Whipple's disease and should lead to appropriate, prolonged antibiotic treatment.

Source: https://www.thelancet.com/journals/lanrhe/article/PIIS2665-9913(20)30005-9/fulltext
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