Background: Spinal cord stimulation (SCS) has recently been reported to be effective for truncal postural abnormalities such as camptocormia and Pisa syndrome in Parkinson's disease. In this case report, we describe a case of a woman with Parkinson's disease in whom SCS was effective for painful camptocormia with Pisa syndrome. Case presentation: A 65-year-old woman was admitted to our hospital because of painful camptocormia. She had noticed resting tremor in the left upper limb and aprosody at 48 years of age. She was diagnosed as having Parkinson's disease (Hoehn & Yahr stage 1) at 53 years of age. Cabergoline was started during that same year, with subsequent addition of selegiline hydrochloride; the symptoms of parkinsonism disappeared. Wearing-off occurred when she was 57 years old, 3 years after starting carbidopa/levodopa, and truncal postural abnormalities-painful camptocormia with Pisa syndrome to the right-appeared at 58 years of age. These symptoms worsened despite adjustment of her oral medications, and deep brain stimulation (DBS) was performed when she was 60 years old. The truncal postural abnormalities improved after DBS, and she could travel abroad at 61 years of age. However, from 62 years of age, painful camptocormia with Pisa syndrome to the right reappeared. The pain was unsuccessfully treated with oral analgesics, radiofrequency coagulation of the dorsal and medial branches of the lumbar spinal nerve, and lumbar epidural block. Finally, SCS was performed for the pain relief. The pain disappeared immediately after SCS and her posture then gradually improved. Unified Parkinson's Disease Rating Scale score improved from 48 to 34 points and Timed Up and Go Test improved from 15 s to 7 s after SCS. Conclusions: This case suggests that SCS may be effective for improving painful truncal postural abnormalities and motor complications of Parkinson's disease. Pain relief or a direct effect on the central nervous system by SCS was considered to explain the alleviation of these symptoms.

Rationale:Subcutaneous implantable electrocardiographs are highly effective in detecting covert atrial fibrillation (AF) in cryptogenic stroke. However, these invasive devices are not indicated for all cryptogenic stroke patients, and noninvasive improvements over conventional Holter-type ambulatory electrocardiography are needed. We evaluated the clinical application and effectiveness of Duranta (ImageONE Co., Ltd.), a wireless patch-type electrocardiographic monitoring system developed in Japan for chronically ill patients or home-based patients at the end of life. A Duranta device was used to detect covert AF in patients with acute ischemic stroke of undetermined source with no sign of AF during cardiographic monitoring ≥24 hours postadmission.Patient concerns:A 72-year-old man with severe aortic stenosis was admitted to our hospital with dysarthria and right upper limb weakness. Diffusion-weighted plain head magnetic resonance imaging (MRI) showed acute cerebral infarctions across the left middle cerebral artery territory. Twelve-lead electrocardiography, Holter-type ambulatory electrocardiography, and cardiographic monitoring for ≥24 hours revealed no AF, indicating a probable diagnosis of artery-to-artery embolism following left common carotid artery stenosis detected by carotid ultrasound imaging and cerebral angiography.Interventions:However, because of high blood brain natriuretic peptide (BNP) and valvular heart disease, continuous monitoring using Duranta was performed from the 2nd to 13th days after onset to exclude possible cardioembolic stroke. Waveform and heart rate trend graph analysis showed paroxysmal AF (PAF) occurred on the 5th and 9th days after onset. PAF did not occur at any other time during the observation period. The quality of the cardiograms sufficed for analysis and diagnosis of AF. The lightweight compact device can be placed quickly with no movement restriction. These features and our findings show the usefulness of the Duranta device for long-term continuous monitoring.Lessons:A noninvasive wireless patch-type electrocardiographic monitoring system, Duranta, placed at the precordium, was useful in detecting covert AF in cryptogenic stroke patients, warranting further investigation.

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Rationale:We report a case of successful diagnosis of POEMS (polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin changes) syndrome based on monoclonality that was confirmed by an osteosclerotic lesion biopsy in a patient without pathognomonic symptoms or monoclonal gammopathy, probably because of comorbidities, which included systemic lupus erythematosus, rheumatoid arthritis, and Sjögren syndrome.Patient concerns:A 57-year-old woman presented with an approximately 2-year history of numbness in the toes that had gradually spread, along with muscle weakness in both arms and legs. She had been receiving immunosuppressant and corticosteroid therapy since being diagnosed with systemic lupus erythematosus and Sjögren syndrome at the age of 31 years and rheumatoid arthritis at the age of 44 years. Neurological examination revealed predominantly distal hypoesthesia and weakness in a typical stocking-and-glove pattern. Immunoelectrophoresis revealed elevated polyclonal immunoglobulin, which was attributed to her known underlying disease.Diagnoses:Biopsy of an osteosclerotic lesion confirmed proliferation of monoclonal plasma cells, leading to a diagnosis of POEMS syndrome.Interventions and outcomes:Lenalidomide therapy was started after the diagnosis and the patient had a favorable outcome.Lessons:Osteosclerotic lesion biopsy can be useful for diagnosis of POEMS syndrome in difficult cases.

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