Hiroaki Fushiki

Respiratory involvement in IgG4-related Mikulicz’s disease

Abstract‘Immunoglobulin G4 (IgG4)-related disease’ is a new clinical concept of multi-organ diseases, with Mikulicz’s disease (MD) being a clinical phenotype of IgG4-related disease. To clarify the clinical characteristics of respiratory involvement associated with IgG4-related MD, we retrospectively assessed 25 patients with MD, 11 (44%) of whom had allergic symptoms, and 7 (28%) of whom complained of respiratory problems. Thirteen patients (52%) presented with pulmonary and/or mediastinal lesions (P-MD) on chest computed tomography (CT), and 11 (44%) had lesions limited to the lacrimal and/or salivary glands (L-MD). Mean serum total protein, IgG, and IgG4 concentrations were significantly higher and CH50 was significantly lower in the P-MD than in the L-MD group. Immune complex was present only in the P-MD group. Chest CT images showed bronchial wall thickening, consolidation, nodule(s), interlobular thickening, ground glass opacity, pleural thickening/effusion, and mediastinal lymphadenopathy. Five of seven patients who underwent histological examination of the lungs had abundant IgG4-positive plasma cell infiltrates (IgG4/IgG-positive plasma cells >40%), but the other two did not. These findings suggest that respiratory lesions are not rare in patients with IgG4-related MD, and that they present with various manifestations. IgG4-related MD should be differentiated from similar diseases, such as sarcoidosis, bronchial asthma, Sjögren’s syndrome, and malignant lymphoma.

Influence of visually induced self-motion on postural stability.

Conclusion Our results indicate that the illusion of self-motion is a significant factor leading to spatial disorientation. Objective Under normal circumstances, self-motion is perceived in response to motion of the head and body. However, under certain conditions, such as virtual reality environments, visually induced self-motion can be perceived even though the subject is not actually moving, a phenomenon known as “vection”. The aim of this study was to examine the possible influence of illusory self-rotation (circular vection) on postural adjustments. Material and methods The subjects were 10 young females with no history of ocular or vestibular disease. Video-motion analysis was applied to measure postural movements during vertical optokinetic stimulation. Results For most subjects, movement of the visual surroundings induced head and body displacements in the same direction as that of the visual stimulus, regardless of the onset of self-motion perception. However, there was a significant increase in postural instability after the subjects began to perceive false self-motion in the opposite direction to that of the visual stimulus.

Mumps virus infection in adults : Three cases of supraglottic edema

Mumps virus infections primarily involve the parotid glands and most frequently affect school‐aged children. We present three unusual adult cases of mumps with dyspnea secondary to severe swelling of the salivary glands and review previously reported cases in the literature. Dyspnea developed progressively after the onset of salivary gland swelling. Laryngoscopy revealed an advanced edematous change in the supraglottis obstructing the airway. In two cases, tracheotomy was needed because of rapid worsening of the supraglottic edema. Questioning regarding breathing problems and laryngoscopic examination is therefore recommended when one encounters a mumps case with combined parotid and salivary gland swelling.

Prognosis of sudden low-tone loss other than acute low-tone sensorineural hearing loss

Conclusion: In clinical practice, sudden low-tone loss other than acute low-tone sensorineural hearing loss (ALHL) shows a potential for recurrence. Objective: ALHL is often associated with recurrence and/or progression to Ménières disease. We examined the prognosis of patients with sudden low-tone loss who were not classified in the audiometric definition of ALHL. Methods: Sixty-three patients diagnosed at the university hospital with idiopathic sudden low-tone loss of sensorineural hearing loss and without subjective vertigo were followed up in the long term with a mean of over 4 years. The rates of recurrence and/or progression to Ménières disease for patients with low-tone loss type other than ALHL (non-ALHL) were compared with those for ALHL patients. Results: The recovery rate at the initial treatment was 47.6% for non-ALHL and 62.9% for ALHL. A Kaplan–Meier plot indicated that cumulative recurrence rates for non-ALHL were 20.2% at 1 year and 43.5% at 5 years, whereas the rates for ALHL were 12.2% at 1 year and 31.3% at 5 years. There was no statistically significant difference between the two patient groups.

Prognostic value of initial electronystagmography findings in idiopathic sudden sensorineural hearing loss without vertigo.

We used electronystagmography (ENG) to characterize recurrent hearing loss and its progression to definite Ménières disease in patients with idiopathic sudden sensorineural hearing loss (SSHL) without subjective vertigo. Methods: We reviewed medical records of 1,334 patients with unilateral hearing loss initially diagnosed with idiopathic SSHL between 1985 and 2003 at our university hospital. Of the 1,334 patients, we examined 127 (86 with low-tone and 41 with high-tone SSHL) who underwent ENG within 30 days of the initial diagnosis and who could be followed up during the long term. Results: Spontaneous nystagmus (SN) was observed in approximately half of the vertigo-unaccompanied group. Long-term follow-up with a mean of 67 months revealed that the recurrence rate of hearing loss was 51.2% in low-tone SSHL patients with SN. The recurrence rate of hearing loss was 27.9% in low-tone SSHL patients without SN. Progression to Ménières disease occurred in 14.0% of the low-tone-type and 12.5% of the high-tone-type patients when SN was detected. In contrast, in both the low-tone-type and high-tone-type groups, none developed Ménières disease when SN was absent. Conclusion: Our results suggest that the initial ENG findings could provide prognostic information for idiopathic SSHL patients, even those who have no vestibular symptoms at the first visit.

MRI of intranasal pleomorphic adenoma

A pleomorphic adenoma in the nasal cavity constitutes a rarity. A 48-year-old woman presented with intranasal pleomorphic adenoma revealed by unilateral continuous obstruction and occasional epistaxis. Nasal endoscopy showed a mucosa-covered and opalescent polypoidal mass arising from the left nasal septum. Unlike cases affecting the major salivary gland, magnetic resonance image (MRI) revealed that the mass showed a low signal intensity on T1-weighted image and a heterogeneous, intermediate signal intensity on T2-weighted image. MRI findings can aid in diagnosis and help exclude the possibility of other neoplasms that occur at this site.

Head and body sway in response to vertical visual stimulation

Conclusions. Postural responses differed according to the stimulus direction, i.e. vertical visual stimulation induced head rather than trunk displacements. Accordingly, it could be that center of foot pressure (COP) responses tended to underestimate the postural sway during visual stimulation. Objectives. To investigate head and body sway in response to vertical visual surround motion, and to examine the correlation between the displacements of head and body segments derived from video-motion analysis and COP measurements. Material and methods. Postural sway was assessed in 10 young female subjects by video-motion analysis of four different head and body segments, and by use of force-plate posturography. Head and body sway in the pitch plane was induced by rotating a random pattern of dots about the subjects inter-aural axis at a constant acceleration of 1°/s2 or a constant velocity of 60°/s in darkness. Results. Generally, head displacement was greater than that of other body parts during vertical optokinetic stimulation (OKS). In most subjects, maximum head displacements were induced in the same direction as the visual motion. Downward OKS induced a forward head and body sway. The COP trajectory correlated well with the displacements of each head and body segment during downward OKS. In contrast, postural responses to upward OKS were complicated in terms of their time course. The correlation coefficient between each head and body segment and the COP varied among individuals for upward OKS.

A low-grade chondrosarcoma presenting as an unusual cervical mass in the hyoid bone: a case report

IntroductionA chondrosarcoma originating from the hyoid bone is very rare. Here, we describe a case of low-grade chondrosarcoma of hyoid origin and discuss its preoperative imaging features, including those on positron emission tomography-computed tomography, and its recurrence rate.Case presentationA 42-year-old Japanese man noticed a mass in the right submandibular region of his neck. A hard 3.0 × 2.8 cm tumor was noted on the right side of his hyoid bone. The mass was immobile and moved with deglutition.ConclusionEven though radiographic studies, including positron emission tomography-computed tomography, were inconclusive, the cartilaginous tumor was surgically removed en bloc, and the tumor was diagnosed based on the results of pathological investigations. Close follow-up is recommended in such cases due to the potential for recurrences, because local recurrence occurred in 50% of the reported cases of grade one chondrosarcomas.

Short-Term Synaptic Plasticity in the Dentate Gyrus of Monkeys

The hippocampus plays an important role in learning and memory. Synaptic plasticity in the hippocampus, short-term and long-term, is postulated to be a neural substrate of memory trace. Paired-pulse stimulation is a standard technique for evaluating a form of short-term synaptic plasticity in rodents. However, evidence is lacking for paired-pulse responses in the primate hippocampus. In the present study, we recorded paired-pulse responses in the dentate gyrus of monkeys while stimulating to the medial part of the perforant path at several inter-pulse intervals (IPIs) using low and high stimulus intensities. When the stimulus intensity was low, the first pulse produced early strong depression (at IPIs of 10–30 ms) and late slight depression (at IPIs of 100–1000 ms) of field excitatory postsynaptic potentials (fEPSPs) generated by the second pulse, interposing no depression IPIs (50–70 ms). When the stimulus intensity was high, fEPSPs generated by the second pulse were depressed by the first pulse at all IPIs except for the longest one (2000 ms). Population spikes (PSs) generated by the second pulse were completely blocked or strongly depressed at shorter IPIs (10–100 or 200 ms, respectively), while no depression or slight facilitation occurred at longer IPIs (500–2000 ms). Administration of diazepam slightly increased fEPSPs, while it decreased PSs produced by the first pulse. It also enhanced the facilitation of PSs produced by the second stimulation at longer IPIs. The present results, in comparison with previous studies using rodents, indicate that paired-pulse responses of fEPSPs in the monkey are basically similar to those of rodents, although paired-pulse responses of PSs in the monkey are more delayed than those in rodents and have a different sensitivity to diazepam.

Correlation between canal paresis and spontaneous nystagmus during early stage of acute peripheral vestibular disorders.

Abstract Conclusions: This study demonstrates that the resolution period of spontaneous nystagmus (SN) may provide an indication of vestibular dysfunction on a particular day in the primary care setting. Objective: We aimed to predict canal paresis using fundamental observations of SN during the early stage of acute peripheral vestibular disorders. Methods: The study involved 87 patients who had recently experienced their first episode of acute spontaneous vertigo and direction-fixed horizontal nystagmus. Although they did not exhibit any other neurological deficits, they had been hospitalized with severe acute symptoms between 2004 and 2007. A correlation between the resolution period of SN and the results of laboratory caloric testing was reviewed. Results: The receiver operating characteristic analysis showed that the resolution period of SN may be a predictive indicator of unilateral vestibular hypofunction in the acute stage. In about half of the patients, SN disappeared on the third day after their initial visit. However, in 20% of the patients SN still persisted on the eighth day. Among the patients with SN, the prevalence of canal paresis increased with the increase in the resolution period of SN. When SN was observed on the fifth day, the prevalence was approximately 70%.