A case report of cryptococcosis in a captive Cape hyrax (Procavia capensis).

Cryptococcosis, a globally distributed mycotic disease caused by Cryptococcus neoformans or C. gattii, has been extensively studied in various domestic animals and humans. However, non-domestic species have often been overlooked in the literature, with limited attention given to their susceptibility and contribution to the epidemiology of the disease. In this study, a captive two-year-old Cape hyrax in a Japanese zoo exhibited neurological symptoms and torticollis, ultimately succumbing to the infection. Necropsy and pathological analyses, including histopathological techniques and PCR, revealed the presence of C. neoformans in the lungs, cerebrum, and internal auditory canal. While cryptococcosis has been reported in various wild animals globally, this case represents the first documented cryptococcosis in Cape hyrax.

Statistical Analysis of the Factors that Affect Postoperative Length of Hospital Stay after Unruptured Intracranial Aneurysm Treatment in Japan: A 20-year Nationwide Multicenter Study.

Treatment strategies for unruptured intracranial aneurysms (UIAs) should be carefully considered with reference to rupture and complication rates. It is also important to minimize the length of hospital stay (LOS) and to ensure a high quality of medical care. In this study, we aim to clarify the factors that affect the LOS of patients treated for UIAs using the Inpatient Clinico-Occupational Database of the Rosai Hospital Group (ICOD-R). This was a nationwide-multicenter study based on ICOD-R data from 2000 to 2019. Patients diagnosed with UIAs who were treated with clipping or coiling were included in the study. Multivariate analysis was performed to identify the factors affecting LOS. LOS was also compared between groups classified by surgical procedure or treatment period. We identified 3294 patients on the database who underwent clipping or coiling of UIAs during the study period. Multivariate analysis revealed hospital admission during the early 2000s and the late 2010s, age, and treating institution to be significantly correlated with LOS (p < 0.05). There was a significant difference between the mean LOS of the clipping group (20.3 days) and the coiling group (9.65 days) (p < 0.001). Compared by treatment period, LOS significantly shortened over time. Our results suggest that the type of treatment, time of treatment, patient age, and the treating institution affect postoperative LOS for UIAs. Although coiling was found to lead to a lower average LOS than clipping, treatment selection should take the characteristics of each patient's aneurysm into consideration.

Apolipoprotein C-III amyloidosis in white lions (Panthera leo).

Apolipoprotein C-III (ApoC-III) amyloidosis in humans is a hereditary amyloidosis caused by a D25V mutation in the APOC3 gene. This condition has only been reported in a French family and not in animals. We analyzed a 19-year-old white lion (Panthera leo) that died in a Japanese safari park and found renal amyloidosis characterized by severe deposition confined to the renal corticomedullary border zone. Mass spectrometry-based proteomic analysis identified ApoC-III as a major component of renal amyloid deposits. Amyloid deposits were also positive for ApoC-III by immunohistochemistry. Based on these results, this case was diagnosed as ApoC-III amyloidosis for the first time in nonhuman animals. Five additional white lions were also tested for amyloid deposition retrospectively. ApoC-III amyloid deposition was detected in 3 white lions aged 19 to 21 years but not in 2 cases aged 0.5 and 10 years. Genetic analysis of white and regular-colored lions revealed that the APOC3 sequences of the lions were identical, regardless of amyloid deposition. These results suggest that ApoC-III amyloidosis in lions, unlike in humans, may not be a hereditary condition but an age-related condition. Interestingly, lion ApoC-III has a Val30 substitution compared with other species of Panthera that have Met30. Structural predictions suggest that the conformation of ApoC-III with Met30 and ApoC-III with Val30 are almost identical, but this substitution may alter the ability to bind to lipids. As with the D25V mutation in human ApoC-III, the Val30 substitution in lions may increase the proportion of free ApoC-III, leading to amyloid formation.

Vitreous hemorrhage in retinal vein occlusion without visible traction from the posterior vitreous membrane: An optical coherence tomography angiography case report study.

Background: We report an unusual case of retinal vein occlusion (RVO) associated with vitreous hemorrhage (VH) without visible traction from the posterior vitreous membrane (PVM) at the bleeding point, challenging our current understanding of VH pathophysiology. Case presentation: A 52-year-old man presented with VH in the right eye. A detailed examination using optical coherence tomography angiography (OCTA) and ultra-widefield fluorescein angiography revealed branch RVO with non-perfused areas (NPAs) extending peripherally and neovascularization elsewhere (NVE). OCTA showed NVE infiltrating the vitreous cavity, leading to substantial bleeding without visible PVM traction at the bleeding point. The NVE was successfully removed following vitrectomy, and visual acuity improved from 20/20 to 20/13 preoperatively, along with a postoperative improvement in floaters. Conclusions: This unique case of RVO suggests the possibility of VH occurring independent of PVM contractions at the bleeding point, challenging the traditional understanding of VH. This finding underscores the potential role of OCTA in diagnosing and managing retinal vascular diseases, underscoring the need for further investigations into the underlying mechanisms, with potential implications for personalized therapeutic strategies.

The Usefulness of Surgical Titanium Microclips for Mucosal Repair in the Frontal Sinus Using ORBEYE: A Technical Note.

Bifrontal craniotomy frequently involves opening the frontal sinus and mucosal injury. We report a new technique for mucosal repair in the frontal sinus using surgical titanium microclips. Six consecutive patients who underwent bifrontal craniotomy with frontal sinus exposure and mucosal injury underwent mucosal repair using surgical titanium microclips between April 2019 and August 2022. In all cases, the frontal sinus mucosa was peeled from the inner walls of the frontal sinus to ensure sufficient mucosal margin for clipping using ORBEYE. The repair was accomplished with the microclips in all cases. We also sealed the mucosal wound using fibrin glue and sufficiently filled the frontal sinus with bone debris, resulting in zero incidence of postoperative liquorrhea in all cases. Repairing the mucosa using surgical titanium microclips using ORBEYE may be a simple and quick technique when the frontal sinus mucosa is injured during craniotomy.

Label-free autofluorescence and hyperspectral imaging of cerebral amyloid-ß lesions in aged squirrel monkeys.

The observation of amyloid-ß (Aß) lesions using autofluorescence in transgenic mice and human Alzheimer disease patients has been reported frequently. However, no reports verify the autofluorescence of spontaneous Aß amyloidosis in animals, to our knowledge. We validated the autofluorescence of Aß lesions in spontaneous squirrel monkey cases under label-free conditions; lesions had intense blue-white autofluorescence in fluorescence microscopy using excitation light at 400-440 nm. Thioflavin S staining and immunohistochemistry of the same specimens revealed that this blue-white autofluorescence was derived from Aß lesions. Hyperspectral analysis of these lesions revealed a characteristic spectrum with bimodal peaks at 440 and 460 nm, as reported for Aß lesions in mice. Principal component analysis using hyperspectral data specifically separated the Aß lesions from other autofluorescent substances, such as lipofuscin. A non-labeled and mechanistic detection of Aß lesions by hyperspectral imaging could provide valuable insights for developing early diagnostic techniques.

Foveal Thickness Fluctuations in Anti-VEGF Treatment for Central Retinal Vein Occlusion.

Purpose: The aim of this study was to examine the effects of foveal thickness (FT) fluctuation (FTF) on 2-year visual and morphological outcomes of eyes with central retinal vein occlusion (CRVO) undergoing anti-VEGF treatment for recurrent macular edema (ME) based on a pro re nata regimen. Design: Retrospective, observational case series. Participants: We analyzed 141 treatment-naive patients (141 eyes) with CRVO-ME at a multicenter retinal practice. Methods: We assessed FT using OCT at each study visit. Patients were divided into groups 0, 1, 2, and 3 according to increasing FTF. Main Outcome Measures: We evaluated the logarithm of the minimal angle of resolution (logMAR) best-corrected visual acuity (BCVA), the length of the foveal ellipsoid zone (EZ) band defect measured using OCT, and the association of FTF with VA and EZ band defect length. Results: The mean baseline logMAR BCVA and FT were 0.65 ± 0.52 (Snellen equivalent range: 20/20-20/2000) and 661.1 ± 257.4 µm, respectively. The mean number of anti-VEGF injections administered was 5.6 ± 3.6. At the final examination, the mean logMAR BCVA and FT values were significantly improved relative to the baseline values (both P < 0.01). During the observation, BCVA longitudinally improved in Groups 0 and 1, remained unchanged in Group 2, and worsened in Group 3. Likewise, the length of the foveal EZ band defect did not increase in Group 0; however, it gradually increased in Groups 1, 2, and 3. Foveal thickness fluctuation was significantly and positively associated with the logMAR BCVA and length of the foveal EZ band defect at the final examination (P < 0.01). The final logMAR BCVA of patients developing neovascular complications was 1.27 ± 0.72 (Snellen equivalent range: 20/50-counting fingers), which was significantly poorer than that of patients without complications (P < 0.001). There was no significant difference in the neovascular complication rate among the FTF groups (P = 0.106, Fisher exact test). Conclusions: In eyes receiving anti-VEGF treatment for CRVO-ME, FTF can longitudinally impair the visual acuity and foveal photoreceptor status during the observation period, thus influencing the final outcomes. However, neovascular complications, which would also lead to a poor visual prognosis, may not be associated with FTF. Financial Disclosures: The authors have no proprietary or commercial interest in any materials discussed in this article.

Pathological Characterization of Spontaneous AA Amyloidosis in Microminipigs.

The minipig has been used as a non-rodent species in nonclinical toxicology studies, but little is known about amyloid A (AA) amyloidosis in this species. Among domestic pigs, reports of AA amyloidosis have been limited to animals with mutations in the N-terminal residue of serum AA (SAA), which is thought to be a primary etiological factor. In this study, we histologically examined 26 microminipigs aged 0.6 to 10 years and observed amyloid deposition in one 0.6-year-old and six 5-year-old or older microminipigs. The amyloid deposits were identified as AA based on mass spectrometry (MS) and immunohistochemistry (IHC). The 0.6-year-old microminipig showed severe deposition in the renal cortex and spleen, whereas 5-year-old or older animals had severe deposition in the renal medulla. MS and IHC detected serum amyloid P-component (SAP) in amyloid deposits in older animals but not in a 0.6-year-old animals. Based on the proteomic analysis and gene sequencing, amino acid mutations of SAA, previously found in domestic pigs, were not involved in the pathogenesis of AA amyloidosis in microminipigs. This study demonstrates that microminipigs with wild-type SAA develop AA amyloidosis and presents the possibility that differences in the environment surrounding amyloid, such as SAP, may influence differences in the pathological phenotype.

Cerebral Venous Sinus Thrombosis Successfully Treated with Mechanical Thrombectomy under Intracranial Pressure Monitoring: A Case Report.

A 54-year-old man with no medical history presented to our hospital with vomiting, left hemiplegia, and seizures. On arrival, he was experiencing generalized tonic-clonic seizures, which required him to be intubated and deeply sedated. Contrast-enhanced computed tomography revealed extensive venous sinus obstruction from the superior sagittal sinus to the bilateral sigmoid sinus and cerebral edema with intracranial hemorrhage. An intracranial pressure (ICP) monitor was immediately placed intracranially, and mechanical thrombectomy (MT) was performed under ICP monitoring. MT was immediately terminated when the venous sinus was partially recanalized enough to decrease the ICP; then, anticoagulation therapy was initiated. Postoperative follow-up angiography revealed that venous sinus obstruction and intracranial venous perfusion improved over time. Although he had intracranial hemorrhage-induced left hemiplegia and sensory deficits, his condition improved with rehabilitation, and the patient was eventually discharged home. The indication criteria and techniques for MT for cerebral venous sinus thrombosis are yet to be established. As in this case, in patients with impaired consciousness due to intracranial hemorrhage or epilepsy, preoperative ICP monitor placement is deemed useful to evaluate venous perfusion during MT and decide the treatment goal.

Inference of Capillary Nonperfusion Progression on Widefield OCT Angiography in Diabetic Retinopathy.

Purpose: The purpose of this study was to explore the spatial patterns of the nonperfusion areas (NPAs) on widefield optical coherence tomography angiography (OCTA) images in diabetic retinopathy (DR) and to investigate their associations with NPA progression and DR severity. Methods: We prospectively enrolled 201 eyes from 158 patients with DR. Widefield images were obtained using a swept-source OCTA device (Xephilio OCT-S1), followed by the creation of 20-mm (1614 pixels) en face images. Nonperfusion squares (NPSs) were defined as 10 × 10-pixel squares without retinal vessels. Eyes with high-dimensional spatial data were mapped onto a two-dimensional space using the uniform manifold approximation and projection algorithm and divided by clustering. The patterns of NPA distribution were statistically compared between clusters. Results: All eyes were mapped onto a two-dimensional space and divided into six clusters based on the similarity of NPA distribution. Eyes in clusters 1 and 2 had minimal and small NPAs, respectively. Eyes in clusters 3 and 4 exhibited NPAs in the temporal and inferotemporal regions, respectively. Eyes in cluster 5 displayed NPAs in both superonasal and inferonasal areas. The unique NPA distributions in each cluster encouraged us to propose eight possible pathways of NPA progression. DR severity was not equal between clusters (P < 0.001), for example, 8 (15.7%) of 51 eyes and 15 (65.2%) of 23 eyes had PDR in clusters 1 and 5, respectively. Conclusions: Dimensionality reduction and subsequent clustering based on the NPA distribution on widefield OCTA enabled the inference of possible NPA progression in DR.

Quantification of dilated deep capillaries in diabetic retinopathy on optical coherence tomography angiography.

Morphological changes in capillaries are one of major clinical signs in diabetic retinopathy (DR). In this study, we quantified the dilated deep capillaries on optical coherence tomography angiography (OCTA) images. Central 3 × 3 mm en face images were obtained using a swept source OCTA device in 105 eyes of 99 patients with DR. Capillaries with a greater diameter in the deep layers were defined as the dilated deep capillaries, using stepwise image processing. The relative areas of automatically selected capillaries with a great diameter were calculated as the index of the dilated deep capillaries. Most eyes with DR had string-like or dot-like dilated deep capillaries in the OCTA images, which appeared to be dilated capillary segments or microaneurysms histologically. They were distributed more densely in the parafovea than in the central sector, while there were no differences between individual quadrants. The index of the dilated deep capillaries was higher in eyes with DR than in nondiabetic eyes. The index in the central subfield was modestly associated with visual acuity, diabetic macular edema, and proliferative diabetic retinopathy. The quantitative dilated deep capillaries are designated as a biomarker of vision-threatening DR.

Senile plaques and phosphorylated tau deposition in a super-aged rhesus monkey (Macaca mulatta).

The brain of a rhesus monkey that died at 43 years of age with symptoms of suspected cognitive dysfunction was analyzed. pathological analyses revealed characteristic Alzheimer's disease-related lesions: the aggregation of amyloid ß (Aß) in the form of senile plaques and phosphorylated tau proteins. We also revealed that Aß43, which is prone to aggregation and toxicity in humans, is involved in senile plaques in the brain of the rhesus monkey, as well as several other Aß species. Comparative studies of neuropathology using aged nonhuman primates lack behavioral descriptions compared to human medicine. This case report showed behavioral abnormalities and the detailed pathological changes that may have caused it in a super-aged rhesus monkey.

Macular retinal circulation in healthy eyes examined by optical coherence tomography angiography extended interscan time analysis.

PURPOSE: To examine whether extended interscan time (IST) on optical coherence tomography angiography (OCTA) can detect slow retinal blood flow, which is undetectable on default IST, in the healthy macula. METHODS: OCTA (OCT-A1, Canon Inc.) scanning of a macular area measuring 4 × 4 mm2 of 14 healthy eyes of 14 healthy volunteers with no history or evidence of systemic and macular diseases was performed. ISTs were set at 7.6 (IST7.6, default setting), 12.0 (IST12.0), and 20.6 msec (IST20.6). Ten OCTA images were acquired at each IST, and an averaged image was created. For each averaged OCTA image obtained at IST7.6, IST12.0, and IST20.6, we defined the area surrounded by the innermost capillary ring as the foveal avascular zone (FAZ). We qualitatively evaluated the delineation of the capillaries consisting of the FAZ and quantitatively measured the FAZ area at each IST. RESULTS: Extensions from IST7.6 to IST12.0 and IST20.6 could newly delineated retinal capillaries that were undetectable at the default IST; new capillaries were detected in 10 (71%) eyes at IST12.0 and 11 (78%) eyes at IST20.0. The FAZ areas were 0.334 ± 0.137 mm2, 0.320 ± 0.132 mm2, and 0.319 ± 0.129 mm2 for IST7.6, IST12.0, and IST20.0, respectively; the FAZ areas at IST12.0 and IST20.0 were significantly decreased compared with that at IST7.6 (p = 0.004 and 0.002, respectively). CONCLUSION: In OCTA for healthy participants, extensions of the ISTs newly detected retinal capillaries with slow blood flow around FAZ. The FAZ shapes varied with different ISTs. Thus, the blood flow dynamics are not physiologically uniform around FAZ. Compared with conventional OCTA, this protocol enables a more detailed evaluation of retinal circulation and provides a better understanding of the physiological circulatory status of the healthy retina, and may enable the assessment of circulation in the very early stages in diseased eyes.

Long-term Patency of Retrograde Bypass Using a Distal Stump of the Parietal Superficial Temporal Artery for Moyamoya Disease.

There have been a number of anastomosis methods of bypass techniques reported for moyamoya disease. However, there are yet no randomized controlled trials conducted on the anastomosis method. Retrograde blood flow of the superficial temporal artery (STA) may be used as one of the donor options. Here, we examined the tolerability of retrograde bypass using a distal stump of the parietal STA (dsPSTA). Anastomosis between the dsPSTA and middle cerebral artery (MCA) was performed for consecutive patients with moyamoya disease whose parietal STA was visualized to be longer than 10 cm using contrast-enhanced computed tomography preoperatively. Retrospectively, we have examined its patency and clinical outcome. Retrograde dsPSTA-MCA bypass was performed in 22 hemispheres of 17 patients. The patency of retrograde dsPSTA-MCA bypass in all 22 anastomoses could be confirmed during follow-up periods (mean: 5.5, range: 2-15 years). No recurrence of ischemic events was observed. The dsPSTA-MCA bypass using retrograde blood flow has been determined as one of the many promising anastomosis methods, and long-term patency was achieved in moyamoya disease.

Overlapped Double-Layer Micromesh Stents for Giant Extracranial Internal Carotid Artery Aneurysm.

Objective: Extracranial internal carotid artery aneurysms (ECAAs) are rare. We herein describe a case of overlapped stenting with two double-layer micromesh stents for a giant ECAA. Case Presentation: A 73-year-old man presented to our hospital with an enlarged right posterior cervical mass. A right internal carotid artery (ICA) angiogram revealed a giant aneurysm of 50 × 60 mm. We chose a carotid double-layer micromesh stent for stenting. With the patient under general anesthesia, the first double-layer micromesh stent (CASPER Rx, 10 × 30 mm; Terumo, Tokyo, Japan) was deployed between the ICA distal to the aneurysm and the common carotid artery (CCA). The second stent was also deployed from a site more proximal than the first one. Ten coils were then placed from a microcatheter that had been placed in the aneurysm. A right CCA angiogram after the procedure revealed a flow-diversion effect for the aneurysm. The patient was discharged with no complications. At the 6-month follow-up angiogram, blood flow into the aneurysm had completely disappeared. Conclusion: A flow-diversion effect using overlapped double-layer micromesh stents can result in thrombosis and healing of giant ECAAs.

Fibrinogen Aα-chain amyloidosis outbreaks in Japanese squirrels (Sciurus lis): a potential disease model.

Fibrinogen Aα-chain amyloidosis is a hereditary systemic amyloidosis characterized by glomerular amyloid depositions, which are derived from the fibrinogen Aα-chain variant in humans. Despite its unique pathology, the pathogenic mechanisms of this disease are only partially understood. This is in part because comparative pathological studies on fibrinogen Aα-chain amyloidosis are currently unavailable as there is a lack of reported cases in animals other than humans. In this study, mass spectrometry-based proteomic analyses of Japanese squirrels (Sciurus lis) that died in five Japanese zoos showed that they developed glomerular-associated fibrinogen Aα-chain amyloidosis with an extremely high incidence rate (29/38 cases, 76.3%). The condition was found to be age-dependent in the Japanese squirrels, with 89% of individuals over 4 years of age affected. Mass spectrometry revealed that the C-terminal region of the fibrinogen Aα-chain was involved in amyloidogenesis in Japanese squirrels as well as humans. No gene variations were identified between amyloid-positive and amyloid-negative squirrels, which contrasted with the available data for humans. The results indicate that fibrinogen Aα-chain amyloidosis is a senile amyloidosis in Japanese squirrels. The results have also provided comparative pathological support that the amyloidogenic C-terminal region of the fibrinogen Aα-chain is involved in the characteristic glomerular pathology, regardless of the animal species. This study elucidates the potential causes of death in Japanese squirrels and will contribute to future comparative pathological studies of fibrinogen Aα-chain amyloidosis. © 2023 The Pathological Society of Great Britain and Ireland.

X-linked Agammaglobulinemia Diagnosed Following Bezold's Abscess: A Case Report.

Bezold's abscess is an extracranial complication of otitis media, in which a cervical abscess forms from the mastoid process through an ostial fistula, and is a rare condition in recent years. In this study, we experienced a X-linked agammaglobulinemia, which was discovered due to Bezold's abscess. Case: A 12-year-old boy suffering from recurrent right suppurative otitis media for three months was treated with tympanostomy and oral antibacterial therapy at a local otorhinolaryngology clinic. The patient visited the clinic due to a recurrence of symptoms. CT showed bony defects in the cortical bone and mastoid process of the lateral side of the right mastoid cell. The patient was referred to our hospital, admitted the same day and underwent emergency surgery. Intraoperative findings led to the diagnosis of acute mastoiditis and Bezold's abscess c aused b y mastoiditis spreading to the s ternocleidomastoid muscle. After drainage and administration of ABPC/SBT, the abscess disappeared, and the patient's general condition improved. Subsequently, a blood typing test performed on admission suggested the influence of low immunoglobulin levels. A close examination by the pediatric department led to a diagnosis of X-linked agammaglobulinemia. As a result, the patient receives regular immunoglobulin therapy and has been free of infection, including Bezold's abscess. CONCLUSIONS: In the case of recurrent otitis media and rare infections, congenital immune abnormalities should be considered.