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Facial distortion due to chronic inflammation of unknown cause in a cat.

ABSTRACT: Case summary:An 8-year-old neutered male indoor cat was presented for evaluation of a year-long history of swelling over the bridge of the nose that extended from the subcutaneous tissue of the right upper eyelid to the dorsum of the skull. Intermittent regression of the mass lesion was reported with antibiotic or corticosteroid therapy; however, progressive swelling, malaise and hiding behavior persisted. CT revealed an aggressive osteolytic mass lesion in the right and left nasal cavities and extending into the frontal sinuses. Rhinoscopy using a 2.8?mm rigid telescope revealed somewhat normal-appearing turbinates rostrally and ventrally on the left side, with turbinate destruction on the right. After obtaining a biopsy from the right side of the nasal cavity, thick material filling the entire nasal cavity was visible caudally and was extracted endoscopically from a rostral approach. Surgical biopsy of the dorsal nasal bridge resulted in protrusion of inspissated material from the incision site. Rhinoscopic exploration revealed that the material extended into both frontal sinuses. Following extensive debridement and medical therapy, marked resolution of facial asymmetry was achieved. Relevance and novel information:Facial distortion is often considered suggestive of a neoplastic process; however, it can also be seen with fungal and mycobacterial infections, and, in this case, an inflammatory condition of unknown etiology. In this cat, aggressive intervention and debridement of necrotic debris resulted in substantial bony remodeling of the skull and return to normal activity levels.

SUBMITTER: Johnson LR

PROVIDER: S-EPMC7498973 | biostudies-literature | 2020 Jul-Dec

REPOSITORIES: biostudies-literature

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Facial distortion due to chronic inflammation of unknown cause in a cat.

Johnson Lynelle R LR Vidal Sarah A SA Brust Kelsey D KD Keel M Kevin MK Steffey Michele A MA

JFMS open reports 20200701 2

<h4>Case summary</h4>An 8-year-old neutered male indoor cat was presented for evaluation of a year-long history of swelling over the bridge of the nose that extended from the subcutaneous tissue of the right upper eyelid to the dorsum of the skull. Intermittent regression of the mass lesion was reported with antibiotic or corticosteroid therapy; however, progressive swelling, malaise and hiding behavior persisted. CT revealed an aggressive osteolytic mass lesion in the right and left nasal cavit ...[more]

PMID: 32983553

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Project description:BackgroundChronic Kidney Disease of unknown cause (CKDu) a disease of exclusion, and remains unexplained in various parts of the world, including India. Previous studies have reported mixed findings about the role of heavy metals or agrochemicals in CKDu. These studies compared CKDu with healthy controls but lacked subjects with CKD as controls. The purpose of this study was to test the hypothesis whether heavy metals, i.e. Arsenic (As), Cadmium (Cd), Lead (Pb), and Chromium (Cr) are associated with CKDu, in central India.MethodsThe study was conducted in a case-control manner at a tertiary care hospital. CKDu cases (n = 60) were compared with CKD (n = 62) and healthy subjects (n = 54). Blood and urine levels of As, Cd, Pb, and Cr were measured by Inductively Coupled Plasma- Optical Emission Spectrometry. Pesticide use, painkillers, smoking, and alcohol addiction were also evaluated. The median blood and urine metal levels were compared among the groups by the Kruskal-Wallis rank sum test.ResultsCKDu had significantly higher pesticide and surface water usage as a source of drinking water. Blood As levels (median, IQR) were significantly higher in CKDu 91.97 (1.3-132.7) µg/L compared to CKD 4.5 (0.0-58.8) µg/L and healthy subjects 39.01 (4.8-67.4) µg/L (p < 0.001) On multinominal regression age and sex adjusted blood As was independently associated with CKDu[ OR 1.013 (95%CI 1.003-1.024) P < .05].Blood and urinary Cd, Pb, and Cr were higher in CKD compared to CKDu (p > .05). Urinary Cd, Pb and Cr were undetectable in healthy subjects and were significantly higher in CKDu and CKD compared to healthy subjects (P = < 0.001). There was a significant correlation of Cd, Pb and Cr in blood and urine with each other in CKDu and CKD subjects as compared to healthy subjects. Surface water use also associated with CKDu [OR 3.178 (95%CI 1.029-9.818) p < .05).ConclusionThe study showed an independent association of age and sex adjusted blood As with CKDu in this Indian cohort. Subjects with renal dysfunction (CKDu and CKD) were found to have significantly higher metal burden of Pb, Cd, As, and Cr as compared to healthy controls. CKDu subjects had significantly higher pesticide and surface water usage, which may be the source of differential As exposure in these subjects.

Project description:Abstract Background A 68-year-old woman with hypogammaglobulinemia and prior treated lymphoma presented with fever and abdominal pain. Evaluation revealed numerous nodules in the lung, eye, brain, and liver (Figure 1). Initial lung and liver biopsies showed necrotizing granulomas with no organisms and negative serology and cultures. After progression while on broad-spectrum antibiotics for 4 months, an open liver biopsy revealed numerous nodular lesions and a mass made up of multifocal coalescing cystic lesions. The mass consisted of a degenerating 3-layered membrane without scoleces characterized by a wavy protuberant ciliated eosinophilic outer layer, subjacent degenerating cells with pyknotic nuclei, and loose connective tissue suggestive of a bladder wall and calcareous corpuscles in a matrix of granulomatous inflammation with areas of necrosis (Figure 2). This was diagnostic of disseminated metacestodes (larval stage) of a cestode (tapeworm). Treatment with praziquantel and albendazole led to improvement of symptoms and lesions. Disseminated cestode infections other than due to Echinococcus species are rare in humans. Sequencing was pursued due to the unusual findings. Methods DNA was extracted from liver tissue followed by targeted amplification of the cestode COX1 gene. PCR products confirmed to be 134 bp, as expected for a cestode COX1 gene, then inserted into a 2.1 Topo vector and cloned. Five separate isolates were sequenced, and 4 were interpretable. The 129-bp consensus sequence is shown in Figure 3. Basic Local Alignment Search Tool (NCBI BLAST) was used to find highly similar sequences. Results The sequence matched to Versteria sp. (T. mustelae) COX1 gene from a mink in Oregon (accession KT223034) with 98% identity. Conclusion Metacestodes have the propensity to proliferate and rarely disseminate. There is one reported case of Versteria sp. causing a lethal disseminated infection of an orangutan. This is the first report of a Versteria sp. disseminated infection in a human and is singular because the patient survived. The patient likely accidentally ingested ova shed from a tapeworm in a mink or similar mammalian host. Histopathologic assessment is crucial in diagnosing cestode infection. COX1 gene sequencing is useful for cestode identification. Disclosures All authors: No reported disclosures.

Project description:IntroductionAicardi-Goutières (AGS) is a rare immune dysregulated disease due to mutations in TREX1, RNASEH2A, RNASEH2B, RNASEH2C, SAMHD1, ADAR1, or IFIH1. Clinical features include basal ganglia calcifications, white matter abnormalities, and cerebral atrophy. Severe systemic inflammation and chronic kidney disease (CKD) are extremely rare in AGS. Herein, we report a patient presenting with systemic inflammation and CKD to broaden the clinical phenotype spectrum of the RNASEH2B defect.MethodsAll testing and molecular genetic analysis were performed after obtaining the informed consent of the parents. Demographic, clinical, and laboratory findings were abstracted from outpatient and inpatient encounters. Cerebral magnetic resonance imaging (MRI), computed tomography (CT) scans, and renal biopsy histopathology reports were reviewed and summarized. Whole exome sequencing (WES) was performed on peripheral blood cells. After exposure to cGAMP in vitro for 24?h, mRNA expression of 12 IFN-stimulated cytokine genes in PBMCs was assessed. Serum cytokine levels were detected by Milliplex.ResultsA 11-year-old girl presented with recurrent aseptic fever, arthritis, chilblains, failure to thrive, mild hearing loss, and neurological manifestations. Laboratory and immunologic findings demonstrated lymphopenia, low complement levels, positive autoantibodies, elevated levels of acute-phase reactants and inflammatory cytokines. Cerebral imaging showed cerebral atrophy, white matter abnormalities, and intracranial calcification. Renal biopsy showed glomerular sclerosis in 3 of 14 glomeruli, infiltration of lymphocytes and other mononuclear cells. WES revealed a homozygous and heterozygous mutations in RNASEH2B. Over-expression of IFN-stimulated cytokine genes was observed, including IFI44, IFI27, IFIT1, IFIT2, IFIT3, ISG15, OAS1, and SIGLEC1.ConclusionsTo date, only two cases with AGS have been reported to have renal disease. Here, we describe a patient with both homozygous and heterozygous variants in RNASEH2B, presenting with neurological manifestations, persistently systemic autoinflammation, and CKD. CKD has never been reported in patients with AGS due to the RNASEH2B defect.Trial registrationNot applicable; this was a retrospective study.

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Facial distortion due to chronic inflammation of unknown cause in a cat.

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Facial distortion due to chronic inflammation of unknown cause in a cat.

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