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1.
Elife ; 122023 02 15.
Article in English | MEDLINE | ID: mdl-36790165

ABSTRACT

Centrosomes are orbited by centriolar satellites, dynamic multiprotein assemblies nucleated by Pericentriolar material 1 (PCM1). To study the requirement for centriolar satellites, we generated mice lacking PCM1, a crucial component of satellites. Pcm1-/- mice display partially penetrant perinatal lethality with survivors exhibiting hydrocephalus, oligospermia, and cerebellar hypoplasia, and variably expressive phenotypes such as hydronephrosis. As many of these phenotypes have been observed in human ciliopathies and satellites are implicated in cilia biology, we investigated whether cilia were affected. PCM1 was dispensable for ciliogenesis in many cell types, whereas Pcm1-/- multiciliated ependymal cells and human PCM1-/- retinal pigmented epithelial 1 (RPE1) cells showed reduced ciliogenesis. PCM1-/- RPE1 cells displayed reduced docking of the mother centriole to the ciliary vesicle and removal of CP110 and CEP97 from the distal mother centriole, indicating compromised early ciliogenesis. Similarly, Pcm1-/- ependymal cells exhibited reduced removal of CP110 from basal bodies in vivo. We propose that PCM1 and centriolar satellites facilitate efficient trafficking of proteins to and from centrioles, including the departure of CP110 and CEP97 to initiate ciliogenesis, and that the threshold to trigger ciliogenesis differs between cell types.


Subject(s)
Centrioles , Cilia , Animals , Female , Humans , Mice , Cell Cycle Proteins/genetics , Cell Cycle Proteins/metabolism , Centrioles/metabolism , Centrosome/metabolism , Cilia/metabolism , Cytoskeletal Proteins/metabolism
2.
J Vet Emerg Crit Care (San Antonio) ; 32(2): 267-273, 2022 Mar.
Article in English | MEDLINE | ID: mdl-35080123

ABSTRACT

OBJECTIVE: To describe the clinical presentation, diagnostic imaging, management, and follow-up of a dog that presented with sequential, bilateral, spontaneous, subcapsular and perirenal hemorrhage (Wunderlich syndrome) due to bilateral renal arterial malformations. CASE SUMMARY: A 9-year-old intact male Field Spaniel presented for acute onset abdominal pain following a possible syncopal episode. Abdominal ultrasonography, contrast-enhanced ultrasound, and computed tomography (CT) revealed right-sided perirenal hemorrhage that extended into the peritoneum. The dog was discharged following stabilization with analgesia, fluid therapy, and tranexamic acid. One month later, the patient presented with identical clinical signs. A CT scan at this stage revealed bilateral kidney infarcts with new left-sided perirenal hemorrhage. Abdominal ultrasound and contrast-enhanced ultrasound were repeated and showed similar findings. Follow up with four-dimensional CT angiography revealed bilateral renal arterial malformations, likely causing spontaneous renal hemorrhage and secondary subcapsular hematomas , retroperitoneal and peritoneal hemorrhage. UNIQUE INFORMATION PROVIDED: This is the first reported case of both bilateral renal arterial malformations and bilateral spontaneous subcapsular and perirenal hemorrhage in the absence of neoplasia, coagulopathy, or trauma in a dog. Advanced imaging modalities, including selective angiography, were required to diagnose this condition successfully. Arterial malformations should be considered as a differential diagnosis in cases of hemoretroperitoneum. Nephrectomy due to perirenal hemorrhage should be cautiously considered, especially in the absence of angiography, because of the sequential bilateral nature of this case. Conservative management in this case resulted in a good long-term outcome 10 months later.


Subject(s)
Dog Diseases , Kidney Diseases , Animals , Dog Diseases/diagnostic imaging , Dog Diseases/therapy , Dogs , Hemoperitoneum/complications , Hemoperitoneum/veterinary , Kidney/diagnostic imaging , Kidney Diseases/diagnostic imaging , Kidney Diseases/etiology , Kidney Diseases/veterinary , Male , Tomography, X-Ray Computed/veterinary , Ultrasonography
3.
Top Companion Anim Med ; 42: 100490, 2021 Mar.
Article in English | MEDLINE | ID: mdl-33115690

ABSTRACT

A 6-year-old, male-neutered, domestic short-haired cat was referred for further management of a 3-month history of uncontrolled diabetes mellitus. The cat visited the hospital on 3 occasions during a 3-week time period. Hyperglycemia was documented at all visits. The cat initially presented with evidence of hypovolemia, cranial abdominal pain, and dehydration. Moderate hyperglycemia, mild ketonemia, and severe hypokalemia were documented. A 3 × 2 cm skin lesion with associated alopecia and erythema was first noticed at a routine follow-up examination (visit 2) 1 week later. A diagnosis of diabetic ketoacidosis was made 6 days later. The previously identified skin lesion now measured 6 × 2.5 cm. Two episodes of respiratory distress were identified at this visit, with no evidence of cardiac or pulmonary pathology. The cat developed a moderate anemia (packed cell volume 16 %, total solids 7.9 g/dL) on the fifth day of hospitalization. Fluid therapy, electrolyte supplementation, regular insulin, anti-emetic, and analgesia medications were administered during visits 1 and 3. Due to development of anemia, suspected pulmonary thromboembolism events and progression of skin lesions, euthanasia was elected. A diagnosis of cutaneous vasculopathy with secondary ischemic necrosis was made postmortem and pulmonary thromboembolism was confirmed. To the authors' knowledge, this is the first report of cutaneous vasculopathy and pulmonary thromboembolism in a cat with confirmed diabetes mellitus, warranting further research to assess if hypercoagulability is common in this patient population, as routine thromboprophylaxis and anticoagulation may be potentially indicated.


Subject(s)
Diabetic Ketoacidosis/diagnosis , Pulmonary Embolism , Venous Thromboembolism , Animals , Anticoagulants , Cat Diseases , Cats , Diabetic Ketoacidosis/veterinary , Male , Pulmonary Embolism/veterinary , Venous Thromboembolism/veterinary
5.
Vet Radiol Ultrasound ; 60(2): 136-144, 2019 Mar.
Article in English | MEDLINE | ID: mdl-30311329

ABSTRACT

Gastric pneumatosis is an imaging finding defined as the presence of gas foci in the gastric wall. In humans, this imaging feature can result from one of two separate clinical entities: life-threatening emphysematous gastritis or clinically benign gastric emphysema. This retrospective case series study describes the clinical and imaging features in five animals diagnosed with spontaneous gastric pneumatosis without gastric dilatation-volvulus. Three canine and two feline cases of spontaneous gastric pneumatosis were identified on radiographic and ultrasonographic examinations. In addition to gastric pneumatosis, one dog and two cats presented concomitant systemic signs such as lethargy, hematemesis, anemia, or leukocytosis. Two dogs remained asymptomatic or presented mild gastrointestinal signs. Portal gas was described in two dogs and one cat, and pneumoperitoneum in one dog. These features were not considered clinically significant. The dog and two cats with systemic signs were euthanized due to clinical deterioration and diagnosed with emphysematous gastritis. The gastric pneumatosis of both dogs without systemic signs resolved while on medical management without antibiotic therapy. These latter cases were interpreted as consistent with gastric emphysema. Findings from the current study indicated that gastric pneumatosis can occur without gastric dilatation-volvulus in cats and dogs and that a combination of clinical and imaging characteristics may help to differentiate between potentially life-threatening emphysematous gastritis and relatively benign gastric emphysema. More studies are needed to determine the etiology and risk factors associated with these conditions.


Subject(s)
Cat Diseases/diagnosis , Dog Diseases/diagnosis , Emphysema/veterinary , Gastritis/veterinary , Stomach Diseases/veterinary , Animals , Cat Diseases/diagnostic imaging , Cat Diseases/etiology , Cats , Dog Diseases/diagnostic imaging , Dog Diseases/etiology , Dogs , Emphysema/diagnosis , Emphysema/diagnostic imaging , Emphysema/etiology , Female , Gastritis/diagnosis , Gastritis/diagnostic imaging , Gastritis/etiology , Male , Retrospective Studies , Stomach Diseases/diagnosis , Stomach Diseases/diagnostic imaging , Stomach Diseases/etiology
6.
J Pediatr (Rio J) ; 79(1): 49-54, 2003.
Article in Portuguese | MEDLINE | ID: mdl-12973509

ABSTRACT

OBJECTIVE: The purpose of this study was to compare the effectiveness of a pre-thickened infant formula (Nan AR) with a conventional homemade formula in the reduction of regurgitation and vomiting in infants with gastroesophageal reflux.\par METHODS: A hundred children, under 12 months, not exclusively breast feeding, were select for the study. Forty-eight were treated with conventional formula with starch and 52 with pre-thickened infant formula.\par RESULTS: There was no statistically significant difference between the two groups in the improvement or cure of symptoms, each treatment having been effective.\par CONCLUSION: Thickened formulas, pre-thickened or home-thickened, have shown the same efficacy in the management of gastroesophageal reflux in children.


Subject(s)
Gastroesophageal Reflux/diet therapy , Infant Formula , Case-Control Studies , Humans , Infant , Infant, Newborn , Prospective Studies
7.
J. pediatr. (Rio J.) ; 79(1): 49-54, jan.-fev. 2003. tab
Article in Portuguese | LILACS | ID: lil-336707

ABSTRACT

Objetivo: o presente estudo foi realizado com o objetivo de comparar a eficácia de uma fórmula infantil pré-espessada (Nan AR) com fórmula convencional, de espessamento caseiro, com amido de milho, na redução de episódios de regurgitações e vômitos de lactentes com refluxo gastroesofágico (RGE). Métodos: foram estudadas 100 crianças, menores de 12 meses de idade, que não faziam uso de leite materno exclusivo. Por sorteio, 48 receberam fórmula convencional, e 52 receberam fórmula infantil pré-espessada (Nan AR), com comparação dos resultados clínicos após 3 meses. Resultados: não houve diferença estatisticamente significativa na melhora ou cura dos sintomas entre os dois grupos, tendo sido ambos os tratamentos eficazes. Conclusões: o espessamento dietético, seja com fórmulas infantis especiais, seja com medidas caseiras, tem eficácia semelhante no tratamento do refluxo gastroesofágico do lactente


Subject(s)
Humans , Male , Female , Infant, Newborn , Infant , Gastroesophageal Reflux/diet therapy
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