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1.
Artigo em Russo | MEDLINE | ID: mdl-33560616

RESUMO

One of the causes of syringomyelia is arachnopathy following an infectious or non-infectious inflammation of the arachnoid membrane. It is extremely rare that adhesive arachnoiditis develops within the occipital cistern and impairs cerebrospinal fluid flow from the foramen of Magendie, along posterior cerebellar surface and into spinal subarachnoid space. These process result syringomyelia. OBJECTIVE: To evaluate postoperative outcomes in patients with syringomyelia following arachnopathy at the craniovertebral level. MATERIAL AND METHODS: A retrospective analysis included 27 patients with syringomyelia and arachnopathy within the occipital cistern for the period from 2013 to 2018. Eight patients (29.6%) underwent primary surgery. In this group, 2 patients had arachnopathy following post-traumatic subarachnoid hemorrhage, 1 - after non-traumatic subarachnoid hemorrhage in posterior cranial fossa, 2 - after bacterial meningitis, 3 - the cause was unclear. Nineteenth patients underwent redo surgery after previous procedures on posterior cranial fossa (tumor resection - 3 patients, suboccipital decompression for Chiari malformation - 16 cases). High-resolution MRI with special protocols for analysis of CSF circulation and mobility of brain structures was used to diagnose arachnopathy (phase contrast ECG-synchronized MRI, TrueFISP). RESULTS: Surgeries were aimed at mobilization of cerebellar tonsils and spinal cord and restoration of CSF circulation at the craniovertebral level. One year later, 13 (48.1%) patients noted improvement in their condition, 8 (29.6%) patients - clinical stabilization. In most cases, stabilization or improvement of sensory and coordination disorders. Syringomyelia symptoms progressed in 6 (22.2%) patients. Mean preoperative mJOA score was 11.86±1.24, in a year after surgery - 14.17±1.19. According to MRI data after 1 year, syringomyelia disappeared in 2 patients (7.4%), reduced in 13 (48.1%) cases, remained the same in 9 (33.3%) cases and deteriorated in 3 (11.1%) patients. Vaquero index reduced from 45.5% to 21.6 within a year after surgery. Early postoperative complications occurred in 3 (11.1%) patients: 1 (3.7%) had hydrocephalus and 2 (7.4%) had aseptic meningitis. CONCLUSION: Modern diagnostic and surgical methods for syringomyelia ensure favorable outcomes in 77.7% of cases.


Assuntos
Malformação de Arnold-Chiari , Siringomielia , Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/diagnóstico por imagem , Malformação de Arnold-Chiari/cirurgia , Fossa Craniana Posterior/cirurgia , Descompressão Cirúrgica , Humanos , Imagem por Ressonância Magnética , Estudos Retrospectivos , Siringomielia/diagnóstico por imagem , Siringomielia/etiologia , Siringomielia/cirurgia , Resultado do Tratamento
3.
Praxis (Bern 1994) ; 109(10): 806-811, 2020 Aug.
Artigo em Alemão | MEDLINE | ID: mdl-32752959

RESUMO

Migraine? Arnold Chiari Malformation? Or Just a Migraine? Abstract. In a young woman with a long-time headache in the sense of a migraine, a neurological reassessment including new findings of the existing MRI images of the skull resulted in the diagnosis of a Chiari malformation type 1. After successful operation of the MRI findings, the headache decreased significantly. However, since minor headaches of a different quality persist, a migraine plus malformation as an incidental finding are most likely.


Assuntos
Malformação de Arnold-Chiari , Transtornos de Enxaqueca , Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/diagnóstico por imagem , Osso e Ossos , Feminino , Cefaleia , Humanos , Imagem por Ressonância Magnética , Transtornos de Enxaqueca/complicações , Transtornos de Enxaqueca/diagnóstico por imagem
4.
PLoS One ; 15(7): e0236357, 2020.
Artigo em Inglês | MEDLINE | ID: mdl-32687509

RESUMO

Adult growth hormone deficiency (GHD) is being increasingly recognized to cause premature mortality exacerbated by oxidative stress. A case-control observational study has been performed with the primary objective of evaluating new parameters of oxidative stress and macromolecular damage in adult GHD subjects: serum nitrotryptophan; Total Antioxidant Capacity expressed as LAG time; urinary hexanoil-lysine; urinary dityrosine and urinary 8-OH-deoxyguanosine. GHD was diagnosed using Growth Hormone-Releasing Hormone 50µg iv+arginine 0,5 g/Kg test, with a peak GH response <9 µg /L when BMI was <30 kg/m2 or <4 µg/L when BMI was >30 kg/m2. Patients affected by adult GHD were divided into three groups, total GHD (n = 26), partial GHD (n = 25), and controls (n = 29). Total Antioxidant Capacity, metabolic and hormonal parameters have been determined in separate plasma samples; nitrotryptophan in serum samples; hexanoil-lysine, dityrosine, 8-OH-deoxyguanosine in urine samples. Assessment of hexanoil-lysine exhibited a trend to increase in comparing total GHD vs partial and controls, although not significant. Values of 8-OH-deoxyguanosine did not significantly differ among the three groups. Significant lower levels of dityrosine in partial GHD vs total and controls were found. No significant difference in nitrotriptophan serum levels was found, while significantly greater values of Total Antioxidant Capacity were showed in total and partial GHD vs controls. Thus, our result confirm that oxidative stress is increased both in partial and total adult GHD. The lack of compensation by antioxidants in total GHD may be connected to the complications associated to this rare disorder.


Assuntos
Antioxidantes/análise , Hormônio do Crescimento Humano/deficiência , Hipopituitarismo/metabolismo , Síndrome Metabólica/metabolismo , Estresse Oxidativo/fisiologia , 8-Hidroxi-2'-Desoxiguanosina/urina , Adulto , Malformação de Arnold-Chiari/sangue , Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/metabolismo , Biomarcadores/sangue , Biomarcadores/urina , Estudos de Casos e Controles , Síndrome da Sela Vazia/sangue , Síndrome da Sela Vazia/complicações , Síndrome da Sela Vazia/metabolismo , Feminino , Humanos , Hipopituitarismo/sangue , Hipopituitarismo/etiologia , Hipopituitarismo/urina , Peroxidação de Lipídeos , Lisina/sangue , Masculino , Síndrome Metabólica/sangue , Síndrome Metabólica/etiologia , Síndrome Metabólica/urina , Pessoa de Meia-Idade , Triptofano/análogos & derivados , Triptofano/sangue , Tirosina/análogos & derivados , Tirosina/urina
5.
J Clin Neurosci ; 77: 1-7, 2020 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-32414621

RESUMO

Chiari Malformation type 1 (CM-I) is congenital or an acquired anomaly of the hind brain; develops when the cerebellar tonsils recede downwards below the foramen magnum. Recurrent post tussive suboccipital headache is the common presentation in a pregnant woman and the diagnosis is usually missed or delayed due to lack of formal understanding of this neurological pathology. Much has been written regarding presentation, morphology and the treatment of CM-I; however, little is known when the etiology is acquired or an iatrogenic in its evolution. Similarly, unknown is the progression of CM-I (diagnosed or undiagnosed) in pregnancy. The objective of this study is to elucidate the causes of progression of CM-I in pregnancy, and how this can be avoided. A detailed literature review has been conducted to find the case reports or case studies on association of CM-I in pregnancy; therefore, the risk factors regarding the progression have been sought. There is a lack of literature on timing, mode of anesthesia, and the management of CM-I. Moreover, authors have sought a questionnaire to screen these patients at pre-conception, intrapartum visits if, the initial diagnosis is delayed. Crucial points of concern including but not limited to the diagnosis, pre-conception counseling, timing of intervention during pregnancy, and mode of anesthesia, have been discussed in detail. In summary, a formal management algorithm has been proposed to avoid the rapid progression of this complex neurological pathology especially, in women of child bearing age and/or during pregnancy.


Assuntos
Malformação de Arnold-Chiari/diagnóstico por imagem , Progressão da Doença , Complicações na Gravidez/diagnóstico por imagem , Adulto , Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/terapia , Tosse/complicações , Tosse/diagnóstico por imagem , Tosse/terapia , Parto Obstétrico/métodos , Feminino , Forame Magno/diagnóstico por imagem , Transtornos da Cefaleia Primários/complicações , Transtornos da Cefaleia Primários/diagnóstico por imagem , Transtornos da Cefaleia Primários/terapia , Humanos , Imagem por Ressonância Magnética/métodos , Gravidez , Complicações na Gravidez/terapia , Fatores de Risco
6.
World Neurosurg ; 139: 70-74, 2020 07.
Artigo em Inglês | MEDLINE | ID: mdl-32298820

RESUMO

BACKGROUND: Chiari 1 malformation is a structural abnormality of the hindbrain and posterior fossa characterized by herniation of the cerebellar tonsils through the foramen magnum. Although asymptomatic in some cases, hindbrain herniation can be associated with disruption of cerebrospinal fluid flow dynamics at the craniovertebral junction and syrinx formation, leading to symptoms. Foramen magnum decompression with or without duraplasty has been the most commonly performed surgical procedure in the management of this condition. The management of syringomyelia associated with Chiari malformation is more challenging and controversial. Although the associated syrinx can significantly improve after craniovertebral decompression and restoration of cerebrospinal fluid flow, in some cases, it will persist despite decompressive surgery and could even continue to enlarge. CASE DESCRIPTION: We have described the case of a 4-year-old boy with non-craniosynostotic Chiari malformation and extensive cervical syrinx who, despite foramen magnum decompression and further revision, continued to deteriorate clinically and radiologically. Posterior calvarial augmentation was performed as a salvage procedure, with resolution of the tonsillar herniation and syrinx. CONCLUSIONS: Posterior calvarial augmentation is a viable option for patients with Chiari malformation refractory to foramen magnum decompression.


Assuntos
Malformação de Arnold-Chiari/cirurgia , Forame Magno/cirurgia , Osso Occipital/cirurgia , Osso Parietal/cirurgia , Procedimentos Cirúrgicos Reconstrutivos/métodos , Terapia de Salvação/métodos , Siringomielia/cirurgia , Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/diagnóstico por imagem , Cerebelo/cirurgia , Vértebras Cervicais , Pré-Escolar , Descompressão Cirúrgica , Progressão da Doença , Forame Magno/diagnóstico por imagem , Humanos , Imageamento Tridimensional , Laminectomia , Masculino , Osso Occipital/diagnóstico por imagem , Osso Parietal/diagnóstico por imagem , Escoliose/etiologia , Crânio/diagnóstico por imagem , Crânio/cirurgia , Siringomielia/complicações , Siringomielia/diagnóstico por imagem , Vértebras Torácicas , Tomografia Computadorizada por Raios X
8.
World Neurosurg ; 136: 454-461.e1, 2020 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-32204297

RESUMO

BACKGROUND: Chiari malformation (CM) is often comorbid with syringomyelia. The treatment of CM via posterior fossa decompression (PFD) may not improve syringomyelia in up to 40% of patients, based on historical cohorts. Management of these patients is problematic, as both reoperation and syrinx shunting have high failure rates in the long term. METHODS: We retrospectively reviewed our cases in which patients with CM type 1 or 1.5 and syringomyelia underwent PFD without postoperative improvement in syringomyelia. Symptomatology and radiographic measurements were collected at presentation and on the first and latest available postoperative scans and analyzed. We present 2 cases to illustrate the challenges in the management of these patients. RESULTS: Our cohort consisted of 48 consecutive patients with CM and syringomyelia who underwent PFD. Of these, 41 patients had postoperative improvement in or resolution of syringomyelia. We subsequently studied the cohort of 7 patients who underwent PFD with (n = 5) or without (n = 2) durotomy and demonstrated worsening of syringomyelia following surgery. This cohort had mean (±SEM) preoperative syrinx area of 23.9 ± 10.0 mm2. Postoperatively, the mean syrinx area increased to 40.5 ± 9.6 mm2 and 57.3 ± 12.5 mm2 on the first and latest postoperative scans available (P = 0.02), for an increase of 106.9% ± 94.4% and 186.0% ± 107.4% (P = 0.04). Presenting symptoms included occipital headache, paresthesias, visual deterioration, and paraspinal pain. On last follow-up (mean 13.9 ± 4.9 months), the majority of symptoms were resolved in this cohort, despite persistence of syringomyelia. CONCLUSIONS: In this small cohort of unique patients, syrinx resolution was not achieved via decompression surgery. Despite "radiographic failure," good symptom control was achieved, with most patients remaining or becoming asymptomatic postoperatively, thus supporting our rationale for what has largely been a conservative approach in this population.


Assuntos
Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/cirurgia , Fossa Craniana Posterior/cirurgia , Descompressão Cirúrgica/métodos , Procedimentos Neurocirúrgicos/métodos , Complicações Pós-Operatórias/etiologia , Complicações Pós-Operatórias/terapia , Siringomielia/etiologia , Siringomielia/terapia , Adolescente , Adulto , Criança , Pré-Escolar , Estudos de Coortes , Feminino , Cefaleia/etiologia , Humanos , Imagem por Ressonância Magnética , Masculino , Pessoa de Meia-Idade , Parestesia/etiologia , Estudos Retrospectivos , Resultado do Tratamento , Adulto Jovem
9.
World Neurosurg ; 137: 174-177, 2020 05.
Artigo em Inglês | MEDLINE | ID: mdl-32028005

RESUMO

BACKGROUND: Chiari I malformation (CIM) is a disorder characterized by caudal displacement of the cerebellar tonsils below the foramen magnum. It is often associated with syringomyelia and occasionally with hydrocephalus. CIM is commonly treated by posterior fossa decompression with or without removal of the posterior arch of C1 and duraplasty, but the treatment for infants with symptomatic CIM is not well established. We present a case of symptomatic CIM in an infant that was successfully treated with a ventriculoperitoneal shunt (VPS) and discuss the importance of the pathophysiology in management decisions. CASE DESCRIPTION: A 6-month-old male with a CIM and a cervicothoracic syrinx presented with stridor, lower cranial nerve dysfunction, and increased tone that worsened with crying. Magnetic resonance imaging studies revealed cerebellar tonsillar displacement extending to the level of C3-C4 with a syrinx extending from C4 to T4. In addition, there was compression of the cervicomedullary junction, fourth ventricular outflow obstruction, and obstructive hydrocephalus. The decision was made to place a ventriculoperitoneal shunt (VPS) instead of performing decompressive surgery as the initial treatment intervention. The infant had significant symptomatic relief at 6-, 9-, and 12-month follow-ups. Postoperative magnetic resonance imaging at 6-month follow-up revealed resolution of the syrinx and ventriculomegaly and ascent of the cerebellar tonsils. CONCLUSIONS: Ventriculoperitoneal shunting alone was successfully used to treat an infant with concurrent CIM, syrinx, and hydrocephalus. This case underscores not only the importance of hydrocephalus as the pathogenesis of CIM in some cases but also the possibility of avoiding the morbidity of decompressive surgery in infants.


Assuntos
Malformação de Arnold-Chiari/cirurgia , Hidrocefalia/cirurgia , Siringomielia/cirurgia , Derivação Ventriculoperitoneal/métodos , Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/diagnóstico por imagem , Atlas Cervical/cirurgia , Vértebras Cervicais , Descompressão Cirúrgica , Humanos , Hidrocefalia/diagnóstico por imagem , Hidrocefalia/etiologia , Lactente , Laminectomia , Imagem por Ressonância Magnética , Masculino , Procedimentos Neurocirúrgicos , Siringomielia/complicações , Siringomielia/diagnóstico por imagem , Vértebras Torácicas , Resultado do Tratamento
10.
World Neurosurg ; 138: e95-e105, 2020 06.
Artigo em Inglês | MEDLINE | ID: mdl-32059967

RESUMO

OBJECTIVE: Foramen magnum decompression (FMD) remains the first-line treatment for Chiari malformation type I associated with syringomyelia, although consistent approaches are not used. However, there are few reports on a persistent or recurrent syrinx or worsening neurologic symptoms after FMD. METHODS: We retrospectively reviewed medical records of patients who attended Xuanwu Hospital between January 2018 and July 2019 with persistent or recurrent syringomyelia after FMD. RESULTS: All 8 patients had syringomyelia preoperatively with a mean disease course of 43 (range, 3-120) months and underwent FMD. The presenting symptoms improved in 1 patient. There was radiologic evidence of decreased syrinx size in 1 patient. Worsening syringomyelia presented at a median of 65 (range, 7-168) months after the primary FMD. Secondary decompression was performed in all patients. A structural, pathological cause for the first FMD failure was identified in all cases at the time of the second decompression. Symptoms were resolved in all patients after repeat decompression surgery. Radiologically, 6 patients had a decrease in syringomyelia size. The level of the upper segment of the syringomyelia was higher in 7 of our revision patients, among which 6 patients had a decrease in syringomyelia size. The median follow-up duration after the secondary decompression was 11.5 (range, 6-22) months. CONCLUSIONS: Adults with persistent syringomyelia after FMD and the higher level of the upper segment of the syringomyelia often have a surgically remediable structural cause. The beneficial effect of a secondary decompression should be considered and guide the decision-making of patients with Chiari malformation type I-related syringomyelia.


Assuntos
Malformação de Arnold-Chiari/cirurgia , Procedimentos Neurocirúrgicos/métodos , Siringomielia/cirurgia , Adulto , Malformação de Arnold-Chiari/complicações , Descompressão Cirúrgica/métodos , Feminino , Forame Magno/cirurgia , Humanos , Masculino , Pessoa de Meia-Idade , Reoperação , Estudos Retrospectivos , Siringomielia/etiologia , Resultado do Tratamento
11.
World Neurosurg ; 137: 304-309, 2020 05.
Artigo em Inglês | MEDLINE | ID: mdl-32058112

RESUMO

BACKGROUND: Little attention has been given to the retroverted dens within the existing medical literature. However, this finding can have a clinical impact, especially in patients with Chiari malformation type I (CM1), as it can have consequences for further treatment. METHODS: Using standard search engines, we performed a literature review of anatomical, radiologic, and clinical studies as well as pathologic and surgical considerations related to the retroverted dens. Key words for our search included retroverted dens; retroflexed dens; odontoid retroflexion; posterior inclination; and tilted dens. RESULTS: A retroverted dens is most commonly found in the pediatric population in relation to CM1. Research has demonstrated that high degree of dens angulation can result in significant anterior brain stem compression with the need for both anterior and posterior decompression in patients with symptomatic CM1. CONCLUSIONS: A greater degree of dens angulation can lead to neurologic symptoms secondary to spinomedullary compression. Therefore, correct measurements are essential as such findings can influence presurgical planning.


Assuntos
Retroversão Óssea/diagnóstico por imagem , Processo Odontoide/anormalidades , Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/fisiopatologia , Malformação de Arnold-Chiari/cirurgia , Retroversão Óssea/complicações , Retroversão Óssea/fisiopatologia , Tronco Encefálico , Descompressão Cirúrgica , Humanos , Processo Odontoide/anatomia & histologia , Processo Odontoide/crescimento & desenvolvimento , Terminologia como Assunto
12.
Spine Deform ; 8(2): 233-243, 2020 04.
Artigo em Inglês | MEDLINE | ID: mdl-31933098

RESUMO

STUDY DESIGN: Retrospective case series. OBJECTIVES: The objective was to assess the long-term outcomes on scoliosis following Chiari-I (CM-I) decompression in patients with CM-I and syringomyelia (SM). A secondary objective was to identify risk factors of scoliosis progression. BACKGROUND: The association between CM-I with SM and scoliosis is recognized, but it remains unclear if CM-I decompression alters the long-term evolution of scoliosis in patients with associated syringomyelia. METHODS: A retrospective review of children with scoliosis, CM-I, and SM during 1997-2015 was performed. Congenital, syndromic, and neuromuscular scoliosis were excluded. Clinical and radiographic characteristics were recorded at presentation, pre-decompression, after 1-year, and latest follow-up. A scale to measure syringomyelia area on MRI was used to evaluate SM changes post-decompression. RESULTS: 65 children with CM-I, SM, and scoliosis and a mean age of 8.9 years (range 0.7-15.8) were identified. Mean follow-up was 6.9 years (range 2.0-20.4). Atypical curves were present in 28 (43%) children. Thirty-eight patients (58%) underwent decompression before 10 years. Syringomyelia size reduced a mean of 70% after decompression (p < 0.001). Scoliosis improved in 26 (40%), stabilized in 17 (26%), and progressed in 22 (34%) cases. Early spinal fusion was required in 7 (11%) patients after a mean of 0.5 ± 0.37 years and delayed fusion in 16 (25%) patients after 6.0 ± 3.24 years. The remaining 42 (65%) patients were followed for a median of 6.1 years (range 2.0-12.3) without spine instrumentation or fusion. Fusion patients experienced less improvement in curve magnitude 1-year post-decompression (p < 0.001) and had larger curves at presentation (43° vs. 34°; p = 0.004). CONCLUSIONS: Syringomyelia size decreased by 70% after CM-I decompression and scoliosis stabilized or improved in two-thirds of patients. Greater curve improvement within the first year post-decompression and smaller curves at presentation decreased the risk of spinal fusion. Neurosurgical decompression is recommended in children with CM-I, SM, and scoliosis with the potential to treat all three conditions. LEVEL OF EVIDENCE: Level IV.


Assuntos
Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/cirurgia , Descompressão Cirúrgica/métodos , Escoliose/complicações , Escoliose/cirurgia , Siringomielia/complicações , Siringomielia/cirurgia , Adolescente , Criança , Pré-Escolar , Progressão da Doença , Feminino , Humanos , Lactente , Masculino , Estudos Retrospectivos , Risco , Fusão Vertebral/métodos , Fatores de Tempo , Resultado do Tratamento
13.
World Neurosurg ; 133: e259-e266, 2020 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-31513955

RESUMO

OBJECTIVE: How Chiari malformation type I (CM-I) affects posterior fossa brain structures and produces various symptoms remains unclear. The fourth ventricle is surrounded by critical structures required for normal function. The foramen of Magendie can be obstructed in CM-I; therefore, fourth ventricle changes may occur. To test this hypothesis, we assessed fourth ventricle volume in CM-I compared with healthy controls. METHODS: Using our database from 2007-2016, we studied 72 patients with CM-I and 30 age-matched healthy control subjects. Fourth and lateral ventricle volumes and posterior fossa volumes (PFV) were assessed and correlated with clinical signs and symptoms. Statistical analysis was performed. RESULTS: Patients with CM-I had larger fourth ventricle volumes compared with control subjects (1.31 vs. 0.95 mL; P = 0.012). There were no differences in lateral ventricle volume or PFV. CM-I fourth ventricle volume was associated with tonsillar descent (P = 0.030). CM-I fourth ventricle volume variance was larger than healthy controls (F71,29 = 8.33; P < 0.0001). Patients with CM-I with severe signs and symptoms had a significantly larger fourth ventricle than patients with CM-I with mild signs and symptoms (1.565 vs. 1.015 mL; P = 0.0002). CONCLUSIONS: The fourth ventricle can be enlarged in CM-I independent of lateral ventricle size and is associated with greater tonsillar descent. Most importantly, fourth ventricle enlargement was associated with a worse clinical and radiographic presentation independent of PFV. Fourth ventricle enlargement can affect critical structures and may be a mechanism contributing to symptoms unexplained by tonsil descent. Fourth ventricle enlargement is a useful adjunct in assessing CM-I.


Assuntos
Malformação de Arnold-Chiari/patologia , Fossa Craniana Posterior/patologia , Quarto Ventrículo/patologia , Adolescente , Adulto , Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/diagnóstico por imagem , Criança , Pré-Escolar , Fossa Craniana Posterior/diagnóstico por imagem , Dilatação Patológica/diagnóstico por imagem , Dilatação Patológica/patologia , Encefalocele/diagnóstico por imagem , Encefalocele/etiologia , Feminino , Quarto Ventrículo/diagnóstico por imagem , Humanos , Imageamento Tridimensional , Imagem por Ressonância Magnética , Masculino , Pessoa de Meia-Idade , Neuroimagem , Tamanho do Órgão , Estudos Prospectivos , Adulto Jovem
14.
J Vet Intern Med ; 34(1): 237-246, 2020 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-31691386

RESUMO

BACKGROUND: Recent studies including an innovative machine learning technique indicated Chiari-like malformation (CM) is influenced by brachycephalic features. OBJECTIVES: Morphometric analysis of facial anatomy and dysmorphia in CM-associated pain (CM-P) and syringomyelia (SM) in the Cavalier King Charles Spaniel (CKCS). ANIMALS: Sixty-six client-owned CKCS. METHODS: Retrospective study of anonymized T2W sagittal magnetic resonance imaging of 3 clinical groups: (1) 11 without central canal dilation (ccd) or SM (CM-N), (2) 15 with CM-P with no SM or <2 mm ccd (CM-P), and (3) 40 with syrinx width ≥4 mm (SM-S). Morphometric analysis assessed rostral skull flattening and position of the hard and soft palate relative to the cranial base in each clinical group and compared CKCS with and without SM-S. RESULTS: Sixteen of 28 measured variables were associated to SM-S compared to CM-N and CM-P. Of these 6 were common to both groups. Predictive variables determined by discriminant analysis were (1) the ratio of cranial height with cranial length (P < .001 between SM-S and CM-N) and (2) the distance between the cerebrum and the frontal bone (P < .001 between SM-S and CM-P). CM-P had the lowest mean height of the maxillary area. CONCLUSIONS AND CLINICAL IMPORTANCE: CKCS with CM-P and SM-S have cranial brachycephaly with osseous insufficiency in the skull with rostral flattening and increased proximity of the hard and soft palate to the cranial base. Changes are greatest with CM-P. These findings have relevance for understanding disease pathogenesis and for selection of head conformation for breeding purposes.


Assuntos
Malformação de Arnold-Chiari/veterinária , Doenças do Cão/congênito , Face/patologia , Dor/veterinária , Siringomielia/veterinária , Animais , Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/patologia , Doenças do Cão/etiologia , Doenças do Cão/patologia , Cães , Feminino , Masculino , Dor/etiologia , Estudos Retrospectivos , Siringomielia/etiologia
15.
World Neurosurg ; 135: 241-244, 2020 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-31881343

RESUMO

BACKGROUND: To the best of our knowledge, the presence of Chiari formation and Hirayama disease in the same patient has not been previously reported. On the basis of the presented case, we have attempted to identify the common pathogenesis of both of these clinical entities. CASE DESCRIPTION: We have reported a case of a 23-year-old male patient who had presented with complaints of pain in the nape of neck and shoulders of 9 months' duration, weakness and stiffness in all 4 limbs, and wasting and weakness of muscles of both hands of 6 months' duration. The investigations showed Chiari formation and syringomyelia. Additional investigations depicted the characteristic radiological features associated with Hirayama disease. The patient was treated by atlantoaxial and multisegmental subaxial cervical spinal fixation. CONCLUSIONS: Spinal instability seems to be the nodal point of the pathogenesis of both Chiari formation and Hirayama disease.


Assuntos
Malformação de Arnold-Chiari/diagnóstico por imagem , Atrofias Musculares Espinais da Infância/diagnóstico por imagem , Siringomielia/diagnóstico por imagem , Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/cirurgia , Articulação Atlantoaxial/diagnóstico por imagem , Articulação Atlantoaxial/cirurgia , Vértebras Cervicais/diagnóstico por imagem , Vértebras Cervicais/cirurgia , Humanos , Masculino , Fusão Vertebral , Atrofias Musculares Espinais da Infância/complicações , Atrofias Musculares Espinais da Infância/cirurgia , Siringomielia/complicações , Siringomielia/cirurgia , Adulto Jovem
19.
Spine (Phila Pa 1976) ; 44(23): 1653-1660, 2019 Dec 01.
Artigo em Inglês | MEDLINE | ID: mdl-31730571

RESUMO

STUDY DESIGN: Retrospective study. OBJECTIVE: To compare syrinx characteristics, scoliotic parameters, and neurological deficits between Chiari I malformation (CIM) and idiopathic syringomyelia (IS) in the scoliotic population. SUMMARY OF BACKGROUND DATA: CIM and IS are common in neuromuscular scoliosis patients; however, differences in syrinx characteristics, scoliotic parameters, and neurological deficits between CIM and IS are unclear. METHODS: Thirty-six patients with scoliosis secondary to CIM were enrolled retrospectively and matched with 36 IS patients for sex, age, scoliosis classification, and Cobb angle. Information on radiographic features of scoliosis and syrinx and neurological deficits was systematically collected. RESULTS: Sex, age, and coronal, and sagittal scoliosis parameters did not differ between the CIM and IS groups. The CIM group had a longer syrinx (12.9 ±â€Š4.0 vertebral levels vs. 8.7 ±â€Š5.5 vertebral levels, P < 0.001), a higher cranial extent (3.6 ±â€Š2.2 vs. 5.2 ±â€Š3.5, P = 0.027), and a lower caudal extent (15.6 ±â€Š2.9 vs. 13.0 ±â€Š4.6, P = 0.006) than the IS group, despite no differences in syrinx/cord (S/C) ratio or syrinx classification. No differences in neurological deficits were identified between the CIM and IS patients. CONCLUSION: With demographic and scoliotic coronal parameters matched, the CIM patients had a longer syrinx, located at a higher cranial and lower caudal level, compared with the IS group. No significant differences in syrinx S/C ratio, sagittal features of scoliosis, or neurological deficits were detected between the two groups. LEVEL OF EVIDENCE: 3.


Assuntos
Malformação de Arnold-Chiari/diagnóstico por imagem , Imagem por Ressonância Magnética/métodos , Escoliose/diagnóstico por imagem , Siringomielia/diagnóstico por imagem , Adolescente , Adulto , Malformação de Arnold-Chiari/complicações , Criança , Estudos Transversais , Feminino , Humanos , Vértebras Lombares/diagnóstico por imagem , Masculino , Estudos Retrospectivos , Escoliose/etiologia , Siringomielia/complicações , Vértebras Torácicas/diagnóstico por imagem , Adulto Jovem
20.
J Vet Intern Med ; 33(6): 2665-2674, 2019 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-31552689

RESUMO

BACKGROUND: Chiari-like malformation (CM) is a complex malformation of the skull and cranial cervical vertebrae that potentially results in pain and secondary syringomyelia (SM). Chiari-like malformation-associated pain (CM-P) can be challenging to diagnose. We propose a machine learning approach to characterize morphological changes in dogs that may or may not be apparent to human observers. This data-driven approach can remove potential bias (or blindness) that may be produced by a hypothesis-driven expert observer approach. HYPOTHESIS/OBJECTIVES: To understand neuromorphological change and to identify image-based biomarkers in dogs with CM-P and symptomatic SM (SM-S) using a novel machine learning approach, with the aim of increasing the understanding of these disorders. ANIMALS: Thirty-two client-owned Cavalier King Charles Spaniels (CKCSs; 11 controls, 10 CM-P, 11 SM-S). METHODS: Retrospective study using T2-weighted midsagittal Digital Imaging and Communications in Medicine (DICOM) anonymized images, which then were mapped to images of an average clinically normal CKCS reference using Demons image registration. Key deformation features were automatically selected from the resulting deformation maps. A kernelized support vector machine was used for classifying characteristic localized changes in morphology. RESULTS: Candidate biomarkers were identified with receiver operating characteristic curves with area under the curve (AUC) of 0.78 (sensitivity 82%; specificity 69%) for the CM-P biomarkers collectively and an AUC of 0.82 (sensitivity, 93%; specificity, 67%) for the SM-S biomarkers, collectively. CONCLUSIONS AND CLINICAL IMPORTANCE: Machine learning techniques can assist CM/SM diagnosis and facilitate understanding of abnormal morphology location with the potential to be applied to a variety of breeds and conformational diseases.


Assuntos
Malformação de Arnold-Chiari/veterinária , Doenças do Cão/sangue , Aprendizado de Máquina , Dor/veterinária , Siringomielia/veterinária , Animais , Área Sob a Curva , Malformação de Arnold-Chiari/sangue , Malformação de Arnold-Chiari/complicações , Malformação de Arnold-Chiari/diagnóstico por imagem , Biomarcadores , Doenças do Cão/diagnóstico por imagem , Doenças do Cão/patologia , Cães , Feminino , Masculino , Dor/etiologia , Curva ROC , Estudos Retrospectivos , Sensibilidade e Especificidade , Siringomielia/sangue , Siringomielia/complicações , Siringomielia/diagnóstico por imagem
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