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2.
Pan Afr Med J ; 42: 257, 2022.
Artigo em Inglês | MEDLINE | ID: mdl-36338550

RESUMO

Anterior dural tears complicated by cerebrospinal fluid (CSF) leakage with anterior meningocele are rare. Indeed, in the literature, cases are described during anterior arthrodesis, but no cases of post-cervical arthroplasty are described. The management of this type of complication is poorly described and not consensual. We present a case of a patient who underwent cervical arthroplasty complicated by an anterior meningocele at 1 month after the first surgery. Imaging revealed a compressive anterior meningocele in relation to the clinically progressive worsening. Revision surgery consisted of a combination of closure of the gap with a fatty patch covered with a TachoSil patch, followed by reinsertion of a new cervical prosthesis. At the last follow-up at 1 year, the patient showed no residual effects of the complication, and the mobility of the disc prosthesis was not impaired by it. Clinical results of the arthroplasty are also very satisfactory. Although these types of complications are rare, it is important to have a consensus on the management of anterior meningocele. In our experience, TachoSil appears to be a satisfactory option for the management of these complications.


Assuntos
Meningocele , Humanos , Meningocele/etiologia , Meningocele/cirurgia , Vértebras Cervicais/cirurgia , Resultado do Tratamento , Discotomia/efeitos adversos , Discotomia/métodos , Artroplastia/efeitos adversos , Artroplastia/métodos , Complicações Pós-Operatórias/etiologia , Complicações Pós-Operatórias/cirurgia
4.
Clin Imaging ; 79: 323-325, 2021 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-34392073

RESUMO

We present two cases of acute and delayed presentations of intraspinal pseudomeningoceles, which is a rare complication of spinal trauma or surgery. Our goal is to discuss the pathophysiology, clinical presentation, imaging findings, and treatment of this rare entity. This diagnosis should be considered in patients with unexplained neurologic symptoms in the setting of prior cervical spine injury or surgery.


Assuntos
Meningocele , Humanos , Meningocele/diagnóstico por imagem , Meningocele/etiologia , Meningocele/cirurgia
5.
Sci Rep ; 11(1): 14801, 2021 07 20.
Artigo em Inglês | MEDLINE | ID: mdl-34285298

RESUMO

Suboccipital decompression with duraplasty is a widely accepted method for treating patients with Chiari malformation type I. However, important details of the duraplasty technique are still controversial. This retrospective study analyzes clinical and radiological outcomes after surgery depending upon the type of graft and methods of graft fixation. Seventy consecutive decompressions with duraplasty were analyzed. Two types of grafts, nonautologous (Non-AutoG; 60.0%) and autologous (AutoG; 40.0%), and two methods of graft fixation, suturing (S; 67.1%) and gluing (G; 32.9%), were used in four different combinations: (Non-AutoG+S: 31.4%; Non-AutoG+G: 28.6%; AutoG+S: 35.7%; AutoG+G: 4.3%) according to surgeon preference. The mean follow-up was 63.4 months. According to gestalt and Chicago Chiari Outcome Scales, satisfactory results were obtained in 72.9% and 78.6% of cases, respectively, in the long term. The outcomes were not related to the kind of graft (p = 0.44), fixation method (p = 0.89) or duraplasty pattern (p = 0.32). Decreased syringomyelia was observed in 88.9% of cases, and no associations with the kind of graft (p = 0.84), fixation method (p = 1) or duraplasty pattern were found (p = 0.96). Pseudomeningocele occurred 5 times more often in the Non-AutoG group than in the AutoG group (52.4% vs. 10.7%; p < 0.05), whereas their formations were not related to the fixation method (p = 0.34). Three cases (12.0%) required reoperation with reduraplasty. Autologous and nonautologous dural grafts can be sutured or glued with similar clinical results; however, the use of nonautologous grafts is linked with a much higher risk of pseudomeningocele formation.


Assuntos
Malformação de Arnold-Chiari/cirurgia , Craniectomia Descompressiva/instrumentação , Dura-Máter/cirurgia , Procedimentos de Cirurgia Plástica/instrumentação , Transplante/classificação , Adulto , Idoso , Craniectomia Descompressiva/efeitos adversos , Feminino , Humanos , Masculino , Meningocele/epidemiologia , Meningocele/etiologia , Pessoa de Meia-Idade , Complicações Pós-Operatórias/epidemiologia , Procedimentos de Cirurgia Plástica/efeitos adversos , Reoperação/estatística & dados numéricos , Estudos Retrospectivos , Transplante/instrumentação , Resultado do Tratamento , Adulto Jovem
7.
Lab Anim ; 55(2): 181-188, 2021 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-32787540

RESUMO

An otherwise healthy two-month-old female C57BL/6J mouse presented with a left-sided head tilt. Differential diagnoses included idiopathic necrotizing arteritis, bacterial otitis media/interna (Pasteurella pneumotropica, Pseudomonas aeruginosa, Streptococcus sp., Mycoplasma pulmonis and Burkholderia gladioli), encephalitis, an abscess, neoplasia, a congenital malformation and an accidental or iatrogenic head trauma. Magnetic resonance imaging (MRI) revealed a large space-occupying right olfactory lobe intra-axial lesion with severe secondary left-sided subfalcine herniation. Following imaging, the animal was euthanized due to poor prognosis. Histopathologic examination revealed a unilateral, full-thickness bone defect at the base of the cribriform plate and nasal conchae dysplasia, resulting in the herniation of the olfactory bulb into the nasal cavity. There was also a left midline-shift of the frontal cortex and moderate catarrhal sinusitis in the left mandibular sinus. The MRI and histopathologic changes are consistent with a congenital malformation of the nasal cavity and frontal aspect of the skull known as an ethmoidal meningoencephalocele. Encephaloceles are rare abnormalities caused by herniation of contents of the brain through a defect in the skull which occur due to disruption of the neural tube closure at the level anterior neuropore or secondary to trauma, surgical complications, cleft palate or increased intracranial pressure. The etiology is incompletely understood but hypotheses include genetics, vitamin deficiency, teratogens, infectious agents and environmental factors. Ethmoidal encephaloceles have been reported in multiple species including humans but have not been reported previously in mice. There are multiple models for spontaneous and induced craniofacial malformation in mice, but none described for ethmoidal encephaloceles.


Assuntos
Encefalocele/diagnóstico , Meningocele/diagnóstico , Animais , Diagnóstico Diferencial , Encefalocele/diagnóstico por imagem , Encefalocele/etiologia , Osso Etmoide/patologia , Evolução Fatal , Feminino , Imageamento por Ressonância Magnética , Meningocele/diagnóstico por imagem , Meningocele/etiologia , Camundongos , Camundongos Endogâmicos C57BL
8.
World Neurosurg ; 144: e693-e700, 2020 12.
Artigo em Inglês | MEDLINE | ID: mdl-32942058

RESUMO

BACKGROUND: Pseudomeningocele is an uncommon but widely recognized complication of spinal surgery that can be challenging to correct. When conservative measures fail, patients frequently require reoperation to attempt primary closure of the durotomy, yet attempts at true watertight closures of the dura or fascia sometimes fall short. We describe a technique of lumbosacral pseudomeningocele repair involving a 2-layer pants-over-vest closure of the pseudomeningocele coupled with mobilization of bilateral paraspinal musculature to create a Z-plasty, or a Z-shaped flap. We have demonstrated a high success rate with our small series. METHODS: The technique used meticulous manipulation of the pseudomeningocele to make a 2-layer pants-over-vest closure. This closure coupled with wide mobilization and importation of paraspinous muscle into the wound effectively obliterated dead space with simultaneous tamponade of the dural tear. The lateral row perforators were left intact, providing excellent vascularity with adequate mobility to the patient. RESULTS: This technique was incorporated into the care of 10 patients between 2004 and July 2019. All wounds were closed in a single stage after careful flap section based on the wound's needs. We demonstrated successful pseudomeningocele resolution in all 10 patients with no observed clinical recurrence of symptomatic pseudomeningocele after at least 6 months of follow-up. CONCLUSIONS: This technique provides a straightforward option for the spine surgeon to manage these challenging spinal wounds with minimal, if any, need for further laminectomy as well as a high fistula control rate with minimal morbidity.


Assuntos
Meningocele/cirurgia , Procedimentos Neurocirúrgicos/métodos , Músculos Paraespinais/cirurgia , Complicações Pós-Operatórias/cirurgia , Adolescente , Adulto , Idoso de 80 Anos ou mais , Criança , Feminino , Humanos , Região Lombossacral/patologia , Região Lombossacral/cirurgia , Masculino , Meningocele/etiologia , Pessoa de Meia-Idade , Reoperação/métodos , Retalhos Cirúrgicos , Resultado do Tratamento , Adulto Jovem
9.
Int J Hematol ; 112(6): 894-899, 2020 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-32772263

RESUMO

Diamond-Blackfan Anemia (DBA) is a congenital pure red cell aplasia caused by heterozygous variants in ribosomal protein genes. The hematological features associated with DBA are highly variable and non-hematological abnormalities are common. We report herein on an affected mother and her daughter presenting with transfusion-dependent anemia. The mother showed mild physical abnormalities and entered spontaneous remission at age 13 years. Her daughter was born with occipital meningocele. Exome sequencing of DNA from the mother revealed a heterozygous novel splice site variant (NM_001011.4:c.508-3T > G) in the Ribosomal Protein S7 gene (RPS7) inherited by the daughter. Functional analysis of the RPS7 variant expressed from a mini-gene construct revealed that the exon 7 acceptor splice site was replaced by a cryptic splice resulting in a transcript missing 64 bp of exon 7 (p.Val170Serfs*8). Our study confirms a pathogenic effect of a novel RPS7 variant in DBA associated with spontaneous remission in the mother and meningocele in her daughter, thus adding to the genotype-phenotype correlations in DBA.


Assuntos
Anemia de Diamond-Blackfan/genética , Aberrações Cromossômicas , Estudos de Associação Genética , Variação Genética/genética , Meningocele/genética , Splicing de RNA/genética , Proteínas Ribossômicas/genética , Adolescente , Adulto , Anemia de Diamond-Blackfan/etiologia , Criança , Éxons/genética , Feminino , Humanos , Meningocele/etiologia , Relações Mãe-Filho , Remissão Espontânea , Análise de Sequência de DNA
10.
World Neurosurg ; 142: 408-412, 2020 10.
Artigo em Inglês | MEDLINE | ID: mdl-32622921

RESUMO

BACKGROUND AND CASE DESCRIPTION: We report a case of an 18-year-old patient who presented with late progressive deterioration of neurologic condition 8 weeks after a penetrating injury to the back. Investigations revealed a dorsally located post-traumatic spinal cord herniation. Urgent exploration, decompression, and repair were performed. We reviewed the literature and found only 19 similar cases previously reported. Pathophysiology and presentations were variable and even poorly understood. CONCLUSIONS: Late-onset post-traumatic spinal cord herniation is a potentially curable cause of neurologic deterioration after spinal trauma and should be considered in all cases with late neurologic deterioration after spinal trauma.


Assuntos
Meningocele/etiologia , Traumatismos da Medula Espinal/complicações , Adolescente , Descompressão Cirúrgica , Humanos , Masculino , Meningocele/cirurgia , Doenças da Medula Espinal/etiologia , Doenças da Medula Espinal/cirurgia
11.
J Feline Med Surg ; 22(12): 1230-1237, 2020 12.
Artigo em Inglês | MEDLINE | ID: mdl-32478635

RESUMO

OBJECTIVES: This study was performed to evaluate retrospectively the clinical signs, complications and postoperative outcomes of feline intracranial meningioma (IM) with concurrent cingulate, transtentorial and foramen magnum herniations. METHODS: The medical records and MRI scans of cats with IM and cerebral herniation were reviewed. Cases involving concurrent cingulate, transtentorial and foramen magnum herniations were included. Owners were contacted to obtain long-term follow-up information. RESULTS: Seven cats (four castrated males and three spayed females) met the inclusion criteria. Median age was 13.0 years (range 9.9-16.1 years) and median duration of clinical signs was 35 days (range 21-163 days). The clinical signs of cats with cerebral herniation included visual impairment (n = 5 [71.4%]), ataxia (n = 4 [57.1%]), impaired consciousness (n = 2 [28.6%]), head pressing (n = 2 [28.6%]), paresis (n = 1 [14.3%]), torticollis (n = 1 [14.3%]) and personality changes (n = 1 [14.3%]). Median tumour volume, cranial cavity volume and tumour volume:intracranial volume ratio before surgery were 3.37 cm3 (range 3.23-11.5 cm3), 32.6 cm3 (range 29.8-78.3 cm3) and 10.4% (range 5.3-35.3%), respectively. Median overall tumour excision rate was 90.6%. Preoperative intracranial pressure (ICP) ranged from 15 to 32 mmHg (median 29 mmHg). In all cases, the ICP dropped to 0 mmHg immediately after tumour removal. No adjuvant therapy was required after surgery. The median survival period was 612 days (range 55-1453 days). CONCLUSIONS AND RELEVANCE: The results of this study indicate that surgical treatment of rostrotentorial IM is effective and allows prolonged survival, even in cats with concurrent cingulate, transtentorial and foramen magnum herniations.


Assuntos
Doenças do Gato/cirurgia , Neoplasias Meníngeas/veterinária , Meningioma/veterinária , Meningocele/veterinária , Animais , Doenças do Gato/patologia , Gatos , Dura-Máter/patologia , Feminino , Forame Magno/patologia , Giro do Cíngulo/patologia , Masculino , Neoplasias Meníngeas/complicações , Neoplasias Meníngeas/patologia , Neoplasias Meníngeas/cirurgia , Meningioma/complicações , Meningioma/patologia , Meningioma/cirurgia , Meningocele/etiologia , Estudos Retrospectivos , Resultado do Tratamento
12.
Am J Otolaryngol ; 41(4): 102560, 2020.
Artigo em Inglês | MEDLINE | ID: mdl-32505907

RESUMO

Along the medical practice of an Otologist he/she will face middle cranial fossa (MCF) bone defects. The purpose of this study is to contribute to the understanding of this possible life threatening condition, and to share and discuss our management approach. A literature review is also presented. STUDY DESIGN: Retrospective case series at García-Ibáñez Otology and Skull base private center referral. METHODS: This study is based on the analysis of data collected from 19 cases of temporal bone meningoencephalic herniations surgically treated from 2006 to 2018. The follow-up ranged from 18 to 162 months with a mean average of 44.5 months. MAIN FINDINGS: Meningoencephalic herniations were divided into four etiologic groups: spontaneous (24.8%), secondary to chronic otitis media (21.8%), iatrogenic (45.9%), and posttraumatic (7.5%). Different surgical techniques were used as treatment: transmastoid (TM) approach (27.8%), MCF approach (27.8%), combined technique (transmastoid plus minicraniotomy, 3%), and middle ear obliteration with blind sac closure of the external auditory canal (41.4%). CONCLUSIONS: Variables like bilateral hearing level, size and location of the bone defect and existence of CSF leak should be analyzed to select the safest and most effective closing surgical approach.


Assuntos
Vazamento de Líquido Cefalorraquidiano , Fossa Craniana Média/cirurgia , Encefalocele/cirurgia , Meningocele/cirurgia , Procedimentos Neurocirúrgicos/métodos , Osso Temporal/cirurgia , Adulto , Idoso , Encefalocele/etiologia , Feminino , Seguimentos , Humanos , Masculino , Meningocele/etiologia , Pessoa de Meia-Idade , Estudos Retrospectivos , Fatores de Tempo , Adulto Jovem
13.
Childs Nerv Syst ; 36(8): 1777-1780, 2020 08.
Artigo em Inglês | MEDLINE | ID: mdl-32314024

RESUMO

Lumbar pseudomeningocele is a common complication of spinal surgery. It is caused by dural defect, and it consists of extradural collection of cerebrospinal fluid communicating with subarachnoid space. Usually it is small, asymptomatic, and self-limiting. In case of persistence, common treatments include bedrest, puncture and compressive dressing, subcutaneous or subarachnoid lumbar drainage, surgical re-exploration for durotomy closure, and blood patch. Fast-resorbing polymer mesh has been recently used for closure osseous gap in adult patients who developed giant pseudomeningocele following laminectomy. We report the case of a 16-year-old girl, affected by spinal dysraphism, who developed a persistent giant lumbar pseudomeningocele after repeated surgery for detethering of the spinal cord and removing of spinal lipomyelomeningocele. She was finally treated with fast-resorbing polymer mesh, to fill the gap of the posterior osseous defect and to provide support for the thecal sac. For our best knowledge, this is the first pediatric case reported in literature on this kind of reconstruction. In complex cases of pseudomeningocele, implant of fast-resorbing polymer mesh may represent an option also in pediatric patients.


Assuntos
Rinorreia de Líquido Cefalorraquidiano , Meningocele , Adolescente , Feminino , Humanos , Meningocele/diagnóstico por imagem , Meningocele/etiologia , Meningocele/cirurgia , Polímeros , Complicações Pós-Operatórias , Coluna Vertebral , Telas Cirúrgicas
14.
Br J Neurosurg ; 34(1): 51-54, 2020 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-31656091

RESUMO

Purpose: In many brain surgeries, dura cannot be primarily repaired and it is necessary to use a graft. Appropriate repair of dura plays a major role in preventing CSF leakage and meningitis. In the cases where sufficient Pericranium is not available for recovery, we need to use graft from other resources. The present study compares the results of amniotic membrane dural graft and pericranium dural graft in terms of CSF leakage and Pseudomeningocele. Materials and methods: This is a semi-empirical research. Thirty cases underwent dural graft with amnion membrane because of inadequate local pericranium for repair. There were 30 other historical cases that had undergone auto-graft dural graft with Pericranium in the same center.Results: The average age of the participants in Pericranium graft group (P, n = 30) and those in amniotic graft group (A, n = 30) was 39.1 ± 15 and 43.9 ± 19 years old, respectively. As for the P and A groups, there were 14 and 17 males, respectively. Two cases of CSF leakage (7%) were observed in P group, while none was reported in A group. There were 3 cases (10%) of meningitis in Pericranium group and 2 cases (7%) were reported in amnion group. Out of 2 cases of meningitis in group P, 2 cases were associated with CSF leak. 5 cases of Hydrocephalus (17%) were observed in group P and 9 cases (30%) were reported in group A. No significant difference was observed between the two groups in any complication. The frequency of hydrocephalus was significantly higher in posterior fossa craniotomy.Conclusion: Amniotic membrane may provide a good source for cases with a large dural defect and can be utilized for all ages. Of course, its application in cases of posterior fossa craniotomy needs to be carefully studied so that the best results may be obtained.


Assuntos
Âmnio/transplante , Vazamento de Líquido Cefalorraquidiano/etiologia , Dura-Máter/cirurgia , Dura-Máter/transplante , Meningocele/etiologia , Procedimentos Neurocirúrgicos/métodos , Adolescente , Adulto , Idoso , Criança , Pré-Escolar , Fossa Craniana Posterior/cirurgia , Feminino , Humanos , Hidrocefalia/etiologia , Masculino , Pessoa de Meia-Idade , Complicações Pós-Operatórias/epidemiologia , Procedimentos de Cirurgia Plástica/métodos , Adulto Jovem
15.
World Neurosurg ; 134: 532-535, 2020 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-31760189

RESUMO

BACKGROUND: An expanding cohort of patients with spina bifida live well into adulthood and pose complex management challenges due to unique combinations of adult health issues overlying congenital problems. CASE DESCRIPTION: We present a case of a 45-year-old woman with an expanding, disfiguring, painful lumbar meningocele more than 40 years after her only surgery as a 3-year-old child. A team of pediatric and adult neurosurgeons as well as plastic/reconstructive surgeons successfully performed surgery to obliterate the meningocele, with preservation of her baseline functional status, and no evidence of recurrence after more than 1 year of follow-up. CONCLUSIONS: Symptomatic meningocele may present in a long-delayed fashion in adult patients with a history of spina bifida. Surgical treatment may provide symptomatic benefit.


Assuntos
Meningocele/etiologia , Meningomielocele/complicações , Feminino , Humanos , Meningocele/cirurgia , Pessoa de Meia-Idade , Disrafismo Espinal/complicações , Disrafismo Espinal/cirurgia
16.
Pediatr Neurosurg ; 54(5): 319-323, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-31533115

RESUMO

BACKGROUND: The incidence of non-traumatic intracranial haemorrhage among neonates is less than among adults. However, the outcomes are generally poor, with high rates of morbidity and mortality. Non-traumatic intracranial haemorrhage can cause a sudden increase in intracranial pressure, requiring emergency craniotomy for evacuation. METHODS: We report 4 cases following craniotomy for non-traumatic acute intracranial haematoma in very young children. The surgical technique and postoperative course are discussed, with emphasis on dural repair to avoid resistant pseudomeningocele. RESULTS: Despite successful management of the acutely raised intracranial pressure, all patients afterwards developed tense pseudomeningoceles and required formal dural repair. This led to a change in institutional practice and modifications to the surgical technique. CONCLUSION: The authors highlight the postoperative occurrence of resistant pseudomeningoceles in young children after clot evacuations and leaving the dura open. They describe the surgical procedures to avoid these pseudomeningoceles.


Assuntos
Craniotomia/efeitos adversos , Encefalocele/diagnóstico por imagem , Hematoma Subdural Intracraniano/diagnóstico por imagem , Meningocele/diagnóstico por imagem , Complicações Pós-Operatórias/diagnóstico por imagem , Pré-Escolar , Craniotomia/tendências , Encefalocele/etiologia , Feminino , Hematoma Subdural Intracraniano/cirurgia , Humanos , Lactente , Recém-Nascido , Masculino , Meningocele/etiologia , Complicações Pós-Operatórias/etiologia
17.
World Neurosurg ; 130: 71-76, 2019 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-31279920

RESUMO

BACKGROUND: Cerebellar cyst formation after surgery is uncommon, and few cases of this condition have been previously reported. These cases had an intraparenchymal cyst in the cerebellar hemisphere that required surgical fenestration of the cyst. We herein present a rare case of a postoperative cerebellar cyst with pseudomeningocele and magnetic resonance images indicating a fistula between the cyst and pseudomeningocele. CASE DESCRIPTION: A patient presented with an intraparenchymal cyst and surrounding edema in the cerebellar hemisphere that developed after a C1 laminectomy and a small suboccipital craniectomy for the removal of an accessory nerve neurinoma at the craniovertebral junction. Fast imaging employing steady-state acquisition images identified the fistula connecting the cyst and extradural cerebrospinal fluid retention. Conservative management with administration of dexamethasone induced spontaneous regression of the cyst, and no recurrence had occurred by the 1-year follow-up. CONCLUSIONS: Watertight dural closure is important for the prevention of this rare complication after posterior fossa surgery. However, an arachnoid tear on the cerebellar fissure and adjacent dural defect are necessary antecedents for this rare condition. High-resolution fast imaging employing steady-state acquisition images could provide additional information for the etiology of postoperative cerebellar cyst.


Assuntos
Cistos do Sistema Nervoso Central/diagnóstico por imagem , Cerebelo/diagnóstico por imagem , Meningocele/diagnóstico por imagem , Complicações Pós-Operatórias/diagnóstico por imagem , Neoplasias da Coluna Vertebral/diagnóstico por imagem , Neoplasias da Coluna Vertebral/cirurgia , Idoso , Cistos do Sistema Nervoso Central/etiologia , Vértebras Cervicais/diagnóstico por imagem , Humanos , Masculino , Meningocele/etiologia , Complicações Pós-Operatórias/etiologia , Base do Crânio/diagnóstico por imagem
18.
Am J Med Genet A ; 179(9): 1799-1814, 2019 09.
Artigo em Inglês | MEDLINE | ID: mdl-31294918

RESUMO

Sacral agenesis is a rare birth defect characterized by partial or complete absence of the sacrum. We sought to (a) describe case characteristics, (b) estimate birth prevalence, and (c) identify risk factors for nonsyndromic sacral agenesis using data from the National Birth Defects Prevention Study (NBDPS). The NBDPS was a population-based, case-control study involving pregnancies with estimated dates of delivery from October 1997 through December 2011. We estimated birth prevalence using all NBDPS eligible cases. Using self-reported maternal exposure information, we conducted multivariable logistic regression analysis to identify potential risk factors overall and among women without diabetes. The birth prevalence of sacral agenesis was 2.6/100,000 live births. In the multivariable analysis, multifetal pregnancy, pre-existing Type 1 diabetes, and pre-existing Type 2 diabetes were positively and significantly associated with sacral agenesis, albeit estimates were imprecise. Preexisting Type 1 diabetes was the strongest risk factor (adjusted odds ratio = 96.6, 95% confidence interval = 43.5-214.7). Among women without diabetes, periconceptional smoking was positively and significantly associated with sacral agenesis. Our findings underscore the importance of smoking cessation programs among women planning pregnancy and the importance of better understanding the role of glycemic control before and during pregnancy when designing interventions for primary prevention of sacral agenesis.


Assuntos
Anormalidades Múltiplas/epidemiologia , Anormalidades Congênitas/epidemiologia , Diabetes Mellitus/epidemiologia , Meningocele/epidemiologia , Malformações do Sistema Nervoso/epidemiologia , Região Sacrococcígea/anormalidades , Anormalidades Múltiplas/etiologia , Anormalidades Múltiplas/genética , Anormalidades Múltiplas/fisiopatologia , Adulto , Estudos de Casos e Controles , Anormalidades Congênitas/genética , Anormalidades Congênitas/fisiopatologia , Complicações do Diabetes/epidemiologia , Complicações do Diabetes/genética , Complicações do Diabetes/fisiopatologia , Diabetes Mellitus/genética , Diabetes Mellitus/fisiopatologia , Feminino , Humanos , Recém-Nascido , Masculino , Exposição Materna , Meningocele/etiologia , Meningocele/genética , Meningocele/fisiopatologia , Malformações do Sistema Nervoso/genética , Malformações do Sistema Nervoso/fisiopatologia , População/genética , Gravidez , Fatores de Risco , Região Sacrococcígea/fisiopatologia , Sacro/anormalidades
20.
Acta Otolaryngol ; 139(5): 421-424, 2019 May.
Artigo em Inglês | MEDLINE | ID: mdl-30861355

RESUMO

BACKGROUND: Marfan syndrome (MFS) is a genetic disorder affecting connective tissue. The composition of the dura can change. Consequently, lumbo-sacral dural herniations and cerebrospinal fluid (CSF) leaks are encountered, however, they have yet to been described in the temporal bone. AIMS/OBJECTIVES: To define the prevalence of temporal bone meningocele or encephalocele among patients with MFS. MATERIALS AND METHODS: Reviewed medical records of all adult patients, diagnosed with MFS, who were treated between 1993 and 2018 at a single academic referral institute. Head targeted CT scans were analyzed. The presence of an anterior or lateral skull base defect was recorded. RESULTS: One-hundred and one patients diagnosed with MFS were identified. Twelve of which had suitable CT scans and were enrolled in the study. The median age of patients with defects was 65 years (range 41-71). Five of the twelve patients (41.6%) had tegmen defect. Of the seven defects found, the median size of the defects was 3 mm (range 2-5 mm). All defects were in the temporal bone, none in the anterior skull base. CONCLUSIONS AND SIGNIFICANCE: The prevalence of radiological evidence of a temporal bone defect among patients with MFS is high. This is a new, important, and potentially life-threatening association with the syndrome.


Assuntos
Encefalocele/etiologia , Síndrome de Marfan/complicações , Meningocele/etiologia , Osso Temporal/anormalidades , Adulto , Idoso , Encefalocele/epidemiologia , Feminino , Humanos , Masculino , Meningocele/epidemiologia , Pessoa de Meia-Idade , Prevalência , Estudos Retrospectivos , Osso Temporal/diagnóstico por imagem
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