Home Life Sciences Combination of a bronchogenic cyst in the thoracic spinal canal with chronic myelocytic leukemia
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Combination of a bronchogenic cyst in the thoracic spinal canal with chronic myelocytic leukemia

  • Hao Zhang , Hai-Feng Li , Hai-Feng Duan , Ke-Feng Huang and Zhi-Hua Tian EMAIL logo
Published/Copyright: September 30, 2023

Abstract

The presented case report describes an incredibly rare instance of an intramedullary bronchial cyst located in the thoracic spinal canal on the dorsal side of the spinal cord, which was observed in a patient with chronic myelogenous leukemia. A 29-year-old man presented with back pain for half a month, along with numbness and pain below the chest and ribs for 1 week. Hypersensitivity was present in the inferior plane of the long xiphoid process in the nervous system. Magnetic resonance imaging (MRI) showed intramedullary cystic lesions in the vertebral body plane of the third to the fourth thoracic vertebra. There was no recurrence during the 6-month postoperative follow-up period. The histopathological findings were consistent with bronchogenic cysts. Cystic lesions were eliminated through the posterior median approach. After the cyst ruptured during surgery, gel liquid was seen, and the majority of the cyst walls were removed. One week after the surgery, the hypersensitivity fully subsided. Six months following surgery, an updated MRI revealed no recurrence. Intramedullary bronchogenic cysts on the dorsal side of the thoracic spine are extremely uncommon. Diagnosis requires histopathological evidence, and it is challenging to diagnose before surgery. Prompt surgical resection is recommended in case of positive diagnosis.

1 Introduction

Bronchogenic cysts are cystic lesions that develop due to abnormal bronchial system development during the embryonic stage. It is an endodermal translocation tumor that frequently develops in the external spinal membrane of the cervical and thoracic spinal cords. Most of the sites are located in the ventral spinal cord. It rarely occurs in the spinal canal, and accounts for 10–15% of mediastinal masses. The first instance of an intraspinal bronchogenic cyst was reported by Yamashita in 1973 [1]. It is incredibly uncommon for cysts to occur anywhere else besides the cervical and thoracic spine. A few cases of intraspinal bronchogenic cysts have been recorded in China over the past several years [2]. However, it should be noted that locating such a cyst in the thoracic spinal cord, as seen in the current case, is extremely rare.

2 Medical record reporting

2.1 History and physical examination

A 29-year-old male patient was admitted to the Jincheng People’s Hospital, Shanxi Province, with back pain for half a month and numbness and pain below the chests and ribs for 1 week. A thorough physical examination was conducted upon admission. The patient was able to move his limbs based on instructions and could maintain autonomous postures. He had no swelling of superficial lymph nodes in the whole body, showed hypersensitivity below the xiphoid plane, and there was a symmetrical degree of hypersensitivity on both sides. The muscle strength of both lower limbs was grade V on the Lovett scale, the muscle tension was normal, the muscles of both lower limbs were not atrophied, and the pathological symptoms of both lower limbs were not induced. Past medical history: the patient was diagnosed with chronic myelocytic leukemia in the chronic stage in the Department of Hematology, Jincheng People’s Hospital in July 2016. The patient was orally administered imatinib mesylate tablets 400 mg/per time per day. Regular blood tests detected no abnormalities in the white blood cells or platelets. There was an intraspinal cystic lesion with hemivertebra deformity in the region of the third to the fourth thoracic vertebra on plain and enhanced magnetic resonance imaging (MRI) scans of the thoracic vertebra.

The plain thoracic vertebra MRI scan is displayed in Figure 1a–c. The result showed that at the thoracic 3–4 vertebral body plane, T1 signal and T2 high signal can be seen in the cystic space, the spinal cord compression was severe, and the cystic fluid signal was homogeneous. The cyst wall was smooth. The enhancement of cyst wall was not obvious in enhanced scan. The enhanced thoracic vertebra MRI scan is displayed in Figure 1d and e. The plain thoracic vertebra computed tomography (CT) scan is displayed in Figure 1f; and the three-dimensional reconstruction is displayed in Figure 2a and b. CT showed malformations of fusion of spinous processes of thoracic 4 and thoracic 5.

Figure 1 
                  (a) T2 image of a sagittal plain MRI scan. On the third to the fourth thoracic vertebral plane, a cystic mass with significant signal intensity could be identified. It is evident that the spinal cord is compressed. (b) T1 image of a sagittal plain MRI scan. On the third to the fourth thoracic vertebral plane, a cystic mass that is T1 isointense or slightly hypointense is visible. (c) A horizontal T2 image from a plain MRI scan. The left dorsal side of the spinal cord has a cystic mass, and the spinal cord is compressed. (d) Enhanced sagittal plain MRI image. An enhanced cystic mass can be seen. (e) Enhanced sagittal plain MRI image. There is no visible cystic space occupying the enhancement. (f) Sagittal plain CT image showing the third to the fourth thoracic vertebrae and spinous process fusion.
Figure 1

(a) T2 image of a sagittal plain MRI scan. On the third to the fourth thoracic vertebral plane, a cystic mass with significant signal intensity could be identified. It is evident that the spinal cord is compressed. (b) T1 image of a sagittal plain MRI scan. On the third to the fourth thoracic vertebral plane, a cystic mass that is T1 isointense or slightly hypointense is visible. (c) A horizontal T2 image from a plain MRI scan. The left dorsal side of the spinal cord has a cystic mass, and the spinal cord is compressed. (d) Enhanced sagittal plain MRI image. An enhanced cystic mass can be seen. (e) Enhanced sagittal plain MRI image. There is no visible cystic space occupying the enhancement. (f) Sagittal plain CT image showing the third to the fourth thoracic vertebrae and spinous process fusion.

Figure 2 
                  (a) and (b) CT three-dimensional reconstruction of the third to the fourth thoracic vertebrae with spinous process fusion.
Figure 2

(a) and (b) CT three-dimensional reconstruction of the third to the fourth thoracic vertebrae with spinous process fusion.

The initial diagnosis was a space-occupying lesion in the spinal canal of the third to the fourth thoracic vertebra.

  1. Informed consent: Informed consent has been obtained from all individuals included in this study.

  2. Ethical approval: The research related to human use has been complied with all the relevant national regulations, institutional policies and in accordance with the tenets of the Helsinki Declaration, and has been approved by the Ethics Committee of Jin Cheng People’s Hospital (JCPH No. 20230629001).

2.2 Surgery and pathology

Cyst lesions were surgically resected using a posterior midline approach. During the surgery, a grinding drill is used to remove the entire vertebral lamina and spinous process, and the dura mater is incised. The partial vertebral plate and spinous process were also resected during the procedure, which also revealed a cystic tumor in the dorsal side of the spinal cord of the third to the fourth thoracic vertebra. There was no spinal cord pulse after the dura mater spinalis was incised. A fine needle was inserted into the cyst, which produced a gel-like fluid. The tumor was entirely resected under a microscope and the cyst wall was thin, grayish white, and translucent (Figure 3). On the second day after surgery, the symptoms of chest and back pain significantly disappeared. The patient did not experience any sensory or motor impairment in both legs, nor did he have any bowel or bowel movements. There were no complications. The examination images indicate that the tumor capsule conforms to the ciliated columnar epithelial structure of the bronchial wall. Postoperative examination showed an intraspinal bronchogenic cyst.

Figure 3 
                  HE staining of a pathological section at 40×. The cyst wall is made of columnar epithelial cells.
Figure 3

HE staining of a pathological section at 40×. The cyst wall is made of columnar epithelial cells.

2.3 Postoperative follow-up

The patient’s hypersensitivity was eliminated 1 week after the operation, and there was no sensation or movement disorder in his limbs. Following a complete MRI, no evident recurrence was discovered 6 months after the operation (Figure 4).

Figure 4 
                  (a) T2 images from a sagittal plain MRI scan re-examined 6 months following surgery. The cystic space that was occupied vanished without a trace. When compared to before surgery, the spinal cord morphology is dramatically improved. (b) T1 images of sagittal plain MRI scan re-examined 6 months following surgery. Cystic space that was occupied vanished without recurrence. When compared to before surgery, the spinal cord morphology is dramatically improved.
Figure 4

(a) T2 images from a sagittal plain MRI scan re-examined 6 months following surgery. The cystic space that was occupied vanished without a trace. When compared to before surgery, the spinal cord morphology is dramatically improved. (b) T1 images of sagittal plain MRI scan re-examined 6 months following surgery. Cystic space that was occupied vanished without recurrence. When compared to before surgery, the spinal cord morphology is dramatically improved.

3 Discussion

Intraspinal bronchial cysts are a rare congenital developmental anomaly of the respiratory system that arise from the pre-embryonic intestine. They account for approximately 0.7–1.3% of all intramedullary spinal cord tumors. The surface of the cyst is covered by respiratory epithelium [3]. There are currently three theories put forth in literature that attempt to explain the exact pathogenesis of bronchial cysts. (1) Isolation insufficiency hypothesis: insufficient isolation occurs when the endoderm and ectoderm are being differentiated into separate cell masses [4,5]. (2) Potential differentiation hypothesis: due to the capacity of the ectoderm to differentiate into endoderm and paraxial mesoderm, it is believed that cysts originate from the ectoderm [6]. (3) Split notochord syndrome: this condition is thought to be caused by incomplete replication or separation of the notochord, which could explain the local defect. This hypothesis can explain the phenomenon that bronchogenic cysts in the spinal canal often combined with other malformations [7,8]. Another abnormal differentiation hypothesis has been added in Chinese literature – bronchogenic cysts originate from abnormal differentiation of the tracheobronchial tree [9]. The notochord fissure syndrome is the currently preferred hypothesis. However, the specific pathogenesis of this disease still needs to be further clarified by accumulating a large amount of case data. In this case, the condition of the patient with the combined spinal malformation was in concordance with the hypothesis of the split notochord syndrome.

Clinical manifestations and symptoms of intraspinal bronchial cysts are generally site-related and non-specific. Neck and back pain, as well as sensory and motor abnormalities in the four limbs are the most typical symptoms. In this case, back and chest pain were the main symptoms of the patient.

On MRI, the majority of intraspinal bronchogenic cysts show hypointense T1 and hyperintense T2 signal, and gadoteric acid meglumine does not clearly augment these signals. As it can be challenging to discern one condition from another using an MRI alone, a CT scan is required. Spina bifida and other congenital spinal abnormalities can be seen on a CT scan [10].

Pathologically, intraspinal bronchogenic cysts are mostly characterized by the single-layer or pseudostratified columnar epithelium of the cyst wall, and glands, cartilage, smooth muscles, and nerve fibers may be present on the cyst wall. The fluid within the cyst is composed of various substances, with most exhibiting a gel-like or yellowish appearance. However, some portions of the fluid may be clear and colorless.

The primary form of treatment for an intraspinal bronchogenic cyst is surgery. During the procedure, the spinal cord and nerve root should be safeguarded, and the total cyst wall resection should not be forced. It is reported that the cyst wall may be washed with diluted iodophor or ignited by local bipolar electrocoagulation micro-current after the cyst wall has been incised to liberate the cyst fluid [11]. Additionally, subtotal resection is also acceptable. According to published data, 84.6% of patients with intraspinal bronchogenic cysts who underwent subtotal resection experienced no recurrence at the end of the follow-up [12]. However, this study also recommended total resection, the recurrence rate after subtotal resection was 15.4%, but there was no recurrence after total resection. In addition, incomplete excision, cystic fenestration, and biopsy associated with recurrence should be avoided [12]. In this case, the cyst wall was completely resected during the operation, and there was no postoperative recurrence. This result was consistent with the conclusions of the above studies.

The patient in our study had a bronchogenic cyst of the spinal canal complicated with chronic myelogenous leukemia. The preoperative surgical plan fully estimated the possibility of intraoperative bleeding, the number of white blood cells, red blood cells, platelets, and the coagulation function of the patient should be paid attention to in preoperative detection. As we all know, patients with leukemia often have coagulation dysfunction, resulting in excessive intraoperative field bleeding, and continuous postoperative bleeding leads to epidural or subdural hematoma, resulting in lower limb dysfunction [13]. Therefore, it is necessary to prepare enough blood before operation, hemostasis during operation should be thorough, and blood routine and coagulation function should be reviewed after operation too. In addition, the patient’s systemic condition, including whether there is ecchymosis and bleeding, as well as lower limb function, should be carefully observed. Once lower limb dysfunction occurs timely surgery is required to remove hematoma.

4 Conclusion

Intraspinal bronchogenic cysts, especially those affecting the dorsal thoracic region, are extremely rare, and the case described in this report falls within this uncommon category. To confirm the diagnosis of this condition, imaging and clinical pathology examinations are essential. Surgery is generally considered the preferred method of treatment.


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Acknowledgements

The authors would like to acknowledge the hard and dedicated work of all the staff that implemented the intervention and evaluation components of the study.

  1. Funding information: Authors state no funding involved.

  2. Author contributions: Conception and design of the research: Z.H. and T.Z.H.; acquisition of data: Z.H. and L.H.F.; analysis and interpretation of the data: D.H.F. and H.K.F.; writing of the manuscript: Z.H. and T.Z.H.; critical revision of the manuscript for intellectual content: D.H.F. and T.Z.H. All authors read and approved the final draft.

  3. Conflict of interest: Authors state no conflict of interest.

  4. Data availability statement: The datasets generated during and/or analyzed during the current study are available from the corresponding author on reasonable request.

References

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Received: 2023-04-11
Revised: 2023-08-11
Accepted: 2023-08-29
Published Online: 2023-09-30

© 2023 the author(s), published by De Gruyter

This work is licensed under the Creative Commons Attribution 4.0 International License.

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  158. A case report of diagnosis and dynamic monitoring of Listeria monocytogenes meningitis with NGS
  159. Effect of autologous platelet-rich plasma on new bone formation and viability of a Marburg bone graft
  160. Small breast epithelial mucin as a useful prognostic marker for breast cancer patients
  161. Continuous non-adherent culture promotes transdifferentiation of human adipose-derived stem cells into retinal lineage
  162. Nrf3 alleviates oxidative stress and promotes the survival of colon cancer cells by activating AKT/BCL-2 signal pathway
  163. Favorable response to surufatinib in a patient with necrolytic migratory erythema: A case report
  164. Case report of atypical undernutrition of hypoproteinemia type
  165. Down-regulation of COL1A1 inhibits tumor-associated fibroblast activation and mediates matrix remodeling in the tumor microenvironment of breast cancer
  166. Sarcoma protein kinase inhibition alleviates liver fibrosis by promoting hepatic stellate cells ferroptosis
  167. Research progress of serum eosinophil in chronic obstructive pulmonary disease and asthma
  168. Clinicopathological characteristics of co-existing or mixed colorectal cancer and neuroendocrine tumor: Report of five cases
  169. Role of menopausal hormone therapy in the prevention of postmenopausal osteoporosis
  170. Precisional detection of lymph node metastasis using tFCM in colorectal cancer
  171. Advances in diagnosis and treatment of perimenopausal syndrome
  172. A study of forensic genetics: ITO index distribution and kinship judgment between two individuals
  173. Acute lupus pneumonitis resembling miliary tuberculosis: A case-based review
  174. Plasma levels of CD36 and glutathione as biomarkers for ruptured intracranial aneurysm
  175. Fractalkine modulates pulmonary angiogenesis and tube formation by modulating CX3CR1 and growth factors in PVECs
  176. Novel risk prediction models for deep vein thrombosis after thoracotomy and thoracoscopic lung cancer resections, involving coagulation and immune function
  177. Exploring the diagnostic markers of essential tremor: A study based on machine learning algorithms
  178. Evaluation of effects of small-incision approach treatment on proximal tibia fracture by deep learning algorithm-based magnetic resonance imaging
  179. An online diagnosis method for cancer lesions based on intelligent imaging analysis
  180. Medical imaging in rheumatoid arthritis: A review on deep learning approach
  181. Predictive analytics in smart healthcare for child mortality prediction using a machine learning approach
  182. Utility of neutrophil–lymphocyte ratio and platelet–lymphocyte ratio in predicting acute-on-chronic liver failure survival
  183. A biomedical decision support system for meta-analysis of bilateral upper-limb training in stroke patients with hemiplegia
  184. TNF-α and IL-8 levels are positively correlated with hypobaric hypoxic pulmonary hypertension and pulmonary vascular remodeling in rats
  185. Stochastic gradient descent optimisation for convolutional neural network for medical image segmentation
  186. Comparison of the prognostic value of four different critical illness scores in patients with sepsis-induced coagulopathy
  187. Application and teaching of computer molecular simulation embedded technology and artificial intelligence in drug research and development
  188. Hepatobiliary surgery based on intelligent image segmentation technology
  189. Value of brain injury-related indicators based on neural network in the diagnosis of neonatal hypoxic-ischemic encephalopathy
  190. Analysis of early diagnosis methods for asymmetric dementia in brain MR images based on genetic medical technology
  191. Early diagnosis for the onset of peri-implantitis based on artificial neural network
  192. Clinical significance of the detection of serum IgG4 and IgG4/IgG ratio in patients with thyroid-associated ophthalmopathy
  193. Forecast of pain degree of lumbar disc herniation based on back propagation neural network
  194. SPA-UNet: A liver tumor segmentation network based on fused multi-scale features
  195. Systematic evaluation of clinical efficacy of CYP1B1 gene polymorphism in EGFR mutant non-small cell lung cancer observed by medical image
  196. Rehabilitation effect of intelligent rehabilitation training system on hemiplegic limb spasms after stroke
  197. A novel approach for minimising anti-aliasing effects in EEG data acquisition
  198. ErbB4 promotes M2 activation of macrophages in idiopathic pulmonary fibrosis
  199. Clinical role of CYP1B1 gene polymorphism in prediction of postoperative chemotherapy efficacy in NSCLC based on individualized health model
  200. Lung nodule segmentation via semi-residual multi-resolution neural networks
  201. Evaluation of brain nerve function in ICU patients with Delirium by deep learning algorithm-based resting state MRI
  202. A data mining technique for detecting malignant mesothelioma cancer using multiple regression analysis
  203. Markov model combined with MR diffusion tensor imaging for predicting the onset of Alzheimer’s disease
  204. Effectiveness of the treatment of depression associated with cancer and neuroimaging changes in depression-related brain regions in patients treated with the mediator-deuterium acupuncture method
  205. Molecular mechanism of colorectal cancer and screening of molecular markers based on bioinformatics analysis
  206. Monitoring and evaluation of anesthesia depth status data based on neuroscience
  207. Exploring the conformational dynamics and thermodynamics of EGFR S768I and G719X + S768I mutations in non-small cell lung cancer: An in silico approaches
  208. Optimised feature selection-driven convolutional neural network using gray level co-occurrence matrix for detection of cervical cancer
  209. Incidence of different pressure patterns of spinal cerebellar ataxia and analysis of imaging and genetic diagnosis
  210. Pathogenic bacteria and treatment resistance in older cardiovascular disease patients with lung infection and risk prediction model
  211. Adoption value of support vector machine algorithm-based computed tomography imaging in the diagnosis of secondary pulmonary fungal infections in patients with malignant hematological disorders
  212. From slides to insights: Harnessing deep learning for prognostic survival prediction in human colorectal cancer histology
  213. Ecology and Environmental Science
  214. Monitoring of hourly carbon dioxide concentration under different land use types in arid ecosystem
  215. Comparing the differences of prokaryotic microbial community between pit walls and bottom from Chinese liquor revealed by 16S rRNA gene sequencing
  216. Effects of cadmium stress on fruits germination and growth of two herbage species
  217. Bamboo charcoal affects soil properties and bacterial community in tea plantations
  218. Optimization of biogas potential using kinetic models, response surface methodology, and instrumental evidence for biodegradation of tannery fleshings during anaerobic digestion
  219. Understory vegetation diversity patterns of Platycladus orientalis and Pinus elliottii communities in Central and Southern China
  220. Studies on macrofungi diversity and discovery of new species of Abortiporus from Baotianman World Biosphere Reserve
  221. Food Science
  222. Effect of berrycactus fruit (Myrtillocactus geometrizans) on glutamate, glutamine, and GABA levels in the frontal cortex of rats fed with a high-fat diet
  223. Guesstimate of thymoquinone diversity in Nigella sativa L. genotypes and elite varieties collected from Indian states using HPTLC technique
  224. Analysis of bacterial community structure of Fuzhuan tea with different processing techniques
  225. Untargeted metabolomics reveals sour jujube kernel benefiting the nutritional value and flavor of Morchella esculenta
  226. Mycobiota in Slovak wine grapes: A case study from the small Carpathians wine region
  227. Elemental analysis of Fadogia ancylantha leaves used as a nutraceutical in Mashonaland West Province, Zimbabwe
  228. Microbiological transglutaminase: Biotechnological application in the food industry
  229. Influence of solvent-free extraction of fish oil from catfish (Clarias magur) heads using a Taguchi orthogonal array design: A qualitative and quantitative approach
  230. Chromatographic analysis of the chemical composition and anticancer activities of Curcuma longa extract cultivated in Palestine
  231. The potential for the use of leghemoglobin and plant ferritin as sources of iron
  232. Investigating the association between dietary patterns and glycemic control among children and adolescents with T1DM
  233. Bioengineering and Biotechnology
  234. Biocompatibility and osteointegration capability of β-TCP manufactured by stereolithography 3D printing: In vitro study
  235. Clinical characteristics and the prognosis of diabetic foot in Tibet: A single center, retrospective study
  236. Agriculture
  237. Biofertilizer and NPSB fertilizer application effects on nodulation and productivity of common bean (Phaseolus vulgaris L.) at Sodo Zuria, Southern Ethiopia
  238. On correlation between canopy vegetation and growth indexes of maize varieties with different nitrogen efficiencies
  239. Exopolysaccharides from Pseudomonas tolaasii inhibit the growth of Pleurotus ostreatus mycelia
  240. A transcriptomic evaluation of the mechanism of programmed cell death of the replaceable bud in Chinese chestnut
  241. Melatonin enhances salt tolerance in sorghum by modulating photosynthetic performance, osmoregulation, antioxidant defense, and ion homeostasis
  242. Effects of plant density on alfalfa (Medicago sativa L.) seed yield in western Heilongjiang areas
  243. Identification of rice leaf diseases and deficiency disorders using a novel DeepBatch technique
  244. Artificial intelligence and internet of things oriented sustainable precision farming: Towards modern agriculture
  245. Animal Sciences
  246. Effect of ketogenic diet on exercise tolerance and transcriptome of gastrocnemius in mice
  247. Combined analysis of mRNA–miRNA from testis tissue in Tibetan sheep with different FecB genotypes
  248. Isolation, identification, and drug resistance of a partially isolated bacterium from the gill of Siniperca chuatsi
  249. Tracking behavioral changes of confined sows from the first mating to the third parity
  250. The sequencing of the key genes and end products in the TLR4 signaling pathway from the kidney of Rana dybowskii exposed to Aeromonas hydrophila
  251. Development of a new candidate vaccine against piglet diarrhea caused by Escherichia coli
  252. Plant Sciences
  253. Crown and diameter structure of pure Pinus massoniana Lamb. forest in Hunan province, China
  254. Genetic evaluation and germplasm identification analysis on ITS2, trnL-F, and psbA-trnH of alfalfa varieties germplasm resources
  255. Tissue culture and rapid propagation technology for Gentiana rhodantha
  256. Effects of cadmium on the synthesis of active ingredients in Salvia miltiorrhiza
  257. Cloning and expression analysis of VrNAC13 gene in mung bean
  258. Chlorate-induced molecular floral transition revealed by transcriptomes
  259. Effects of warming and drought on growth and development of soybean in Hailun region
  260. Effects of different light conditions on transient expression and biomass in Nicotiana benthamiana leaves
  261. Comparative analysis of the rhizosphere microbiome and medicinally active ingredients of Atractylodes lancea from different geographical origins
  262. Distinguish Dianthus species or varieties based on chloroplast genomes
  263. Comparative transcriptomes reveal molecular mechanisms of apple blossoms of different tolerance genotypes to chilling injury
  264. Study on fresh processing key technology and quality influence of Cut Ophiopogonis Radix based on multi-index evaluation
  265. An advanced approach for fig leaf disease detection and classification: Leveraging image processing and enhanced support vector machine methodology
  266. Erratum
  267. Erratum to “Protein Z modulates the metastasis of lung adenocarcinoma cells”
  268. Erratum to “BRCA1 subcellular localization regulated by PI3K signaling pathway in triple-negative breast cancer MDA-MB-231 cells and hormone-sensitive T47D cells”
  269. Retraction
  270. Retraction to “Protocatechuic acid attenuates cerebral aneurysm formation and progression by inhibiting TNF-alpha/Nrf-2/NF-kB-mediated inflammatory mechanisms in experimental rats”
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