Multilocular bronchogenic cyst of the bilateral adrenal: report of a rare case and review of literature

Purpose: Bronchogenic cysts are rare benign congenital anomalies, originating from the embryonic foregut ventral segment. Adrenal bronchogenic cyst is a rare form of this anomaly. One extremely rare case of bilateral adrenal multilocular bronchogenic cyst in our hospital was reported and the relevant literatures were reviewed. Significant findings: A 51-year-old man suffered from an intermittent vague headache, fatigue and hypertension history for 2 years, which were gradually worsened in a week. Imaging tests showed bilateral suprarenal mass and left renal cysts. After underwent two retroperitoneal laparoscopic adrenal gland tumor separately, they were all proved to be both the multilocular bronchogenic cyst located in bilateral adrenal gland by histopathological examination. Conclusions: This report confirms the bronchogenic cyst that can be involved bilateral joint in the adrenal gland. And we demonstrated retroperitoneoscopic surgical management is effective in the treatment of the disease.     

A single re-implanted ventral root exerts neurotropic effects over multiple spinal cord segments in the adult rat

Spinal cord injuries, particularly traumatic injuries to the conus medullaris and cauda equina, are typically complex and involve multiple segmental levels. Implantation of avulsed ventral roots into the spinal cord as a repair strategy has been shown to be neuroprotective and promote axonal regeneration by spinal cord neurons into an implanted root. However, it is not well known over what distance in the spinal cord an implanted ventral root can exert its neurotropic effect. Here, we investigated whether an avulsed L6 ventral root acutely implanted into the rat spinal cord after a four level (L5–S2) unilateral ventral root avulsion injury may exert neurotropic effects on autonomic and motor neurons over multiple spinal cord segments at 6 weeks postoperatively. Using retrograde labeling techniques and stereological quantification methods, we demonstrate that autonomic and motor neurons from all four lesioned spinal cord segments, spanning more than an 8 mm rostro-caudal distance, reinnervated the one implanted root. The rostro-caudal distribution suggested a gradient of neurotropism, where the axotomized neurons closest to the implanted site had the highest probability of root reinnervation. These results suggest that implantation of a single ventral root may provide neurotropic effects to injured neurons at the site of lesion as well as in the adjacent spinal cord segments. Our findings may be of translational research interest for the development of surgical repair strategies after multi-level conus medullaris and cauda equina injuries, in which fewer ventral roots than spinal cord segments may be available for implantation.     

Bronchogenic cyst of the stomach masquerading as benign stromal tumor

Gastric bronchogenic cyst is an extremely rare lesion. A 62-year-old Korean woman presented with an incidentally found perigastric nodule. An abdominal ultrasonogram and computed tomography showed a 1.5 cm solid, homogeneous, low-density nodule in the hepatogastric ligament near the gastric lesser curvature. At the time of surgery, a soft 1.7 cm nodule was found in the lesser curvature side just below the gastroesophageal junction, and a gelatinous mass was protruded from the nodule during the resection. Microscopically, the gastric subserosa showed cystic structures lined by pseudostratified ciliated columnar epithelium, seromucinous gland, connective tissue and complete layers of smooth muscle bundles. Neither cartilage nor gastrointestinal epithelium was identified. A mucocele was found near the cyst and foamy cells and faintly bluish mucinous substances were found near the cystic structures. All the mucinous substances were stained by alcian blue at pH 2.5 with varying degrees of staining intensity. The present case is the smallest reported cyst among the gastric bronchogenic cysts, and these lesions are unusually located at the lesser curvature side of the stomach. Although these lesions are very rare, gastric bronchogenic cyst should be included in the differential diagnosis of gastric wall mass.     

Neurenteric cyst: case report and a review of the potential dysembryology

We report a 13-year-old female who presented to an outside emergency room following blunt trauma to the head. As part of her evaluation, an unenhanced CT of the face and radiographs of the cervical spine were performed. She was referred to our clinic with "abnormal" imaging. Radiographs revealed dysmorphic cervical spine and ventral clefting of the vertebral bodies from C7-T2. A subsequent MRI was obtained that demonstrated a large spinal cord mass. MRI demonstrated an unenhanced intramedullary mass of the cervicothoracic spinal cord measuring approximately 1.5 cm in greatest axial diameter. Surgical exploration of her intraspinal mass revealed a neurenteric cyst. Spinal neurenteric cysts are categorized in the spectra of occult spinal dysraphism and most likely arise from incomplete separation of the developing notochord and foregut in the embryo. We discuss potential etiologies for the formation of such cysts. The clinician should consider neurenteric cysts in their differential diagnosis of patients with spinal dysraphism and/or intraspinal masses.     

Descriptive study of the differences in the level of the conus medullaris in four different age groups

In performing neuraxial procedures, knowledge of the location of the conus medullaris in patients of all ages is important. The aim of this study was to determine the location of conus medullaris in a sample of newborn/infant cadavers and sagittal MRIs of children, adolescents, and young adults. The subjects of both the samples were subdivided into four developmental stages. No statistical difference was seen between the three older age groups (P > 0.05). A significant difference was evident when the newborn/infant stage was compared with the other, older stages (P < 0.001 for all comparisons). In the newborn/infant group the spinal cord terminated most frequently at the level of L2/L3 (16%). In the childhood stage, the spinal cord terminated at the levels of T12/L1 and the lower third of L1 (21%). In the adolescent population, it was most often found at the level of the middle third of L1 and L1/L2 (19%). Finally, in the young adult group, the spinal cord terminated at the level of L1/L2 (25%). This study confirmed the different level of spinal cord termination between newborns/infants less than one‐year‐old and subjects older than one year. In this sample the conus medullaris was not found caudal to the L3 vertebral body, which is more cranial than the prescribed level of needle insertion recommended for lumbar neuraxial procedures. It is recommended that the exact level of spinal cord termination should be determined prior to attempting lumbar neuraxial procedures in newborns or infants. Clin. Anat. 28:638–644, 2015. © 2015 Wiley Periodicals, Inc.     

An investigation of the conus medullaris termination level during the period of fetal development to adulthood.

The spinal cord fills the length of the vertebral canal at the early period of intrauterine term. It is reported to extend to the level of the third lumbar vertebra at birth, because the vertebral column is growing more rapidly in the longitudinal direction than the spinal cord. The present investigation aimed to determine the changes in the termination level of conus medullaris (TLCM) from fetus to adulthood in a total of 285 individuals who had no defects in the central or peripheral nervous system, and were obtained from our Faculties of Medicine and Konya Maternity Hospital between 1992-1995. The age distribution was as follows: 36 fetuses, 20 prematures and 50 neonates, 51 children aged 1 to 7 years and 128 adults aged 15 to 68 years. In this study, for fetuses, prematures, neonates and children the TLCM was determined using ultrasonography. In addition, microdissection was used in fetuses to confirm the results obtained from the above technique. Also, magnetic resonance imaging was used in adults. During fetal life the end of the conus altered its levels from S5 to L3 vertebrae. The tip of the conus medullaris of the prematures and neonates ranged from L1 to L3 vertebrae. The tip of the conus medullaris in the children lay between the Th12 and L3 vertebrae, and in the adults it was between the Th12 and L2 vertebrae. There were slight differences between the prematures and neonates in terms of the TLCM. We concluded that there are differences in the TLCM between the age groups and therefore, especially in prematures and infants the determination of the tip of conus medullaris might be important for preventing postoperative neurological complications.     

Spinal dysraphism: genetic relation to neural tube malformations.

The families of 207 index patients treated for spinal dysraphism at The Hospital for Sick Children were studied to discover whether the condition was aetiologically related to the classical neural tube malformation--spina bifida cystica and anencephaly. The index patients had all had a tethered conus medullaris and one or more of a variety of anomalies of the spinal cord, vertebrae, or skin overlying the vertebral column. Of 364 sibs of index patients, 9 had an encephaly and 6 spina bifida cystica, a pro-proportion of 4.12%. This approximates to the proportion of sibs affected by neural tube malformations in the London region when the index patients themselves have spina bifida or anencephaly. It is, therefore, appropriate that the mothers of children with spinal dysraphism should be offered prenatal screening for neural tube malformations.     

Bronchogenic cyst in the hepatogastric ligament masquerading as an esophageal mesenchymal tumor: a case report

Lesions occur in hepatogastric gap is common, but most of these lesions are from stomach, lower esophagus and pancreas lesions extending or transferring to the gap. Lesions occurred in the hepatogastric ligament are rare, especially bronchogenic cysts in the hepatogastric ligament. So far, there were no relevant cases reported. Here, we report a case of bronchogenic cyst in the hepatogastric ligament that masquerading as an esophageal mesenchymal tumor. A 24-year-old young man presented with abdominal bloating was diagnosed as esophageal mesenchymal tumor in previous hospital by gastroscopy, endoscopic ultrasonography (EUS) and computed tomography (CT) examination. For the sake of endoscopic submucosal dissection (ESD), the man was transferred to our hospital. During surgery, we found no lesions in lower esophagus, but external pressure ridge lesions in the distal esophagus right side wall. Then laparoscopic surgery and pathology confirmed as bronchogenic cyst in the hepatogastric ligament. We report what is, to the best of our knowledge, the first case of a bronchogenic cyst in the hepatogastric ligament masquerading as an esophageal mesenchymal tumor.     

Retroperitoneal bronchogenic cyst presenting as an adrenal mass

Subdiaphragmatic bronchogenic cysts are rare, and those located retroperitoneally are exceptional. A review of the English-language literature revealed only three reported cases. We describe an additional case of a retroperitoneal bronchogenic cyst that presented uniquely as a symptomatic adrenal mass and discuss the cases of subdiaphragmatic bronchogenic cysts reported in the English-language literature.     

Sacral intraspinal bronchogenic cyst: a case report

Intraspinal bronchogenic cysts are rare congenital cystic lesions. In all the reported cases, the cysts have been located in the cervical, upper thoracic or thoracolumbar segments. We report the case of an intraspinal bronchogenic cyst in the sacral location. We present the case of a 5-month-old female with a skin dimple in the midline over the sacral vertebra. Magnetic resonance image of the lumbar and sacral vertebra revealed a dermal sinus tract and an epidural cystic mass at the S2 level. The patient underwent the removal of the dermal sinus tract and the cyst. The cystic mass was shown to be connected to the subarachnoid space through a slender pedicle from the dura. The cyst was diagnosed to be a bronchogenic cyst based on the results of the histopathological examination. We conclude that intraspinal bronchogenic cysts may appear in the sacral location.     

Spinal cord termination in adult Africans: Relationship with intercristal line and the transumbilical plane

The level of cord termination and level of vertebral intersection of intercristal line and transumbilical plane (TUP), frequently used landmarks, show ethnic variation. The relationship of the spinal cord termination to these lines is vital in spinal surgery and anesthesia, but data on these parameters are scarce in the African population. The purpose of this work is to determine the level of cord termination and establish its relationship with intercristal line and TUP. One hundred and twelve specimens obtained from the department of Human Anatomy at the University of Nairobi were used in this study. The conus medullaris was exposed by laminectomy and its vertebral level together with those of intercristal line and TUP recorded. The distance of conus medullaris from intercristal plane was measured in millimeters. Data obtained were coded and analyzed using Statistical Package for Social Sciences (SPSS) for windows version 16.0 Chicago, Illinois, 2002. Student's t‐test was used in the statistical assessment of gender differences. A P value of less than 0.05 was considered significant. The median level of termination of the cord was the upper third of L2, 51.9% of cases terminating below this. There was no statistically significant gender difference in the level of termination of the cord. The intercristal plane passed through L4/L5 disc (70.9%) and below (29.1%). The TUP corresponded with intercristal line in 78.2% of subjects. The mean distance of the spinal cord termination from intercristal line was 99 ± 24 mm. The spinal cord terminates at or below the upper third of L2. Care should be exercised during lumbar punctures and spinal epidural anesthesia among Africans. Intecristal line and TUP are safe landmarks to use in location of conus medullaris. Clin. Anat. 23:563–565, 2010. © 2010 Wiley‐Liss, Inc.     

What is the true location of Jacoby's line?

We investigated 60 cadavers to examine the accurate location of conus medullaris, lower end of dura mater, and intercrestal line (Jacoby's line). Jacoby's line was established using the method described by Jacoby GW (1899). Then the intersection of this line and vertical line through the spinous processes was marked by a fine nail. After a laminectomy was performed to expose the spinal cord, spinal level of the conus medullaris, the lower end of dura mater, and Jacoby's line were examined, and marked and recorded these locations of nailing. In 2 of 48 cases (2.1%), Jacoby's line passed through the level superior to 3rd lumber vertebra (L3). The Conus medullaris was at the level beneath L3 in 2 of 57 cases (3.5%). And lower end of dura mater was located beneath the superior margin of S3 in 1 of 28 cases (3.6%). When Jacoby's line is used as a guide-point, the site for subarachnoid puncture is established in the L3-4 interspinous space. But when we consider the risk of spinal cord injury cannot be completely excluded, in case that conus medullaris is located inferior to L3 in 3.5% of cases. Conseqently, it should be important to avoid performing subarachnoid puncture at L2-3 or higher lumbar vertebral level especially in the elderly.     

Foramen magnum arachnoid cyst induces compression of the spinal cord and syringomyelia: case report and literature review

It is very rare that a foramen magnum arachnoid cyst induces compression of the spinal cord and syringomyelia, and currently there are few treatment experiences available. Here we reported the case of a 43-year-old male patient who admitted to the hospital due to weakness and numbness of all 4 limbs, with difficulty in urination and bowel movement. MRI revealed a foramen magnum arachnoid cyst with associated syringomyelia. Posterior fossa decompression and arachnoid cyst excision were performed. Decompression was fully undertaken during surgery; however, only the posterior wall of the arachnoid cyst was excised, because it was almost impossible to remove the whole arachnoid cyst due to toughness of the cyst and tight adhesion to the spinal cord. Three months after the surgery, MRI showed a reduction in the size of the arachnoid cyst but syrinx still remained. Despite this, the symptoms of the patient were obviously improved compared to before surgery. Thus, for the treatment of foramen magnum arachnoid cyst with compression of the spinal cord and syringomyelia, if the arachnoid cyst could not be completely excised, excision should be performed as much as possible with complete decompression of the posterior fossa, which could result in a satisfying outcome.     

Subphrenic bronchopulmonary foregut malformation with pulmonary-sequestration-like features

A retroperitoneal bronchopulmonary foregut malformation in a 62-year-old man is reported. The lesion was composed of mature lung tissue with randomly distributed bronchial structures and ciliated epithelium-lined cysts, some of which were lined with gastric mucosa. The histological features of this lesion were of both pulmonary sequestration and a bronchogenic, or foregut, cyst, and thus were a unique example of bronchopulmonary foregut malformation with pulmonary differentiation. This case is important in understanding the pathogenesis of foregut anomalies (i.e. bronchopulmonary foregut malformations), which range from pulmonary sequestrations to bronchogenic cysts and foregut duplication cysts.     

Caudal end of the rat spinal dorsal horn

We have previously demonstrated that the transformation of the caudal spinal cord through the conus medullaris to the filum terminale takes place in three steps. In the conus medullaris the twin layers of CGRP-immunoreactive and IB4-labeled primary afferent fibers as well as the translucent portion of the superficial dorsal horn equivalent to the substantia gelatinosa discontinue before the complete removal of the dorsal horn. Parallel with these changes VGLUT1-immunoreactive myelinated primary afferent fibers arborize not only in the deep layers but also in the entire extension of the remaining dorsal horn, while scattered CGRP fibers still remains at the margin of and deep in the dorsal horn. PKCgamma-immunoreactive dorsal horn neurons discontinue parallel with the disappearance of the IB4-labeled nerve fibers. These observations suggest that in the dorsal horn certain neurons are linked to the substantia gelatinosa, while others are substantia gelatinosa-independent neurons.     

Bronchogenic cyst in the abdomen

Summary A bronchogenic cyst was found in the abdomen, in the retroperitoneum adjacent to the superior pancreatic body. The cyst was unilocular and contained about 100 ml pale yellow mucinous fluid. Microscopic examination revealed a pseudostratified columnar ciliated or cuboidal epithelium, seromucous glands, smooth muscle and cartilage, the distinctive features of bronchogenic cysts. This aberrant location of the cyst is explicable if abnormal buds of the tracheobronchial tree are pinched off and migrate into the abdomen in an early embryonic stage before the canal linking the abdominal with the thoracic cavity is closed by fusion of the future components of the diaphragm.     

Carcinoid tumor arising in a thymic bronchogenic cyst associated with thymic follicular hyperplasia

Bronchogenic cysts are congenital malformations of the embryonic tracheobronchial tree and are the most common cause of mediastinal cysts, encountered mainly in pediatric patients and young adults. Conservative treatment has been proposed for asymptomatic patients. However, malignant transformation occurs occasionally and the clinicopathological features of secondary malignancy are not well characterized. In this report, we present a carcinoid tumor found in the thymic bronchogenic cyst of a 41-year-old female complaining of mild chest pain. The thymic tissue also shows follicular lymphoid hyperplasia. Atypical imaging features were found in her chest computed tomography. Additional 22 cases of bronchogenic cysts with malignant transformation were also reviewed from the literature. The clinicopathological data were summarized. The tumorigenesis of these tumors is unclear, but bronchogenic cysts of the lung may undergo malignant changes at a younger patient age and more frequently than mediastinal ones. Carcinoid tumors were also seemingly overrepresented in mediastinal cases. Some atypical imaging features may serve as clues for early detection and guide clinical management.     

Plasma cell neoplasm in conjunction with glioblastoma of the conus medullaris

We report a plasma cell neoplasm in conjunction with a glioblastoma multiforme (GBM) of the conus medullaris in a 42-year-old man. Glioblastoma is a World Health Organization (WHO) grade IV neoplasm that requires surgical intervention, radiation, and possibly chemotherapy. Astrocytomas of the spinal cord are rare neoplasms, with intramedullary glioblastomas comprising only 1% to 3%. Plasma cell neoplasms result from monoclonal proliferation of mature B cells; they have been reported as a primary malignancy with gliomas arising after treatment. Secondary plasma cell neoplasms arising within glioblastomas have not previously been described. However, there have been reports of glioblastomas related to other plasma cell and hematopoietic diseases such as Waldenstrom's macroglobulinemia and myeloid sarcomas.     

Foregut cyst of the oesophageal wall in a cynomolgus monkey (Macaca fascicularis)

Congenital oesophageal cysts of foregut origin are rare in animals and human beings. This report describes a case in a 4-year-old cynomolgus monkey with no clinical symptoms. The cyst, which was located within the oesophageal submucosal tissue near the mid-point of the oesophagus, was lined with pseudostratified ciliated epithelium and had a thin layer of submucosal tissue. The cyst was surrounded by a smooth muscle layer which was partly intermingled with the circular muscle layer of the oesophagus. The muscularis mucosae of the oesophagus was not shared with the cyst wall. Simple tubular glands were present, opening into the cyst lumen. No communication between the cyst lumen and the oesophagus was observed. Cartilaginous tissue, which is a diagnostic feature of bronchogenic cysts, was not identified in the cyst wall. On the basis of the histopathological features, a foregut cyst of the oesophagus was diagnosed.     

Reimplantation of avulsed lumbosacral ventral roots in the rat ameliorates injury-induced degeneration of primary afferent axon collaterals in the spinal dorsal columns

Injuries to the cauda equina/conus medullaris portion of the spinal cord can result in motor, sensory, and autonomic dysfunction, and neuropathic pain. In rats, unilateral avulsion of the motor efferents from the lumbosacral spinal cord results in at-level allodynia, along with a corresponding glial and inflammatory response in the dorsal horn of the spinal cord segments immediately rostral to the lesion. Here, we investigated the fate of intramedullary primary sensory projections following a motor efferent lesion. The lumbosacral (L6 and S1) ventral roots were unilaterally avulsed from the rat spinal cord (VRA; n=9). A second experimental group had the avulsed roots acutely reimplanted into the lateral funiculus (Imp; n=5), as this neural repair strategy is neuroprotective, and promotes the functional reinnervation of peripheral targets. A laminectomy-only group served as controls (Lam; n=7). At 8 weeks post-lesion, immunohistochemical examination showed a 42% reduction (P<0.001) in the number of RT97-positive axons in the ascending tracts of the dorsal funiculus of the L4-5 spinal segment in VRA rats. Evidence for degenerating myelin was also present. Reimplantation of the avulsed roots ameliorated axon and myelin degeneration. Axons in the descending dorsal corticospinal tract were unaffected in all groups, suggesting a specificity of this lesion for spinal primary sensory afferents. These results show for the first time that a lesion restricted to motor roots can induce the degeneration of intramedullary sensory afferents. Importantly, reimplantation of the lesioned motor roots ameliorated sensory axon degeneration. These data further support the therapeutic potential for reimplantation of avulsed ventral roots following trauma to the cauda equina/conus medullaris.