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A 1-year-old boy with a bronchopulmonary foregut malformation presented with a large mediastinal bronchogenic cyst associated with pulmonary sequestration, a cervical esophageal duplication cyst, a bronchial communication between these cysts, and 2 small bronchogenic cysts around the communication. These lesions were resected followed by an uneventful recovery.
We report the first case of pachyonychia congenita (PC) associated with both eruptive vellus hair cyst (EVHC) and epidermoid cyst. The patient is a 12-year-old Japanese girl who presented with two natal teeth at birth. She had thickening and discoloration of the fingernails and toenails, plantar hyperkeratosis, palmar-plantar hyperhidrosis and multiple cutaneous cysts. Histologic examination revealed EVHC and epidermoid cyst.
The expression of mRNA encoding the inflammatory cytokines interleukin-1alpha (IL-1alpha), interleukin-1beta (IL-1beta), interleukin-6 (IL-6), interleukin-8 (IL-8) and tumor necrosis factor alpha(TNF-alpha) have been examined in radicular cysts by in situ hybridization. Furthermore, the biological activity of the contents of radicular cysts (RCC) has been assayed by adding extracts of RCC to cultured human gingival fibroblasts (HGFs) and analyzing the culture medium for the release of inflammatory cytokines. In the epithelial layer, keratinocytes expressed all cytokine mRNAs examined at various levels. Basal layer cells expressed mRNA for each cytokine. In the subepithelial granulation tissue of the cysts, fibroblasts and macrophages expressed mRNA for IL-6, IL-8, IL-1beta and TNF-alpha mRNA at varying levels; especially clear expression of TNF-alpha and IL-1beta mRNA was detected on macrophages. The infiltrating lymphoid cells, largely composed of T cells and plasma cells, expressed these cytokine mRNAs, especially those encoding IL-6 and IL-8, at various levels. In vitro analysis indicated dose-dependent release of both IL-6 and IL-8 by HGFs in response to RCC. After heating to 100 degrees C for 10 min, RCC almost completely failed to stimulate IL-6 release from HGFs. Furthermore, anti-IL-1beta antibody (neutralization test) did not prevent the stimulation of IL-6 release by RCC. Significant amounts of IL-6 and IL-8 were detected in RCC in two cases, and a trace amount of IL-1beta was detected in one case. This study demonstrated the wide expression of mRNA encoding inflammatory cytokines in radicular cyst tissues, and RCC itself was capable of stimulating IL-6 and IL-8 production from HGFs.
A case of a 16-year-old male with both a nasal dermoid sinus cyst (NDSC) and a third ventricle colloid cyst is presented. The NDSC was excised via a single-stage combined intracranial-extracranial approach and the third ventricle colloid cyst was resected endoscopically. The pathogenetic theories of NDSC and third ventricle colloid cyst are discussed, and an embryological explanation for the simultaneous development of the two lesions in this patient is explored. This case is best classified among congenital developmental malformations in a category we propose to call 'anterior neuropore corridor defects.'
The distinction among craniopharyngioma (CR), Rathke's cleft cyst (RCC), and intrasellar arachnoid cyst (AC) remains a difficult preoperative problem. Accurate diagnosis of these rare pituitary lesions is important to determine the type of treatment and predict prognostic outcome. The majority of the literature describes the clinical manifestations and management of only one of CR, RCC, or AC, rendering comparisons difficult. We conducted a study to 1) investigate distinguishing preoperative clinical, biochemical, and radiographic features of patients with CR, RCC, and AC; and 2) identify clinicopathological features that independently predict recurrence in CR and RCC in adults. Fifty-two adult patients included 21 patients with CR (mean age at initial surgery, 35 +/- 14 yr), 26 patients with RCC (mean age, 37 +/- 14 yr), and 5 patients with AC (mean age, 53 +/- 12 yr). Mean follow-up duration was 70 +/- 13 months. Patients with CR presented with hypopituitarism in 95% of cases and hyperprolactinemia in 38%. These patients also had more preoperative neurological deficits (67%), ophthalmological complaints (67%), and significantly higher psychiatric manifestations (33%; P = 0.003) than those with RCC or AC. Patients with AC presented with headaches (60%), visual field deficits (60%), or impotence (50%) in the absence of other specific endocrine dysfunction symptoms. Using biochemical criteria, the percentage of patients with two or more pituitary hormonal axes impaired preoperatively was 67% for CR and 62% for RCC, significantly greater (P = 0.03) than that for the AC patients who had pituitary dysfunction of only one axis. The composition of CR lesions was cystic (38%), solid (10%), or mixed solid and cystic (43%). Patients with RCC or AC groups had a significantly greater proportion (P = 0.006) of purely cystic lesions (88% and 100%, respectively). Calcification detectable on computed tomographic scanning was present in 87% of patients with CR, a significantly greater proportion (P < 0.001) compared to those with RCC (13%) or AC (0%). No significant differences were found between the groups based on computed tomography density, the presence of postcontrast enhancement, or magnetic resonance imaging. Recurrence rate was 62% for CR, 19% for RCC, and 20% for AC. Surgical intervention statistically improved most neurological, ophthalmological, and psychiatric manifestations; in contrast, galactorrhea, menstrual dysfunction, and diabetes insipidus (52% CR; 31% RCC) did not improve or became worse postoperatively. A significantly higher percentage of patients with CR required postoperative hormone replacement. Similarly, there was a biochemical trend suggesting that a smaller proportion of patients with CR improved in at least one pituitary axis after surgery (P = 0.08) compared to those with RCC or AC. There was a positive correlation between cyst size and recurrence rate (r = 0.689; P < 0.01) and between cyst size and time to recurrence (r = 0.582; P = 0.037) for all three groups. We describe the largest clinical, biochemical, radiographic, and histological series of adult patients with cystic disease of the sella turcica. Patients with AC tended to be older at initial diagnosis than CR or RCC patients. Mass effects, such as visual problems and headaches, are common symptoms of all three cystic lesions, but psychiatric deficits favor a diagnosis of CR. Calcification or solid components on neuroimaging characterize CR. Endocrinological deficits, especially diabetes insipidus, had the worst prognosis after surgery. Low recurrence rates can be expected for RCC and AC. These data have direct implications for the management and monitoring of patients with cystic lesions of the sella turcica.
BACKGROUND: Mesenteric cysts are rare intraabdominal lesions of childhood that may vary in presentation from an asymptomatic mass to an acute abdomen. CASE: We encountered a rare case of peritonitis caused by a ruptured, infected mesenteric cyst of the mesocolon in a female child that had been initially interpreted as a ruptured ovarian cyst. CONCLUSION: When confronted with a large unilocular or septated echolucent mass in the lower abdomen in female children, gynecologists should consider a mesenteric cyst in the differential diagnosis.
Mesenteric cystic tumors are very rare. They may simulate the ovarian cysts. We report a case of a 38 year-old woman with large cystic tumor suggesting ovarian cyst. The patient was operated on and we found that genital organs were normal. Besides there was a large cyst of small intestine mesentery with 10 liters of clear fluid. The cyst was removed. Histological material showed mucinous cystadenoma.
Soft tissue ganglion cysts are the most common benign tumours of the wrist; their pathogenesis remains controversial. We prospectively screened the radiographic appearance of the wrists of 51 patients presenting to a single surgeon with dorsal wrist ganglions during a one-year period. Postero-anterior and lateral radiographs were systematically performed looking for possible associated intraosseous ganglion cysts. There were 51 dorsal soft tissue ganglion cysts in 51 patients. We detected 29 associated intraosseous ganglia in 24 patients (47%): 16 ganglia in the lunate bone (55%), 5 in the capitate bone, 7 in the scaphoid and 1 in the trapezoid. Mean size of the intraosseous ganglia was 3 mm (range, 2 to 5 mm). This high prevalence of intraosseous ganglia in association with soft tissue ganglia has to our knowledge never been reported previously. A common aetiology for these two types of ganglion cysts may explain this high association rate.
A 25-year-old man underwent extirpation of a cystic mass of the esophageal wall. It proved to be an unilocular cyst surrounded by a pseudo-stratified, ciliated columnar epithelium with occasional goblet cells. Outside of the epithelium, a smooth muscle layer was seen and a small foci of seromucous glands were scattered in the wall. Electron microscopic observations of the epithelial layer revealed 5 types of the cells: ciliated columnar cells, mucous cells, brush cells, intermediate cells, and basal cells. Our results suggest that electron microscopic observations are useful for distinguishing the bronchogenic cyst from the esophageal cyst. Nine cases of para-esophageal bronchogenic cysts that have been reported in Japan also are briefly reviewed.
The case of a woman patient with an extended prepapillary choledochal cyst and pancreaticobiliary reflux shows that it is necessary to consider the possibility of a choledochal cyst on detecting a cystic structure in the head of the pancreas (condition after acute pancreatitis); besides this, differential diagnosis will have to consider a pancreatic pseudocyst, hydrops of the gallbladder, a retroperitoneal cyst or even vascular aneurysm. If necessary, the physician should take suitable diagnostic measures before performing any percutaneous cyst drainage which he may have planned, since this may entail the risk of biliary peritonitis.
Nineteen transvaginal, sono-guided ovarian cyst punctures were performed in 18 patients. These patients, who were high surgical risks, were diagnosed to possess benign ovarian tumors as revealed with sonography. Aspiration curettage of the inner surface of the cyst present during aspiration was also performed to facilitate cytologic diagnosis. The recurrence rate was 11.1%. Sono-guided transvaginal cyst aspiration has been demonstrated here to be the treatment of choice for dealing with an ovarian cyst in patients at high surgical risk.
Aprosencephaly is a very rare brain malformation that occurs in isolated and sydromatic forms. The syndromatic form has been named "XK-aprosencephaly", and is characterized by near total absence of prosencephalon with a midline oculofacial defect similar to the most severe forms of holoprosencephaly, in association with limb and genital anomalies. We present a case of syndromatic aprosencephaly with absence of thumb and abnormal external genitalia. A previously undescribed finding was a Rathke's cleft cyst. Two other cystic structure were also identified--an ependymal cyst, which may represent a dorsal cyst as in holoprosencephaly, and a pigmented epithelial cyst, which may represent a rudimentary eye. Additional findings were extensive calcific vasculopathy in the rudimentary prosencephalon, absence of pituitary gland, forking of the aqueduct of Sylvius and marked cerebellar hypoplasia. Since calcific vasculopathy is a common accompaniment of other inflammatory diseases of the central nervous system, its presence in this case suggests that destructive processes may be involved in the genesis of some cases of aprosencephaly.
We describe two patients with cyst-like lesions of bone that developed at sites of healed or healing fractures. One case showed histological features of a unicameral bone cyst, which, to the best of our knowledge, is a previously unreported finding in a post-traumatic cyst. It is suggested that there are two principal clinical and radiological types of post-traumatic cyst, of which each of our cases represents an example: (1) asymptomatic transient cortical lesions, found only in children, and (2) more central expanding lesions, found in a wider age group and associated with pain, swelling, and pathological fractures.
Arachnoid cysts (ACs) are congenital cystic brain malformations associated with epilepsy. The purpose of this study was to determine the effect of surgical intervention of ACs on cyst size and seizure outcome. We reviewed the world's medical literature dealing with surgically treated ACs in epilepsy patients. Our study included only cases, in which the relationship between pre-and postoperative CT-size of the AC and seizure outcome was described. We also included six patients with ACs and epilepsy treated surgically at the University of Mainz. We analyzed postoperative AC size and seizure outcome with respect to mode of operation, cyst location, and patients' age. A total of 76 patients was reviewed. Sixty (79%) patients had a smaller AC postoperatively. Forty-six of those 60 (76.6%) experienced seizure improvement. Thirteen patients (21.6%) remained unchanged and one patient (1.8%) worsened. In 16 of the 76 patients (21%) the postoperative AC size was unchanged. Eight of those 16 patients improved. Six patients (37.5%) remained unchanged and two (12.5%) worsened. A positive correlation between postoperative AC size and seizure outcome was well demonstrated among patients treated by cyst fenestration, needle aspiration, or internal shunting. Among patients treated by cystoperitoneal shunting this direct correlation was less clear. Seizure outcome correlates directly with postoperative AC size. Seizure reduction is associated with decreased AC size postoperatively and depends on the mode of operation. Based on these data we would expect that patients with epilepsy secondary to ACs would demonstrate improved seizure control with lower AC volume. Conversely, we might expect increasing AC size to correlate with worse seizure control. This relationship may guide physicians in efficacy and timely patient management.
We describe two girls with abdominal cysts which demonstrated the "double wall" or "muscular rim" sign. On the basis of this, the diagnosis of duplication cyst was made in each case. At surgery, both were found to have ovarian cysts, and histology revealed prominent haemorrhage within the cyst wall. To our knowledge, there has been no previous report of a false-positive "double wall sign".
Primary splenic cysts are a rare finding. Some are large and require surgical removal. The epidermoid type has an epidermal lining, and prevention of recurrence is dependent on complete resection of the cyst wall, preserving the splenic tissue. Several open, laparoscopic, or percutaneous procedures have been proposed with or without splenic resection, but few give completely satisfactory results. Five consecutive splenic epithelial cysts in pediatric patients were treated by parenchyma-sparing complete removal of the cyst wall, which was gently peeled off the splenic tissue without major bleeding in all but one case. Long-term follow-up showed freedom from recurrence.
Congenital bile duct cysts are observed in any part of the bile duct from the liver to the duodenum. Reports of cases of cancer arising from it are increasing. Excision of the choledochal cyst seems to be the treatment of choice and partial resection of the intrahepatic cyst followed by intrahepatic cystoenterostomy at the porta hepatis is necessary for type IV-A cysts.
Various prostaglandins, particularly PGE2 and PGI2, appear to play a major role in osteolytic processes. Radiochromatographic studies have demonstrated that 6-oxo-PGF1-alpha is a major product of exogenously added arachidonic acid in human dental cysts. As platelets may also act as inflammatory cells, platelet-derived growth factor might also have a PGI2-stimulating influence in such cysts. Eleven human dental cysts were examined by a radioimmunoassay and bioassay which can show PGI2 synthesis in human dental cysts without addition of PDGF. Incremental PDGF addition caused a highly significant increase in the rate of PGI2 synthesis. PGDF thus stimulates PGI2 synthesis in chronic inflammatory processes in vitro and may thereby elicit or accelerate osteolysis.