Traumatic sacral dermoid cyst rupture with intracranial subarachnoid seeding of lipid particles: illustrative case

BACKGROUND Intracranial deposits of fat droplets are an unusual presentation of a spinal dermoid cyst after spontaneous rupture and are even more uncommon after trauma. Here, the authors present a case with this rare clinical presentation, along with a systematic review of the literature to guide decision making in these patients. OBSERVATIONS A 54-year-old woman with Lynch syndrome presented with severe headache and sacrococcygeal pain after a traumatic fall. Computed tomography of the head revealed multifocal intraventricular and intracisternal fat deposits, which were confirmed by magnetic resonance imaging (MRI) of the neuroaxis; in addition, a ruptured multiloculated cyst was identified within the sacral canal with proteinaceous/hemorrhagic debris, most consistent with a sacral dermoid cyst with rupture into the cerebrospinal fluid (CSF) space. An unruptured sacral cyst was later noted on numerous previous MRI scans. In our systematic review, we identified 20 similar cases, most of which favored surgical treatment. LESSONS Rupture of an intraspinal dermoid cyst must be considered when intracranial fat deposits are found in the context of cauda equina syndrome, meningism, or hydrocephalus. Complete tumor removal with close postoperative follow-up is recommended to decrease the risk of complications. CSF diversion must be prioritized if life-threatening hydrocephalus is present.

The predicting factors for indication of surgery in patients with hemoperitoneum caused by corpus luteum cyst rupture

The aim of the study was to determine the risk factors for surgery in patients with hemoperitoneum caused by corpus luteum cyst rupture. A retrospective review of medical records of 155 patients diagnosed with hemoperitoneum caused by corpus luteum cyst rupture was conducted between January 2010 and March 2015. The patients were divided into two groups: surgical and conservative management. The differences in characteristics between the two groups were compared. The indicators that determine the need of a surgery at the initial visit were also compared between the two groups. Initial hemoglobin level was lower (11.3 ± 1.4 g/dL vs. 12.2 ± 1.2 g/dL; p = 0.007) in the surgery group. There were significant differences in posterior cul-de-sac (PCDS) fluid collection depth (6.2 ± 2.5 cm vs. 4.5 ± 1.6 cm, p = 0.000), total fluid collection depth (8.4 ± 1.8 cm vs. 6.5 ± 2.1 cm, p = 0.000), single deepest pocket depth (6.7 ± 2.2 cm vs. 5.1 ± 1.5 cm, p = 0.006), liver-dome fluid (78.9% vs. 35.6%; p = 0.002), and estimated intrapelvic bleeding amount (325 ± 250 cc vs. 206 ± 146.5 cc, p = 0.002). The extravasation over grade 2 was more often in surgery group (68.4% vs. 30.1%; p = 0.001). PCDS fluid collection depth, the presence of liver-dome fluid, and the severity of contrast extravasation through ultrasonography and computed tomography are good indicators for determining the management of hemoperitoneum resulting from corpus luteum cyst rupture in healthy women.

Biliary peritonitis due to liver cyst rupture in autosomal dominant polycystic kidney disease

Background Autosomal dominant polycystic kidney disease (ADPKD) is the most frequent genetic kidney disease and polycystic liver disease is its major extrarenal manifestation, however biliary peritonitis due to a liver cyst rupture is extremely rare. Case presentation The patient was a 71-year-old Japanese woman who was diagnosed with ADPKD 3 years previously and developed right abdominal pain suddenly 1 month previously. As abdominal computed tomography (CT) showed a ruptured liver cyst in the right lobe, she was admitted to our hospital. Her symptoms improved with conservative management and she was discharged from the hospital after 1 week. Although she was asymptomatic for a while, she noticed abdominal distension and general malaise at 1 month after hospital discharge. Since abdominal CT showed massive ascites, she was admitted to our hospital again. A physical examination revealed abdominal distention without tenderness. Her serum creatinine, alkaline phosphatase, γ-glutamyl transpeptidase, total bilirubin, and CA19-9 were elevated. Abdominal paracentesis revealed amber transparent ascites and the bilirubin and CA19-9 concentrations were high. She was diagnosed with biliary peritonitis due to a ruptured liver cyst. Hemodialysis treatment was initiated with drainage of the ascites. The outflow of the ascites was no tendency to decrease and drip infusion cholangiography (DIC)-CT revealed a communication between the ruptured cyst and an intrahepatic bile duct. On day 31, she was transferred to a university hospital and abdominal surgery was performed. After removing the necrotic roof of the ruptured cyst on the right liver lobe, the orifice of the bile leakage was sutured. Cholecystectomy was performed and cholangiography showed no stones in the common bile duct. Abdominal CT one month after the operation showed no recurrence of ascites and she was discharged on day 49. Hemodialysis treatment was discontinued immediately after discharge because urine volume increased and her creatinine level decreased. There has been no recurrence of ascites since then. Conclusions While rare, biliary peritonitis can occur in association with the rupture of a liver cyst in ADPKD patients due to communication between the cyst and the intrahepatic bile duct, and DIC-CT should be recommended when biliary cyst rupture is suspected.

Twenty-six years of involvement with cystic echinococcosis: a case report

Introduction Spinal hydatidosis, a zoonotic disease caused by infection with Echinococcus spp. larvae, is rare, but its treatment remains a significant medical challenge. Approximately 70% of patients with spinal hydatidosis have lesions in their liver, 0–15% have lung involvement, and only 0.5–2% have bone involvement. Case presentation Here we report a 38-year-old Iranian man with spinal hydatidosis, who had a history of eight times surgery in over of 26 years due to hydatid cyst in the liver, lungs, and chest wall. At the most recent admission to hospital he presented with chest pain, paraplegia, and urinary incontinence. Magnetic resonance imaging revealed thoracic spinal hydatid disease. He underwent surgery, and the hydatid cysts were completely removed. Lower extremity forces recovered dramatically and completely within 4 weeks. Conclusion Spinal hydatidosis is a rare disease, but it is associated with a high degree of morbidity, mortality, and poor prognosis. Because of the infiltrative nature of hydatid disease, surgery alone is rarely curative. The current case study demonstrates the importance of a suitable surgical approach, adequate intraoperative prophylaxis to prevent cyst rupture, and prolonged complete paraplegia.

Evaluation of the Apoptotic and Scolicidal Effects of Crude and Flavonoid Extracts of Allium noeanum on Protoscolices and Hydatid Cyst Wall

Background: Hydatidosis is a disease, caused by the larval phase of Echinococcus granulosus. It is considered a serious condition, as cyst rupture can release protoscolices into the host body, leading to the formation of secondary cysts. Surgery is the main approach for the treatment of this disease. Some natural compounds, which are safe options with few side effects, have been assessed for their scolicidal activities. Objectives: Allium noeanum is a native herb of Markazi Province, Iran, which has been used for the traditional treatment of certain diseases. However, its anti-parasitic property has not been investigated so far. The present study aimed to evaluate the anti-parasitic property of this plant by evaluating the scolicidal and apoptotic effects of two types of this herbal extract (crude and flavonoid) on hydatid cysts. Methods: The hydatid cysts were obtained from abattoirs, and protoscolices were drained under sterile conditions. The protoscolices suspension and cyst walls were used for scolicidal and immunohistochemical evaluations. In addition, A. noeanum was collected from Shazand Mountains. The crude and flavonoid extracts of this plant were prepared by maceration and chromatography methods, respectively. Immunohistochemistry was conducted for the detection of caspase-3 activity using a commercial kit. Data were analyzed in Excel and SPSS, and statistical significance was defined as P < 0.05. Results: The 100% concentration (0.49 gr/mL) of the crude A. noeanum extract caused the death of 100% of protoscolices. On the other hand, all concentrations of the flavonoid extract led to the death of 100% of parasites. According to the statistical analyses, each type of the extract showed different dose- and time-dependent rates of scolicidal activity (P < 0.001). Immunohistochemistry showed lower caspase-3 activity in cyst walls exposed to flavonoids, compared to those exposed to the crude extract. Conclusions: In the present study, the scolicidal activity of the flavonoid extract of A. noeanum against protoscolices was confirmed. It was also found that the apoptotic effect of the crude extract of this herb was more than its flavonoid extract.

Extensive pulmonary artery embolisation caused by cardiac hydatid cyst rupture

This case study is a rare example of cardiac hydatidosis in a high-income country, where a middle-aged man presented with a ruptured right ventricular cyst causing anaphylaxis, pulmonary emboli and dissemination of Echinococcus throughout the lung. He survived the cyst rupture and underwent cardiac surgery but had incomplete resection and experienced progressive cardiopulmonary hydatidosis despite antihelminthic therapy. As a result, he experienced an array of cardiopulmonary sequelae over his lifespan. This case report highlights rare clinical manifestations of hydatid disease and potential complications of its treatment.