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Venous sinus dilatation syndrome ? A case report
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     [Abstract] A case of multiple venous sinus dilatation is reported in this paper. A 40-year-old women with varicose veins of the both lower extremities accepted the Linton's operation in 1998. Two years later, she felt fatigue, heaviness and swell of the leg again. The examination revealed that varicose veins and multiple small apophysis, 0.4 cm in diameter on both feet. The phlebography revealed string-beads-like dilatations of all the deep venous sinus of the both legs and the remained great and small saphenous veins of the right leg. A large aneurysm (5 cm×7 cm in size) located in the popliteal vein.The parents and 2 brothers of the patient also have varicose veins of the lower extremities, but her brothers refuse to have surgical procedure and her parents passed away. Venous sinus dilatations have not been reported yet in the literature. Perhaps the venous sinus dilatations are a new syndrome. It is just about the normal function of the valves, and due to development bug of the venous wall is presents, mostly the fibrin in the venous wall is lack, so the increased venous pressure makes the part of the venous sinus dilatation. This syndrome may be congenital.

    [key words] venous sinus dilatations,congenital;varicose veins

    INTRODUCTION Venous aneurysm of the lower limbs is rare. As originally described by Schild in 1992, 311 cases were reported from 1939 to 1992. There is no multiple venous sinus dilatation is written. The authors treated a patient who has multiple venous sinus dilatations of lower limbs and underwent three different surgical procedures during 6 years. This report reviews the diagnosis and management of multiple venous sinus dilatations.

    CASE REPORT

    A 40-year-old women with varicose veins of the both lower extremities accepted the Linton's operation in 1998. Two years later, she felt fatigue, heaviness and swell of the leg again. The examination revealed that varicose veins and multiple small apophysis, 0.4 cm in diameter on both feet (Figure 1). The phlebography revealed string-beads-like dilatations of all the deep venous sinus of the both legs and the remained great and small saphenous veins of the right leg. A large aneurysm (5 cm× 7 cm in size) located in the popliteal vein (Figure 2). The parents and 2 brothers of the patient also have varicose veins of the lower extremities, but her brothers refuse to have surgical procedure and her parents passed away.

    TP1

    Figure 1 There were varicose veins and multiple small apophysis (remained),0.4 cm in diameter on both feet (arrow)

    Department of Vascular Surgery, Peking University Third Hospital, Beijing 100083,China

    Correspondence to DONG Guo-xiang,MD.Address: N.49, Garden Road, Haidian District, Beijing 100083,China

    Tel: +86-10-84814556, Fax: +86-10-62017700,E-mail: dong_guoxiang@126.com

    TP2

    Figure 2 The phlebography revealed string-beads-like dilatations of all the deep veins of the both legs and the remained great and small saphenous veins (arrow) of the right leg. A large aneurysm (5 cm×7 cm in size) located in the popliteal vein

    All the varicose veins and the remained great and small saphenous veins of the right leg were removed and the popliteal aneurysm was repaired at second surgical procedure. Ten days later, the patient felt pain in the multiple small apophysis on both feet, and thrombosis of the apophysis veins was detected and partial apophysis veins were removed and thrombi fullfiled the multiple small apophysis. Post-operatively, the patient was found there was a mass in the popliteal vein in 2004. The phlebography was repeated and also revealed string-beads-like dilatatations of all the deep veins. A venous aneurysm (4 cm×6 cm in size) was located in the popliteal vein again (Figure 3).

    TP3

    Figure 3 The repeated phlebography also revealed string-beads-like dilatatations of all the deep venous sinus,A venous aneurysm (4 cm×6 cm in size) was located in the popliteal vein again The popliteal aneurysm was repaired around with ePTFE graft. The aim of the operation is to prevent the thrombosis in the venous aneurysm and to decrease the risk of pulmonary embolism. The patient was advised to wear the compression stockings lifetime.

    DISCUSSION

    Venous aneurysm is rare. Schild et al[1] collected 311 cases of venous aneurysms reported from 1939 to 1992 in the literature. Sixty two percent of the patients suffering from venous aneurysms were less than 40 years of age. The most frequently involved vessels were the internal jugular vein (n=56), the portal vein (n=28), the saphenous vein (n=30), the popliteal vein (n=23), the azygos vein (n=22) and the vena cava superior (n=20). The diameters of the aneurysms were between 1 cm and 16 cm. Symptoms depend on the localization of the aneurysm, but they are unspecific in the majority of cases. Pulmonary embolisms originating from venous aneurysms were seen in 19/311 (6.1%). Due to the diameter of the venous aneurysm is much bigger than normal veins, the flow in the aneurysm is lower than that in normal veins, therefore it is easily to be a cause of pulmonary embolism. Actually, it is reported in many literatures[1~6].One of the purposes to treat venous aneurysms is to prevent thrombosis and pulmonary embolism. So the large aneurysm, the popliteal aneurysm, was repaired again with ePTFE graft around it so as to prevent thrombosis in the aneurysm and to prevent pulmonary embolism, and other small ones were left alone. Also the patient was suggested to wear compression stocking lifetime.

    Venous aneurysm is rare, venous sinus dilates have not been reported yet in the literature. Perhaps the venous sinus dilate are a new syndrome. The phlebography revealed that all the venous sinuses of the lower extremities are dilatations, the function of the venous valves are normal. It is just about the function of the valves, and due to development bug of the venous wall is presents, mostly the fibrin in the venous wall is lack, so the increased venous pressure makes the part of the venous sinus dilatation. This syndrome may be congenital, because the parents and 2 brothers of the patient have varicose veins of the lower extremities, although they have not been treated yet till now. It is in a dark if they have venous sinus dilatations or not, because there are not any evidence of imageology except the patient history.

    REFERENCES

    1. Schild H, Berg S, Weber W, et al.The venous aneurysm. Aktuelle Radiol 1992,2 (2):75-80.

    2. Sigg P, Koella CH, Stobe C, Jeanneret CH.Popliteal venous aneurysm, a cause of pulmonary emdolism. Vasa,2003,32(4): 221-224.

    3. Farah I, Sessa C, Villenur B, et al.Venous aneurysm of unusual localization revealed by a pulmonary embolism. Report of two cases. J Mal Vasc,1999,24(1): 49-52.

    4. Biessaux Y, Van Damme H, Raskinet B, et al.Popliteal venous aneurysm: unusual source of pulmonary embolism. Acta Clini Belg,1994,49(2): 92-94.

    5. Leung JC, Coel M, Ko J, et al.Pulmonary embolism from a popliteal vein aneurysm. Clin Nucl Med,1993,18(1): 7-10

    6. Hurwitz RJ, Geladert H: Thrombosed iliac venous aneurysm: a rare cause of left lower extremety venous obstruction. J Vasc Surg,1989,9(6): 822-824.

    (Editor Jaque )

    REVIEW

    Surgical treatment of insulinoma

    LIU Wei-nan

    Insulinomas are the most common tumors arising from the pancreatic islets of Langerhans.In 1927, Wilder[1] established the association between hyperinsulinism and a functional islet cell tumor.In 1929, Graham[2] achieved the first surgical cure of an islet cell adenoma.

    In the past, delayed or missed diagnosis often resulted in treatment for psychomotor epilepsy or in prolonged psychiatric care.

    In the US insulinomas are the most common pancreatic endocrine tumor. The incidence is about 1 case per 250,000 patient-years or 1~4 per million people. Approximately 50% of islet cell tumors are insulinomas[3]. Internationally, exact data are not available. One source from Northern Ireland reported an annual incidence of 1 case per million persons. This report may be an underestimate. In China,500 cases of insulinoma were reported[4].

    The postoperative morbidity rate in a recently published series was 14%, mainly due to local complications, such as fistula formation after pancreatic resections. The postoperative mortality rate in another series of 117 insulinoma patients was 7.7%[5]. The median survival in metastatic disease to the liver ranges from 16 to 26 months.

    The male-to-female ratio is 2∶3. The median age at diagnosis is about 50 years, except in insulinoma patients with MEN 1, in whom the median age is the mid 20s. In one series, patients with benign disease were younger (mean age of 38 y) than those with metastases (mean age of 52 y)[6].

    Because insulinoma resection achieves cure in 90% of patients, it is currently the therapy of choice[7].

    GENERAL CONCERN OF SURGICAL

    TREATMENT

    The presence of a hormonal syndrome may give rise to a singular problem for surgical strategy, namely, making an early clinical diagnosis, without the presence of an evident lesion at imaging techniques. This is especially true for the most common functioning pancreatic tumors, such as insulinoma. In this situation, when a team of experts reaches a diagnosis, the surgeon can perform a laparotomy with a good certainty of identifying the tumor. The surgical procedures have to be quite rigorous [8,9,10]. After a careful abdominal exploration, the pancreatic gland should be widely exposed using the Kocher maneuver, and dissection of both the superior and the inferior margins of the gland and of the splenic ligament. In this way, the pancreas will become accessible for a bi-digital manual examination and the entire parenchyma can be studied using intra-operative ultrasound with a 10 or 7.5 MHz probe. Insulinomas appear visually as gray-reddish masses, with a higher consistency in comparison to the surrounding parenchyma. Ultrasound reveals a hypoechogenic aspect. The use of intra-operative ultrasound allows the identification of 88% of insulinomas[11,12].

    In consideration of the fact that insulinomas do not have a particular prevalence for any specific localization within the pancreas and their low grade of malignancy (only 10% of cases) [14], a surgeon who fails to identify the lesion during laparotomy, should refrain from pancreatic resection. A postoperative Dopmann test must be carried out in order to localize the disease [15,16]. When even the latter fails to localize the tumor, close follow-up of the patient is recommended until the lesion is identified. Although data regarding this issue are incomplete, it would seem that a blind resection of the pancreas should be considered only in those cases in which venous sampling allows the localization of

    Department of Surgery,Peking Uion Medical College Hospital,Beijing 100730,China

    the tumor and when medical therapy does not result in satisfactory control of the clinical symptoms [17].

    After identification, there is no doubt about the surgical indication in all localized tumors. The choice of procedure will depend on the risk of malignancy based on parameters such as type, size and features of the mass. Atypical resection (enucleation or middle pancreatectomy) has the advantage of preserving the pancreatic parenchyma as much as possible, thereby reducing the risk of late exocrine/endocrine insufficiency. However, in these procedures, a lymphadenectomy is not usually performed, they are not “foolproof” in oncological terms. Enucleation can usually be performed in those cases where the lesion is single, capsulated, of limited dimensions (less than 4 cm in diameter) and does not involve, or is “sufficiently far” from the main pancreatic duct [18]. When the lesion is in the body and/or nearby the Wirsung duct, a middle pancreatectomy should be the procedure of choice. In these instances, enucleation would be at very high risk for a postoperative fistula. Insulinomas and small non-functioning tumors are the most indicated cases. Authors advocating atypical resection report a 10-year survival rate of 94% but with a low disease-free postoperative rate (51%) [8]. This percentage is reported to be close to 90% by authors who routinely perform typical resections [19].

    When radicality is doubtful or only achievable by the demolition of nearby organs (stomach, colon, kidney, adrenal gland) or by further vascular resection, an intra-operative histological diagnosis is indicated [13, 20, 21, 22, 23]. If the endocrine nature of the tumor is confirmed, an aggressive treatment is justified even if other organs and/or portal-mesenteric vein confluence resection are necessary. The involvement of the superior mesenteric artery and/or the celiac trunk is less frequent [20, 22].

    LAPAROSCOPIC APPROACH

    The insulinoma is a treatable laparoscopic tumor given its unique characteristics. There are three surgical options: enucleation, distal pancreatectomy with or without preserving the spleen. The most important complication is the pancreatic fistula. The pancreatic surgery is a feasible and safe one, although, its superiority over the classic open approach has not been demonstrated yet[24,25].

    Resection of the pancreas can be performed safely via the laparoscopic approach with all the potential benefits to the patients of minimally invasive surgery[26].Laparoscopic pancreatic surgery is safe and feasible for patients with benign tumors and cystic lesions[27].The procedure is safe and feasible for tumors located at the body or tail of the pancreas. Its application for tumors located at the pancreatic head needs further evaluation[28,29].

    And surgical resection of insulinomas is the preferred treatment in order to avoid symptoms of hypoglycaemia. During the past years, advances in laparoscopic techniques have allowed surgeons to approach the pancreas and treat these lesions laparoscopically[30].

    A study performed between January 1998 and September 2003, 11 consecutive patients with sporadic insulinoma and two female patients with multiple insulinomas associated with multiple endocrine neoplasia type 1 (MEN-1) were operated using the laparoscopic approach. Of 11 patients with sporadic insulinoma, laparoscopic enucleation (LapEn) was planned in 8 patients, but in 1 patient, the use of LapUS missed the tumor and the patient was converted to open surgery. Mean operating time after LapEn was 180 minutes, and the mean blood loss was 200 ml. The mean hospital stay was 5 days. In 3 of the 11 patients, laparoscopic spleen-preserving distal pancreatectomy (LapSPDP) was performed; the mean operative time was 240 minutes, and the mean blood loss was 360 ml. Postoperative complications occurred in 3 of 7 patients after LapEn (3 pancreatic fistulas managed conservatively, and 1 case of bleeding requiring reoperation). LapSPDP was performed in 2 patients with MEN-1; in 1 patient with splenic vessel preservation (SVP), the operating time was 210 minutes and blood loss was 650 ml, with a hospital stay of 6 days. In another patient without SVP, the operating time was 150 minutes and blood loss was 300 ml. The latter patient developed a 4 cm splenic infarct managed conservatively, and the hospital stay was 14 days. LapEn and LapSPDP are feasible and safe and achieved cure in patients with sporadic insulinoma and multiple insulinomas associated with MEN-1. However, the risk of pancreatic leakage after LapEn remains high, and LapSPDP without SVP may be associated with splenic infarct[31].

    A group of researchers suggested that laparoscopic pancreatic procedures are still at an evaluation stage regard to their indications and techniques. Svab J,et al.give report about first experience with successful laparoscopic enucleation of insulinoma in a case of 83-year-old woman. The operative time was 90 min[32].

    Insulinomas are usually small, benign tumors of the pancreas, often found in obese patients, which require an incision that is out of all proportion to the size of the lesion. A laparoscopic technique for enucleation of a pancreatic insulinoma is described in a study.Nowadays, laparoscopic enucleation of such tumours and tail resections become feasible and safe[33] Laparoscopic resection of the tumor was attempted and accomplished in 2 patients. The young man underwent laparoscopic enucleation of the tumor, while the 50 year old man underwent left sided laparoscopic resection of the pancreas because the tumor was adjacent to the pancreatic duct. Postoperative course was uneventful in both cases. It is to be expected, that this approach will receive increasing attention in the near future[34].Four patients with clinical and radiological diagnosis of insulinoma were treated between January 2000 and September 2005. All the patients were laparoscopically approached to attempt the tumor enucleation. In 3 cases complete enucleation was possible; the remaining case was converted to perform the resection of the middle portion of the pancreas. One patient developed a pancreatic fistula that closed spontaneously. Blood glucose levels returned to normality[35].Advances in laparoscopic techniques have recently enabled the safe resection of pancreatic islet cell tumors and may provide patients with the benefits of minimally invasive surgery. Another study demonstrates the technique of laparoscopic enucleation of a pancreatic insulinoma[36] too.

    A retrospective multicenter study was conducted in 25 European surgical centers concerning their experience with LPR. During the study period, 127 patients with presumed pancreatic neoplasms were enrolled in this series. In laparoscopically successful operations, the median postoperative hospital stay was 7 days (range, 3~67 days), decreased compared with patients requiring conversion to open pancreatectomy. During a median follow-up of 15 months (range, 3-47 months), 23% of the patients with pancreatic malignancies had tumor recurrence. Long-term outcome was satisfactory in all patients with benign diseases. LPR is feasible and safe in selected patients with presumed benign and distal pancreatic tumors. The management of the pancreatic stump remains a challenge[37].

    Laparoscopic distal pancreatectomy for neuroendocrine tumors of the pancreas may be accomplished safely, with preservation of the spleen and splenic vessels in benign disease, and with benefits to the patients in terms of postoperative recovery[38].

    In cases of distal pancreatectomy, splenic salvage, preferably with preservation of splenic vessels, is feasible albeit more demanding and can be achieved in most cases[40].

    The laparoscopic distal pancreatectomy was performed using a harmonic scalpel (Ethicon EndoSurgery/UltraCision), without mechanical suturing. There were no intra- or postoperative complications or hypoglycemias during the 6 months of follow-up. When it is performed by experienced laparoscopic surgeons, this is a technically feasible procedure, safe for the treatment of benign lesions of the pancreas body and tail[39,40].

    A 37-year-old woman with a history of syncope was hospitalized with a diagnosis of hypoglycemia due to insulinoma. Because the tumor was located deep in the pancreatic parenchyma, laparoscopic distal pancreatectomy was performed. Spleen-preserving laparoscopic distal pancreatectomy by division of splenic vessels is a feasible treatment option for benign pancreatic disease[41].

    In general,laparoscopic localization and resection of insulinomas are feasible and safe and may decrease morbidity and the length of hospitalization. Laparoscopic localization and resection of insulinomas are feasible and safe. Excluding patients with pancreatic leaks (observed in both groups), hospital stay and time to recovery may be shortened by using laparoscopic insulinoma resection[42].

    METASTASIS AND ABLATION

    A 78-year-old woman was referred with typical symptoms of an insulinoma. Diagnosis was confirmed by laboratory findings, EUS, and EUS-guided ablation may become a minimally invasive alternative for patients with insulinomas in whom surgery is not feasible[43].

    Laparoscopic thermal ablation of hepatic tumors is a novel, minimally invasive method of providing effective cytoreduction of neuroendocrine tumors metastatic to the liver[44].

    A 51-year-old man who had 2 large insulinomas resected from the body of the pancreas and 19 years later, having again become symptomatic, was found to have hepatic metastases. Malignant insulinoma may follow a rather indolent course and symptoms respond well to locally destructive therapies. Hepatic embolization is less traumatic than hepatic lobe resection and radio-frequency ablation offers an alternative if vascular access to the tumour is no longer possible[45].

    A case of 76-year-old woman with liver metastases due to a malignant insulinoma, which had been diagnosed as benign after its complete removal 9 years earlier.This case showed the difficulty of distinguishing between benign and malignant insulinomas when there is no initial evidence of metastases. This fact poses the need to conduct a long follow-up period in order to detect any possible recurrence even if the initial diagnosis was one of a benign insulinoma[46].

    As a minimally invasive, readily repeatable procedure that can be used to ablate small tumors, preferably before patients become severely symptomatic, radiofrequency ablation can provide effective control of liver tumor volume in most patients over many years[47].

    A 64-year-old nondiabetic woman presented with spells of lightheadedness and diaphoresis associated with lethargy and hunger of 2 weeks' duration. She underwent resection of the pancreatic mass, radiofrequency ablation, and cauterization of hepatic lesions. Histology confirmed pancreatic insulinoma. Ten months later, she was free of hypoglycemic symptoms, with normal plasma insulin C-peptide and significantly decreased proinsulin levels.It is proved that surgery is the only curative treatment. Octreotide can be used to control hormone secretion and tumor growth[48].

    A report on an unusual occurrence of pancreatic metastases from a previously diagnosed Merkel cell carcinoma with the discovery of a concomitant insulinoma.An 82-year-old lady,furthermore, in the pancreatic body there was a hypervascularized area, that was suspicious for an insulinoma. With a working clinical diagnosis of symptomatic insulinoma not responding to medical measures, a decision for surgical resection of this large lesion was inevitable, the age of the patient and the previous history of palliative radiation just 6 months prior notwithstanding[49].

    SUMMARY

    Surgery is the preferred treatment for insulinoma. The tumor is localized with diagnostic testing or surgical exploration. Solitary tumors are removed, but patients with multiple tumors usually require partial removal of the pancreas (partial pancreatectomy). At least 15% of the pancreas is left to avoid malabsorption due to lack of pancreatic enzymes.

    In enucleation procedure the surgeon removes only the tumor preserving the rest of the pancreas. Since insulinomas usually occur on the surface of the pancreas and are covered by a capsule, they can be completely removed without having to remove any of the surrounding pancreas. The recurrence rate after enucleation of the insulinoma is very low and this is the treatment of choice if it is technically possible to perform this operation.

    For pancreatic resection (removal), less commonly the surgeon may remove part of the pancreas where the tumor is located by utilizing operative procedures such as a distal pancreatectomy or a Whipple operation. These operations are performed as an exception rather than commonly since the vast majority of the tumors can be removed with enucleation.

    Laparoscopic operations for removing pancreatic insulinoma. The operation of enucleation of the insulinoma is performed laparoscopically. With laparoscopic surgery the post-operative recovery period and amount of pain is reduced, hospital stay is shorter and patients are able to return to their normal activity much faster.

    If no tumor is found at surgery or a patient is not a candidate for surgery, the drug diazoxide may be given to lower insulin secretion and avoid hypoglycemia. A diuretic is always given with this medication to avoid retaining too much salt. Octreotide has also been used to suppress insulin secretion in some patients. Medical treatment is also used to stabilize the person prior to surgery.

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    (Editor Jaque)(DONG Guo-xiang)