10.1245/s10434-006-9133-6
Annals of Surgical Oncology 14:1065-1069 (2007)
© 2007 Society of Surgical Oncology
Laparoscopic Distal Pancreatectomy in Children: Case Report and Review of the Literature
Gianluigi Melotti, MD1,
Alvise Cavallini, MD2,
Giovanni Butturini, MD, PhD2,
Micaela Piccoli, MD1,
Andrea Delvecchio, MD3,
Cesare Salvi, MD3 and
Paolo Pederzoli, MD2
1 General Surgery, Baggiovara Hospital, Modena, Italy
2 General Surgery B, University Hospital GB Rossi, Policlinico GB Rossi, Borgo Roma, Piazzale LA Scuro, 10 37134, Verona, Italy
3 Paediatric Clinic, Desenzano Hospital, Desenzano del Garda, (BS), Italy
Correspondence: Address correspondence and reprint requests to: Alvise Cavallini, MD; E-mail: alvise.cavallini{at}tiscali.it
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ABSTRACT
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Background: Laparoscopic resection of benign tumors of the pancreas has been reported in adults, but only four cases of partial laparoscopic pancreatectomy in children have been described in the English-language literature.
Methods: We describe the case of an 11-year-old girl with a solid pseudopapillary tumor who was treated with a laparoscopic, spleen-preserving, distal pancreatectomy. The specimen was extracted in an endoscopic bag retrieval system through a Pfannenstiel incision. Operative time was 120 minutes, and minimal blood loss occurred. The literature is reviewed.
Results: The postoperative course was uneventful. Twenty-two months after the operation, clinical follow-up (including assessment of exocrine and endocrine pancreatic function) revealed nothing abnormal. The functional and aesthetic results were satisfactory.
Conclusions: The technique used for our case is simple and reproducible, was completed safely within a reasonable operative time, and yielded a good result.
Key Words: Laparoscopy • Children • Pancreatectomy • Solid pseudopapillary tumor
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INTRODUCTION
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Laparoscopic pancreatic resection was initially described in both experimental and human studies in the early 1990s. Thereafter, thanks to improvements in instrumentation, such as the development of endoscopic staplers and expanded training in laparoscopy, great strides have been made in the treatment of adults. The first laparoscopic pancreatoduodenectomy was performed in 1994,1 and distal pancreatectomy (DP) was first performed in 1996.2 During open procedures, access to the pancreas requires wide exposure. Laparoscopy offers the advantage of large magnification and benefits to the patients in terms of postoperative recovery and acceptable rates of perioperative morbidity. DP is well suited to the laparoscopic approach because of the absence of anastomosis. The magnified view of the operative field facilitates separation of splenic vein and artery from the pancreatic parenchyma. However, the published series are based on a relatively few cases and operating times are relatively long (mean, 4.5 hours).3 A recent large multicenter experience suggests that a pancreatic laparoscopic approach be taken to treat benign tumors.4
To our knowledge, in children, only four cases of partial laparoscopic pancreatectomy have been described in the English-language literature, one of them for solid pseudopapillary tumor (SPT). This rare neoplasm (1% to 2% of exocrine pancreatic tumors) was first described by Frantz in 1959 as a "papillary tumor of the pancreas, benign or malignant."5 Since the report of five cases by Kloppel et al. in 1981, according to Lam et al.,6 452 cases have been reported in the English-language literature. Synonyms include solid and cystic tumor, solid and papillary epithelial neoplasm, papillary-cystic neoplasm, papillary cystic epithelial neoplasm, papillary-cystic tumor, and Frantz tumor. In 1996, the World Health Organization renamed this tumor solid pseudopapillary tumor.7 This uncommon benign neoplasm is found mainly in women between the second and third decades of life, although rare cases have been reported in children and men.6,8 Most SPTs exhibit benign behavior, but malignant degeneration sometime does occur.
We report the case of an 11-year-old girl with an SPT who was treated with a laparoscopic, spleen-preserving, distal pancreatectomy, emphasizing the good results obtained regarding operating time and the quick discharge of the patient.
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PATIENTS AND METHODS
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The patient, an 11-year-old, previously healthy girl, was admitted to our hospital with a 3-day history of nonbilious emesis and abdominal pain. At admission, her temperature and vital signs were normal. She weighed 41.5 kg and was 139 cm tall. She complained of periumbilical, intermittent, constrictive pain not related to eating. Urine and stools were normal; no recent abdominal trauma was reported. Physical examinations revealed nothing abnormal.
Laboratory data (including tumor markers), chest x-ray, and electrocardiogram were normal. Transabdominal ultrasound revealed a not homogeneous, round, hypoechoic mass, with cystic and solid components, 42 mm in diameter, in the tail of the pancreas. Computed tomographic scan confirmed the solid fairly homogeneous mass with little cystic areas in the tail with atrophy of the distal part of the pancreas; the rest of the gland was unaffected. The lesion showed no marked contrast enhancement and compressed the splenic artery and the stomach. Liver, spleen, abdominal aorta, kidneys, and bladder were normal, and no abdominal nodes were observed (Fig. 1
). All these findings were according with the diagnosis of SPT of the pancreas. The patient was referred to the operating unit "Chirurgia B," Verona, Italy, for further diagnostic and preoperative evaluation. Before surgery, the patient’s relatives’ consent was obtained for both a laparoscopic and an open approach.
The patient was placed supine in the reverse 30° Trendelenburg position, with the surgeon standing between the patient’s legs (Fig. 2). The screen was on the left side of the patient’s head. A four-port laparoscopic technique was used. A 10-mm camera port was placed at the umbilicus. Two additional working 5-mm ports, along with a 12-mm port for the linear stapling device, were placed in the xiphoid area and the right and left flank, respectively (Fig. 3). Electrocautery and LigaSure Atlas were used for dissection. A wide window was made in the gastrocolic ligament. A forceps was used to raise the stomach from the epigastric trocar, enabling good exposure of the body tail of the pancreas. The inferior border of the gland was dissected and the tail detached from the retroperitoneum. This mobilization enabled the surgeon to visualize the posterior wall of the gland with the splenic vessels (Fig. 4). A spleen-preserving distal pancreatectomy was performed, freeing the tail from the splenic vessels. The pancreatectomy was achieved by transection with an endoscopic linear 45-mm stapler (Fig. 5). The dissected part was extracted in an endoscopic bag retrieval system through a Pfannenstiel incision. A drain had been placed on the pancreatic stump and drawn out through the 12-mm port site. Operative time was 120 minutes, and there was little blood loss.
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FIG. 4. The inferior border of the pancreas was dissected to visualize the posterior wall of the gland with the splenic vein.
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The postoperative course was uneventful, with recovery of alimentation and normal serum amylase level at postoperative day 2. The girl was discharged on postoperative day 5.
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RESULTS
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Pathologic diagnosis was of 40-mm solid lobulated neoplasm with regular margins. Three lymph nodes were free of disease. At immunohistochemical analysis, the tumor cells were positive for 
-1-antitrypsin, progesterone receptor and ß-catenin, CD10, CD56, and vimentin. The cells were negative for chromogranin A, synaptophysin, S100 protein, CD34, CD68, and keratin 8/18/19 (5D3). These findings helped to establish the diagnosis of SPT of the pancreas.
Twenty-two months after the operation, clinical follow-up (including exocrine and endocrine pancreatic function) were normal, and her body weight and height were normal for her age. The functional and aesthetic result are satisfactory (Fig. 6).
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DISCUSSION
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SPT of the pancreas is a very rare clinical entity, representing 2% to 3% of all primary pancreatic tumors occurring at any age, with a relatively low grade of malignant potential. Lam et al.6 found that 66 (15%) of 452 reported tumors were malignant as a result of the presence of metastases or invasion of adjacent structures. Malignant forms had a male and elderly age predilection. According to the literature, recurrence is possible after 10 years, so all patients should be submitted to follow-up. However, the prevalence is in young female patients, suggesting a possible role of sex hormones.9 The diagnosis is incidental in approximately 60% of the patients. Whenever present, abdominal mass or vague abdominal pain are the common complaints; obstructive symptoms may occur if the tumor compresses adjacent viscera. Because of indistinct symptoms and low incidence, SPTs are often misdiagnosed. In general, in a young population, neoplastic diseases tend to be ignored, and abdominal discomfort is usually interpreted as gastritis.10 Once a neoplastic disease is recognized, a mass is often palpable. In this situation, differential diagnosis should take into account pancreatoblastoma (although this is a solid tumor), or inflammatory or neoplastic cysts arising either from the pancreas or the spleen. In this respect, SPT usually has a solid component. This latter finding is present in endocrine nonfunctioning tumors, but they usually affect an older population.11 The clinician should consider SPT in young women, especially women <25 years old.
Imaging techniques may help in differential diagnosis. Computed tomographic scan usually demonstrates a well-encapsulated lesion with a solid-cystic component as a result of hemorrhagic degeneration. After contrast medium is added, enhancing solid areas are typically peripherally noted.12 Magnetic resonance imaging usually demonstrates a well-defined lesion with heterogeneous signal intensity on T1- and T2-weighted images, which reflects the complex nature of the mass. Areas of high signal intensity on T1-weighted images and low or inhomogeneous signal intensity on T2-weighted images can help identify blood products.13,14 Lee et al.15 retrospectively evaluated ultrasound findings in 11 cases of pathologically proven solid and papillary epithelial neoplasms. Well-encapsulated cystic and solid masses were typically seen, but sometimes a mass was pure and solid-looking or had internal septa or calcifications. In this regard, contrast-enhanced ultrasonography is promising because it shows the typical rim enhancement in the early dynamic phases.16 Laboratory tests (i.e., serum amylase levels) and tumoral markers (e.g., CA19-9, carcinoembryonic antigen, -fetoprotein) are usually normal. Fewer than 100 cases have been reported in children,17 and only four cases have been laparoscopically treated.18–21 In 2001, Blakely et al.18 reported a 4-week-old baby with persistent hyperinsulinemic hypoglycemia that required a spleen-preserving pancreatectomy. The infant required complementary open surgery within a few days because his symptoms persisted, and the authors concluded that laparoscopic pancreatectomy can be performed safely, even in a newborn, without prolonged operative time or unnecessary risk.
Laparoscopic DP for blunt injury of the pancreas in a 10-year-old boy has also been reported.19 The spleen and its vessels were preserved. The patient was sent home on postoperative day 3 without any postoperative complications, and the authors concluded that in the trauma setting, advanced laparoscopic pancreatic procedure is feasible, particularly in children. In 2003, researchers reported a laparoscopic distal pancreatic resection of an insulinoma.20 In the only previous report of laparoscopic pancreatic resection for SPT,21 operative time was 3 hours, 30 minutes. The postoperative course was uneventful, with recovery of alimentation and normal serum amylase level at postoperative day 2. The boy was discharged on postoperative day 6, and 6 months later, he was free of disease. The specimen was extracted in an endoscopic bag retrieval system through a minimally enlarged umbilical trocar incision. This was the main difference with our technique because we prefer to perform a Pfannenstiel incision to avoid breaking the mass. This fact is important because the same authors reported a recurrence in a 12-year-old girl with SPT that was initially approached by laparoscopic biopsy because it was thought that she had a lymphoma; disease was eventually treated by open pancreatic resection. Two years later, the child developed a recurrence in the form of a peritoneal carcinomatosis. The authors could not exclude the possibility that the laparoscopic biopsy had contributed to the disease’s evolution; consequently, they stopped using the laparoscopic approach for solid tumors of the pancreas. Nowadays, on the basis of several reported experiences, we believe that the laparoscopic approach is feasible, provided that the tumor is not broken open, as is done in an open technique.
After distal laparoscopic pancreatectomy, the main reported complication is the occurrence of a pancreatic fistula (8% to 30%), but the rate and overall morbidity are comparable to recent large open series of distal open pancreatectomy. No perioperative deaths have been reported. However, with the laparoscopic approach, spleen preservation seems more feasible and is achievable in up to 80% of cases.4 As regards the conversion rate, it presumably depends the surgical team’s experience with the procedure.
The technique used for our case is simple and reproducible, was completed safely within a reasonable operative time, and yielded a good result. However, it should be considered an advanced laparoscopic procedure, and its application in children is still being developed. Only surgical teams with advanced laparoscopic skills should attempt it. As experience with this technique continues to grow, laparoscopic distal pancreatectomy may well become the approach of choice in selected patients for benign tumors, and it would seem ideal for treatment of SPT in children, with the recommendation that breaking the mass be avoided.
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ACKNOWLEDGMENTS
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Supported by grants from "Fondazione Zanotto" and COFIN 2005060715_004, Rome, Italy.
Received for publication June 8, 2006.
Accepted for publication June 14, 2006.
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- Frantz VK. Tumors of the pancreas. In: Atlas of Tumor Pathology. Washington, DC: Armed Forces Institute of Pathology, 1959:32–3.
- Lam KY, Lo CY, Fan ST. Pancreatic solid-cystic-papillary tumor: clinicopathologic features in eight patients from Hong Kong and review of the literature. World J Surg 1999; 23:1045–50.[CrossRef][Medline]
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- Blakely ML, Lobe TE, Cohen J, et al. Laparoscopic pancreatectomy for persistent hyperinsulinemic hypoglycemia of infancy. Surg Endosc 2001; 15:897–8.[Medline]
- Sayad P, Cacchione R, Ferzli G. Laparoscopic distal pancreatectomy for blunt injury to the pancreas: a case report. Surg Endosc 2001; 15:759.[CrossRef][Medline]
- Lo CY, Tam PK. Laparoscopic pancreatic resection of an insulinoma in a child. Asian J Surg 2003; 26:43–5.[Medline]
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ABSTRACT
INTRODUCTION
