|Year : 2021 | Volume
| Issue : 5 | Page : 225-229
Aggressive pancreatic resections for Frantz tumours in a low-volume Caribbean hospital
Shamir O Cawich1, Lian Sampath1, Sherrise Sandy1, Neil W Pearce2, Vijay Naraynsingh1, 3
1 Department of Surgery, Port of Spain General Hospital, Port of Spain, Trinidad and Tobago
2 Department of Surgery, Southampton General Hospital, Southampton, UK
|Date of Submission||28-Apr-2021|
|Date of Decision||04-Aug-2021|
|Date of Acceptance||17-Sep-2021|
|Date of Web Publication||30-Oct-2021|
Dr. Shamir O Cawich
Department of Surgery, Port of Spain General Hospital, Port of Spain
Trinidad and Tobago
Source of Support: None, Conflict of Interest: None
Background: Frantz tumours are uncommon lesions worldwide and many surgeons would not encounter this lesion commonly throughout their practices.
Aims: This study sought to document the therapeutic outcomes after pancreatic resections for Frantz neoplasms in a low-volume, resource-poor setting in the Caribbean.
Materials and Methods: Data were retrospectively collected from patients who had pancreatic resections for Frantz tumours in Trinidad and Tobago between 1 June 2012 and 1 July 2017.
Results: There were 12 patients with confirmed Frantz tumours, treated by one of four surgeons at a low volume of 0.6 cases per surgeon per year. The patients were all females at a mean age of 24.83 years (standard deviation [SD] ±7.6; Range 8–35; Median 25.5). A Whipple's resection was performed in 1 patient with a pancreatic head lesion, 2 had central pancreatectomies for lesions in the body and 6 had distal pancreatectomies, 2 with splenic preservation. Histologically, clear margins were achieved in all cases and none of the nodes examined contained metastatic disease. Nine (75%) patients had poor prognostic features present on pre-operative computed tomography scans and/or histopathologic examination. The sole complication was a Grade 1 post-operative pancreatic fistula. No deaths were recorded. All patients were alive without disease recurrence after a median of 34 months (Mean 36.75; SD ± 12.8; Range 12–52).
Conclusion: Surgeons practicing in the Caribbean treat patients with pancreatic Frantz tumours at minimal volumes, performing aggressive resections with good outcomes. We advocate aggressive surgical resections for pancreatic solid pseudopapillary neoplasms, considering that outcomes are good and 75% of patients have unfavourable radiologic and/or histopathologic features present at the time of diagnosis.
Keywords: Frantz, hamoudi, margin, pancreas, pathology, resection
|How to cite this article:|
Cawich SO, Sampath L, Sandy S, Pearce NW, Naraynsingh V. Aggressive pancreatic resections for Frantz tumours in a low-volume Caribbean hospital. Curr Med Res Pract 2021;11:225-9
|How to cite this URL:|
Cawich SO, Sampath L, Sandy S, Pearce NW, Naraynsingh V. Aggressive pancreatic resections for Frantz tumours in a low-volume Caribbean hospital. Curr Med Res Pract [serial online] 2021 [cited 2021 Dec 8];11:225-9. Available from: http://www.cmrpjournal.org/text.asp?2021/11/5/225/329702
| Introduction|| |
Frantz tumours (solid pseudopapillary neoplasms [SPN]) of the pancreas are uncommon lesions worldwide., Many surgeons in this setting would not encounter the lesion during their practice. There are data to suggest that therapeutic outcomes after pancreatic resections are better when performed in high-volume centres,,,,,,, but most pancreatic resections for Frantz tumours in the Caribbean are performed by general surgeons at low volumes. We sought to document the therapeutic outcomes after resection of pancreatic Frantz tumours in a low-volume, resource-poor setting in the Caribbean.
| Materials and Methods|| |
The local institutional review board granted ethical approval (CEC027/10/16) to collect data on patients with Frantz tumours who had pancreatic resections over a 5-year period from 1 June 2012 to 1 July 2017. The patients were retrospectively identified through a nationwide search of operating theatre records and pathology lab registers in Trinidad and Tobago.
The clinical records for these patients were retrieved and the following data were extracted: Patient demographics, clinical presentations, details of operative treatment, blood loss, operative time, duration of hospital stay, morbidity, mortality, margin clearance, nodal status and histologic features. All data were entered into a Microsoft Excel spread sheet and analysed with the Statistical Package for the Social Sciences [(SPSS) version 12.0. (IBM Corporation, Armonk, New York, United States). Descriptive statistics were generated as appropriate.
| Results|| |
There were 12 patients with confirmed Frantz tumours across the nation, treated by one of four surgeons. The individual surgeon's case volume was low-amounting to 0.6 cases per surgeon per year. The patients were all females at a mean age of 24.83 years (standard deviation [SD] ±7.6; Range 8–35; Median 25.5). Four patients had incidental lesions on imaging for unrelated conditions and 8 (67%) were symptomatic with obstructive jaundice (1) and abdominal pain.
One patient had a pancreatic head Frantz tumour and underwent an open Whipple's operation. Reconstruction was achieved with Roux-en-Y hepaticojejunostomy, gastro-jejunostomy and pancreatico-gastrostomy. This operation was completed in 390 min with 650 ml blood loss and no recorded complications. She required 36 h stay in the high dependency unit stay and hospitalisation for a further 7 days. Histology confirmed the presence of an SPN at the pancreatic head with 9 mm margin clearance and none of the 9 nodes involved.
Two (17%) patients underwent central pancreatectomy for small lesions (mean diameter 2.6 cm) in the pancreatic body. In both cases, the distal pancreatic duct was restored by pancreatico-gastrostomies and the proximal duct was oversewn with 3/0 polydioxanone sutures. In this group, there was 275 min mean operation time, 310 ml mean blood loss and no complications. Both resection margins were clear, with a mean distance from the pancreatic transection line of 1 cm. The mean nodal harvest in this group was 3 and no nodal metastases were present.
Six patients underwent distal pancreatectomy: Two with splenic preservation and 4 without. The operations were completed via the laparoscopic approach in 4 cases, with 206 ml mean blood loss and 265 min mean operating time. Two patients had open operations completed in 237 min mean operating time with 375 ml mean blood loss. The patients who underwent spleen-preserving distal pancreatectomy had smaller mean tumour diameters (3.175 vs. 6.83 cm) and lower mean node harvest (3.5 vs. 5.25 nodes) compared to those who had distal pancreatectomy plus splenectomy. However, no nodal metastases were identified in any of these patients. All patients were reported to have microscopically clear margins, with a mean distance of 16 mm from the pancreatic transection line after spleen-preserving pancreatectomy and 13.25 mm after pancreatico-splenectomy. One patient developed a Grade 1 pancreatic fistula after spleen-preserving distal pancreatectomy-this was successfully managed expectantly.
Three patients required multi-visceral resections (>2 contiguous organs removed en-bloc) to achieve clearance. In these patients, pre-operative staging computed tomography (CT) scans predicted involvement of multiple structures: Spleen (3), stomach (2), left adrenal (2), small bowel (1). These patients had open operations with 265 min mean operating time and 816 ml mean blood loss. These patients had larger tumours (mean diameter 11.3 cm) and greater nodal harvest (mean of nodes harvested), but there were no histologically involved nodes. Resection margins were clear in all cases, with a mean reported distance of 4 mm from the pancreatic transection line. No complications or deaths were recorded in this group.
Histopathologic examination revealed classic features that were diagnostic of Frantz tumours in all cases [Figure 1] and [Figure 2]. Nine (75%) patients had poor prognostic features: Capsular invasion [Figure 3] in 5 cases and lymphovascular invasion [Figure 4] in six patients. Immunohistochemistry (IHC) was not routinely available, but when it was, there was positive staining for neuron-specific enolase in 7/7 cases, alpha-1-antitrypsin in 4/7, vimentin in 3/6, CD56 in 3/7, progesterone in 3/7 and CD10 in 1/7 cases.
|Figure 1: A low power view at ×10 that shows classic tumour architecture. The images demonstrate sheets of uniform, poorly cohesive cells with eosinophilic cytoplasm and bland nuclei surrounding delicate blood vessels (arrow)|
Click here to view
|Figure 2: A high power view at ×10 that demonstrates cholesterol clefts (arrow) which are a classic feature of this tumour|
Click here to view
|Figure 3: A low power view at ×4 showing a solid pseudopapillary neoplasm with evidence of capsular invasion by tumour (arrow)|
Click here to view
|Figure 4: A medium power view at ×20 that demonstrates lymho-vascular invasion by a solid pseudopapillary neoplasm (arrow)|
Click here to view
We were unable to comment on the 5-year survival rate for these patients because many patients had not yet reached the 5-year mark after their operations at the time of this study. However, all were alive without disease recurrence after a median of 34 months (Mean 36.75; SD ± 12.8; Range 12–52).
| Discussion|| |
The first description of this lesion is credited to Frantz in 1959. These are uncommon lesions across the globe, and in the Caribbean, where pancreatic SPNs occur at an annual incidence of 0.17 cases/100,000 population.
These are usually indolent tumours with good long-term prognoses once histologically clear margins are achieved.,,, The ideal therapeutic approach remains controversial. Some authorities recommend an aggressive approach with formal resections (Whipple's or distal pancreatectomy) to achieve clear margins.,,,,, Others suggest that tumour enucleation alone is sufficient., Yet, others have recommended selective enucleation guided by intra-operative frozen section,, invasion of adjacent structures and/or tumour position. While the ideal surgical approach remains unsettled, most authorities agree on the need to achieve histologically clear margins. We adopted an aggressive approach, with routine formal resections and multi-visceral resections when dictated by the involvement of adjacent structures. Using this strategy, we achieved histologically clear resection margins in all cases.
Another controversial aspect of treatment is the need for lymphadenectomy. Some authorities advocate aggressive nodal dissection to minimise nodal recurrence. Others suggest that extensive lymphadenectomy is unnecessary due to the low incidence of nodal metastases.,, Machado et al. reported nodal metastases in none of 34 patients and Bostancı et al. reported nodal metastases in none of 16 patients with Frantz tumours. In our series, a total of 70 harvested nodes were examined and there were no nodal metastases. This lends support to the theory that extensive lymphadenectomy for pancreatic Frantz tumours is probably unnecessary.,,
It is well documented that clinical outcomes are improved when pancreatic lesions are resected in high-volume centres.,,,,,, Therefore, pancreatic surgeons practicing in high-volume centres should have the relevant skill sets to resect pancreatic Frantz tumours, even if they encounter this lesion infrequently. However, in the Caribbean setting most resections are performed by general surgeons in low-volume centres. Even the existing Caribbean hepatobiliary referral hospitals qualify as low-volume centres with 12–15 major pancreatic resections per year. Therefore, we expected that there would be greater morbidity and mortality after treatment in this setting. However, there were good outcomes with 8% overall morbidity and no mortality. These outcomes reinforce our belief that experience in pancreatic surgery, and not case volume alone, is a key determinant of therapeutic outcomes.
We are unable to comment on long-term survival as many patients would not have reached the 5-year interval post-resection, but the short-term surgical outcomes are promising. A longer follow up period is required to accurately comment on long-term survival with this aggressive surgical strategy.
The Royal College of Pathologists published cancer datasets for reporting on pancreatic neoplasms to allow standardised, accurate reporting consistently. Reporting of the core data items in each dataset allows clinicians to interpret relevant pathologic information to ensure accurate staging and treatment. Unfortunately, in this series, the pathologists did not routinely report all margins required by the Royal College of Pathologists standardised datasets. The only margin routinely reported was that at the pancreatic transection line. The absence of the remaining posterior, anterior and radial margins is a limitation of this study.
Other limitations include the absence of routine intra-operative frozen section and IHC. Therefore, these data were not available for interpretation in this study. Finally, the duration of follow-up in this series was short, with a mean follow-up 36 months. Therefore, we could not comment adequately on survival with this lesion. Further studies are required to determine long-term survival in this setting.
| Conclusion|| |
Although surgeons at this resource-poor centre treated patients with pancreatic Frantz tumours at low volumes of only 0.6 cases annually, there were good therapeutic outcomes with aggressive resections. We suggest that experience in pancreatic surgery, and not case volume alone, is a key determinant of therapeutic outcomes.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Papavramidis T, Papavramidis S. Solid pseudopapillary tumors of the pancreas: Review of 718 patients reported in English literature. J Am Coll Surg 2005;200:965-72.
De Castro SM, Singhal D, Aronson DC, Busch OR, van Gulik TM, Obertop H, et al.
Management of solid-pseudopapillary neoplasms of the pancreas: A comparison with standard pancreatic neoplasms. World J Surg 2007;31:1130-5.
Gordon TA, Burleyson GP, Tielsch JM, Cameron JL. The effects of regionalization on cost and outcome for one general high-risk surgical procedure. Ann Surg 1995;221:43-9.
Lieberman MD, Kilburn H, Lindsey M, Brennan MF. Relation of perioperative deaths to hospital volume among patients undergoing pancreatic resection for malignancy. Ann Surg 1995;222:638-45.
Begg CB, Cramer LD, Hoskins WJ, Brennan MF. Impact of hospital volume on operative mortality for major cancer surgery. JAMA 1998;280:1747-51.
Gouma DJ, van Geenen RC, van Gulik TM, de Haan RJ, de Wit LT, Busch OR, et al.
Rates of complications and death after pancreaticoduodenectomy: Risk factors and the impact of hospital volume. Ann Surg 2000;232:786-95.
Birkmeyer JD, Finlayson SR, Tosteson AN, Sharp SM, Warshaw AL, Fisher ES.
Effect on hospital volume on in-hospital mortality with pancreaticoduodenectomy. Surgery 1999;125:250-6.
Glasgow RE, Mulvihill SJ. Hospital volume influences outcome in patients undergoing pancreatic resection for cancer. West J Med 1996;165:294-300.
Ihse I. The association between higher volume and better outcome for pancreatoduodenectomy. HPB (Oxford) 2001;3:153-5.
Shukla PJ, Sakpal SV, Naraynsingh V. Centralization of surgery: Is it applicable to less populous nations? HPB (Oxford) 2008;10:506-7.
Frantz VK. Tumours of the pancreas. Atlas Tumour Pathol 1959;1:32-3.
Cawich SO, Ledesma Z, Sampath L, Sandy S. Clinico-pathologic features of solid pseudopapillary pancreatic neoplasms in an eastern caribbean population. Trop Doct 2018;48:224-7.
Bostancı EB, Öter V, Binarbaşı C, Turhan N, Özer İ, Ulaş M, et al.
Surgical outcomes of solid pseudopapillary neoplasm of the pancreas: A single institution's experience of 16 cases. Arch Iran Med 2016;19:30-4.
El Nakeeb A, Abdel Wahab M, Elkashef WF, Azer M, Kandil T. Solid pseudopapillary tumour of the pancreas: Incidence, prognosis and outcome of surgery (single center experience). Int J Surg 2013;11:447-57.
Yin Q, Wang M, Wang C, Wu Z, Yuan F, Chen K, et al.
Differentiation between benign and malignant solid pseudopapillary tumor of the pancreas by MDCT. Eur J Radiol 2012;81:3010-8.
Ji S, Xu J, Zhang B, Xu Y, Liu C, Long J, et al.
Management of a malignant case of solid pseudopapillary tumor of pancreas: Case report and literature review. Pancreas 2012;41:1336-40.
Lubezky N, Papoulas M, Lessing Y, Gitstein G, Brazowski E, Nachmany I, et al.
Solid pseudopapillary neoplasm of the pancreas: Management and long-term outcome. Eur J Surg Oncol 2017;43:1056-60.
Reddy S, Cameron JL, Scudiere J, Hruban RH, Fishman EK, Ahuja N, et al.
Surgical management of solid-pseudopapillary neoplasms of the pancreas (Franz or Hamoudi tumors): A large single-institutional series. J Am Coll Surg 2009;208:950-7.
Lam KY, Lo CY, Fan ST.
Pancreatic solid cystic papillary rumour: Clinicopathologic feature in 8 patients from Hong Kong and review of the literature. World J Surg 1999;23:1045-50.
Butte JM, Brennan MF, Gonen M, Tang LH, D'Angelica MI, Fong Y, et al
. Solid pseudopapillary tumours of the pancreas. Clinical features, surgical outcomes and long term survival in 45 consecutive patients from a single centre. J Gastrointest Surg 2011;15:350-7.
Morikawa T, Onogawa T, Maeda S, Takadate T, Shirasaki K, Yoshida H, et al
. Solid psudopapillary neoplasms of the pancreas: An 18-year experience at a single Japanese institution. Surg Today 2012;41:91-6.
Li Z, Zhang Z, Liu X, Hu W, Mai G, Zhang Y, et al.
Solid pseudopapillary tumor of the pancreas: The surgical procedures. Surg Today 2011;41:91-6.
Garcea G, Ong SL, Rajesh A, Neal CP, Pollard CA, Berry DP, et al.
Cystic lesions of the pancreas. A diagnostic and management dilemma. Pancreatology 2008;8:236-51.
Machado MC, Machado MA, Bacchella T, Jukemura J, Almeida JL, Cunha JE. Solid pseudopapillary neoplasm of the pancreas: Distinct patterns of onset, diagnosis, and prognosis for male versus female patients. Surgery 2008;143:29-34.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]