Heterotopic gastric mucosa of the proximal esophagus (HGMPE) is a congenital island of salmon-colored, abnormally located gastric epithelium, commonly present distally to the upper esophageal sphincter (UES). It can vary from microscopic and small foci to extensive and circumferential patches11.

Most are largely asymptomatic8, found incidentally during esophagogastroduodenoscopies (EGD), with an underestimated prevalence ranging from 2.6 to 21%1,9.

However, it can lead to complications such as bleeding, ulceration, neoplastic transformation, acid production, and laryngopharyngeal reflux, specifically chronic cough, throat discomfort, hoarseness, globus sensation, and regurgitation3.

Secretion can be acidic, as demonstrated by pH monitoring, and proton pump inhibitor may improve pharyngeal manifestations7, but non-acidic mucus can also lead to symptoms2.

The inlet patch is commonly a potential site for Helicobacter pylori infection. It is closely related to active inflammation and associated with H. pylori infection in the stomach5. The fact that HGMPE is commonly missed in EGDs can be explained, since it is located in the upper esophagus, a difficult area to examine due to the UES contraction, and is commonly neglected during device removal.

INTRODUCTION

Endoscopic stent placement has become a well-established treatment in the management of biliary tract obstruction8. Complications such as pancreatitis, hemorrhage, duodenal perforation, or cholangitis can occur in up to 10% of insertions4,6,8. Iatrogenic perforation of the portal vein during the procedure or stent migration to the portal system are unusual complications, and due to scarce reports in the literature, there is no consensus on how to manage it1,2,11.

Here, we present a case of portal thrombosis secondary to an accidental portal insertion of a biliary stent. The study was approved by the Ethics Committee of the Institution (number 76693423.1.0000.0068).

CASE REPORT

A 44-year-old female presented in another medical institution with acute upper abdominal pain, jaundice, and vomiting, and was diagnosed with mild acute biliary pancreatitis. Magnetic resonance imaging (MRI) showed choledocholithiasis. As the jaundice persisted, she underwent endoscopic retrograde cholangiopancreatography (ERCP). After undergoing papillotomy and plastic stent placement, the clinical jaundice subsided. The clinical report did not mention technical difficulties.

After 4 months, the patient was referred to our outpatient facility due to upper abdominal pain and generalized pruritus. Blood tests showed: total bilirubin, 2.06 mg/dL (reference range, 0.2-1.2 mg/dL); direct bilirubin, 1.0 mg/dL (up to 0.5 mg/dL); indirect bilirubin, 1.1 mg/dL (0.2-0.7 mg/dL); alkaline phosphatase, 270 U/L (40-150 U/L), and gamma-glutamyl transferase, 431 U/L (12-64U/L).

BACKGROUND:

Pancreas divisum is an anatomical abnormality where the junction of the main and accessory pancreatic duct fails to occur and the smaller-caliber duct acts as dominant, resulting in overload during the drainage of the organ’s secretion through the minor duodenal papilla.

AIMS:

To report a case of recurrent acute pancreatitis due to symptomatic pancreas divisum who underwent pancreatoduodenectomy.

CASE REPORT:

A 21-year-old male patient presented with intermittent painful crises, located in the upper abdomen, with radiation to the back, associated with nausea and vomiting, for the past three years. Magnetic resonance imaging and endoscopic retrograde cholangiopancreatography revealed pancreas divisum, subsequently confirmed by endoscopic ultrasound. An attempt was made through endoscopic intervention but failed to catheterize the minor papilla; therefore, a pancreaticoduodenectomy was indicated. The organ was identified as hard and atrophied, with moderate peripancreatic inflammation. The histopathological findings also identified a focal well-differentiated G1-type neuroendocrine tumor measuring 0.4 cm.

CONCLUSIONS:

In patients with pancreas divisum, rare cases may progress to recurrent acute pancreatitis. Pancreaticoduodenectomy is an option in symptomatic patients who had no success with endoscopic treatment.

It is essential to reevaluate old concepts that, over time, become unquestionable truths. This is particularly important in the treatment of asymptomatic cholelithiasis, where an expectant, non-surgical approach is commonly recommended. We should therefore commend and congratulate the authors of the article “ASYMPTOMATIC CHOLELITHIASIS: EXPECTANT OR CHOLECYSTECTOMY. A SYSTEMATIC REVIEW,” published in the Brazilian Archives of Digestive Surgery (Arq Bras Cir Dig. 2023;36:e1747), for promoting the discussion of one of the most prevalent digestive disorders in the adult population.

 

Although it is a controversial topic in clinical practice, we agree with the authors’ conclusion that “the majority of evidence points to the safety and feasibility of a conservative (clinical follow-up) management of asymptomatic cholelithiasis”1. However, we need to consider that some variables are not always taken into consideration in systematic reviews, which can prejudice obtaining more robust conclusions.

 

It is important to note that there are no clinical trials directly comparing surgical and expectant treatments for asymptomatic cholelithiasis. In most cases, prophylactic cholecystectomy is not recommended due to the low risk of serious complications and the generally mild initial symptoms. The evidence supporting this recommendation, however, largely originates from studies conducted in the 1970s and 1980s, before the advent of laparoscopic surgery. For instance, in 1983, Ransohoff et al.7 concluded that conventional prophylactic cholecystectomy decreased patient survival based on a model analyzing the natural history of cholelithiasis. This type of study is now considered outdated considering the advancements in surgical techniques.

 

The standardization of laparoscopic surgery has drastically reduced the risks and complications associated with cholecystectomy, making it the prime example of the benefits of minimally invasive surgical techniques. Currently, serious complications of laparoscopic cholecystectomy, such as bile duct injuries, bleeding, and inadvertent bowel injuries, have an incidence of less than 0.5%. Surgical wound infection rates are also significantly lower compared to the open technique4,9. Additionally, the “critical view of safety” approach has been an ally in reducing harm and training new surgeons2.

Insulinomas are rare neoplasms of the endocrine pancreas. Minimally invasive treatment options for insulinomas have gained prominence, replacing surgical resection due to its associated morbidity and mortality. Radiofrequency ablation (RFA) has emerged as a relevant treatment option. We present a case of a female patient with neuroglycopenic symptoms and severe hypoglycemic crises. The abdominal magnetic resonance imaging (MRI) showed a small nodular lesion in the pancreatic body. Laparotomy was performed, followed by RFA using a 15-mm active-tipped needle. No complications transpired, and no hypoglycemic episodes were observed during 12 months of follow-up.

ABSTRACT

Despite endoscopic eradication therapy being an effective and durable treatment for Barrett’s esophagus-related neoplasia, even after achieving initial successful eradication, these patients remain at risk of recurrence and require ongoing routine examinations. Failure of radiofrequency ablation and argon plasma coagulation is reported in 10–20% of cases.

INTRODUCTION

Large tumors located in the right liver lobe may preclude the safe performance of right trisectionectomy due to a small left lateral segment remaining. First reported by Makuuchi et al. in 1980, embolization of the right portal vein (PV) is performed to trigger contralateral hypertrophy and enable the performance of right trisectionectomy6. The obstruction of the left PV due to inadvertent migration of embolizing agents following right portal vein embolization (PVE), as preparation for resection of large unresectable colorectal liver metastasis (CRLM), may preclude curative liver resection1.

The ALPPS (Associating Liver Partition and Portal vein Ligation for Staged hepatectomy) procedure usually induces fast hypertrophy of the left liver remnant8,10. Inclusion criteria are patients with extensive bilobar colorectal liver metastases and a predicted future liver remnant <30%. A rare case of utilization of the ALPPS procedure as rescue surgery after a failed PVE is reported herein.

CASE REPORT

A 54-year-old female presented with a large metachronous CRLM involving right hemiliver and segment IVa, 22 months after a left hemicolectomy. After a 4-cycle oral FOLFIRI chemotherapy, she was evaluated for a right trisectionectomy, but the future liver remnant (FLR=22%) was deemed as insufficient. To induce FLR hypertrophy, right PVE guided by ultrasound was performed through peripheral right PV branches and superselective catheterization of anterior and posterior portal branches employing polyvinyl alcohol particles (ContourT, Boston Scientific, Cork, Ireland®). A mixture of cyanoacrylate glue and lipiodol was injected under fluoroscopic guidance. Portography revealed adequate right PVE.

Four weeks later, a computed tomography (CT) scan revealed thrombosis of left PV small branches secondary to migration of embolizing material inside LPV (Figures 1A1B), explaining the lack of hypertrophy of the left hemiliver (FLR=25%). Balloon angioplasty and stent placement in the LPV were carried out (Figures 2A2B).

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Figures 1
(A) Computed tomography scan revealed thrombosis of left portal vein small branches (B) secondary to migration of embolizing material inside left portal vein.

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Figures 2
(A) Balloon angioplasty and (B) Stent placement in the left portal vein.

ALPPS10 was taken out seven weeks later (Figure 3A) (predicted FLR=29% two weeks thereafter). Then, a right trisectionectomy was operated as ALPPS completion (Figure 3B).

657aa2bb5d0fe5f0db9fa6d9ee1cf931c8c1e6d3

Figures 3
(A) Associating liver partition and (B) Portal vein ligation for staged hepatectomy.

One month later, a CT scan identified a non-occlusive thrombosis compromising the inferior vena cava (from the left hepatic vein to the left common iliac vein). Rivaroxaban was initiated and kept for 6 months, and patient was discharged home 41 days after the hepatectomy. The patient received an 8-cycle FOLFIRI adjuvant scheme. She remains stable, with no signs of recurrent disease or thrombosis 21 months after the procedure. This study was approved by the Institutional Board (number 2017-0271), and the patient signed informed consent for this report.

DISCUSSION

Complete resection of CRLM is the best treatment to achieve long term survival3,5. Resection of adjacent structures may be necessary in order to achieve free tumor margins2. In some cases, whatsoever insufficient FLR due to a small left lateral segment may preclude such approach.

PVE is used in order to induce contralateral lobe hypertrophy, aiming to achieve a safe FLR volume. However, such hypertrophy is not always reached. PVE generally triggers a 20 to 40% increase in FLR. With the advent of ALLPS, patients previously considered non-operative candidates are now being able to benefit from a complete tumor resection. Even if the patient experiences a failed or insufficient PVE, “salvage ALPPS” can still be carried out9. A higher morbi-mortality should be expected in such cases, though4. Due to previous PVE, only liver partition is generally performed without the need for PV ligation.

Rolinger et al. presented a case of inadvertent nontarget portal thrombosis of the FLR following PVE. An ALPPS procedure with concomitant thrombectomy of the left portal vein was used as a rescue strategy for this patient, concluding that it can be used in the event of technical failures or complications following PVE, even in patients with perihilar carcinoma7. In the case reported herein, ALPPS was utilized in a scenario of failed PVE. Salvage ALPPS may be a therapeutic option for failed PVE even in the event of inadvertent contralateral migration of embolizing material.

  • Editorial Support: National Council for Scientific and Technological Development (CNPq).
  • Financial source: None

REFERENCES

  • 1
    Basilio L, Steinbrück K, Fernandes R, D’Oliveira M, Cano R, Vasconcelos H, et al. Re-hepatectomy means more morbidity? A multicentric analysis. ABCD Arq Bras Cir Dig. 2022;35:e1647. https://doi.org/10.1590/0102-672020210002e1647
    » https://doi.org/10.1590/0102-672020210002e1647
  • 2
    Chedid AD, Chedid MF, Kruel CRP, Girardi FM, Kruel CDP. Extended right hepatectomy with total caudate lobe resection and biliary tree resection for a large colorectal liver metastasis involving both the right and left hepatic lobes and the umbilical fissure: a case report. Am Surg. 2005;71(5):447-9. PMID: 15986980.
  • 3
    Chedid AD, Villwock MM, Chedid MF, Rohde L. Prognostic factors following liver resection for hepatic metastases from colorectal cancer. Arq Gastroenterol. 2003;40(3):159-65. https://doi.org/10.1590/s0004-28032003000300005
    » https://doi.org/10.1590/s0004-28032003000300005
  • 4
    Chow FCL, Chok KSH. Colorectal liver metastases: an update on multidisciplinary approach. World J Hepatol. 2019;11(2):150-72. https://doi.org/10.4254/wjh.v11.i2.150
    » https://doi.org/10.4254/wjh.v11.i2.150
  • 5
    Gössling GCL, Chedid MF, Pereira FS, Silva RK, Andrade LB, Peruzzo N, et al. Outcomes and prognostic factors of patients with metastatic colorectal cancer who underwent pulmonary metastasectomy with curative intent: a Brazilian experience. Oncologist. 2021;26(9):e1581-e1588. https://doi.org/10.1002/onco.13802
    » https://doi.org/10.1002/onco.13802
  • 6
    Makuuchi M, Thai BL, Takayasu K, Takayama T, Kosuge T, Gunvén P, et al. Preoperative portal embolization to increase safety of major hepatectomy for hilar bile duct carcinoma: a preliminary report. Surgery. 1990;107(5):521-7. PMID: 2333592.
  • 7
    Rolinger J, Li J. Surgical removal of inadvertent portal thrombosis by rescue-ALPPS for perihilar cholangiocarcinoma. Int J Surg Case Rep. 2019;1(1):1-4. https://doi.org/10.31487/j.IJSCR.2019.01.06
    » https://doi.org/10.31487/j.IJSCR.2019.01.06
  • 8
    Santibañes E, Clavien PA. Playing Play-Doh to prevent postoperative liver failure: the “ALPPS” approach. Ann Surg. 2012;255(3):415-7. https://doi.org/10.1097/SLA.0b013e318248577d
    » https://doi.org/10.1097/SLA.0b013e318248577d
  • 9
    Sparrelid E, Gilg S, Brismar TB, Lundell L., Isaksson B. Rescue ALPPS is efficient and safe after failed portal vein occlusion in patients with colorectal liver metastases. Langenbecks Arch Surg. 2017;402(1):69-75. https://doi.org/10.1007/s00423-016-1524-y
    » https://doi.org/10.1007/s00423-016-1524-y
  • 10
    Torres OJM, Fernandes ESM, Herman P. ALPPS: past, present and future. ABCD Arq Bras Cir Dig. 2015;28(3):155-6. https://doi.org/10.1590/S0102-67202015000300001
    » https://doi.org/10.1590/S0102-67202015000300001

LETTER TO THE EDITOR

 

Extrahepatic biliary cystadenocarcinoma mimicking Klatskin tumor

 

Sergio Renato Pais-Costa; Sandro Jose Martins; Sergio Luiz Melo Araujo; Olímpia Alves Teixeira Lima; Marcio Almeida Paes; Marcio Lobo Guimaraes

 

Hospital Santa Lucia, Brasília, DF, Brazil

 

Correspondence

 

INTRODUCTION

 

Biliary cystadenocarcinoma (BCAC) is a very rare malignant cystic neoplasm. Some authors think this particular neoplasm was conversion of biliary cystadenoma over several years of evolution. In the majority of the cases, it habitually occurs in the liver parenchyma (intrahepatic cystadenocarcinoma); sometimes can be observed extrahepatic biliary origin3,4,6,7,8,10,11 extrahepatic biliary cystadenocarcinoma, (EBCAC), which generally leads jaundice associated with palpable mass1,2,5,6,9. In the past, the diagnosis was done by means of endoscopic retrograde cholangiopancreatography. However, actually the computed tomography or even nuclear magnetic resonance are preferred due non-invasive approaches1,2,3,4,5,6,7,8,9,10.

 

Sometimes, EBCAC has difficulties on differential diagnosis with cholangiocarcinoma, mainly when occurs in the hepatic hilum6. The surgical treatment provides good prognosis; however, en-bloc biliary tract resection with hepatectomy may be necessary to get free margins3,6,10. To date, was not found any case reported in Brazil, nor successful operation using a en-bloc biliary resection with hepatectomy to treat it.

 

CASE REPORT

 

A 54 year-old caucasian man presented with painless obstructive jaundice and hepatic palpable mass which began one month before. Liver functions tests showed elevated bilirubin levels of 14,8 ng/dl, alkaline phosphatase of 1067 U/l, gamma-glutamyl transferase of 550 U/l, AST of 175 U/l, and ALT 143 U/l. The alpha-fetoprotein was normal while Ca19,9 was high about 345 U/l. The patient underwent a computed tomography that showed a cystic lesion with irregularity and thickened wall in conjunction with dilated intrahepatic bile ducts mainly left side with atrophy of left lobe. Subsequently, he was submitted a cholangioresonance that showed dilatation of intrahepatic biliary tree more significant at the left side biliary duct with irregularity and thickened wall close to hepatic confluence (Figure 1). The patient underwent radiological examination without signals of systemic dissemination. Therefore, main diagnosis was hilar cholangiocarcinoma or extrahepatic biliary cystadenoma or cystadenocarcinoma. Surgical treatment was indicated, and resection of the suprapancreatic biliary tree including hilar confluence and en-bloc extended left hepatectomy with caudate lobectomy was performed. A formal hilar lymphadenectomy was also realized during surgical resection (Figure 2). One self limited biliary leakage was observed, and treated by conservative management. The patient was discharged at 15th postoperative day. Definitive histological examination showed cystadenocarcinoma of left hepatic duct with free margins (Figure 3). Immunohistochemical studies showed positive reactions for carcinoembryonic antigen (CEA), cytokeratin 19 and CA 19.9. No postoperative adjuvant treatment was performed. To date, one year of follow-up, the patient is alive without tumor recurrence.

 

DISCUSSION

 

BCAC is a rare malignant epithelial neoplasm of bile ducts. Takayasu et al.9 reported an incidence of 0,41 % among all hepatic malignant epithelial tumors. Almost all of these tumors are intrahepatic while the minority arise in the major bile ducts or even gallbladder (extrahepatic origin). EBCAC arise more frequently hepatic confluence or even sectorial duct (left or right) than gallbladder1,3,6,7,8,10. There are few reports on literature about these uncommon neoplasms1,2,3,4,5,6,7,8,9,10. Especially in Brazil, no case was reported. Azambuja et al.1 reported a single case but this tumor was an intrahepatic BCAC1. This tumor is more frequently observed in middle-aged people and may occur in both gender having a less favorable prognosis in man3. In accordance to Devaney et al.4 there are at least two well-defined types of hepatobiliary cystadenocarcinoma. One developing exclusively in female patients, usually accompanied by an "ovarian-like" stroma, which follows an indolent course and the other, lacking the distinct cellular stroma, seen in males, follows an aggressive biological behavior that may result in the patient's death from tumor10.

 

While intra-hepatic BCAC leads abdominal mass or pain, EBCAC leads generally obstructive jaundice that may eventually be accompanied by symptoms. More lately, it may lead weight loss and ascites that denotes advanced disease1,3,6,7,8,10.

 

Despite its rarity, EBCAC should remind as differential diagnosis with hilar cholangiocarcinoma in patients who present high obstructive jaundice as the present case10. Like this tumor present more loco-regional dissemination than distant metastasis1,10.

 

Histological, BCAC are characterized by papillary projections with a multilayered cell lining with foci of dysplasia and moderate mitotic activity10. In BCAC most tumor cells were positive on immunohistochemical staining for cytoqueratin, epithelial membrane antigen, CA 19.9 and carcinoembryonic antigen4.

 

Although the diagnosis is histological and may be confirmed by immunohistochemistry, image examinations may contribute for suspect diagnosis. Computed tomography and more recently magnetic cholangioresonance may reveal multilocular cystic lesion with internal septations or nodularities. More rarely, as the case described, an unilocular lesion may be observed6. Some authors have suggested that presence of nodules or coarse calcifications along the wall or septa favor the BCAC2,6. However, as reported Tseng et al.10, EBCAC may be confounded with hilar cholangiocarcinoma because may lead dilatation of the intrahepatic biliary ducts and atrophy of the affected lobe such as observed in present case. Different from Tseng et al.10, the present authors observed no hepatolithiasis in this case. The real cause of atrophy of the left hepatic lobe was unknown.

 

Tseng et al.10 suggested that the tumor would lead compression of the left trunk of the portal vein that could deviate hepatic flow for right lobe. As this case of the EBCAC, it presents a difficult differential diagnosis with common hilar cholangiocarcinoma because on image examinations EBCAC may be confounded due intense dilatation of intra-hepatic biliary tree. Independently of the diagnosis as EBCAC as hilar cholangiocarcinoma, the treatment has been total resection of the lesion with free margins, because partial resection has been associated a high levels of recurrence and dismal prognosis1,2,3,4,5,6,7,8,9. Generally, surgical resection may be associated with hepatectomy, due proximity of the hepatic parenchyma3,10. Radical resection is the only curative treatment and may be associated either cure or long-term survival1,2,3,4,5,6,7,8,9. The overall prognosis may be good since a radical resection is performed, as in the present case.

 

REFERENCES

  • 1. Azambuja E, Batista RG, Waetcher FL, Sampaio JA, Alvares-da-Silva MR, Fleck JF. Biliary cystoadenocarcinoma. Revista AMRIGS 2004; 48(3): 186-9.
  • 2. Buetow PC, Buck JL, Pantograg-Brown L. Biliary cystadenoma and cystadenocarcinoma: clinical-imaging-pathologic correlations with emphasis on the importance of ovarian stroma. Radiology 1995; 196: 805-10.
  • 3. Davies W, Chow M, Nagorney D. Extrahepatic Biliary Cystadenomas and Cystadenocarcinoma. Report of Seven Cases and Review of the Literature. Ann Surg 1995; 222(5), 619-25.
  • 4. Devaney K, Goodman ZD, Ishak KG. Hepatobiliary Cystadenoma and Cystadenocarcinoma: A Light Microscopic and Immunohistochemical Study of 70 patients. Am J Surg Pathol 1994; 18 (11): 1078-91.
  • 5. Erdogan D, Busch ORC, Van Delden OM, Gouma DJ, Van Gulik T. World J Gastroenterol 2006; 12 (35): 5735-8.
  • 6. Korobkin M, Stephens DH, Lee JK. Biliary cystadenoma and cystadenocarcinoma: CT and sonographic findings. A J R 1989; 153: 507-11.
  • 7. Lauffer JM, Baer HU, Maurer CA, Stoupis CH, Buchler MW. Biliary cystadenocarcinoma of the liver: the need for complete resection. Eur J Cancer 1998; 34 (12): 1845-51.
  • 8. Owono P, Scoazec JY, Valete PJ, Dumortier J, Gouysse G, Berger F, Boulez J, Partensky C. Cystadénomes et cystadénocarcinomes hépatobiliaires. Etude clinique, radiologique et anatomopathologique de 7 cas. Gastroenterol Clin Biol 2001; 25: 414-21.
  • 9. Takayasu K, Muramatsu Y, Moriyama N. Imaging diagnosis of bile duct cystadenocarcinoma. Cancer 1988; 61: 941-6.
  • 10. Tseng C, Pan YS, Chen CY, Liu CS, Wu DC, Wang WM et al. Biliary cystadenocracinoma associated with atrophy of the left hepatic lobe and hepatolithiasis mimicking intrahepatic cholangiocarcinoma: A case report. Kaohsiung J Med Sci 2004; 20(4): 198-202.
  • 11. Yu Q, Chen T, wan YL, Min J, Guo H. Intrahepatic biliary cystadenocarcinoma: clinical analysis of 4 cases. Hepatobiliary Pancreat Dis Int 2009; 8 (1): 71-4.

LETTER TO THE EDITOR

 

Approach to upper digestive hemorrhage with diagnosis of cavernous lymphangioma

 

Carlos Rafael Lima Monção

 

Santa Casa de Montes Claros, Minas Geral, MG, Brazil

 

Correspondence

 

INTRODUCTION

 

Lymphangioma consists of lymph vessels-like structures. Are considered benign tumors of such vessels. Presents three histological types: cystic cavernous and simple and can occur in different regions of the body such as head and neck, mesentery and gastrointestinal tract1,5,6. This publication aims to expose a rare case of cavernous lymphangioma, diagnosed after extensive workup for upper gastrointestinal bleeding.

 

CASE REPORT

 

Men 68 years, hypertensive patient, with a history of ischemic stroke two years ago. He sought medical attention with severe microcytic and hypochromic anemia and several episodes of melena. Underwet to endoscopy evidencing on the second duodenal portion granulous inflammatory process, friability and presence of blood residues, mild distal esophagitis and mucosal pallor. Biopsy revealed a diagnosis of chronic nonspecific duodenitis. He was submitted two more endoscopies with biopsies that showed the same endoscopic and histological changes without adding any other data.

 

Proceeding with colonoscopy, mucosa presented with color, brightness and transparency usual, but with the presence of blackened secretion in the cecum. Image examinations found in abdominal ultrasonography mesogastric inferiorly in the pancreas region two nodules above the aorta measuring about 3.7 x 1.9 and 2.0 x 1.5 cm (Figure 1).

 

In total abdominal CT contrast, it was noted thickening of the third portion of the duodenum and proximal jejunum with massive retroperitoneal lymph node, and several lymph nodes diffusely distributed (Figure 2).

 

The intestinal transit showed irregularities in mucosal, thickening at the third portion of the duodenum and proximal jejunum of unknown etiology. Was further performed abdominal aortography and selective arteriography of the superior and inferior mesenteric artery, not being evidenced by angiography signs of active bleeding.

 

The treatment was performed by laparotomy with inventory of abdominal cavity, where it was found vegetating lesion infiltrating the proximal jejunum and mesentery. Histology showed portions of tissue with blood and lymphatic vascular spaces lined by endothelium with fibrofatty stroma sometimes forming cysts; the final diagnosis was cavernous linfoangioma

 

DISCUSSION

 

Linfagiomas are benign lesions that are usually congenital malformations, especially in regions of the head and neck, but can affect various topographies and organs4. Therefore, symptoms and complications depend on the site and size. Three histologic subtypes are presented separately for the most part, however there may be different classes within a single tumor.

 

The diagnosis may be suspected by ultrasonography, computed tomography and magnetic resonance imaging of the affected segment, and do not present any characteristic in these exams7. Confirmation must be made by histopathologic or immunohistochemistry2.

 

A surgical excision of the lesion and adjacent tissues consists the better treatment5. A partial resection is associated with high recurrence rate, reaching to 50% at two years3.

 

REFERENCES

  • 1. Christison-Lagay ER, Fishman SJ. Vascular Anormalies. Surg Clin N Am 2006; 86: 393-425.
  • 2. Glasgow SC, Birnbaum EH, Lowney JK, et al. Retrorectal Tumors: A diagnostic and Therapeutic Chalenge. Dis Colon Rectum 2005; 48: 1581-87.
  • 3. Hebra A, Brown M, McGeehin K, Ross A - Mesenteric, omental, and retroperitoneal cysts in children: the clinical study of 22 marries. South Med J, 1993; 86: 173-6.
  • 4. Legienh GM, Heran MKS. Classification, Diagnosis and Interventional Radiologic Management of Vascular Malformations. Orthop Clin N Am 2006; 37: 435 -74.
  • 5. Perrot M, Rostan O, Morel P, et al - Abdominal lynphangioma in adults and children. Br J Surg, 1998; 2:385-95.
  • 6. Schefter RP, Olsen KD, Gaffey TA. Cervical lymphangioma in the adult. Otolaryngol Head Neck Surg 1985;93:65-9.
  • 7. Vara-Thorbeck C, Méndez RT, Hidalgo RH, et al Laparoscopy resection of a giant mesenteric cystic lymphangioma. Eur J Surg 1997; 163: 395-6.

LETTER TO THE EDITOR

 

Giant fibrovascular polyp of the esophagus: a diagnostic challenge

 

Filipe P. Madeira; Jonatan William R. Justo; Cacio R. Wietzycoski; Lucas M. Burttet; Cleber Dario Pinto Kruel; André Pereira da Rosa

 

Department of Digestive Surgery, Hospital de Clinicas de Porto Alegre, Universidade Federal do Rio Grande do Sul, Porto Alegre, RS, Brazil

 

Correspondence

 

INTRODUCTION

 

Fibrovascular polyps of the esophagus are rare benign tumors, with just over 100 cases reported in the literature. Are pedunculated, intraluminal, covered by normal mucosa and composed of vascular, adipose and fibrous tissue3. It originates usually on the proximal third of the esophagus and can reach large dimensions. The symptoms only appear when the polyp becomes large. There are reports of sudden death from asphyxiation after regurgitation of the tumor11. The incidence is higher in men, 50 to 60 years3,10. In view of the debilitating symptoms and the risk, resection is indicated and usually performed by cervical esophagotomy3. Endoscopic surgery is reserved for cases of small polyps.

 

CASE REPORT

 

Data were collected from medical records of the patient after written consent and approval by the Ethics Committee of the Hospital de Clínicas de Porto Alegre (HCPA).

 

Man of 47 years came to the clinic of Digestive Tract Surgery of HCPA complaining of dysphagia for solids and liquids, regurgitation and odynophagia in about six months. Endoscopy showed extensive dilatation, without injury or luminal stenosis. Contrast radiography of esophagus, stomach and duodenum showed dilation of the thoracic esophagus without signs of stenosis of the lower esophagus or hiatal hernia. Chagas serology and esophageal manometry were requested to investigate chagasic megaesophagus and achalasia. He returned with worsening of the dysphagia, frequent regurgitation with loss of 18 kg in a few months. The Chagas serology was negative and manometry was normal to lower esophageal sphincter with incomplete relaxation; the esophageal body had aperistalsis. PH monitoring was also performed which revealed no pathologic reflux. Upon diagnosis of achalasia was performed Heller esophagomyotomy and Dor fundoplication by laparoscopy. The patient progressed well, but remained feeding only liquids, with partial relief of dysphagia, requiring nasoenteric probe for proper nutrition.

 

New endoscopy (Figure 1) after five months showed important bulging, not allowing the adequate distension of the organ, extending about 3 cm of the esophagogastric junction. The conclusion was suggestive of esophageal intramural hematoma, or extrinsic compression. Radiographic study showed megaesophagus and suggestive tumoral image on the distal esophagus. New endoscopy after seven months visualized tumoral image just below the upper esophageal sphincter, whose biopsy in ulcerated surface at 25 cm of the upper dental arch showed spindle mesenchymal cell neoplasm. Chest tomography scan showed distended esophagus, heterogeneous to the cardia and compressing the adjacent structures. Due to the presence of advanced esophageal disease, and no improvement after esophagomyotomy, was suspected the existence of mesenchymal neoplasm (GIST); was indicated esophagectomy with gastric replacement. The operation was performed by transthoracic approach, without complications. The specimen visualized intraluminal giant pedunculated polyp (Figure 2), originated near the upper esophageal sphincter and protruded through the esophageal body about 23 cm in length and up to 9 cm in diameter (Figure 3). Pathological examination showed fibrovascular polyp of the esophagus.

 

On the 3rd postoperative day the patient developed pneumothorax and atelectasis. He underwent bronchoscopy and chest drainage. On the 7th day was done contrast radiography, which showed good transit and absence of contrast extravasation. He was discharged on the 15th day with good acceptance of diet. In follow-up of three months was asymptomatic recovering 11 kg.

 

DISCUSSION

 

Benign tumors of the esophagus are relatively rare. Among them, only 1-2% are giant fibrovascular polyps 3,11. According to their location, benign esophageal tumors are divided into intramural, extramural and intraluminal. Among the intraluminal, fibrovascular polyps are the most common. It consists of a network of lipomas in conjunction with loose or dense connective tissue, interspersed by a network of vessels and covered by squamous epithelium2,9.

 

Usually these polyps arise from the cervical esophagus, inferiorly to the cricopharyngeal muscle, the triangle of Laimer3,8,12,13. Redundant mucous folds of the region result in a polypoid formation. The peristaltic activity from the esophagus through constant traction over the years, leads to the growth of this lesion11. Is believed to have low capacity for malignant transformation3.

 

Polyps are fibrovascular indolent behavior, so that, with their slow growth rate, these lesions may reach masses of large volume, but without developing symptoms for many years. When they appear, they tend to be nonspecific2,4,6,7,8,11,13,14. However, the most characteristic symptom is tumoral regurgitation to the oral cavity9,10,11. It is infrequent and feared, because can obstruct the larynx and suffocate the patient11.

 

For purposes of diagnostic investigation, barium studies are commonly used2,4,5,11,15. However, it should be noted that they have low sensitivity to demonstrate the pedicle of the lesion5. In this case, the x-ray showed no tumor, however, suggested a foreign body obstruction in the distal esophagus. Endoscopy visualizes polypoid mass, elongated, emerging from the upper esophagus1,7,8,13,15, however due to the squamous epithelium covering the lesion it can be false negative or inconclusive6,12,14 The endoscopic ultrasound may be useful as adjunct imaging examination, since it provides information about the size and origin of the lesion on the vascularization of polyps13. It was not performed in this case due it is not available on the service. Tomography and resonance are more accurate in evaluating these lesions2,4,5,7,12. This is considered adjunct tool which may be useful both for diagnosis and for surgical planning3,15.

 

Despite benign polyps resection is usually recommended11. There are two options: surgery or endoscopic resection, based on lesion size and vessels that nourish it. Usually polyps less than 2 cm in diameter with thin pedicles may be removed by endoscopic pedicle ligation and electrocautery10. Surgical resection is preferred in larger sizes for cervical esophagotomy. In this case, the lesion had more than 20 cm, practically eliminates any possibility of endoscopic resection.1. Thoracotomy is considered to more caudal lesion or larger lesions, thus obtaining the best possible management of intraoperative bleeding4,8,15. Esophagectomy can be used in bulky tumor6 or in doubt about benign behavior12. In thiscase, was opted esophagectomy because the patient already had advanced disease, esophageal dilation and aperistalsis, associated with suspected mesenchymal neoplasm.

 

REFERENCES

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