LETTER TO THE EDITOR

 

Textiloma in abdominal cavity: 35 years later

 

Francisco Venditto Soares; Luciano Vicentini; Alfredo Rafael Dell'Aringa; Luís Carlos de Paula e Silva

 

Hospital das Clínicas, Medical Faculty of Marília, Marília, SP, Brazil

 

Correspondence

 

INTRODUCTION

 

The description of foreign body in patients who have undergone surgical procedures is increasing. This fact has contributed to the development of strategies to promote tools to prevent these events, professional qualification and early detection.

 

The first report of a case with the presence of a foreign body in the abdominal cavity was described in 1884. Since then several others are being described and indicate a higher frequency of textile products such as tampons and surgical gauze1,2.

 

Recent studies have shown that the incidence of foreign body in the postoperative period is one in a thousand cases, predominantly after gynecological and obstetric operations. Prevails those performed in emergency conditions. Mortality can reach 35%3,4,5.

 

The presence of foreign material in the body can lead to septic fibrosis, adhesions, encapsulation becoming granuloma and abscess with bacterial colonization or not.

 

These manifestations can appear in different periods after surgery. From the first 24 hours to 8th to 13th days granulomatous inflammation emerge that aim to destroy the foreign body. After five years can be absorbed, or even be calcified6.

 

The surgical gauze are made from cotton material that rarely produces reactions in contact with the body. However, after a period of exposure can trigger reactions that lead to formation of granulomas4.

 

This study reports a case of intestinal blockage by gauze after surgical procedure held 35 years before.

 

CASE REPORT

 

Man 68 years sought assistance with abdominal discomfort that began 12 hours with localized abdominal pain in the epigastric region after food intake. Subsequently, the pain progressed to the whole abdomen accompanied by bloating, postprandial weight, nausea and lack of bowel movements. Said that underwent Billroth I gastrectomy at age 35 in a public hospital due to duodenal peptic ulcer bleeding.

 

On physical examination, he was dehydrated, abdomen distended and painful on palpation, decreased bowel sounds. Laboratory examinations that had relevance only to amylase with 760; ultrasonography showed distension of the small bowel loop, and computed tomography revealed dilated loop in paraduodenal region with wall suffering signs and air in the portal system.

 

Laparotomy with supraumbilical incision was done. Showed adherence to the wall, distention of the jejunal loop about one meter from the duodenojejunal angle with intraluminal content of spongy consistency completely occluding the lumen. Opening was held and a foreign body (surgical gauze) was seen inside that grouped and encapsulated. They were removed.

 

The intestinal wall was preserved without blockages at the site of obstruction. The incision was sutured and the abdomen closed in layers. The patient was discharged on day 7 postoperatively, with amylase of 85.

 

DISCUSSION

 

It is imperative that some considerations should be made in relationship the possibilities to prevent this situation. It is fundamental to routinely use detailed revision of the cavity in order to minimize risks.

 

There are several procedures that should be considered intraoperatively, and the literature is wide in the issue. Reports of foreign body accidentally left in the abdominal cavity are increasing. This fact has contributed to more reflective analysis, seeking professional better qualification and implementation of measures to promote prevention to become less frequent these serious complications.

 

REFERENCES

  • 1. Debnath D, Buxton JK, Koruth NM. Two Years of Wait and 7000 Miles of Journey: The Tale of a Gossypiboma. Int Surg. 2005;90:130-3. [ Links ]
  • 2. Filho G. Lopes. Corpos estranhos intra-abdominais. Jornal do Conselho Regional de Medicina do Estado São Paulo. Edição 219 - 11/2005.
  • 3. Iglesias AC, Salomão RM. Gossipiboma intra-abdominal análise de 15 casos. Rev Col Bras Cir. [periódico na Internet] 2007; 34(2). Disponível em URL: http://www.scielo.br/rcbc
  • 4. Jane Bunahum de Carvalho, José Carlos Vinhaes, Corpo estranho retido na cavidade abdominal durante onze anos. Rev. Col. Bras. Cir. Vol. 31 - Nº 1: 68-70, Jan. / Fev. 2004.
  • 5. Rappaport W, Haunes K. The retained surgical sponge following intra- abdominal surgery: a continuing problem. Arch Surg. 1990;125(3):405-7.
  • 6. Tinker MA, Burdman D, Deysine M, Teicher I, Platt. N, Aufses AH. Granulomatous peritonitis due to cellulose fibers from 121Rev Colomb Cir. 2009;24:114-22.

INTRODUTION

Situs inversus totalis is a rare congenital condition that occurs in one out of 4000-20000 people, characterized by complete transposition of the thoracic and abdominal viscera1 - 14. In contrast, situs solitus is a term that refers to the normal arrangement of body organs. Any disposition of organs between these two extremes is designated by situs ambiguous, situs transversus or situs inversus partialis8.

The etiologic nature of this anomaly is not known. This condition is typically associated with normal life expectancy unless a gastrointestinal or cardiac anomaly is present2 , 8. The typical cardiac anomaly has from 3-5% incidence, is transposition of the great vessels, and 80% of these patients have a right sided aortic arch5. Other vascular anomalies are variation of celiac trunk and superior mesenteric artery4. There are also anomalies of the gastrointestinal system to include biliary tree atresia, duodenal atresia, preduodenal portal vein, colonic aganglionosis, malrotation of the intestine, polysplenia/asplenia, anular pancreas, diaphragmatic hernia and others4 , 5. Moreover, it may be associated with clinical syndromes, like Kartagener's (situs inversus, chronic rhinosinusitis and bronchiectasias)14.

This anomaly is not a premalignant condition. However, many cases of malignant neoplasms and situs inversus totalis have been reported, especially gastric cancer8. Association between colorectal cancer and situs inversus totalis is rare.

CASE REPORT

Man with 74 years-old, white, ex-alcoholic and smoker had family history essentially negative for either situs inversus totalis, familial and hereditary disease or colorectal cancer. The patient didn´t know he had situs inversus totalis. He had a history of abdominal pain in left hemiabdome, asthenia and mucocutaneous pallor for the last two years. He had normal physical examination, except for pale mucous membranes, heart sounds audible in the right chest and slightly painful on palpation of the left abdomen. Abdominal ultrasound showed only abdominal situs inversus and colonoscopy demonstrated sub-oclusive lesion in hepatic angle of the colon, which biopsy revealed moderately differentiated adenocarcinoma. His carcinoembryonic antigen was 1,8 ng/dL. ECG and chest radiography indicated dextrocardia. Computerized tomography showed complete transposition of abdominal viscera, confirming situs inversus totalis (Figure 1).

 

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Figure 1 -
Chest x-ray film shows dextrocardia and findings of inversion of the abdominal organs in abdominal computed tomography Li=liver; St=stomach; Sp=spleen; Ao=aorta

 

According to these findings, a proximal hemicolectomy was carried out with lymph nodal dissection followed by ileocolic anastomosis. The cavity inventory at laparotomy showed situs inversus abdominal and presence of spherical and hard lesion in the hepatic flexure of colon (Figure 2) with absence of macroscopic metastatic involvement.

 

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Figure 2 -
Surgical abdominal findings: A) liver in the left upper part; B) appendix in lower left; C) duodenojejunal angle to the right; D) gallbladder in the left upper part; E) spleen and great curvature of the stomach in the right upper position; F) resected specimen showing a ulcerated mass in hepatic flexure of the colon.

 

Anatomopathological evaluation (Figure 2) confirmed tubular adenocarcinoma, moderately differentiate; presence of perineural invasion; vascular and angiolymphatic involvement were absent. TNM staging was T3N0M0, stage IIA.

Postoperative course was uneventful and he was discharged from the hospital on the 4th day after operation. He began adjuvant Mayo Clinic regimen, but stopped in the 4th cycle due toxicity of the gastrointestinal tract. Up to the moment of this writing, no sign of recurrence or metastasis has been observed.

DISCUSSION

In the literature, there are 13 cases recognized about this issue, making a total of 14 cases, by adding this report. Enrolling all papers, colorectal cancer was more frequent in women (n= 9; 64%) than men (n=5; 36%). The age ranged from 41- 78 years, mean of 63,71 and median of 61,5 (SD=±10,40). Adenocarcinoma was the histological type present in all cases. Regarding the location of the tumor, there was a predominance of the transverse colon (n=6; 43%), with emphasis on hepatic flexure of the colon (n=5; 36%), followed by ascending colon (n=4; 29%), rectum (n=3; 21%) cecum and sigmoid colon (n=1; 7% in both topographies). Grouping the tumors in the right colon (proximal to splenic flexure of the colon) and left (from the splenic flexure of the colon), noted a prevalence of 79% and 21%, respectively, with statistical significance (p=0.029), according to an exact test for proportion and level of significance was α=0.05. Regarding the surgical procedure, 10 (71%) patients underwent proximal hemicolectomy, and one case each (7%) of rectosigmoidectomy, abdomino-perineal amputation, transversectomy and decompression colostomy. Laparotomic surgical procedure accounted for 93% and only one case was operated by laparoscopic approach (7%)8.

Surgical procedures are considered more difficult in patients with situs inversus than other patients because of different anatomic position of organs, especially in laparoscopic surgery3.

The preoperative evaluation for situs inversus includes two main objectives: evaluation for gastrointestinal and cardiac anomalies and orientation of the viscera. The extent of evaluation should be based on the complexity of the procedure. Anomalies should be defined by using various imaging technologies to determine appropriate surgical treatment and decrease surgical difficulties and time4 , 5. Furthermore, the risk of occurrence of intra-operative complications is higher in comparison with the procedures of patients without situs inversus totalis1. Besides, incorrect surgical incision and a second operation are avoided2.

REFERENCES

  • 1
    Bielecki K, Gregorczyk M, Baczuk L. Visceral situs inversus in three patients. Wiad Lek. 2006; 59(9-10): 707-9.
  • 2
    Blegen HM. Surgery in situs inversus. Ann Surg. 1949; 129:244-59.
  • 3
    Fujiwara Y, Fukunaga Y, Higashino M, Tanimura S, Takemura M, Tanaka Y and Osugi H. Laparoscopic hemicolectomy in a patient with situs inversus totalis. World J Gastroenterol. 2007; 13(37): 5035-37.
  • 4
    Goi T, Kawasaki M, Yamazaki T, Koneri K, Katayama K, Hirose K and Yamaguchi A. Ascending colon cancer with hepatic metastasis and cholecystolithiasis in a patient with situs inversus totalis without any expression of UVRAG mRNA: report of a case. Surg Today. 2003; 33:702-6.
  • 5
    Greene QJ, Cheadle WG. Ascending colon cancer in a patient with situs inversus. Am Surg. 2007; 73(8):831-2.
  • 6
    Grigorjew A, Andrzejczak L. Colonic cancer in situs inversus. Wiad Lek. 1982; 35:901-2.
  • 7
    Ikenaga M, Mishima H, Hirao M, Fujitani K and Tsujinaka T. A case of multiple colorectal cancer with situs inversus totalis. J Jpa Coll Surg. 2005; 30(1):52-6.
  • 8
    Iwamura T, Shibata N, Haraguchi Y, Hasashi Y, Nishikawa T, Yamada H, Hayashi T and Toyoda K. Synchronous double cancer of the stomach and rectum with situs inversus totalis and polysplenia syndrome. J Clin Gastroenterol. 2001; 33(2):148-153.
  • 9
    Nagase T, Adachi I, Yoshino Y, Morita K, Murakami N and Yamada T. A case of ascending colon cancer with total situs inversus and intestinal malrotation. J Jpn Surg Assoc. 2003; 64(7):1773-6.
  • 10
    Reiff PS, Mathias LR, Souza IES, Reif ABM. Surgery in situs inversus. A case report (acute cholecistitis and carcinoma of the colon). Rev bras Cir. 1992; 82(4):149-50.
  • 11
    Shibuya J, Nakakuma T, Oshima Y, Kakita A. A operation case of situs inversus totalis associated with a transverse colon cancer. Nippon Gekakei Rengo Gakkaishi. 2003; 28(2):271-4.
  • 12
    Takizawa Y, Maeda K, Shimizu J, Kawaura Y. A case of colonic cancer associated with situs inversus totalis. Surgery. 2002; 64(2):245-8.
  • 13
    Wright CB, Morton CB. Situs inversus totalis with adenocarcinoma of the cecum: case report. Am Surg. 1971; 37(2): 65-6.
  • 14
    Yoshida J, Tsuneyoshi M, Nakamura K, Murakami T, Akamine Y. Primary ciliary dyskinesia with transverse colon carcinoma. Am J Clin Pathol. 1986; 85:101-104.

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INTRODUCTION

Endometrial tissue localized outside the uterine cavity is defined as endometriosis. It commonly has been demonstrated in the ovaries, peritoneal surfaces, vagina, scar tissues, cervix, fallopian tubes, rectum, urinary tract, pouch of Douglas and possibly any organ in the abdomen1. The estimated prevalence reported in literature ranges from 8-15%2. Extra-pelvic localization of endometriomas are relatively rare. Especially abdominal wall placements are very infrequent. Usually such cases are associated with surgical scars3. The proposed mechanisms that have been put include retrograde menstruation, venous or lymphatic dissemination or metastasis, and mechanical transplantation into scars at the time of surgery4.

Herein, is presented a case of endometrioma localized in the rectus abdominis muscle.

CASE REPORT

A 31 year old female with a history of two cesarean sections, the last one of them three years prior, presented with a painful mass in the left lateral side of the Pfannenstiel incision which had been steadily growing. The same patient went a month ago to our institution's urology clinic with pain in the inguinal region. After a detailed history and physical examination an ultrasonography was performed that revealed a 20X12 mm hypo-echoic nodular mass, neighboring the rectus abdominis muscle, with minimal vascularization. The differential diagnosis included endometriosis and a possible desmoid tumor. The patient was referred to our clinic for further evaluation. The pain started on the left lower quadrant and radiated towards the inguinal region, and was associated with menstruation. In physical examination a 2 cm wide mass was palpated in the previously described localization. Magnetic resonance imaging was performed which revealed a 20x11 mm mass which is slightly hyper-intense in the T1 sequence, and contrast enhanced after IV gadolinium injection in the T2 sequence, with increased signal intensity and nodular appearance in diffusion weighted sequences. These were found to be consistent with an endometrial implant (Figures 1A and 1B). Examination of the uterine cavity showed effusion which was at most 15 mm in width when measured. A little free fluid, indicating peritoneal irritation was present in between the intestinal loops. No pathological lymph nodes were present in the lower abdomen.

 

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Figure 1 -
Arrows show the area of endometriosis in rectus abdominis muscle: A) CT scan in

 

The patient was admitted for surgical removal of the mass. A 2 cm wide fibrotic appearing mass was excised (Figure 2) and was sent for pathological examination. The patient's complaints resolved after the surgery. She was discharged with no complications two days after the operation. Four months after the surgery, the patient came in for a follow-up visit, and had no complaints or complications. Histopathological examination was consistent with glandular structures of the endometrium with accompanying endometrial stroma within muscle and connective tissue (Figure 3).

 

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Figure 2 -
The mass excised in the operation

 

 

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Figure 3 -
Typical endometrium glands and spindle endometrium stroma existed in the area of endometrosis (H&Ex50)

 

DISCUSSION

Ectopic endometrial tissue localized in the rectus abdominis tissue is a very rare occurrence. Previously there have been only 20 reported cases in literature5. The first one of these cases was presented by Amato et al, in 19846. Giannella et al. has reviewed the previously reported cases extensively; their clinico-pathological characteristics, summarized, were: endometriosis with rectus abdominis placement usually is seen in premenopausal women, aged 27-42y, and history of previous surgery (77%), similarly to this patient. The average size of the endometriomas were 4X4 cm in diameter. This case had a much smaller dimension. The imaging studies' measurements were 2 cm at the greatest width. While CT scan has most commonly been used, in this case imaging diagnosis preferred to use ultrasound followed by an MRI7. Fine needle aspiration has also been tried in these cases; however, failed to prove effective in establishing diagnosis8.

Cesarean section is very frequently associated with abdominal wall endometriosis. The incidence, as reported in previous literature, can be as high as 1%. One of the proposed theories for how this occurs, takes into consideration the possibility that during the operation, endometrial cells may escape through the incision in the uterus and implant themselves within the abdominal incision site8.

This patient presented with cyclic symptoms that were associated with menstruation. The differential diagnosis of cyclic pain in general include lymphadenopathy, mesenteric lymphadenitis, lipoma, abscesses, hernias, hematomas, soft tissue sarcomas, desmoid tumors (which was considered in differential diagnosis) and even metastatic cancer. Previously some studies have looked into the utility of biochemical markers for tracking endometriosis. These markers include CA-125, C-reactive protein, anti-mullerian hormone, follistatin9 - 12.

Previously conducted sonographic studies have determined that abdominal wall endometriomas (which were first demonstrated via ultrasound in 197913) are commonly hypo-echoic, well defined, solid masses; this is consistent with sonographic findings of this case14. Medical treatment for these conditions, which have been previously utilized in literature, include danazol and progesterone; however, this treatment is frequently inefficacious, and therefore must be reserved for cases in which surgical treatment is not preferred15.

To summarize, in masses which present with cyclic pain and growth, localized to the abdominal surface, endometriomas must be considered in the differential diagnosis. Surgical removal, as evidenced previously reported cases, is successful, especially when limited and localized within in the rectus abdominis muscle. Sonography followed by resonance, provides the most definitive imaging. Molecular markers are currently not established enough to be considered as a standard of diagnosis. Further large-scale studies or reviews are necessary to determine which approach is the best, with consideration of the patient's request.

REFERENCES

  • 1
    Woodward PJ, Sohaey R, Mezzetti TP. Endometriosis: radiologic-pathologic correlation. Radiographics 2001;21: 193-216.
  • 2
    Horton JD, Dezee KJ, Ahnfeldt EP et al.Abdominal wall endometriosis: a surgeon's perspective and review of 445 cases.Am J Surg. 2008 Aug;196(2):207-12
  • 3
    Erkan N, Haciyanli M, Sayhan H.Abdominal wall endometriomas.Int J Gynaecol Obstet. 2005 Apr;89(1):59-60.
  • 4
    Gunes M, Kayikcioglu F, Ozturkoglu E.Incisional endometriosis after cesarean section, episiotomy and other gynecologic procedures.J Obstet Gynaecol Res. 2005 Oct;31(5):471-5.
  • 5
    Calò PG, Ambu R, Medas F et al.Rectus abdominis muscle endometriosis Report of two cases and review of the literature.Ann Ital Chir. 2012 Jun 20 .
  • 6
    Amato M, Levitt R.Abdominal wall endometrioma: CT findings.J Comput Assist Tomogr. 1984 Dec;8(6):1213-4.
  • 7
    Giannella L, La Marca A, Ternelli G. et al.Rectus abdominis muscle endometriosis: case report and review of the literature.J Obstet Gynaecol Res. 2010 Aug;36(4):902-6
  • 8
    Bumpers HL, Butler KL, Best IM.Endometrioma of the abdominal wall.Am J Obstet Gynecol. 2002 Dec;187(6):1709-10.
  • 9
    Luisi S, Gabbanini M, Sollazzi S Surgical scar endometriosis after Cesarean section: a case report.Gynecol Endocrinol. 2006 May;22(5):284-5.
  • 10
    Lermann J, Mueller A, Körber F. Evaluation of high-sensitivity C-reactive protein in comparison with C-reactive protein as biochemical serum markers in women with endometriosis. Fertil Steril. 2010 May 1;93(7):2125-9.
  • 11
    Lemos NA, Arbo E, Scalco R. Decreased anti-Müllerian hormone and altered ovarian follicular cohort in infertile patients with mild/minimal endometriosis. Fertil Steril. 2008 May;89(5):1064-8.
  • 12
    Florio P, Reis FM, Torres PB. High serum follistatin levels in women with ovarian endometriosis. Hum Reprod. 2009 Oct;24(10):2600-6
  • 13
    Miller WB Jr, Melson GL.Abdominal wall endometrioma.AJR Am J Roentgenol. 1979 Mar;132(3):467-8.
  • 14
    Aleem F, Pennisi J, Zeitoun K. The role of color Doppler in diagnosis of endometriomas. Ultrasound Obstet Gynecol. 1995 Jan;5(1):51-4.
  • 15
    Koger KE, Shatney CH, Hodge K.Surgical scar endometrioma.Surg Gynecol Obstet. 1993 Sep;177(3):243-6.

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INTRODUCTION

Benign tumors of the hepatoduodenal ampulla are rare, with few cases reported in the world literature. Among these tumors adenomas, lipomas, hemangiomas, carcinoid and leiomyomas have been described. The villous adenoma remains the most frequent2 ,5. Necropsy studies have shown an incidence of around 0.04% to 0.12%, and for this rarely remembered in the periampullary lesions differential diagnosis2.

The adenoma of it affects more frequently women, an age group between 5th and 7th decades, and is often associated with colonic polyposis. Initially, the patient may be asymptomatic and clinical findings begin to emerge with the growth of the tumor2 , 5.

The most common symptoms are biliary colic pain type and gastrointestinal bleeding; in some cases there may be pancreatitis and obstructive jaundice5. The diagnosis is given by histopathological lesion obtained by upper endoscopy with duodenal papilla vision and always colonoscopy should be performed to identify adenomatous colonic polyposis.

Some authors2 , 3 , 5 believe in the existence of a pre-malignant lesions, where as malignant degeneration occurs in 30-40% of cases. Because of this potential for considerable degeneration, treatment is still controversial, with numerous discussions about the best approach, endoscopic, surgical with it transduodenal local excision or pancreaticoduodenectomy.

CASE REPORT

Woman of 77 years was admitted in General and Digestive Surgery Service, Walter Cantídio University Hospital,Fortaleza, CE, Brazil. Three months prior to admission, the patient began exhibiting abdominal pain in the epigastrium without irradiation and not associated with other symptoms. She denied weight loss, diarrhea, appetite loss or icterus. On examination she was in good general condition, normal colored and hydrated, focused and normal vital signs. Cardiac auscultation, pulmonary and abdominal examination without changes. Suffered from systemic hypertension, diabetes mellitus type 2 and dyslipidemia in regular medication use.

Laboratory investigation showed normal serum levels of transaminases and bilirubin. Endoscopy visualized polypoid lesion in the duodenal papilla, measuring about 5 cm, which was biopsied at seven different sites. Histopathology was compatible with tubulo-villous adenoma with low grade dysplasia. Abdominal ultrasonography showed normal common bile duct and gallbladder and bile ducts without dilatation. Computed tomography of the abdomen showed no changes. The colonoscopy was normal, not evidencing any other polypoid lesion.

The patient was taken to the operating room for the purpose of a transduodenal local resection of the tumor (ampulectomy), but with all the pre-operative support for a possible PD. During surgery, after duodenotomy, polypoid lesion of approximately 6 cm in the duodenal papilla (Figure 1) was observed.

 

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Figure 1 -
Polypoid lesion o f the papilla: laparotomic transduodenal viewing

 

The lesion was completely resected and the main pancreatic duct and common bile duct were fixed with synthetic absorbable suture. The tumor was sent for frozen biopsy, which resulted in tubulo-villous adenoma with high-grade dysplasia. Ampulectomy was done and was also performed catheterization with good visualization of the common bile duct and main pancreatic duct (Figure 2).

 

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Figure 2 -
Catheterization of the terminal common bile duct with output of bile and catheterization of main pancreatic duct with output of pancreatic secretion

 

With the resected lesion was necessary to reconstruct the gap left. The top wall of the pancreatic duct was sutured to the bottom wall of the bile duct with interrupted stitches in synthetic absorbable sutures, thus forming a common channel. The duodenum and the "new hepatopancreatic ampulla" were united with separated 4-0 absorbable synthetic stiches to create an anastomosis of the mucosal lining of the ducts with the duodenal wall. Afterwards, followed the duodenorrhaphy with separate 3-0 silk sutures.

Postoperatively, the patient stayed clinically stable, with physiological parameters and good diet acceptance. He was discharged on the 6th postoperative day.

Currently, the patient is asymptomatic and at six months follow-up. The final histopathology of the surgical specimen was tubulo-villous adenoma with high-grade dysplasia and free surgical margins. Postoperative endoscopy showed only papilla surgical scar without tumor recurrence.

DISCUSSION

Benign tumors of the duodenal papilla have a very low incidence around 0.04 to 0.12%. And so, hardly enter the differential diagnosis of jaundice by periampullary diseases. Most patients remained asymptomatic and develops symptoms of biliary colic, gastrointestinal bleeding, or jaundice in accordance with tumor growth2 , 3 , 5.

The patient in this case complained of pain in the upper abdomen, but at no time presented jaundiced. The diagnosis was confirmed with histopathology obtained by endoscopy.

The treatment of benign tumors of the papilla is still controversial. There is no consensus in the literature about the best approach to these tumors, endoscopic, if surgical resection under local or pancreaticoduodenectomy1 , 3 , 5. The proper approach to the patient should be thoroughly discussed, it is known that there is a likelihood of malignancy of these tumors of approximately 30-40%. In this case, patients undergoing endoscopic treatment or surgical treatment with local resection should be rigorously followed in the clinic so that recurrences are detected early2 , 3 , 5.

The higher probability of malignant transformation of these tumors depends on a few factors: the larger the tumor, the greater the chance of harboring "islands" of adenocarcinoma; villous tumors are more associated with degeneration to carcinoma; multicentric tumors have a higher risk of malignancy and lesions located in the ampulla are more prone to malignancy than lesions located in the duodenum and small intestine2. Some small adenomas (<1 cm) can be removed endoscopically with stenting of the biliar and pancreatic ducts to allow the resolution without stenosis. Options for endoscopic resection of these lesions include papillectomy or ampulectomy with snare and thermal ablation with laser, argon plasma coagulation and electrosurgery. Four criteria for eligibility endoscopic papillectomy are needed: the injury must have less than 4 cm and cannot be endoscopic malignancy appearance (must have regular margin, absence of ulceration and soft consistency); minimum of six biopsies confirmed the benign histology and absence ductal invasion by endoscopic ultrasound3 , 5. However, endoscopic resection has a recurrence rate of 30% and often multiple attempts to be necessary before complete tumor eradication. Morbidity about 20% have been reported, including pancreatitis and duodenal perforation5.

Surgical options for ampullary tumors include transduodenal local excision or pancreaticoduodenectomy. In the case of villous adenomas, the risk of malignancy and the loss of opportunity to cure must be weighed against the complications of pancreaticoduodenectomy 1 , 2 , 3 , 5.

Thinking of the considerable risk of malignancy, one can agree that the best approach would be the pancreaticoduodenectomy, but one should be aware of the significant morbidity and mortality of this procedure1. The mortality rate of pancreaticoduodenectomy in centers specializing in pancreatic surgery is in the range of 2-3%. Despite low mortality rates, the incidence of postoperative complications remains high. Yeo et al. 4 showed a series of 650 consecutive pancreaticoduodenectomy in the John Hopkins Hospital. The mortality rate was 1.4%. The three most common complications were delayed gastric emptying in 19%, pancreatic fistula in 14% and surgical wound infection in 10%. These conditions also significantly increase the duration of hospitalization for the patient and hospital costs3 , 4 , 5.

Even with so much discussion about the best surgical approach, it is known that most surgeons agree that patients should be brought to the operating room with pre-operative support for a wide resection for pancreaticoduodenectomy5. This patient preparation is essential if freezing demonstrate malignancy. If all these precautions are taken, it is known that local excision transduodenal can be an excellent treatment for benign tumors of the bulb, especially in patients with high surgical risk for PD12 , 5. After resection of the local ampullary tumors should perform endoscopic control 6-12 months after surgery to assess local recurrence. Patients who have pancreatitis or jaundice months or years after local excision should undergo endoscopy with endoscopic retrograde cholangiopancreatography due to strong suspicion of relapse1 , 5.

In this patient, it was decided to hold the local surgical resection with ampulectomy, because it showed the tumor>4 cm, histopathological (obtained for seven biopsies) was compatible with villous adenoma, freezing intraoperatively confirmed the nature benign lesion and clinically the patient had comorbidities and high surgical risk.

REFERENCES

  • 1
    COELHO, Júlio Cezar Uili. APARELHO DIGESTIVO - CLÍNICA E CIRURGIA. 3rd edition. Atheneu.
  • 2
    WIEDERKEHR, Julio Cesar. COELHO, Julio Cezar Uili. SIGWALT, Marcos Fabiano. NASSIF, Aissar Eduardo. Adenoma viloso da ampola de Vater. Revista do Colégio Brasileiro de Cirurgiões. Volume XXV. Número 6.
  • 3
    YEO, Charles. DEMPSEY, Daniel. KLEIN, Andrew. PEMBERTON, John. PETERS, Jeffrey. SHACKELFORD´S SURGERY OF THE ALIMENTARY TRACT. 2007. 6th edition.
  • 4
    YEO, CJ. CAMERON, JL. Six hundred fifty consecutive pancreaticoduodenectomies in the 1990s: pathology, complications, and outcomes. Ann Surg 1997; Sep; 226(3): 248-57.
  • 5
    ZINNER, Michael. ASHLEY, Stanley. MAINGOT - Abdominal Surgery. 2011. 11th edition.

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INTRODUCTION

Anatomical variations of the hepatic artery are commonly found during radiological examinations and abdominal operations1 , 89 It is estimated that the variation index reach 45% of the population8. The significant prevalence gives this type of variation major medical importance, and justifies the surgeon know it to avoid iatrogenic injury.

The usual blood vessel anatomy is the common hepatic artery ascend from the celiac trunk8; however, this arrangement may change due to embryonic variations. These variations, from most to least frequent are: 1) the right hepatic artery ascend from the superior mesenteric artery; 2) the left hepatic artery ascend from the left gastric artery; 3) the two events occur simultaneously; and 4) the common hepatic artery ascend from the superior mesenteric artery.

In this article, is reported the occurrence of the fourth situation. According to the literature, there is no consensus on the criteria for designation of this variation. Therefore, it can be described in two ways: consider it a hepatomesenteric trunk - giving rise to the superior mesenteric artery and common hepatic artery - or it can be said that the common hepatic artery arises as a branch of the superior mesenteric artery.

The objective of this paper is to present a case and emphasize the prevalence of anomalous positions of the hepatic arteries and their possible implications.

CASE REPORT

It was observed in a male corpse, common hepatic artery originating from the superior mesenteric artery, located 3.5 cm lower and lateral from the celiac trunk, forming a hepatomesenteric trunk. The other branches of the celiac trunk were normal and exhibited the typical path (Figure 1).

 

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Figure 1 -
A) Photograph of the abdominal cavity of the corpse with some vessels in evidence: the left renal vein (double arrow), celiac trunk (single arrow) and the hepatomesenteric trunk (indicated by *); B) illustration of the same photo, highlighting the hepatomesenteric trunk (indicated by *).

 

DISCUSSION

Variations of the hepatic artery have embryological basis10. During intrauterine development, there is the formation of four ventral splanchnic vessels, connected by a ventral longitudinal anastomosis. With the maturation of these, the two central roots degenerate. Thus, the first and fourth roots which form respectively the celiac artery and superior mesenteric artery anastomoses remain. If the separation between them occurs at different level of the standard pattern, any vessel in the celiac trunk can be shifted to the superior mesenteric artery. This situation appears in this case report: with the anomalous roots separation, there was formation of a hepatomesenteric trunk and other gastrosplenic.

Several studies1 , 2 , 3 , 4 , 5 , 6 , 7 , 8 , 9 , 10 , 11 report different variations. The most representative of them is to Hiatt et al.5 with a sample of 1000 people. The variation here presented (common hepatic artery + superior mesenteric artery) is of uncommon occurrence with an average of 2%. This value agrees well with the values ​​found in other articles, ranging from 1.6% to 3.5%.

ACKNOWLEDGEMENTS

The authors thank Priscilla Vieira Ely Hattori, technique laboratory of the Federal University of Grande Dourados (Dourados, Mato Grosso do Sul) for logistical support offered to the writing of this Letter to the Editor.

REFERENCES

  • 1
    Arjhansiri K, Charoenrat P, Kitsukjit W. Anatomic variations of the hepatic arteries in 200 patients done by angiography (resumo). J Med Assoc Thai. 2006 Sep; 89 Suppl 3: S161-8,.
  • 2
    Bertevello P L, Chaib E. Variações do sistema arterial hepático e sua aplicabilidade na bipartição do fígado: estudo anatômico em cadáveres. Arq. Gastroenterol. 2002, vol.39, n.2 [cited 2010-12-04], pp. 81-85.
  • 3
    Chen H, Yano R, Emura S, Shoumura S. Anatomic variation of the celiac trunk with special reference to hepatic artery patterns (resumo). Ann Anat. 2009 Oct; 191(4): 399-407.
  • 4
    Freitas A C T, Coelho J C U, Matias J E F et al. Anatomia arterial hepática: estudo em 150 transplantes hepáticos. Rev. Col. Bras. Cir. 2001, vol.28, n.1 [cited 2010-12-04], pp. 13-16.
  • 5
    Hiatt J R, Gabbay J, Busuttil R W. Surgical anatomy of the hepatic arteries in 1000 cases (resumo). Ann Surg., 1994 Jul, 220(1): 50-2.
  • 6
    Koops A, Wojciechowski B, Broering D C. Anatomic variations of the hepatic arteries in 604 selective celiac and superior mesenteric angiographies. Surg Radiol Anat, 2004 Jun, 26(3): 239-44.
  • 7
    Lopez-Andujar R, Moya A, Montalva E, Berenguer M Et Al. Lessons learned from anatomic variants of the hepatic artery in 1,081 transplanted livers (resumo). Liver Transpl, 2007 Oct 13(10): 1401-4.
  • 8
    Soares R V, Coelho J C U, Matias Jef, Zeni Neto C, Freitas A C T, Godoy J L. Anatomia da artéria hepática em doadores e receptores de transplante hepático intervivos. Rev Col Bras Cir. 2006 Mar-Abr; 33(2) .
  • 9
    Mburu K S, Alexander O J, Hassan S, et al. Variations in the Branching Pattern of the Celiac Trunk in a Kenyan Population. Int. J. Morphol. 2010, vol.28, n.1 [citado 2010-12-05], pp. 199-204 .
  • 10
    Ottone N E, Agustín A M, Domínguez M L et al. Arterias Hepáticas Aberrantes: Estudio en 64 Cadáveres Disecados. Int. J. Morphol. 2006, vol.24, n.4 [citado 2010-12-05], pp. 581-585 .
  • 11
    Song S Y, Chung J W, Yin Y H et al. Celiac axis and common hepatic artery variations in 5002 patients: systematic analysis with spiral CT and DSA. Radiology, , 2010 Apr 255(1): 278-88.

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INTRODUCTION

The presence of a vermiform appendix inside a hernial sac is not a common condition7. In the literature, the reported incidence is around 1% of all hernias6. It is even rarer to find an acute appendicitis inside the inguinal hernia4.

When the cecal appendix, inflamed or not, is found in the inguinal sac, it is called an Amyand hernia5. This kind of hernia is more frequent in men and pre-operative diagnosis is not easy9. It must be suspected in patients with a tense inguinal hernia with no signs of intestinal obstruction. The appendectomy will always be carried out at the same time as the repair of the hernia.

The aim of the present study is to present a case of acute appendicitis within a right inguinoscrotal hernia and to review the literature.

CASE REPORT

A 35-year-old male farmworker arrived at the General Surgery Service of the Hospital Universitário Oswaldo Cruz, Recife, Pernambuco, Brazil. He reported the appearance of a mass in the right inguinoscrotal region for around one month without pain. Two days previously he had begun to experience epigastric pain with nausea and vomiting. He visited his local health service and received treatment for gastritis. As the pain continued and was located in the right iliac fossa, he was admitted to hospital. A physical examination revealed a heart rate of 100 bpm, a respiratory rate of 21 ipm, PA=130x80 mmHg and an inguinoscrotal hernia on the right side with slight irritation of the peritoneum. He was referred for surgery and the procedure revealed an inflamed appendix with purulent secretion at its apex within the hernial sac. As surgical access was by transverse incision of the inguinal hernia, it was decided to perform the appendectomy and the Bassini repair of the hernia simultaneously (Figure 1). Antibiotic prophylaxis with metronidazole and ceftriaxone was carried out for 24 hours. After two days, the patient was discharged from hospital with no complications. The result of a biopsy confirmed the appendicitis.

 

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Figure 1 -
Inflamed cecal appendix in a right inguinoscrotal hernia

 

DISCUSSION

Some authors believe that a cecal appendix in an inguinal hernia was first described by De Garengeot in 173110. Claudius Amyand (1681-1740), a French surgeon, who was a refugee in England, was the first to perform an appendectomy13 , 11. The appendix is found in the hernial sac in around 1% of inguinal hernias and an inflamed appendix is found in only 0.13% of cases.

A variant of this, an appendix inside a femoral hernia, is called a Garengeot hernia4. In 1937, Ryan described 11 cases of acute appendicitis (within an inguinal hernia) among 8,692 cases of appendicitis12. Another author1 reported 10 cases of appendicitis within an inguinal hernia over nine consecutive years.

The etiopathogenesis of acute appendicitis is unclear. Many authors believe there is an association between incarceration and inflammation of the cecal appendix in the hernial sac, that is, an ischemic phenomenon deriving from compression of the organ by the hernial ring leading to appendicitis14. Typical symptoms of acute appendicitis, such as initial epigastric pain settling later in the right iliac fossa, nausea, vomiting and anorexia may also be seen in patients with an Amyand hernia. According to the literature, fever and leukocytosis are not common in these patients13. Pre-operative diagnosis is unusual. In an article reviewing 50 cases of Amyand's hernia, only one case was diagnosed prior to surgery14.

The presence of peritoneal irritation and early pain in an incarcerated hernia may suggest appendicitis inside the hernial sac. The use of imaging methods may assist diagnosis4.

Surgery is mandatory. However, the kind of surgery recommended subject to controversy. In most circumstances, treatment involves an emergency appendectomy and repair of the hernia8. When there is a risk of complications, such as a pericecal abscess, the appendectomy should be pre-peritoneal to minimize possible infection of the wound and recurrence of the hernia2.

REFERENCES

  • 1
    Carey LC. Acute appendicitis occurring in hernias: a report of 10 cases. Surgery 1967; 61:236-8.
  • 2
    Doyle GS, McCowan C. Amyand hernia: a case of an unusual inguinal herniace. Am J Emerg Med 2008; 26(5):637. e5-6.
  • 3
    Franko J, Sulkowki R. A rare variation of Amyand's Hernia. Am J Gastroenterol 2002; 97(10):2684-5.
  • 4
    Gillion JF, Bornet G, Hamrouni A, Jullès MC, Convard JP. Amyand and de Garengeot' hernias. Hernia 2007; 11(3):289-90
  • 5
    Hiatt JR, Hiatt N. Amyand's hernia. N Engl J Med 1988; 318(21):1402.
  • 6
    Hotiana MM, Kundu S, Ahmad I. Complicated inguinal hernia of Amyand. South Med J 2007; 100(4):411.
  • 7
    Lippolis PV, Barlettai M, Filidei F, Seccia M. The Amyand's hernia. Case report and review of the literature. Ann Ital Cir 2007; 78(2):153-7.
  • 8
    Logan MTBS, Nottingham JM. Amyand's hernia: a case report of an incarcerated and perforated appendix within an inguinal hernia and review of the literature. Am Surg 2001; 67(7)628-9.
  • 9
    Losanoff JE, Basson MD. Amyand hernia: what lies beneath--a proposed classification scheme to determine management. Am Surg 2007; 73(12):1288-90.
  • 10
    Priego P, Lobo E, Moreno I, Sánchez-Picot S, Gil Olarte MA, Alonso N, Fresneda V. Acute appendicitis in an incarcerated crural hernia: analysis of our experience. Rev Esp Enferm Dig (Madrid) 2005; 97(10):707-715.
  • 11
    Rodríguez Montes JA. Historias de la cirugía. AstraZeneca 2003; 87-102.
  • 12
    Ryan WJ. Hernia of the vermiform appendix. Ann Surg 1937; 106:135-9.
  • 13
    13. Torres Hernández D, Roselló Fina JR, del Campo Abad R, Canals Rabasa PP, Enríquez Weinmann ES. Hernia de Amyand: presentación de un caso y revisión de la literatura. Arch Cir Gen Dig 2003; 22 Sep. Available at: www.cirugest.com.
    » www.cirugest.com
  • 14
    Weber RV, Hunt ZC, Kral JC. Amyand's hernia. Etiologic and therapeutic implications of two complications. Surg Rounds 1999; 22:552-6.

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INTRODUCTION

The actual incidence of foreign bodies retained in the abdominal cavity is not well known, as such cases are under-reported5. They occur even with highly experienced surgeons and may cause serious consequences. Related risk factors require the adoption of systematic preventive measures5.

This paper aims to report a case involving a surgical sponge abandoned after cholecystectomy that migrated into the duodenum and was successfully removed by upper digestive endoscopy.

CASE REPORT

A 26-year-old female patient underwent videolaparoscopic cholecystectomy converting to open surgery due to choledocholithiasis. Choledocholithotomy plus Kehr drainage was then performed. The patient had a good recovery, but after nine months she sought medical care presenting antropyloric obstruction syndrome (epigastric pain, recurrent postprandial vomiting, and weight loss).

Upper digestive endoscopy revealed the presence of a foreign body, probably a surgical sponge, in the gastric cavity, in the transpyloric region, blocking the passage of the equipment (Figure 1A). Abdominal CT scan (Figure 1B) revealed a well-defined mass located between the liver and the stomach, with mixed density, air bubbles in its inside, and spiral radiopaque stripes representing the sponge markers.

 

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Figure 1 -
A) Upper digestive endoscopy showing the surgical sponge; B) CT scan aspect

 

With a diagnostic hypothesis of pyloric obstruction caused by a foreign body, a new upper digestive endoscopy was performed in an attempt to remove the sponge, which was successfully done by snare polypectomy (Figure 2A). After the removal of the foreign body (Figure 2B) superficial esophageal lacerations were observed with self-limited bleeding and a blocked deep ulcer occupying almost all the anterior wall of the duodenal bulb, with no signs of cavity perforation.

 

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Figure 2 -
A) Moment of the endoscopic removal; B) removed sponge

 

The patient had a good recovery. Medicated with proton-pump inhibitors, she accepted oral feeding in the room one day after endoscopy. On the 8th day, a control upper digestive endoscopy showed that the ulcer size decreased with signs of cicatrization. The patient was then discharged from the hospital.

A new control endoscopy performed two months after discharge revealed undeformed duodenum and intact normal mucosa.

The patient presented no symptoms in the last appointment, 10 months after the removal of the foreign body.

DISCUSSION

Foreign bodies retained in the abdominal cavity are not always reported, as this may carry legal medical implications. As a consequence, their real incidence is unknown. It is estimated that there is one case in every 500 to 1500 intra-abdominal surgeries, that is, an incidence of approximately 0,15% to 0,2%.1 , 5

Textile materials (gauze dressings and sponges) are the most commonly abandoned or unintentionally left foreign bodies in the abdominal cavity. The set comprising the foreign body and the surrounding tissue reaction is called gossypiboma or textiloma3.

Risk factors for foreign objects retained in the abdominal cavity are said to include: emergency surgeries, hemorrhage, operatory procedures altered from those initially proposed, participation of more than one surgical team during the procedure, the absence of number listings of surgical sponges and instruments, unsatisfactory anesthesia, inadequate material and infrastructure, surgeon's or team's tiredness, incomplete surgical teams, and obesity2. This case report presents an initially videolaparoscopic cholecystectomy converted to laparotomy due to choledocholithiasis.

There are three possibilities of evolution in the natural history of foreign bodies retained in the abdominal cavity: 1) to be encapsulated by the reactive inflammatory fibrotic process with our without the formation of an abscess or fistula; 2) to be removed by surgical incision; or 3) to migrate into the lumen of a hollow viscera (intestines, bladder, or vagina)3 , 5.

The clinical picture varies greatly, as it depends on the type of reaction triggered by the organism in response to the presence of the foreign body. If a foreign body becomes encapsulated by the inflammatory process, it may have an asymptomatic evolution and be found in an imaging test in 30% of the cases. It may manifest itself as a poorly-defined palpable tumor or present intra-cavity abscess signs and symptoms5.

If the foreign body migrates to the intestinal lumen, the sick patient may present abdominal pain, or show signs of intestinal occlusion or sub-occlusion, or even excrete it via feces5.

CT scan is the gold standard diagnostic test for gossypiboma. Its features include spiral radiopaque stripes found in sponge markers and the spongiform appearance of the tumor with small air bubbles in its inside3 , 4. A Ultrasonography and simple abdominal radiography can also suspect a diagnosis of a retained foreign object.

The treatment for retained foreign bodies in the abdominal cavity may be expectant when concomitantly the patient is asymptomatic and the surgery was performed a long time ago, with no signs of abscesses, or when its migration to the intestinal lumen indicates a spontaneous resolution1. The removal of the foreign body is recommended when it is associated with relevant signs and symptoms or when diagnosis is made on the first days after surgery5. This procedure may be done by conventional surgery, using a laparoscopic or even endoscopic approach4, as in the case described here.

Preventive measures should be implemented to reduce its occurrence, such as: 1) the placement of Pean clamps on surgical sponge tapes, positioning them out of the abdominal cavity; 2 surgical sponge counting procedures (although this is not a fool-proof indicator as studies show cases in which counting was regarded as correct and even so sponges remained in the abdominal cavity); 3) after being given to a surgeon, a gauze dressing is supposed to be immediately returned to the surgical technologist's hands; 4) cavity inventory before closure; 5) use of radiopaque markers for textile materials; and 6) in case of doubt about a possible retention, radiography must be done in the operating room2.

As for legal medical implications, abandoning foreign bodies in surgical procedures may result in civil and criminal charges. Naturally, medical errors must be investigated and considered in all dimensions. Generally, they are characterized as negligence5. Peer group attitudes must not cover up grave medical errors and negligent professionals.

On the other hand, it is unacceptable to prejudge and publicly execrate the medical professional without taking into account critical emergency situations, extensive surgeries, unsatisfactory conditions of the surgical environment, etc., all of which factors that may influence the conduction of the surgical procedure.

REFERENCES

  • 1
    Alis H, Soylu A, Dolay K, Kalayci M, Ciltas A. Surgical intervention may not always be required in gossypiboma with intraluminal migration. World J Gastroenterol. 2007;13(48):6605-7.
  • 2
    Gawande AA, Studdert DM, Orav EJ, Brennan TA, Zinner MJ. Risk factors for retained instruments and sponge after surgery. N Engl J Med. 2003;348(3):229-35.
  • 3
    Iglesias AC, Salomão RM. Gossipiboma intra-abdominal - análise de casos. Rev Col Bras Cir. 2007;34(2):105-13.
  • 4
    Kansakar R, Hamal BK. Cystoscopic Removal of an intravesical gossypiboma mimicking a bladder mass: a case report. J Med Case Reports. 2011 Dec 16;5(1):579.
  • 5
    Schanaider A, Manso JEF. Corpos estranhos provenientes de acesso cirúrgico à cavidade abdominal. Aspectos fisiopatológicos e implicações médico legais. Rev Col Bras Cir. 2006;33(4):250-5.

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ABSTRACT

The introduction of chatbots has been one of the most intriguing advances in artificial intelligence. There are numerous potential uses for artificial intelligence in clinical research. However, there are also some issues that require attention. Everyone agrees that AI requires a more stable foundation and that a cutting-edge approach is necessary for AI to operate effectively.

INTRODUCTION

Neuroendocrine tumors of the pancreas are rare neoplasms representing approximately 2% of all pancreatic tumors9. Due to the progress of diagnostic imaging and radioimmunoassay, its diagnosis has become more frequent. Recent epidemiological studies suggest increased frequency12. Among the functioning tumors, vipoma (tumor cells producing vasoactive intestinal polypeptide) is also known as WDHA syndrome (watery diarrhea, hypokalaemia, and achlorhydria), Werner Morrison´s syndrome and pancreatic cholera; it is still rare neoplasia, mainly characterized by profuse diarrhea with hydro-electrolytic disorders. It has an estimated incidence of 0,2 to 0,5 per million inhabitants per year3. Approximately 90% of these tumors originate in the pancreas; however, there are descriptions located in other segments of the gastrointestinal tract, bronchus, adrenal, sympathetic ganglia and liver. There are few cases described in the medical literature of extra-pancreatic location in adults8. Due to its low incidence, it is unknown the true epidemiological data of this unique neoplasm.

The objective of this report is to present another case of extra-pancreatic vipoma.

CASE REPORT

Man of 54 years old had for four months diarrhea, watery stools, often in every three hours lasting 15 days; had 14 kg weight loss during this period, with asthenia, anorexia, malaise, muscle weakness and cramps. Laboratory tests showed VIP (intestinal polypeptide active vessel) 242 pg/ml (<75); Na=138; K=1.6; and creatinine=1.53. Ultrasonography showed hypoechoic image on segment IV of 4.2 cm, confirmed by CT with the presence of several hypervascular images in liver segments IV, III, II (Figure 1). Ultrasonography guided biopsy was performed in one liver nodule that revealed metastatic neuroendocrine carcinoma by immunohistochemistry. Laparotomy confirmed multiple liver metastases (Figure 2). Intraoperative ultrasonography showed nodule in pancreatic body. Bodycaudal pancreatectomy with splenectomy (Figure 3) and left hepatic trisegmentectomy were performed (Figure 4). No tumor was found in the pancreatic parenchyma. Histopathology showed acinar atrophy and hyperplasia of islets in the caudal region. In peri-pancreatic adipose tissue was confirmed the presence of five nodes with well-differentiated neuroendocrine carcinoma infiltrating the adipose tissue adjacent the neoplastic infiltration beyond perineural and angiolymphatic (Figure 5). Hepatic lesions confirmed the diagnosis of metastatic well-differentiated neuroendocrine carcinoma. Immunohistochemical analysis showed positivity for sinaftofisin, chromogranin and intestinal polypeptide active vessel (VIP). Ki-67 was positive in 10%. Patient had no major complications. There was immediate regression of diarrhea and electrolyte abnormalities. After 12 months, returned again with the same initial clinical picture. Liver CT images showed multiple metastatic nodulation diffusely distributed. Therapy with somatostatin analogue with prolonged action (LAR) and chemotherapy with inhibitors of mTOR (everolimus) was started with regression of liver lesions and clinical symptoms. At the third year postoperatively he was without clinical signs of disease recurrence.

 

DISCUSSION

Although there is a previous report, this neoplasm was first described by Werner and Morrison in 1958 in two patients with profuse diarrhea and hypokalemia associated with malignancy of non insulin-producing pancreatic islets. Its pathophysiological aspects were more well-known since 1973, the time in which Bloom et al.2 associated WDHA syndrome with increased serum vasoactive intestinal polypeptide5, a fact later confirmed by the studies of Kane et al.7 reproducing this syndrome after intravenous administration of VIP in five volunteers6. It is an aminoacid peptide produced by the delta-2 pancreatic islet cells and is also present in the central and peripheral nervous system and considered as a neurotransmitter. High concentrations are found in the gastrointestinal tract. Among its effects are described: stimulation of the smooth muscles of the gastrointestinal tract; increasing intestinal and pancreatic secretions; vasodilation; inhibition of gastric acid secretion; increased glycogenolysis and hypercalcemia4. Classically, vipomas present profuse diarrhea with consequent electrolyte repercussions, weight loss, and, more rarely, skin lesions, tachycardia and low back pain. Relatively often, these patients are initially investigated by a number of more common diseases whose main symptom is diarrhea. Much of this neoplasm originates in the pancreas and is sporadic; but may also be associated with multiple endocrine neoplasia11. However, these tumors can arise in the ganglia of the sympathetic nervous system, especially in children. Extra-pancreatic vipomas can be classified by their origin in neurogenic and non-neurogenic, the latter very little reported in literature5. From the clinical, laboratory and histopathological findings, it is not possible to differentiate neurogenic extra-pancreatic tumors. The neurogenic appears to have less severe clinical picture, lower VIP levels when compared to gastrointestinal disturbances, lack of production of pancreatic polypeptide and histopathological different characteristics8. The clinical diagnosis is confirmed by the increase in the level of serum VIP and radiological findings, mostly performed by computed tomography or magnetic resonance, localizing the tumor in the pancreas topography in most cases, due to the fact that most of these tumors have more than 3 cm. Likewise, more than 60% of these tumors have liver or lymph node metastasis at the time of diagnosis1. The therapeutic approach is to initially control the electrolyte disturbances, use of somatostatin analogues and subsequently surgical approach. Similar to others gastroenteroneuroendocrine diseases, also to other cancers, surgical resection is the best way to control the clinical symptoms and prolong survival10; it also can be performed cytoreductive operation, resection of liver metastases and even liver transplantation. The completion of adjuvant chemotherapy appears to play an important role in controlling the disease and have been described in addition to biotherapy with somatostatin analogues for long-term use to increase survival with good quality of life6 , 9. In this patient, besides the surgical treatment of liver metastases and the recurrence of the disease, there was considerable gain in life expectancy with the use of adjuvant chemotherapy.

REFERENCES

  • 1
    Amir A. Ghaferi & Karen A. Chojnacki & William D. Long & John L. Cameron & Charles J. Yeo. Pancreatic VIPomas: Subject Review and One Institutional Experience. J Gastrointest Surg 2008; 12:382-93.
  • 2
    Bloom SR, Polak JM, Pearse AG. Vasoactive intestinal peptide and watery-diarrhoea syndrome. Lancet 1973;2:14-6.
  • 3
    Friesen SR. Update on the diagnosis and treatment of rare neuroendocrine tumors. Surg Clin North Am 1987;67:379.
  • 4
    Holst JJ, Fahrenkrug J, Knuhtsen S, Jensen SL, Poulsen SS, Nielsen OV. Vasoactive intestinal peptide (VIP) in the pig pancreas: role of VIPergic nerves in the control of fluid and bicarbonate secretion. Regul Pept 1984;8:245.
  • 5
    Jo JH, Lim S, Han MS, Cho IR, Kim GJ, Ahn JB, Roh K, Shin SJ. VIPoma that arose from the rectum in a 65-year-old male patient. Int J Colorectal Dis 2012; 27:1385-86.
  • 6
    Joyce dl, Hong k, Fishman EK, Wisell J, Pawlik TM. Multi-visceral resection of pancreatic VIPoma in a patient with sinistral portal hypertension. World J Surg Oncol 2008; 6:80.
  • 7
    Kane MG, O'Dorisio TM, Krejs GJ. Production of secretory diarrhea by intravenous infusion of vasoactive intestinal polypeptide. N Engl J Med 1983;309:1482-5.
  • 8
    Long RG, Bryant MG, Mitchell SJ, Adrian TE, Polak JM, Bloom SR. Clinicopathological study of pancreatic and ganglioneuroblastoma tumours secreting vasoactive intestinal polypeptide (vipomas). Br Med J 1981; 282:1767 - 71.
  • 9
    Massironi S, Sciola V, Peracchi M, Ciafardini C, Spampatti MP, Conte D. Neuroendocrine tumors of the gastro-entero-pancreatic system. World J Gastroenterol 2008, 21; 14(35): 5377-84.
  • 10
    Nikou GC, Toubanakis C, Nikolaou P, Giannatou E, Safioleas M, Mallas E, Polyzos A. VIPomas: an update in diagnosis and management in a series of 11 patients. Hepatogastroenterology. 2005 Jul-Aug;52(64):1259-65.
  • 11
    Soga J, Yakuwa Y. Vipoma/diarrheogenic syndrome: a statistical evaluation of 241 reported cases. J Exp Clin Cancer Res. 1998;17(4):389-400.
  • 12
    Yao JC, Hassan M, Phan A, Dagohoy C, Leary C, Mares JE, Abdalla EK. One hundred years after "carcinoid": epidemiology of and prognostic factors for neuroendocrine tumors in 35.825 cases in the United States. J Clin Oncol 2008; 26:3063-72.

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INTRODUCTION

The primary retroperitoneal cystadenoma is very rare tumor, described by Handfield-Jones in 1924 and observed almost exclusively in women1. The symptoms are usually nonspecific, hampering its differential diagnosis with other retroperitoneal masses and makes them with imaging and surgical approach for diagnosis and treatment2.

The present report is of one case of a primary benign retroperitoneal mucinous cystadenoma.

CASE REPORT

Woman 51 year old referred abdominal pain since one year ago, located on the right flank and radiating to the lumbar region, with progressive worsening. Physical examination revealed a palpable mass in the right flank, painless. Was submitted to ultrasound examination which identified a bulky abdominal cystic lesion. Computed tomography (Figures 1 and 2) revealed homogeneous retroperitoneal cystic lesion measuring 15x12, 5x5, 5 cm and medially displacing the ascending colon, suggesting the diagnosis of cystic lymphangioma. The patient underwent exploratory laparotomy (Figures 3 and 4), which identified massive retroperitoneal cystic lesion which was dissected from adjacent structures with ease, allowing complete resection. The pancreas and ovaries showed no alterations or contiguity with the injury. No complications were observed during the postoperative course. Pathological examination of the surgical specimen revealed retroperitoneal mucinous cystadenoma benign (Figures 5 and 6).

 

DISCUSSION

Most patients have a palpable mass, asymptomatic or accompanied by mild abdominal pain that may be associated with nonspecific gastrointestinal complaints2. The differential diagnosis must be made with cystic lymphangioma, cystic teratoma, cystic neoplasms of the pancreas and ovary. The assessed by CT or MRI identifies retroperitoneal cystic lesion, but does not define its exact nature3; so, confirmation of the diagnosis can be established only after histological examination of the surgical specimen. Thus, the surgical approach is indicated for proper evaluation of the topography and resection of the lesion.

Histologically primary retroperitoneal mucinous cystadenoma can be classified in three types: benign, borderline and malignant4. Benign, the most common, with no recurrence after surgical resection; borderline, with adjacent proliferative columnar epithelium and small malignant potential; malignant, which can be recurrent and metastatic5. It shares histological similarities with ovarian mucinous cystadenoma and can be located anywhere in the retroperitoneum without connections to the ovary. The histogenesis remains incompletely understood. However, there are two main assumptions. According to the first, as there is similarity with ovarian mucinous cystadenoma, it is possible originated from an ectopic ovarian tissue6; however, ovarian tissue was found in the cyst wall only in few cases7 and have been described in men8. The second hypothesis suggests that they originate from an invagination of multipotent mesothelial cells with subsequent mucinous metaplasia of the mesothelial cells6.

Complete surgical resection of the lesion, as well as allowing adequate diagnostic evaluation, represents the best treatment9.

REFERENCES

  • 1
    Calo PG, Congiou A, Ferreli C, Nikolosi A, Tarquin A. Primary retroperitoneal tumors. Our experience. Minerva Chirurgica 1994;49:43-9.
  • 2
    Roma AA, Malpica A: Primary retroperitoneal mucinous tumors: a clinicopathologic study of 18 cases. AmJ Surg Pathol 2009;33:526-33.
  • 3
    Falidas E, Konstandoudakis S, Vlachos K, Archontovasilis F, Mathioulakis S, Boutzouvis S et al. Primary retroperitoneal mucinous cystadenoma of borderline malignancy in a male patient. Case report and review of the literature. World Journal of Surgical Oncology 2011;9:98.
  • 4
    Navin P, Meshkat B, McHugh S, Beegan C, Leen E, Prins H, Aly S. Primary retroperitoneal mucinous cystadenoma - A case study and review of the literature. International Journal of Surgery Case Reports 3 2012;486-488.
  • 5
    Min BW, Kim JM, Um JW, Lee ES, Son GS. Kim SJ et al. The First Case of a Retroperitoenal Mucinous Cystadenoma in Korea: A Case Report. The Korean Journal of Internal Medicine 2004;19:282-284.
  • 6
    Matsubara M, Shiozawa T, Tachibana R, Hondo T, Osasda K, Kawaguchi K, et al. Primary retroperitoneal mucinous cystadenoma of borderline malignancy: a case report and review of the literature. International Journal of Gynecological Pathology 2005;24:218-23.
  • 7
    Pennell TC, Gusdon Jr JP. Retroperitoneal mucinous cystadenoma. American Journal of Obstetrics and Gynecology 1989;160:1229-31.
  • 8
    Lai KKT, Chan YYR, Chin ACW, Ng WF, Huang YHH, Mak YLM, et al. Primary retroperitoneal mucinous cystadenoma in a 52-year-old man. Journal of Hong Kong College Of Radiologists 2004;7:223-5.
  • 9
    Tapper EB, Shrewsberry AB, Oprea G, Majmudar B. A unique benign mucinous cystadenoma of the retroperitoneum: a case report and review of the literature. Archives of Gynecology and Obstetrics 2010;281:167-9.

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ABCD – BRAZILIAN ARCHIVES OF DIGESTIVE SURGERY is a periodic with a single annual volume in continuous publication, official organ of the Brazilian College of Digestive Surgery - CBCD. Technical manager: Dr. Francisco Tustumi | CRM: 157311 | RQE: 77151 - Cirurgia do Aparelho Digestivo

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