cases with focus on atypical locations of Leishmania. Clin Infect

Dis 2000;31:1093–5.

2. Mc Bride MO, Fisher M, Skinner CJ, et al. An unusual

gastrointestinal presentation of leishmaniasis. Scand J Infect Dis

1995;27:297–8.

3. M L Alvarez-Nebreda, E Alvarez-Fernandez, S Rada, F Branas,

E Maranon, M T Vidan, J A Serra-Rexach. Unusual duodenal

presentation of leishmaniasis. J Clin Pathol 2005;58:1321–2.

4. Berman JD. Human leishmaniasis: clinical, diagnostic and

chemotherapeutic developments in the last 10 years. Clin Infect

Dis 1997;24:684–703.

Transperineal excision: A novel andalternative surgical approach forpelvic recurrence of rectal cancer

Introduction

Colorectalcancer (CRC) is the third most frequently diagnosed

cancer in both men and women and the second most fatal

cancer.1 The incidence of CRC is greater in men than in women.2

CRCs are often seen in the elderly population and approximately

30% of these are localized in the rectum.2 Distal rectal cancer is

a surgical and oncological challenge. Various surgical

operations, such as abdomin operineal resection and low

anterior resection with total mesorectal excision (TME), are

performed in the management of distal rectal cancer. Although

recurrence rates have decreased to about 10% after using the

method of TME, local recurrence remains an important clinical

issue.3 Surgery is the treatment of choice but it is a difficult

procedure, with very poor prognosis. Here, we report a case of

pelvic recurrent tumor that was entirely excised by a novel and

alternative surgical method, transperineal approach, with CT-

guided wire marking.

Case Report

A 71-year-old female patient, who had undergone abdominal

operineal resection for a low rectal cancer with T2N0

pathological stage before fifteen months, was admitted to our

unit for routine follow-up. She had received a full course of

adjuvant chemoradiation following the operation. On blood

tests, cancer antigen 19.9 (CA 19-9) level was high (163 IU/mL)

with normal carcinoembryonic antigen (CEA) level (5ng/mL).

As shown in Figure 1, CT scan showed a heterogeneous

pelvic mass, 38 mm x 35 mm in size, at the left posterolateral

side of bladder. Similar lesion within definite borders was also

seen on magnetic resonance imaging. In PET/CT, an increased

18F - FDG uptake (SUVmaks: 5.1), approximately 29 mm x 25

mm in size, at the inferior left para-iliac zone was observed. CT-

guided trucut biopsy was performed. Biopsy identified an

adenocarcinoma. The pelvic mass was marked with a

CT-guided wire and was excised totally by transperineal

approach under spinal anesthesia as shown in

Figure 2. On histopathological examination,

an adenocarcinoma, evaluated as a recurrent lesion of the

Figure 2: The appearance of the area after excision of the mass,

localized between iliac bone (blue arrow) and obturatory

vein (white arrow)

Figure 1: The tomographic appearance of the pelvic mass, marked

with a wire, localized at the left posterolateral site of

bladder

Tropical Gastroenterology 2015;36(2):123–125

primary rectal cancer, was reported as shown in

Figure 3.

Discussion

The most important component for obtaining long term local

control and survival is R0 resection with sharp TME.

Additionally, the use of adjuvant radiotherapy has been shown

to reduce loco-regional recurrence (LR).4 However, patients,

treated with a curative rectal cancer resection, have a 10% risk

of developing LR.3 Close anatomic relation of the rectum to the

small pelvis makes rectal cancer proneto recurrence. LR of rectal

cancer usually occurs within the first few years following

resection of the primary.5 Similarly, the pelvic recurrence

developed fifteen months after primary surgery in our case.

The stage of primary tumor and a positive circumferential

resection margin infers an LR risk and poor prognosis.6 Rates

of LR or distant metastases are 5 - 10% in stage A, but 40-70%

in stage C.7 Our patient had a T2N0 tumor with clear radial

margins and no perforation occurred during the surgery. In

addition, the patient had adjuvant radiotherapy and

chemotherapy. As is well known, the CEA level is a useful

marker for follow-up of colorectal cancer recurrence. In our

patient, the CEA level was normal but CA 19.9 level was

increased. Morales-Gutierrez et al also showed in their study

that elevated level of CA 19.9 was an independent risk factor

for recurrence and was associated with a poor prognosis.8 The

surgical treatment of LR is particularly challenging, however

patients with LR have a median survival of approximately 8

months without treatment.9 The 5-year overall survival rate after

the operation for LR is approximately 50% even where surgery

is possible.10 Unfortunately, only a small proportion of patients

with LR have a chance of surgery. These patients are usually

operated via laparotomy ortransanally. But these methods have

their own limitations. In selected patients, the transperineal

approach may be the most easy and appropriate way for

resection of recurrent pelvic lesions. Thus, the difficulties of

laparotomy are avoided. However, adequate preoperative

radiological evaluation must be done. In addition, marking the

lesion is very helpful for the success of the operation in a

transperineal approach. As far as we know, this is the first case

of rectal cancer recurrence treated via the transperineal

approach. Finally, LR is a major cause of morbidity and mortality

in patients undergoing curative resection of rectal cancer.

Surgical resection is the recommended treatment modality and

can be performed safely by the transperineal approach in

selected patients.

MURAT ÖZGÜR KILIÇ1,

GÜRKAN DEGIRMENCIOGLU1,

CENAP DENER1,

NUR ARSLAN2

Correspondence: Dr. Murat Özgür KILIÇ

Department of General Surgery1 and Pathology2,

Faculty of Medicine, Turgut Özal University,

Ankara, Turkey

Email: murat05ozgur@hotmail.com

References

1. Siegel R, Naishadham D, Jemal A.Cancer statistics, 2012. CA

Cancer J Clin. 2012;62:10–29.

2. Rim SH, Seeff L, Ahmed F, King JB, Coughlin SS. Cancer

Colorectal cancer incidence in the United States, 1999–2004: an

updated analysis of data from the National Program of Cancer

Registries and the Surveillance, Epidemiology, and End Results

Program. 2009;115:1967–76.

3. Das P, Skibber JM, Rodriguez-Bigas MA, Feig BW, Chang GJ,

Hoff PM, et al. Clinical and pathologic predictors of locoregional

recurrence, distant metastasis, and overall survival in patients

treated with chemoradiation and mesorectal excision for rectal

cancer. Am J Clin Oncol. 2006;29:219–24.

4. Nissan A, Guillem JG, Paty PB, Douglas Wong W, Minsky B,

Saltz L, et al. Abdominoperineal resection for rectal cancer at a

specialty center. Dis Colon Rectum. 2001;44:27–35

5. Davies M, Harris D, Hirst G, Beynon R, Morgan AR, Carr ND,

et al. Local recurrence after abdomino-perineal resection.

Colorectal Dis. 2009;11:39–43.

6. Kelly SB, Mills SJ, Bradburn DM, Ratcliffe AA, Borowski DW;

Northern Region Colorectal Cancer Audit Group. Effect of the

circumferential resection margin on survival following rectal cancer

surgery. Br J Surg. 2011;98:573–81.

Figure 3: Microscopic appearance of adenocarcinoma with

distortioned tubules in the stroma (HE X 200)

124 Tropical Gastroenterology 2015;36(2):123–125

7. Burdy G, Panis Y, Alves A, Nemeth J, Lavergne-Slove A, Valleur

P. Identifying patients with T3-T4 node negative colon cancer at

high risk of recurrence. Dis Colon Rectum. 2001;44:1682–8.

8. Morales-Gutierrez C, Vegh I, Colina F, Gomez-Camara A,

IgnacioLanda J, Ballesteros D, et al. Survival of patients with

colorectal carcinoma: possible prognostic value of tissular

carbohydrate antigen 19.9 determination. Cancer.

1999;86:1675–81.

9. Moriya Y. Treatment strategy for locally recurrent rectal cancer.

Jpn J Clin Oncol. 2006;36:127–31.

10. Tanaka K, Noura S, Ohue M, Seki Y, Yamada T, Miyashiro I, et

al. Doubling time of carcinoembryonic antigen is a significant

prognostic factor after the surgical resection of locally recurrent

rectal cancer. Dig Surg. 2008;25:319–24.

Blue rubber bleb nevus syndrome –Role of aggressive surgical resection

Introduction

Blue rubber bleb nevus syndrome is a rare multifocal venous

malformation primarily involving the skin, soft tissues and the

gastrointestinal tract but may involve any tissue. We present

the case of a young girl who developed recurrent melena

necessitating surgical resection.

Case report

The patient was a 12-year old girl, a known case of blue

rubberbleb nevus syndrome (BRBNS) who had numerous

lesions (Figure 1) numbering 65, over scalp, lips, trunk,

abdominal wall, all four extremities, gluteal region and vulva

for which she had received four sittings of injection

sclerotherapy (sodium tetradecyl sulphate) with satisfactory

regression of the lesions injected.

She developed intermittent episodes of small volume melena

over the one year prior to presentation, leading to anemia and

chronic fatigue necessitating ten blood transfusions. Blood

pool scan showed no evidence of active intestinal bleed but

showed multiple pooling in the intestine along with numerous

musculoskeletal uptakes.

Wireless capsule endoscopy (Figure 2) revealed multiple

intraluminal blebs from duodenojejunal flexure to the cecum

with no active bleed.

In view of recurrent episodes of melena requiring multiple

blood transfusions, surgery was planned. On examination she

had pallor. Abdominal examination was unremarkable. The

abdomen was accessed through a right upper transverse

Figure 1 Cutaneous manifestation of BRBNS

Figure 2 Capsule endoscopy revealing intraluminal blebs

Tropical Gastroenterology 2015;36(2):125–127