PRIMARY SUBCUTANEOUS HYDATID CYST ON THE INCISIONAL SCAR FOLLOWING OPEN CHOLECYSTECTOMY: A RARE CASE REPORT AND REVIEW OF THE LITERATURE

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1 Acta Medica Mediterranea, 2015, 31: 329 PRIMARY SUBCUTANEOUS HYDATID CYST ON THE INCISIONAL SCAR FOLLOWING OPEN CHOLECYSTECTOMY: A RARE CASE REPORT AND REVIEW OF THE LITERATURE SINAN HATIPOGLU 1, SAMI AKBULUT 2, UMUT GULACTI 3, UGUR LOK 3, FILIZ HATIPOGLU 4 1 Department of Surgery, Adiyaman University Faculty of Medicine, Adiyaman - 2 Department of Surgery, Diyarbakir Education and Research Hospital, Diyarbakir - 3 Department of Emergency Medicine, Adiyaman University Faculty of Medicine, Adiyaman - 4 Department of Obstetrics and Gynecology, Adiyaman Education and Research Hospital, Adiyaman, Turkey ABSTRACT Aims: Primary subcutaneous hydatid cyst (pshc) in the abdominal wall is very seldom clinical presentation, even in endemic area including Turkey. Herein we present a new case of pshc on incision scar of abdominal wall following cholecystectomy, which is the first in the literature. Materials and methods: We conducted a case presentation and a literature review of studies published in the medical literature on pshc of the anterior abdominal wall, accessed via PubMed, Medline, and the Google Scholar databases. Keywords used were hydatidosis, echinococcosis, hydatid disease, subcutaneous, abdominal wall, and soft tissue. Case presentation: A 57-year-old female patient applied to emergency department with complaint of abdominal pain and swelling on right upper quadrant of abdominal wall. She underwent cholecystectomy with right subcostal incision 9 years ago. The patient was hospitalized in the General Surgery Clinic. Radiological findings revealed a lesion resembling a hydatid cyst. Prior to surgery, patient was treated with albendazole for 10 days. Cystic lesion was completely excised under the local anesthesia without cyst rupture. Macroscopic and microscopic examination of the specimens confirmed hydatid cysts. No recurrence had occurred after 9 months of follow up. Conclusion: pshc in the abdominal wall should be well-considered in the differential diagnosis of every subcutaneous cystic mass, especially in countries endemic for hydatidosis. In order to avoid complications such as anaphylaxis and cyst spillage, the best surgical approach should be the total excision of the cyst without opening the cyst cavity. Key words: Hydatid cyst, primary, subcutaneous tissue, abdominal wall, incisional scar, abdominal pain. Received June 18, 2014; Accepted October 02, 2014 Introduction Hydatid disease is an important public-health problem in endemic areas such as South America, the Middle East, Africa, Australia, and the Mediterranean countries, including Turkey. It is a zoonotic infestation caused by the larval stage of a Echinococcal parasite (1,2). Humans are infested either by direct contact with definitive hosts or indirectly by ingestion of eggs due to contaminated water and food sources (3). The prevalence of the hydatid disease worldwide is 1-500/100,000 individuals and the incidence is 5-20/100,000. In Turkey, the prevalence is /100,000 and the incidence is 3.4/100,000 (4). In literature of Turkey, approximately more than 21,303 cases with hydatid disease were reported between 1987 and 1994 in Turkey (4). The disease occurs in almost all regions of our country. There are a lot of factors, such as general unhygienic conditions, country s socioeconomic condition, and lack of public education about hydatid cyst disease prevention affect the high prevalence of the infection in Turkey (4). Liver is the first and the more frequent involved organ, followed by the lung, spleen and kidney (1). Moreover, the primary subcutaneous hydatid cyst (pshc) in the abdominal wall without

2 330 Sinan Hatipoglu, Sami Akbulut et Al involving thoracic or abdominal organs remain extremely rare even in endemic regions, its incidence is unknown, and only a few cases are reported in medical literature till date (5-12). However, there is not any reported pshc of abdominal wall on the incisional scar. Herein we share an interesting case of pshc on incision scar of abdominal wall, which is the first in medical literature. We also reviewed the medical literature to provide an update on pshc of the abdominal wall. Materials and methods We conducted a case presentation and a literature review of studies published in the medical literature on pshc of the anterior abdominal wall, accessed via PubMed, Medline, and the Google Scholar databases. Keywords used were hydatidosis, echinococcosis, hydatid disease, subcutaneous, abdominal wall, and soft tissue. We only included and selected papers related to pshc disease from the search. The patients were evaluated according to age, sex, country, symptoms, diagnostic radiology, localization and size of cyst, serology, surgical modality, adjuvant medications, recurrences and complications that developed during the follow-up period. A literature search, using the above-mentioned review criteria, retrieved eight case reports (5 female and 3 male, aged years) about subcutaneous hydatid cyst on the anterior abdominal wall. All the publications were as a case report between 2004 and 2012 (5-12). The detail informations about these cases are summarized in Table 1. Table 1: Clinical and demographic characteristics of patients. RLQ: Right lower quadrant, RPU: Right paraumbilical, NA: No-available, NS: No-stated, * Case report Case presentation A 57-year-old female patient admitted to Emergency Department of Adiyaman University Training and Research Hospital with complaints of abdominal pain, gradually increasing local abdominal swelling and smoothly outlined mass on the subcostal incisional scar site in the right upper quadrant of abdominal wall and intermittent nausea. Patient stated that these complaints started about 18 month ago. She reported history of open cholecystectomy 9 years ago. She lived in the rural areas of southeastern part of Turkey, endemic for hydatid disease. 1 A 1 B Figure 1. A-B: Shows 57-year-old female patient with local abdominal swelling and smoothly outlined mass in the right upper quadrant of abdominal wall. She had no history of surgery for a hydatid cyst in another organ. On the physical examination, a swelling of 6 x 4 cm was found on incisional scar area of right upper quadrant of abdominal wall (Figure 1A,1B). The mass on the incisional scar was smooth, not freely mobile, uni-lobed with well-defined margins, unreducible, no compressible, and no change in dimensions on lying down position. The patient was hospitalized in the General Surgery Clinic. Her liver and renal function tests measured within normal limit. Anti-echinococcus IgG and Enzyme-Linked Immunosorbent Assay (ELISA) were negative. Abdominal ultrasonography (US) revealed a uniloculated

3 Primary subcutaneous hydatid cyst on the incisional scar following open cholecystectomy rounded cystic structure without solid component of the size of approximately 55x37 mm in the subcutaneous area of the anterior abdominal wall (Figure 2). Figure 2: Transabdominal ultrasound image of right anterior abdominal wall. Shows well-defined circumscribed spherical cystic lesion. 3 A US showed a lesion resembling a hydatid cyst and according to Gharbi s classification (13), the cystic mass was type I. Magnetic resonance imaging (MRI) revealed a well-defined round cystic lesion in the antero-lateral subcutaneous area right upper abdominal wall (Figure 3A, 3B, 3C). Radiological findings revealed a lesion resembling a hydatid cyst. Radiological investigations (US and MRI) for all of body were performed for the other organ involvement and no other hydatic cyst lesion was found. For definitive treatment, surgery was planned under local anesthesia. During the surgical exploration, the skin and subcutaneous layers were incised and the cyst was reached. Surgical exploration revealed that the cystic mass was in the subcutaneous adipose tissue but was not associated with any part of skin, fascia, and muscular tissue. The cyst was found to be well capsulated and was excised without rupture. The diagnosis was confirmed on the macroscopic examination of the resected piece and the cyst was unilocular filled with hydatid membrane and clear fluid. The postoperative period was uneventful and the patient was discharged on the first post-operative day. The histopathological examination of the resected specimen confirmed hydatid cyst of subcutaneous abdominal wall. No recurrence occurred after 9 months of follow up. Discussion 3 B 3 C Figure 3: Shows MRI scans (3A- T2W Axial MRI, 3B- T2W Coronal MRI, 3C-Axial MRI): Hydatid cyst involving the subcutaneous tissue of the right anterior abdominal wall. Hydatid disease, also known as echinococcal disease, is a zoonotic disease caused by the parasite species echinococcus belonging to the taeniidae family of the cestode. The most common species encountered in humans are E. Granulosus causing cystic echinococcosis and E. Multilocularis causing alveolar echinococcosis (2,3). In the biological life cycle of hydatid disease carnivores are the definitive hosts while herbivores have a role of intermediary host. Having no role in the biological life cycle of the hydatid disease, humans are infected by inadvertently ingesting the echinococcus eggs containing live oncospheres in canine feces. The ingested eggs first penetrate intestinal wall, then pass to portal system and ultimately reside in hepatic sinusoids. Larvae escape liver s filtering system to reach lungs where they are entrapped by a second capillary filtering system. Larvae that escape lung filters may pass to distant organs such as kidney, spleen, brain, retroperi-

4 332 Sinan Hatipoglu, Sami Akbulut et Al toneum, musculoskeletal stricture and subcutaneous tissue. Subcutaneous hydatid cyst is a rare disease even in area where hydatid disease is endemic. Subcutaneous hydatid cyst may develop as primary (involving the subcutaneous tissue only) or secondary (with multiple organ involvement) disease. The reported subcutaneos hydatid cyst disease is usually secondary. A number of hypotheses regarding the mode of larvae dissemination to subcutaneous tissue of abdominal wall have been put forward. But exact mechanism by which larvea from the liver and lung capillary barrier enter to the systemic circulation, and how they identify which tissue or organ in which to settle still remains a controversial topic. Possible occurrence mechanisms for primary hydatid cyst disease of abdominal wall to right upper quadrant, paraumbilical, and iliac fossa are direct entry of parasite into inferior vena caval system via connection between systemic veins and portal veins and subsequent reflux implantation of parasites during periods of daily activity associated with Valsalva maneuver (8). Secondly, penetration of larvea from intestine into peritoneal space and direct invasion of peritoneum in most dependent areas of right paracolic gutter (8). Thirdly, penetration of larvea into peritoneal lymphatic route and localization into abdominal wall (6-8,14). Fourthly, direct subcutaneous contamination or spread from adjacent regions may be another mechanism of infection whenever a cyst microrupture has occurred (6,15,16). Main question of our case was represented by the exclusive involvement of the incision scar of abdominal wall. We cannot find definitive answers to this question. We proposed an airborne transmission and/or direct implantation during the previous open abdominal surgery. The subcutaneous layer of anterior abdominal wall is a very seldom site even in our country in which hydatid disease is endemic. In our literature search, using the above-mentioned review criteria retrieved eight case reports about subcutaneous hydatid cyst on the anterior abdominal wall. The detail information about these cases is summarized in Table 1. In the literature, presentations of hydatid cyst disease in the anterior and posterior muscle of abdominal wall are also available (14,17-23). Making the preoperative diagnosis of hydatid cyst disease is important. But especially diagnosing hydatid cysts in unusual localizations is very difficult. Clinically, a hydatid cyst in the soft tissues might mimic teratomas, sarcomas, abscesses, sebaceous cyst, lipoma, tuberculous abscess, aneurysm, hernia, chronic hematoma, synovial cyst, necrotic soft tumor, or fibromatosis (14,15,24,25). Moreover, exposure to the contents of the cyst can cause complications such as anaphylactic reaction and local recurrence. The finding of a hydatid cyst disease is usually incidentally during the US, computerized tomography (CT), MRI that show the size, localization, relationship to adjacent organs, and type of the cyst (3,6-8,26). They can also be used to search for other hydatid locations. At the end of combinations of specific radiological findings, the type of hydatid cyst can be diagnosed in all cases (27). Our pre-diagnosis for this mass was soft tissue mass or incisional hernia. And then, abdominal ultrasound was made and it showed a cystic lesion in subcutaneous layer with echogenic content within. The diagnosis of our case was based on US and MRI that showed a cystic mass in subcutaneous anterior abdominal wall with an uncalcified cystic wall. No other intraabdominal cystic lesions were found. US is main radiologic tool both to diagnose and to follow-up hydatid disease. Nevertheless, the atypical localization of the cysts in the unusual sites demanded further radiologic investigations in the form of CT or MRI to better delineate the anatomy and help in differential diagnosis. US is the examination of choice because of its high diagnostic accuracy, low cost and non-invasiveness and it shows both the assessment of the state and activity within the cysts (3,6,20,23). Cyst wall calcification, cyst infection and peritoneal seeding are well showed by CT with high sensitivity and specificity (3). The multiplanar imaging and the excellent soft tissue contrast of MRI provide valuable information on the extent of the disease (27-29). Enhancement of the pericystic soft tissues can be considered an MRI feature suggestive of soft-tissue hydatid disease (7,28,29). Serologic tests such as immuno-electrophoresis, specific IgG, indirect hemagglutination (IHA) tests and complement fixation tests are useful tools that confirm the correct diagnosis (8,14). However, they may not always be helpful in diagnosing of pshc disease. ELISA (Enzyme-Linked Immuno Sorbent Assay) /Western blood serology is % sensitive and 88-96% specific for liver cyst infestation, but less sensitive for lung (50-56%) or other organ involvement (25-26%) (8). The IHA test

5 Primary subcutaneous hydatid cyst on the incisional scar following open cholecystectomy is positive in more than 80% of liver hydatid cysts. However, false negative IHA results can be higher in other located hydatid cyst (7,20). Negative test results do not indicate the absence of the disease (30). The risk of false-positive or false-negative results should be taken into consideration. Preoperative and postoperative values of IgG- ELISA and IHA serology in our case were negative. Because of the variable clinical presentation of pshc, uniform guidelines for evaluation and treatment are lacking. The surgical treatment can be useful for making an accurate and prompt diagnosis of suspicious on hydatid cyst. The best curative treatment of pshc of anterior abdominal wall is complete surgical excision of the intact cyst and surrounding tissue without opening the cyst, which avoids leakage of cyst material that can cause anaphylaxis and local recurrence (6-8,20). The aim of complete resection of hydatid cyst removes all parasitic and pericystic tissues. An incomplete resection (subtotal pericystectomy) or a percutaneous puncture-aspiration-injection-reaspiration (PAIR) procedure can be considered if the total resection of cyst is impossible (7,8,25). PAIR is an effective non-surgical approach in hydatid cyst as well as medical treatment with the use of albendazole (7). Fine needle aspiration biopsy is not recommended, since there is the risk of spillage and allergic reactions. We performed total cyst excision with local anesthesia in the operating room and irrigated the surgical areas with protoscolicidal agents. Rupture of hydatid cyst occurs usually secondary to trauma and surgery, but it may also occur spontaneously (31,32). Cysts s content spilled after rupture are considerably allergenic, which can damage surrounding tissues. Anaphylaxis mediated by IgE in hydatid cyst is a life-threatening complication. 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