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52 TMJ 2012, Vol. 62, No. 1 - 2

INTRODUCTION

Echinococcus granulosus, which causes cystic echinococcosis, is one of the smallest members of the Taenia family. In the larval stage, it causes zoonosis in humans.1

The hydatid disease is widespread around the globe but is unevenly concentrated, being more common in rural areas, and more prevalent among shepherds, butchers and cattle breeders. The disease

ORIGINAL ARTICLES

ABSTRACT

REZUMAT

TREATMENT OF THE HEPATIC HYDATID CYST BY ULTRASOUND-GUIDED TRANSCUTANEOUS PUNCTURE

Abdulah Salim1, Doru Bordos1, Ciprian Duta1, Delia Zahoi2, Ecaterina Daescu2

Introduction: The hepatic hydatid cyst, a parasitic disease caused by Taenia Echinococcus, can nowadays be successfully treated using ultrasound-guided transcutaneous puncture, a minimally invasive method, described in the literature as early as the ‘90s. Objectives: This study aims to evaluate the results of the percutaneous puncture treatment of the hepatic hydatid cyst, a method that is more cost effective and often safer than surgery. Material and methods: The study was conducted on 319 patients (aged between 16 and 67 years), treated between April 1996 and December 2011. Out of the 319, 46 were operated laparoscopically, 56 by classical surgery and 217 were subjected to ultrasound-guided transcutaneous puncture. Results: The average duration of hospitalization was 3.5 days. The reduction in cyst diameter was: 58-100% in the first month; 69-100% after three months; 72-100% after 6 months and 75-100% after 12 months. A number of 5 (2.3%) patients had mild allergic reactions, 86.56% of the cysts disappeared, 5.97% needed to undertake the puncture procedure again and 7.46% of the cysts relapsed. Conclusions: The advantages of this method are obvious in terms of surgery trauma, post-operative morbidity, duration of hospitalization, surgically difficult to reach areas and post-surgery relapses. Key Words: hepatic hydatid cyst, Taenia Echinococcus, ultrasound-guided transcutaneous puncture.

Introducere: Chistul hidatic hepatic, afecţiune provocată de Taenia Echinococcus, este în prezent tratat cu succes şi prin puncţie transcutanată ghidată ecografic, metodă minim invazivă cu rezultate bune şi foarte bune, descrisă în literatura de specialitate începând cu anii ‘90. Obiective: În această lucrare am avut ca scop evaluarea rezultatelor tratamentului chistului hidatic hepatic prin puncţie percutană, metodă comparabilă şi adesea mai sigură decât chirurgia şi care se poate realiza cu costuri minime. Material şi metode: În perioada aprilie 1996 – decembrie 2011 au fost incluşi în studiu un număr de 319 pacienţi dintre care 46 au fost operaţi laparoscopic, 56 au fost operaţi pe cale clasică iar la 217 s-a efectuat puncţia transcutanată ghidată ecografic. Vârsta pacienţilor a fost cuprinsă între 16 şi 67 ani. Rezultate: Durata de spitalizare medie a fost de 3,5 zile. Scăderea diametrului chistelor a fost: în prima lună – între 58 şi 100%; la trei luni – între 69 şi 100%; la 6 luni – între 72 şi 100%; după 12 luni – între 75 şi 100%. Un număr de 5 (2,3%) pacienţi au prezentat reacţii alergice uşoare ; 86,56% din chiste au dispărut; 5,97% au necesitat repuncţionare; 7,46% din chiste au recidivat. Concluzii: Avantajele metodei sunt evidente în ceea ce priveşte traumatismul operator, morbiditatea postoperatorie, durata spitalizării, în localizările greu accesibile chirurgical, în recidivele postoperatorii. Cuvinte cheie: chist hidatic hepatic, Taenia Echinococcus, puncţie transcutanată ghidată ecografic

Received for publication: Nov. 24, 2011. Revised: May 20, 2012.

1 Department of General Surgery No. 2, Clinical Emergency County Hospital Timisoara, 2 Department of Anatomy and Embryology, Victor Babes University of Medicine and Pharmacy, Timisoara

Correspondence to: Abdulah Salim, Emergency County Hospital, 10 I. Bulbuca Blvd, 300736, Timisoara, Romania. Tel. +40-72-223-9078. Email: surg20032003@yahoo.com

is considered endemic in the following areas: the Mediterranean, Middle East, Eastern Europe, Africa, Argentina, Australia, Chile, China and New Zealand.2,3 In Romania, the number of cases is increasing due to increases in livestock numbers and to current diagnostic possibilities.4

Diagnosis is usually the result of paraclinical investigations such as ultrasound, X-ray, CT, hematologic and immunohistochemical tests. It is rarely established by clinical examination as the disease has a slow, insidious evolution and, in the absence of complications, is accompanied by minimum and non-specific clinical manifestations.5,6

The treatment of hepatic hydatid cysts includes drugs such as albendazole or mebendazole, classical or laparoscopic surgery and the PAIR method (puncture, aspiration, injection, re-aspiration).7

The treatment is usually mixed, but in the case of small, young cysts with thin walls, only medical methods can be used. Some studies have shown that high doses and long term use of drugs can lead to reversible

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Abdulah Salim et al 53

neutropenia and, in some cases, to modifications of the hepatic and renal functions.8-11

Open surgery is especially useful in difficult to reach locations, in the cases of giant cysts or in the presence of adhesions, but it has a higher rate of complications and leads to longer hospitalization. Laparoscopy, recommended only when the cyst is easily accessible, ensures a high visibility, a safe hemostasis and a shorter hospitalization.10-12

The ultrasound-guided transcutaneous puncture is a method that caught on very quickly due to its relatively easy execution and to its incontestable advantages compared to surgery, in terms of post-operative mortality and duration of hospitalization.10,11

However, the method also involves a number of risks, such as bleeding accidents, injuries of other viscera, secondary seeding caused by leaks of the hydatid fluid into the abdominal cavity and infections. Also, allergic reactions including anaphylactic shock may occur.

We report here the preliminary results of this treatment used on 217 patients presenting 268 hepatic hydatid cysts.

MATERIAL AND METHODS

Between April 1996 and December 2011, 319 patients with hepatic hydatid cysts were admitted to the Department of General Surgery No. 2, of the Timisoara County Emergency Hospital. Out of these, 46 were operated laparoscopically, 56 were operated through classical methods and 217 were treated using ultrasound-guided transcutaneous punctures. (Fig. 1)

Figure 1. Distribution of cases according to the type of surgery.

As a detection method for hydatid cysts, all patients undertook abdominal ultrasounds. The hepatic cysts were classified according to the criteria developed by Gharbi and Niron.13,14

The abdominal ultrasound was followed by computed tomography for a more accurate cyst evaluation, with superior results regarding size and location. Casona intradermoreactions were also performed in 67 cases, with serological diagnosis based on echinococcosis antibodies.

The 217 patients were selected for treatment by ultrasound-guided transcutaneous puncture according

to the following criteria:- Types I and II according to the Gharbi

classification;- Patients for whom drug treatment had failed;- Patients for whom surgical treatment was

contraindicated due to the multiple comorbidities;- Cysts in difficult to reach locations;- Patients who accepted this treatment as an

alternative to the surgical treatment;This treatment was unsuitable for:- Patients with hydatid cysts classified from the

ultrasound point of view as Gharbi types III, IV and V;- Patients whose cysts were infected or broken in

the bile ducts or in the peritoneum.After the certitude diagnosis of hepatic hydatid

cysts, therapeutic protocols must be prepared. At the moment, the protocol for the percutaneous puncture is standardized and is unanimously approved and applied by specialists in all the countries that have accepted it.

The minimally invasive PAIR treatment protocol of the hydatid cyst15-17

The puncture site was chosen under ultrasound guidance. Most often, it was the same intercostal space recommended by most of the authors as having the lowest risk of peritoneal seeding.18-20

The steps of the PAIR minimally invasive treatment protocol of hydatid cysts were observed.

2

Chirurgia deschisa este utila mai ales in localizarile greu accesibile ale chistelor hidatice hepatice, in cele gigante, in prezenta aderentelor, dar are o rata mai mare a complicatiilor si o spitalizare crescuta, pe cand cura laparoscopica este indicata doar in localizarile accesibile ale chistelor, insa asigura o vizibilitate foarte buna, o hemostaza sigura si o spitalizare mai scurta.10,11,12

Punctia transcutanata ghidata ecografic e o metoda ce a prins foarte repede, pe de o parte datorita executiei relativ facile si pe de alta parte datorita avantajelor incontestabile in raport cu tratamentul chirurgical, in ceea ce priveste morbiditatea postoperatorie si durata spitalizarii.10,11 Metoda are insa si o serie de riscuri, respectiv accidente hemoragice, leziuni ale altor viscere, insamantari secundare determinate de scurgera lichidului hidatic in cavitatea abdominala, infectii. De asemenea pot apare reactii alergice mergand pana la soc anafilactic.

In acest articol raportam rezultate preliminare ale acestui tratament aplicat in cazul a 268 de chiste hidatice hepatice, la 217 pacienti.

MATERIAL ŞI METODĂ În perioada aprilie 1996 - decembrie 2011, au fost internaţi şi operaţi în Clinica Chirurgie II a Spitalului Clinic Judeţean de

Urgenţă, Timişoara un număr de 319 cazuri cu chist hidatic hepatic, dintre care 46 de bolnavi au fost operati laparoscopic, 56 pe cale clasică, iar în 217 cazuri s-a efectuat puncţia transcutanată ghidată ecografic (Grafic 1);

56 46

217

0

50

100

150

200

250

Open surgery Laparoscopic surgery Transcutaneous puncture

No. of cases

Grafic 1: Distribuţia cazurilor în funcţie de tipul intervenţiei chirurgicale Ca şi metodă de detectare a chistului hidatic, toţi pacienţii au efectuat ecografie abdominală si chistele hepatice au fost

clasificate dupa criteriile enuntate de Gharbi13 si Niron14.

7

The minimally invasive PAIR treatment protocol of the hydatid cyst 15,16,17

The puncture site was chosen under ultrasound guidance. Most often, it was the same

intercostal space recommended by most of the authors as having the lowest risk of peritoneal

seeding.18,19,20

The steps of the PAIR minimally invasive treatment protocol of hydatid cysts were

observed.

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54 TMJ 2012, Vol. 62, No. 1 - 2

RESULTS

The age of the 319 patients under study was between 16 and 67 years, with an increased incidence in 21 – 30 and 31 - 40 years age groups. (Fig. 2) The female/male ratio was 1.4/1 (187 cases found in females and 132 cases found in males). (Fig. 3)

Figure 2. Distribution of cases according to age group.

Figure 3. Distribution of cases according to gender.

Table 1. Ultrasound classification of hepatic hydatid cysts of the entire

group, according to Gharbi criteria.13

Out of the 319 patients, we shall further refer only to those selected for the PAIR method (217 patients, presenting 268 cysts).

Table 2. Clinical parameters of the study group.*

Table 3. Cysts parameters.

The cysts’ diameter varied from 2.5 cm up to giant size of 15.8 cm. The average diameter of the treated cysts was 5.8 cm.

All cysts were successfully treated using the PAIR method. The results are presented in Table 4.

Table 4. Results.

Following this procedure, five (2.3%) cases of allergic reactions were recorded, which responded very well to treatment with hydrocortisone hemisuccinate and anti-allergic medication. In this series 232 cysts (86.56%) disappeared, 16 (5.97%) needed to undertake the puncture procedure again, due to the stagnation of diameters for 3 months, and 20 cysts (7.46%) relapsed, so surgical interventions were subsequently required.

DISCUSSIONS

Transcutaneous ultrasound-guided puncture entered the therapeutic arsenal for hydatid cysts starting with the mid 80’s.21

Today, most experts believe that the first choice in the treatment of the univesicular hydatid cyst is drug therapy with albendazole in the usual doses. If this treatment fails, the PAIR method is to be associated, and if the technique cannot be used, traditional laparoscopic treatment or conventional surgery shall be applied.22-24

5

Din intreg lotul de studiu, 319 pacienti, reiese ca vârsta pacienţilor a fost cuprinsă între 16 – 67 ani, cu incidenţă crescută în grupele de vârstă 21 – 30 ani şi 31 - 40 ani (Grafic 2), iar raportul sex feminin/sex masculin=1,4 (187 cazuri întâlnite la sexul feminin şi 132 cazuri întâlnite la sexul masculin) - Grafic 3.

21

104

81

57

31 25

0

60

120

16-20 21-30 31-40 41-50 51-60 >61

No. of cases

Grafic 2: Distribuţia cazurilor în funcţie de grupele de vârstă

6

187

132

0

50

100

150

200

Female Male

No. of cases

Grafic 3: Distribuţia cazurilor în funcţie de sex

Tabel 1 - Clasificarea ecografica a chistelor hidatice hepatice la intregul lot dupa criteriile Gharbi13:

Tipul ecografic Pacienti (nr.) Pacienti (%)

I = chist bine delimitat, lichid limpede în interior;

120 37,61

II = chist bine delimitat, cu dublu contur al pertilor, cu lichid limpede

97 30,4

8

RESULTS

The age of the 319 patients under study was between 16 and 67 years, with an

increased incidence in 21 – 30 and 31 - 40 years age groups (Fig. 2). The female/male ratio

was 1.4/1 (187 cases found in females and 132 cases found in males) – (Fig. 3).

Figure 2: Distribution of cases according to age group

Figure 3: Distribution of cases according to gender

Table 1 – Ultrasound classification of hepatic hydatid cysts of the entire group, according to

Gharbi criteria:13

Out of the 319 patients, we shall further refer only to those selected for the PAIR method

(217 patients, presenting 268 cysts).

9

Table 2 – Clinical parameters of the study group*

10

Table 3 - Cysts parameters:

The cysts’ diameter varied from 2.5 cm up to giant size of 15.8 cm. The average

diameter of the treated cysts was 5.8 cm.

All cysts were successfully treated using the PAIR method. The results are presented

in Table 4.

11

Table 4 – Results.

Following this procedure, 5 (2.3%) cases of allergic reactions were recorded, which

responded very well to treatment with hydrocortisone hemisuccinate and anti-allergic

medication. In this series 232 cysts (86.56%) disappeared, 16 (5.97%) needed to undertake

the puncture procedure again, due to the stagnation of diameters for 3 months, and 20 cysts

(7.46%) relapsed, so surgical interventions were subsequently required.

Figure 4 – Hepatic hydatid cyst before puncture (ultrasound)

Figure 5 - Hepatic hydatid cyst before puncture (ultrasound)

Figure 6 - Hepatic hydatid cyst 5 days after the puncture (ultrasound)

Figure 7 - Hepatic hydatid cyst 5 days after the puncture (ultrasound)

Figure 8 - Hepatic hydatid cyst 9 months after the puncture (ultrasound)

DISCUSSIONS

Transcutaneous ultrasound-guided puncture entered the therapeutic arsenal for hydatid

cysts starting with the mid 80s.21

Today, most experts believe that the first choice in the treatment of the univesicular

hydatid cyst is drug therapy with albendazole in the usual doses. If this treatment fails, the

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Abdulah Salim et al 55

Figure 4. Hepatic hydatid cyst before puncture (ultrasound).

Figure 5. Hepatic hydatid cyst before puncture (CT).

Figure 6. Hepatic hydatid cyst 5 days after the puncture (ultrasound).

PAIR benefits are related to the fact that this is a minimally invasive maneuver, with a low risk compared to surgery, as well as much lower costs and hospitalization times. Also, it improves the effectiveness of chemotherapy before and after the puncture, most likely by increasing the penetrability of the drug substance within the cystic cavity.

Figure 7. Hepatic hydatid cyst 5 days after the puncture (CT).

Figure 8. Hepatic hydatid cyst 9 months after the puncture (ultrasound).

As with any puncture, there is a risk of hemorrhagic accidents, injuries of adjacent viscera and, if the recommended protocol is not observed, secondary seeding due to the rupture of the pericyst and leakage of hydatid fluid into the abdominal cavity. Also, rupture of the hydatid cysts can lead to allergic reactions, ranging from mild symptoms to anaphylactic shock. The frequency of their occurrence is however not sufficiently studied.25 Due to the allergic potential of echinococcosis, prophylactic antihistamines and anti-inflammatory steroids were administered to the patients in our study before anesthesia, and only 5 cases (2.3%) of mild allergic reactions were recorded. In similar studies, authors also recorded small numbers of mild allergic reactions and no cases of anaphylactic shock, giving 20 minutes before the puncture H1 antihistamines and an anti-inflammatory steroid (Prednisone).26

As with conventional surgery, this method brings into discussion the possibility of sclerosing cholangitis, through the injection of a parasiticide into a cyst with a cystic-biliary communication. Thus, if a cystic-biliary fistula is suspected following the analysis of the aspirated fluid, the procedure is aborted.

8

Fig.1 - Chist hidatic hepatic inainte de punctie(ecografie)

9

Fig.2 - Chist hidatic hepatic inainte de punctie(tomografie)

10

Fig.3- Chist hidatic hepatic la 5 zile dupa punctie(ecografie)

11

Fig.4- Chist hidatic hepatic la 5 zile dupa punctie(tomografie)

12

Fig.5- Chist hidatic hepatic la 9 luni dupa punctie(ecografie)

Discutii Punctia transcutanata ghidata ecografic a intrat in arsenalul terapeutic al chistului hidatic

hepatic incepand cu mijlocul anilor 80. 21 In prezent majoritatea specialistilor considera ca prima alegere in chistul hidatic

univezicular este terapia medicamentoasa cu albendazol in dozele uzuale. In cazul esecului se asociaza PAIR, iar daca tehnica este imposibil de aplicat se recurge la tratamentul laparoscopic sau prin abord clasic.22, 23, 24

Beneficiile PAIR sunt legate de faptul că este o manevră minim invazivă, cu un risc scăzut în comparaţie cu chirurgia, cu o spitalizare mult redusă faţă de chirurgia clasică şi cu un cost mult mai mic decât intervenţia chirurgicală clasică. De asemenea ea îmbunătăţeşte eficacitatea chimioterapiei pre şi postpunctională, probabil prin creşterea penetrabilităţii substanţei medicamentoase în interiorul cavităţii chistice.

Ca in orice punctie exista risc de accidente hemoragice, de leziuni a vicerelor adiacente, iar daca nu se respecta protocolul recomandat, pot apare insamantari secundare determinate de ruperea perichistului si scurgerea lichidului hidatic in cavitatea abdominala.Tot prin ruptura chistului hidatic pot aparea reactii alergice usoare mergand pana la soc anafilactic, dar frecventa declansarii acestora nu este suficient studiata.25 Astfel datorita potentialului alergic al echinococozei, in cazul lotului nostru de pacienti, s-au administrat profilactic preanestezic antihistaminice si antiinflamatoare steroidiene, inregistrandu-se doar 5 cazuri(2,3%) de reactii alergice usoare. In studii similare si alti autori au inregistrat un numar mic de reactii alergice

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56 TMJ 2012, Vol. 62, No. 1 - 2

In such cases, some authors recommend the introduction of drainage tubes into the cavity, by means of dilators on the initial tract of the puncture. Drainage tubes are maintained until the externalized secretion diminishes and disappears.26 Their results were mostly good, but in some cases surgery was still necessary in order to perform an external biliary drainage before closing the biliary fistulas of the residual cavity. Today, this disadvantage can also be eliminated by performing an endoscopic papilosphincterotomy.26,27

Mueller et al. were the first to report a case of a patient who, after the PAIR procedure, was left with a drainage tube for three months. Subsequently, it turned out that maintaining a catheter for a longer period of time can lead to superinfection and clogging of the lumen drainage tube with fragments of the germinative membrane.21 Also, the routine use of catheterization for the drainage of the remaining cavity largely extended the duration of hospitalization, that reached to an average of 8.73 days with limits between 2 and 30 days, and, in the case of concurring infection, to an average of 25 days with limits between 20 and 30 days.26

All cysts punctured by us were of types I and II according to the Gharbi classification, and their treatment was performed in a single step, without subsequent drainage.

The cavity that results from the procedure was found to gradually reduce in diameter. This is due to the use of alcohol, which ensures the sclerosis of the germinative membrane and its fibrous transformation. Through content aspiration, the intracystic pressure disappears and the hepatic parenchyma expands, leading to a gradual collapse of the cavity.

There are cases in which, due to the large size of the cavity, liquid secretion increases from transudation through the cyst walls, impeding the cavity’s collapse. In these cases, a puncture repetition may be required if the diameter of the cavity appears constant for over 3-6 months.

Other authors used the PAIR method for some cysts of types III and IV under the Gharbi classification and experienced complications – both major (anaphylactic shock in 0.1 to 0.2% of cases) and minor (rash, jaundice, fever, cyst superinfection or biliary system rupture in 10-30% of cases).28

Even with cysts of types I and II and no complications, cysts were found to relapse in up to 4% of patients.29,30

The results of our study are only preliminary, as the monitoring period necessary to draw safe conclusions about the effectiveness of the PAIR treatment has not ended.

We believe that the PAIR method is not sufficient in the case of multivesicular, infected or calcified cysts, but it represents an efficient and safe solution for the treatment of the hydatid cyst in carefully selected cases. Surgical treatment remains the main alternative.

CONCLUSIONS

The PAIR method is increasing in popularity for the treatment of hydatid cysts because it is easy to apply and is linked to low mortality.

The advantages of this method are most obvious when compared to surgery, in terms of trauma, post-operatory mortality, hospitalization duration, use in difficult to reach locations and post-surgery relapses. The combination of medical treatment using anthelmintic drugs with percutaneous puncture is a viable alternative to surgery for removing hepatic hydatid cysts.31

Still, the PAIR method is limited in its’ applicability and requires careful patient selection.

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