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Long-term outcome of minimal access surgery for hydatidosis in children: a single institutional experience

  • Gowri ShankarEmail author
  • S. Ramesh
  • K. R. Srimurthy
Original Research
  • 184 Downloads

Abstract

Background

Human cystic echinococcosis continues to be a public health problem worldwide. The standard treatment of hydatidosis in children is open surgical intervention for removal of the cyst completely without spillage. The limited use of minimal invasive surgery (MIS) in hydatidosis has been due to the concern of inadequate removal and spillage, leading to recurrence or dissemination of the disease. Recently, a few authors have reported the successful use of minimal invasive surgery (MIS) for management of hydatid disease in children. We present our experience with successful use of MIS in the management of hydatid disease involving lung and liver in children.

Methods

Between 2006 and 2017, data of 22 children treated for hydatid disease in our institute were reviewed. The diagnosis was made radiologically with Computerised Tomography scan of chest and abdomen. All children received albendazole therapy prior to and after the surgery. Thoracoscopic/laparoscopic procedures were undertaken sequentially in all children. Children with synchronous disease involving lung and liver had an interval of 2 weeks between procedures. The techniques of puncture, aspiration, injection and re-aspiration (PAIR) and also our modifications of cyst removal are described.

Results

There were a total of 22 children with liver and or lung involvement. Four children had synchronous lung and liver involvement and four children had bilateral lung involvement. The duration of the procedure ranged from 60 to 90 min. There were 3 (18) conversions in the thoracic group and 1 (12) in the laparoscopic group. PAIR technique with our modification of cyst extraction was used in all except in one child. Capitonnage of the cyst wall in lung hydatidosis was not done. There were no postoperative events. Recurrence of the lesion at original site was seen in two children, one each in laparoscopic and thoracoscopic group. Occurrence of new lesion or dissemination of the disease was not identified on a mean follow-up of 7 years.

Conclusion

Our series demonstrates the safe and effective utility of MIS in management of pulmonary and liver hydatid in children. Use of MIS does not lead to dissemination of the disease. Albendazole therapy is an useful adjunct prior to surgery. Single lung ventilation with isolation of uninvolved lung is useful during surgery for lung hydatid.

Keywords

Pulmonary hydatid Liver hydatid Children MIS 

Introduction

Human cystic echinococcosis, or hydatid cyst disease, is a zoonosis caused by the larval cestode Echinococcus granulosus and man being an accidental host.

It still continues to be a public health problem in sheep rearing regions around the world [1]. In India, endemic areas described are in Andhra Pradesh and Tamilnadu [2]. The main therapeutic modalities presently available for management are drug therapy with albendazole or mebendazole, percutaneous drainage and surgery.

Management of hydatid cysts has involved open surgical approach with care taken to avoid spillage [3, 4, 5, 6]. Percutaneous aspiration of a hydatid cyst was considered as a contraindication to avoid the risk of leakage of cyst contents, and possibility of anaphylaxis or seeding.

Since the advent of effective drug therapy against Echinococcus; the use of pre- and postoperative therapy with albendazole or mebendazole combined with percutaneous drainage (consisting of puncture, aspiration, injection, and re-aspiration, or PAIR) of hydatid cysts is now being widely advocated with good outcomes [7, 8].

Becaumer [9] initially described the use of thoracoscopy for hydatid. With the development of laparoscopic techniques over the past decades, minimally invasive surgery has been investigated as a feasible approach for the treatment of hydatid disease for intra-abdominal lesions as well.

Since then, many investigators have reported utility of MIS in the management of these lesion in children [9, 10, 11].

We present our experience in the use of MIS in the management of lung and liver hydatid in children. The technique of PAIR with our technique of cyst management is described. Ours series also discusses the management of synchronous lesion and bilateral lung lesions.

Materials and methods

The records of 22 children undergoing thoracoscopy/laparoscopy for lung and liver hydatid between 2006 and 2017 were reviewed. There were 15 boys and 7 girls. The mean age was 12.5 years. Presenting symptoms included cough, fever and chest pain in children with lung involvement and pain abdomen in children with liver hydatidosis. All children underwent chest X-ray, CT scan thorax, abdomen and pelvis. The diagnosis was made on clinical and radiological studies (Fig. 1). Specific anti-echinococcal serological tests were not undertaken due to poor specificity. Ten children had unilateral isolated lung involvement and four had bilateral lung involvement. Seven children had isolated liver involvement. Four had synchronous lung and liver involvement. The location and site are discussed in Table 1. The size of the cysts ranged from 4 cm to the largest being 11 cm in dimension.
Fig. 1

CT Scan showing the synchronous lesions involving lung and Liver

Table 1

XXXX

No

Age

Sex

Organ involved

Laterality and size (cm)

1

13

M

Lung and liver

Liver segment-7 (5.0 × 5.9 × 6.4)

Lung-right-5.8 × 5.1 × 5.6, Left- 4.3 × 4.1 × 4.4

2

9

F

Lung

Left lower lobe (6.0 × 5.3 × 5.9)

3

5

M

Liver

Liver segment-7,8 (7.2 × 6.4 × 4.8)

4

7

M

Lung

Left lower lobe- (6 × 7.2 × 8)

5

10

M

Lung

Right middle lobe- (4.2 × 4.8 × 5.0)

6

12

F

Liver

Liver segment-6, 7, 8 (10.6 × 8), Liver segment- 3 (4.4 × 4.0)

7

10

M

Liver

Liver segment- 6 (10 × 8)

8

5

M

Lung

Left lower lobe (5.8 × 6.2 × 6.4)

9

8

M

Lung

Bilateral lower lobe; left- (4.3 × 4.4 × 5.0), right-(3.4 × 4.3 × 4.3)

10

12

F

Liver

Liver segment-7, 8 (10 × 9 × 11)

11

4

M

Lung and liver

Left lower lobe

Liver segment-6 (4.3 × 4.1 × 3.5)

12

8

M

Liver

Liver segment-6 (4.3 ×  4.8 ×  4.5)

13

12

M

Liver

Liver segment-3 (7.2 ×  6.5 ×  6.0)

14

6

M

Liver

Liver segment-7, (6.3 × 5.5 ×  5.0)

15

10

M

Lung and liver

Liver segment-6, 7 (11.5  ×  5.5  ×  6.7)

16

10

F

Liver

Liver segment- 7, 8 (6.1 ×  5.5 ×  6.0)

17

7

M

Lung

Bilateral: Left upper lobe and right lower lobe,

4.1 ×  4.5 ×  5.0 (L)

5.2 × 5.6 × 6.1 (R)

18

10

M

Lung and liver

Left upper lobe lung (5.3 × 4.8 × 6.4)

Right upper lobe lung (4.8 × 4.3 × 5.6)

Liver segment-3, 4 (4.0 × 6.4 × 3.4)

19

8

F

Lung

Right middle lobe (4.3 × 4.1 × 4.4)

20

4

F

Lung

Left lower lobe (4.3 × 4.1 × 4.4)

21

10

M

Lung

Right lower lobe (5 × 8)

22

9

F

Lung

Right middle lobe (5.1 × 5.0 × 4.5)

All children with lung and liver hydatidosis were pretreated with albendazole. This was given in a dosage of 10 mg/kg/day (with a maximum dose of) for 2–4 weeks prior to surgery.

Operative technique

Thoracoscopic technique

Single lung ventilation was used in all. Bronchoscopy for placement of Fogarty balloon catheter was used to occlude the bronchus on the involved side. The child was placed in lateral position with the lesion side up. Three ports were used in all. Initial optical port was placed farthest form the cyst. A pressure of 5 mmHg with a flow rate of 1.5 L/min was used. Additional port was placed in relation to the optical port to achieve a triangular configuration. 18G spinal needle was inserted percutaneously to puncture the cyst with aspiration of cystic fluid. Equal amount of 3% normal saline was instilled into the cyst taking care to avoid spillage. A suction canula was kept close to the site of puncture during aspiration or instillation of saline (Fig. 2). A total dwell time of 15 min with 3 instillations was used. Care was taken not to aspirate the cyst completely.
Fig. 2

Showing PAIRE technique. a Liver hydatid; b Lung hydatid

After the third instillation, a 5-mm/10-mm port was inserted directly into the cyst. A disposable rigid suction cannula (Fig. 3a) was passed through the 10-mm trocar. This was used to aspirate the endogerminal contents. In all cases, the germinal layer could be completely aspirated (Fig. 3b). The remnant cyst was evaluated for completeness of removal with a 10-mm telescope placed directly into the cyst cavity. It was evaluated for air leak and completeness of removal of the germinal layer. The pleural cavity was irrigated with normal saline. An intercostal drain was placed with the tip into the cyst and side holes in the pleural cavity. Capitonnage of the cyst was not done in any of the cases as it was not deemed necessary.
Fig. 3

a External picture showing the rigid suction cannula and Port paced into cyst with suction. b Specimen of cyst completely removed. c Intra-cystic view after removal of endogerminal layer. d X-ray chest in immediate post operative period showing resolution of the lesion

Laparoscopic technique

The patient was placed in modified lithotomy position. The optical port (5 mm) was placed at the umbilicus. Pneumoperitoneum was induced with a pressure of 8–10 mmHg, with a flow rate of 1.5 L/min. Working ports were placed in the epigastrium/left hypochondrium and right anterior axillary line. 18G spinal needle was inserted percutaneously to puncture the cyst under laparoscopic visualisation. The sequence of aspiration and instillation of hypertonic saline was done three times with a total dwell time of 15 min. A 10-mm trocar was inserted directly into the cyst followed by the technique of suction aspiration as mentioned in thoracoscopic section (Fig. 2). The remnant cyst was evaluated for completeness of removal with a 10-mm telescope placed directly into the cyst cavity (Fig. 3c).

In one child, the cyst size was small. After the PAIR technique the cyst wall was incised over 4 cm. The endo-germinal layer was extracted and placed into an endo bag for retrieval.

Results

The procedure was well tolerated. There were no anaesthesia-related complications.

The operative time for thoracoscopy group ranged from 60 to 90 min and for the laparoscopy group ranged from 60 to 100 min.

There were 4 intra-operative events. There were three conversions in children undergoing thoracoscopy and one conversion in laparoscopy group. This was due to development of air leak at the completion of the procedure in two children and in one child after extubation. The site of air leak (not seen during thoracoscopy) could be identified at open thoracotomy through the cyst. This bronchial communication was closed using monofilament suture (Prolene® Ethicon Inc.) in a purse string configuration. In one child with liver involvement, there was bleeding after PAIR technique and aspiration requiring laparotomy and completion of the procedure.

Post-operative recovery was uneventful. In children undergoing thoracoscopy, the intercostal drain was removed between days 3 and 5. There was no air leak identified in the post operative period nor was there any requirement of prolonged intercostal drainage. Normalisation of lung parenchymal could be identified in the chest X-ray done in postoperative period (Fig. 3d).

In the laparoscopy group, the drain was removed by day 3. We did not encounter bile leak through the drain in any of the children. Of the four children with lung and liver involvement, three underwent sequential surgeries with lung lesion treated first followed by liver lesion. In one child in the later part of our series, both lesions were managed simultaneously.

The follow-up period ranged from 3 to 11 years with a mean follow-up of 7 years. There was one recurrence seen in each group. One child with liver hydatid cyst had recurrence in the same site (segment − 7) at 5 years of follow-up. There were no new lesions. He underwent open surgery as it was deeply seated and posterior in the liver parenchyma. The child with lung recurrence underwent thoracoscopy again with successful clearance of the lesion.

There was no appearance of new lesions or dissemination seen in the follow-up period. There were no adverse reactions encountered to the use of albendazole.

Discussion

MIS for management of hydatid lesions in children is safe and effective as seen in our series. There have been several concerns with the use of MIS in management of hydatid disease. The main concern is of spillage of the cyst fluid leading to possibility of recurrence/dissemination of the disease. This has been addressed effectively by the use of albendazole in the pre- and post-procedure period.

Single lung ventilation has been used by most authors to secure the uninvolved lung [9, 12]. The issue of aspiration of cyst contents is taken care by isolation of the affected side by use of bronchial blockage. This is necessary during the management of lung hydatid.

Some authors have used hypertonic saline in the pleural cavity to take care of spillage during procedure. There have been reports of “near drowning” by the use of this technique [9]. We have used hypertonic saline irrigation only at the completion of the procedure.

In our series, there has been one recurrence in each group and no formation of new lesion on follow-up; the longest being 11 years.

Our technique of insertion of port into cyst and suction aspiration may help in preventing spillage. The endogerminal layer gets seperated off easily and is easily handled by suction. Even large cyst could be easily aspirated without breakage. Similar technique of direct port insertion into the cyst and aspiration has been described in literature [13].

Completeness of removal of the germinal layer can be assessed by placing another telescope directly into the cyst. Also any bile or air leak can be identified at the same time. This was not apparent in three children undergoing thoracoscopy requiring conversion.

Capitonnage has been mentioned in the management of hydatid cysts. Turna et al. [14] compared patients undergoing capitonnage with patients undergoing drainage. There was no significant difference between the two. We did not do capitonnage of the cyst. Follow-up chest X-ray did not reveal any persisting cyst/parenchymal lesion (Fig. 3d).

Our recurrence rates were less than those mentioned in other series [12, 15].

In patients with hydatid disease involving liver, there may be difficulty in identifying the lesions. Proper orientation by pre-operative imaging is necessary to avoid damage to the liver parenchyma.

The cosmetic result has been excellent avoiding thoracotomy or laparotomy.

There has been no port-related problems.

Conclusion

MIS for hydatid involvement of liver and lung in children is safe. Similar results as of open surgery with excellent cosmesis can be achieved with MIS. Bronchial isolation prior to surgery is mandatory for lung hydatidosis. Pre-surgery administration of albendazole helps in sterilizing the cyst and prevention of recurrence or complications of spillage.

Notes

Funding

None.

Compliance with ethical standards

Conflict of interest

The authors declare that they have no conflicts of interest.

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© Springer Nature Singapore Pte Ltd 2019

Authors and Affiliations

  1. 1.Department of Pediatric SurgeryIndira Gandhi Institute of Child HeathBangaloreIndia

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