Pediatr Surg Int (2005) 21: 841–842 DOI 10.1007/s00383-005-1526-2
CASE REPORT
David C. van der Zee Æ Klaas (N)MA Bax
Laparoscopic resection of intra-abdominal extralobar pulmonary sequestration
Accepted: 5 June 2005 / Published online: 26 August 2005 Springer-Verlag 2005
Abstract The intra-abdominal localization of extralobar pulmonary sequestration (EPS) is an uncommon entity, although there are an increasing number of publications in literature on EPS over recent years. There seems to be a predominance of left-sided suprarenal positioning of the sequester and so far resection has been undertaken by way of laparotomy. This paper describes the laparoscopic resection of EPS in two patients. In both instances the procedure was successful and the postoperative course was uneventful. It is concluded that EPS should be included in the differential diagnosis of suprarenal masses, particularly on the left side. Laparoscopic resection is the method of choice for EPS. Keywords Extralobar pulmonary sequestration Æ Intra-abdominal Æ Laparoscopy Æ Children
Introduction The intra-abdominal localization of extralobar pulmonary sequestration (EPS) with congenital cystic adenomatoid malformation (CCAM) type II is an uncommon entity. Until 1994 only five cases have been described [1]. Since then there have been more case reports on EPS containing CCAM type II [2, 3] or congenital pulmonary airway malformation (CPAM) type II as it is nowadays called [4]. Prenatal ultrasound may be responsible for the increased awareness in recent years. It is nowadays believed that intra-abdominal extralobar pulmonary sequestration should be kept in differential diagnosis in cases of masses in the suprarenal location, especially on the left side [5]. D. C. van der Zee (&) Æ K. (N)MA Bax Department of Pediatric Surgery, KE 04.140.5, Wilhelmina Children’s Hospital, University Medical Center Utrecht, P.O. Box 85090, 3508 AB Utrecht, The Netherlands E-mail:
[email protected] Tel.: +31-30-2504004 Fax: +31-30-2505348
This paper describes the laparoscopic approach of EPS in two cases where an intra-abdominal mass was diagnosed prenatally on ultrasound.
Case reports Case 1 A 14-month-old child in whom antenatally an intraabdominal mass was detected, was explored through a leftsided lumbotomy by a pediatric urologist under the diagnosis of adrenal mass. During exploration it became obvious that the process was not in connection with the kidney. The consulted pediatric surgeon advised to terminate the procedure and to perform further diagnostic workup. Postoperatively an MRI scan was performed, which demonstrated a cystic process in close connection with the adrenal gland. There was no connection with the intestines. Alpha-fetoprotein, VMA/HVA were within normal ranges. On laparoscopic approach after mobilization of the splenic flexure of the colon a cystic tumor was seen in close connection with the left adrenal gland. The process could be dissected from the gland and removed in an endobag. Histopathology demonstrated a CPAM type II extralobar pulmonary sequester. Recovery was uneventful and the child was discharged the second day after surgery. Case 2 At 30 weeks of gestation a solid tumor was found in the upper left abdomen. The postnatal ultrasound confirmed a hyperdense tumor with a diameter of 2.5·3 cm. The process was located between the medial side of the kidney and the spleen and posteriorly from the pancreas.
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Fig. 1 Intra-abdominal extralobar pulmonary sequestration, located between kidney (K), spleen and behind pancreas (P)
The probable diagnosis was set at intra-abdominal extralobar pulmonary sequestration. At the age of 2 weeks the child underwent a laparoscopic resection of the process. The approach was through the lateral mesocolon, posterior from the pancreas and medial from the kidney (Figs. 1, 2). By lifting the colon against the ventral abdominal wall with a superficial stay suture the view was excellent. The tumor had the aspect of lung tissue. Vascularization came from the splenic vessels. The child tolerated the laparoscopic approach very well. Postoperative recovery was uneventful and the patient was discharged the second day after surgery. Histology displayed CPAM type II tissue.
Discussion Approximately 9.7% of extralobar sequestration occur intra-abdominally [2]. Vascularization usually comes from the aorta abdominalis but may arise from other intra-abdominal vessels like in our second patient [2]. Excision is indicated to ascertain diagnosis. Malignant degeneration has been described but is anecdotal [2]. CPAM type II is an infrequent occurring lesion in extralobar pulmonary sequestration, but has been described more often in recent years. The term congenital pulmonary airway malformation is more appropriate for the lesion as described by Stocker [4]. Resection so far has always been a major surgical procedure for the neonate. Like in adrenal gland surgery the approach warrants major exposure of the area. The introduction of laparoscopic adrenal surgery has greatly facilitated fast recovery, also in the very small child. The
Fig. 2 Intra-abdominal extralobar pulmonary sequestration of 2.5·3 cm
two cases presented demonstrate that the laparoscopic approach to the lesion is simple. With the large magnification of the endoscope meticulous dissection of the lesion can be carried out with identification and ligation of the feeding vessels. The tumor can be evacuated through a slightly enlarged umbilical port site with the use of an endobag. Recovery therefore is quick and uneventful. In conclusion the laparoscopic approach can be used for a continuum of new indications reducing the major impact of open surgery.
References 1. Aulicino MR, Reis ED, Dolgin SE, Unger PD, Shah KD (1994) Intra-abdominal pulmonary sequestration exhibiting congenital cystic adenomatoid malformation. Report of a case and review of the literature. Arch Pathol Lab Med 118:1034–1037 2. Gross E, Chen MK, Lobe TE, Nuchtern JG, Rao BN (1997) Infradiaphragmatic extralobar pulmonary sequestration masquarading as an intra-abdominal suprarenal mass. Pediatr Surg Int 12:529–531 3. Morad NA, al-Malki T, e-Tahir M (1997) Intra-abdominal pulmonary sequestration: diagnostic difficulties. Pathology 29:218–220 4. Stocker JT (2002) Congenital pulmonary airway malformation: a new name and an expanded classification of congenital cystic adenomatoid malformations of the lung. Histopathology 41:424–431 5. White J, Chan YF, Neuberger S, Wilson T (1994) Prenatal sonographic detection of intra-abdominal extralobar pulmonary sequestration: report of three cases and literature review. Prenat Diagn 14:653–658
