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Journal of Medical C<strong>as</strong>e Reports<br />

C<strong>as</strong>e report<br />

<strong>Low</strong> <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong> <strong>in</strong> a 6-<strong>year</strong>-<strong>old</strong> <strong>boy</strong> present<strong>in</strong>g <strong>as</strong> <strong>an</strong><br />

<strong>in</strong>carcerated <strong>in</strong>gu<strong>in</strong>al <strong>hernia</strong>: a c<strong>as</strong>e report<br />

Efstratios Christi<strong>an</strong>akis 1 , Nikolaos P<strong>as</strong>chalidis 2 , Georgios Filippou 2 ,<br />

Spiros Rizos 2 , Dimitrios Smailis 2 <strong>an</strong>d Dimitrios Filippou* 2,3<br />

BioMed Central<br />

Open Access<br />

Address: 1Department of Pediatric Surgery, Pendeli Children's Hospital, Palaia Pendeli, Athens, Greece, 2First Department of Surgery, Piraeus<br />

General Hospital "Tz<strong>an</strong>eio", Piraeus-Athens, Greece <strong>an</strong>d 3Department of Anatomy, Histology <strong>an</strong>d Embryology, Nurs<strong>in</strong>g Faculty, University of<br />

Athens, Galatsi-Athens, Greece<br />

Email: Efstratios Christi<strong>an</strong>akis - xristi<strong>an</strong>akis@<strong>in</strong>.gr; Nikolaos P<strong>as</strong>chalidis - webdocgr@hotmail.com;<br />

Georgios Filippou - georgiosfilippoumd@hotmail.com; Spiros Rizos - srizos@otenet.gr; Dimitrios Smailis - d_smailis@yahoo.gr;<br />

Dimitrios Filippou* - d_filippou@hotmail.com<br />

* Correspond<strong>in</strong>g author<br />

Published: 29 J<strong>an</strong>uary 2009<br />

Received: 19 May 2008<br />

Journal of Medical C<strong>as</strong>e Reports 2009, 3:34 doi:10.1186/1752-1947-3-34<br />

Accepted: 29 J<strong>an</strong>uary 2009<br />

This article is available from: http://www.jmedicalc<strong>as</strong>ereports.com/content/3/1/34<br />

© 2009 Christi<strong>an</strong>akis et al; licensee BioMed Central Ltd.<br />

This is <strong>an</strong> Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0),<br />

which permits unrestricted use, distribution, <strong>an</strong>d reproduction <strong>in</strong> <strong>an</strong>y medium, provided the orig<strong>in</strong>al work is properly cited.<br />

Abstract<br />

Introduction: <strong>Low</strong>er <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong> is a very rare entity. The cl<strong>in</strong>ical f<strong>in</strong>d<strong>in</strong>gs are similar to those<br />

of <strong>in</strong>gu<strong>in</strong>al <strong>hernia</strong>s <strong>an</strong>d <strong>in</strong> m<strong>an</strong>y c<strong>as</strong>es may be misdiagnosed. In the literature, only a few references<br />

to this entity have been reported <strong>in</strong> children. To the best of our knowledge, this is the first c<strong>as</strong>e<br />

report of a lower <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong> <strong>in</strong> a child who presented with <strong>an</strong> acute pa<strong>in</strong>ful scrotum.<br />

C<strong>as</strong>e presentation: We discuss the c<strong>as</strong>e of a 6-<strong>year</strong>-<strong>old</strong> Greek <strong>boy</strong> who presented to our<br />

emergency department compla<strong>in</strong><strong>in</strong>g of severe pa<strong>in</strong> <strong>in</strong> the left <strong>in</strong>gu<strong>in</strong>al area <strong>an</strong>d scrotum. The acute<br />

pa<strong>in</strong>ful swell<strong>in</strong>g started suddenly, without <strong>an</strong>y obvious cause. The <strong>in</strong>itial diagnosis w<strong>as</strong> <strong>in</strong>carcerated<br />

<strong>in</strong>gu<strong>in</strong>al <strong>hernia</strong> which w<strong>as</strong> reduced with difficulty. Five days later, the patient still experienced mild<br />

pa<strong>in</strong> dur<strong>in</strong>g palpation <strong>an</strong>d he w<strong>as</strong> operated on. Dur<strong>in</strong>g the operation, a large lower <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong><br />

w<strong>as</strong> revealed <strong>an</strong>d reconstructed.<br />

Conclusion: Although <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong>s are rare <strong>in</strong> children <strong>an</strong>d difficult to diagnose, physici<strong>an</strong>s<br />

should be aware of them <strong>an</strong>d <strong>in</strong>clude them <strong>in</strong> the differential diagnosis.<br />

Introduction<br />

<strong>Low</strong>er <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong> is a rare entity <strong>in</strong> pediatric surgery,<br />

<strong>an</strong>d is often misdiagnosed <strong>as</strong> <strong>an</strong> <strong>in</strong>gu<strong>in</strong>al <strong>hernia</strong> [1]. Less<br />

th<strong>an</strong> 50 c<strong>as</strong>es have been presented <strong>in</strong> the literature, <strong>an</strong>d<br />

only half of them occurred <strong>in</strong> younger children. In most<br />

of these c<strong>as</strong>es, <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong>s are considered congenital,<br />

although there is not enough evidence to support this<br />

suggestion [2,3]. We report a rare c<strong>as</strong>e of a lower <strong>Spigeli<strong>an</strong></strong><br />

<strong>hernia</strong> <strong>in</strong> a child which presented <strong>as</strong> <strong>an</strong> <strong>in</strong>carcerated<br />

<strong>in</strong>gu<strong>in</strong>al <strong>hernia</strong>.<br />

C<strong>as</strong>e presentation<br />

A 6-<strong>year</strong>-<strong>old</strong> Greek <strong>boy</strong> presented with a pa<strong>in</strong>ful <strong>in</strong>tumescence<br />

of the left <strong>in</strong>gu<strong>in</strong>al area. The child w<strong>as</strong> <strong>in</strong>itially<br />

referred to a general hospital where the physici<strong>an</strong>s suggested<br />

that the problem w<strong>as</strong> <strong>an</strong> <strong>in</strong>carcerated left <strong>in</strong>gu<strong>in</strong>al<br />

<strong>hernia</strong>. The <strong>hernia</strong> w<strong>as</strong> reduced with difficulty by the local<br />

surgeon <strong>an</strong>d the next day, the child w<strong>as</strong> referred to our<br />

hospital for further evaluation <strong>an</strong>d treatment. In the physical<br />

exam<strong>in</strong>ation, the left <strong>in</strong>gu<strong>in</strong>al area appeared normal<br />

without signs of <strong>in</strong>tumescence. The only f<strong>in</strong>d<strong>in</strong>gs were<br />

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Journal of Medical C<strong>as</strong>e Reports 2009, 3:34 http://www.jmedicalc<strong>as</strong>ereports.com/content/3/1/34<br />

subcut<strong>an</strong>eous edema <strong>an</strong>d a hematoma, probably due to<br />

the m<strong>an</strong>ipulations for the reduction. Dur<strong>in</strong>g palpation,<br />

the child presented with mild discomfort <strong>an</strong>d pa<strong>in</strong> <strong>in</strong> the<br />

left testicle without cl<strong>in</strong>ical evidence of testicular torsion.<br />

These symptoms persisted for 3 days <strong>an</strong>d f<strong>in</strong>ally we<br />

decided to operate on the patient. A lower <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong><br />

w<strong>as</strong> identified dur<strong>in</strong>g the operation. The <strong>hernia</strong> sac<br />

w<strong>as</strong> found to penetrate from a small defect (with <strong>an</strong> estimated<br />

diameter of about 1.5 cm) <strong>in</strong> the <strong>Spigeli<strong>an</strong></strong> f<strong>as</strong>cia<br />

with<strong>in</strong> Hesselbach's tri<strong>an</strong>gle (Figure 1).<br />

The lower <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong> orifice is usually located <strong>in</strong> a<br />

well-def<strong>in</strong>ed area. The defect develops <strong>as</strong> a r<strong>in</strong>g-like open<strong>in</strong>g<br />

through the fibers of the tr<strong>an</strong>sversalis f<strong>as</strong>cia <strong>an</strong>d the<br />

f<strong>as</strong>cia of the tr<strong>an</strong>sversalis abdom<strong>in</strong>us <strong>an</strong>d <strong>in</strong>ternal oblique<br />

abdom<strong>in</strong>us muscles. <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong>s usually present just<br />

lateral to the rectus muscle <strong>in</strong> the lower left quadr<strong>an</strong>t.<br />

In our patient, the <strong>hernia</strong> sac w<strong>as</strong> that of a typical <strong>Spigeli<strong>an</strong></strong><br />

<strong>hernia</strong>. The orifice diameter w<strong>as</strong> about 1.5 cm, while<br />

the sac extended up to 7 cm surrounded by preperitoneal<br />

fat (Figure 1). The <strong>hernia</strong> sac conta<strong>in</strong>ed a small part of the<br />

Intra-operative photograph of the <strong>hernia</strong> show<strong>in</strong>g the opened sac<br />

Figure 1<br />

Intra-operative photograph of the <strong>hernia</strong> show<strong>in</strong>g the opened sac.<br />

large omentum which w<strong>as</strong> reduced. Reconstruction of the<br />

<strong>hernia</strong> w<strong>as</strong> performed with non-absorbable sutures. Eight<br />

<strong>year</strong>s after the operation, the patient rema<strong>in</strong>s free of symptoms<br />

<strong>an</strong>d recurrence.<br />

Discussion<br />

<strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong>s occur through a congenital or usually<br />

acquired defect of the <strong>Spigeli<strong>an</strong></strong> f<strong>as</strong>cia, lateral to the rectus<br />

muscle sheath [1]. In adults, almost 90% of acquired <strong>hernia</strong>s<br />

occur with<strong>in</strong> the <strong>an</strong>terior superior iliac sp<strong>in</strong>e <strong>an</strong>d the<br />

umbilicus, while the majority of congenital <strong>hernia</strong>s occur<br />

at the level of the arcuate l<strong>in</strong>e (f<strong>old</strong> of Dougl<strong>as</strong>) [1-3]. Herni<strong>as</strong><br />

that are protrud<strong>in</strong>g through the <strong>Spigeli<strong>an</strong></strong> f<strong>as</strong>cia<br />

with<strong>in</strong> Hesselbach's tri<strong>an</strong>gle, caudally <strong>an</strong>d medially to the<br />

<strong>in</strong>ferior epig<strong>as</strong>tric vessels, are called low or lower <strong>Spigeli<strong>an</strong></strong><br />

<strong>hernia</strong>s [4]. Direct <strong>in</strong>gu<strong>in</strong>al <strong>hernia</strong>s c<strong>an</strong> also occur <strong>in</strong><br />

this area, although <strong>in</strong> most of these c<strong>as</strong>es, the <strong>hernia</strong>'s orifice<br />

is difficult to identify. This is the re<strong>as</strong>on why m<strong>an</strong>y<br />

<strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong>s are misdiagnosed <strong>an</strong>d usually considered<br />

<strong>as</strong> direct <strong>in</strong>gu<strong>in</strong>al <strong>hernia</strong>s, para-<strong>in</strong>gu<strong>in</strong>al <strong>hernia</strong>s or<br />

<strong>hernia</strong>s through the conjo<strong>in</strong>ed tendon [5,6]. The diagnosis<br />

is usually made on the b<strong>as</strong>is of the location of the her-<br />

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Journal of Medical C<strong>as</strong>e Reports 2009, 3:34 http://www.jmedicalc<strong>as</strong>ereports.com/content/3/1/34<br />

nia's orifice. The orifice of the <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong> is usually<br />

located <strong>in</strong>side the fibers of the tr<strong>an</strong>sversalis f<strong>as</strong>cia. The orifice<br />

is sharp <strong>an</strong>d rigid <strong>in</strong> palpation <strong>an</strong>d its diameter r<strong>an</strong>ges<br />

from 2 to 3 cm [2,3,6]. It extends through this defect <strong>in</strong>terstitially<br />

or penetrates the entire abdom<strong>in</strong>al wall [7,8]. The<br />

sac of these <strong>hernia</strong>s usually conta<strong>in</strong>s preperitoneal fatty<br />

tissue, <strong>an</strong>d rarely omentum or <strong>in</strong>test<strong>in</strong>e. Some authors<br />

have suggested that, <strong>in</strong> <strong>in</strong>f<strong>an</strong>ts, <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong>s may<br />

coexist with undescended testes.<br />

The small size of the <strong>hernia</strong> neck may be a possible expl<strong>an</strong>ation<br />

for the <strong>in</strong>cre<strong>as</strong>ed <strong>in</strong>cidence of <strong>in</strong>carceration. The<br />

<strong>in</strong>cidence of <strong>in</strong>carceration varies signific<strong>an</strong>tly between<br />

various series <strong>an</strong>d authors. Some authors have suggested<br />

that <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong> <strong>in</strong>carceration is a rare entity while<br />

others have suggested that it is very common with <strong>an</strong> estimated<br />

<strong>in</strong>cidence of approximately 22% [9,10].<br />

The pre-operative diagnosis of lower <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong>s is<br />

difficult, especially <strong>in</strong> non-<strong>in</strong>carcerated c<strong>as</strong>es. In most of<br />

the c<strong>as</strong>es, diagnosis c<strong>an</strong>not be achieved by physical exam<strong>in</strong>ation<br />

<strong>an</strong>d the <strong>hernia</strong> c<strong>an</strong> be e<strong>as</strong>ily misdiagnosed <strong>as</strong> congenital<br />

<strong>in</strong>gu<strong>in</strong>al <strong>hernia</strong>. Recent studies have reported that<br />

the use of ultr<strong>as</strong>ound <strong>an</strong>d computed tomography (CT)<br />

may contribute to the pre-operative diagnosis <strong>in</strong> selected<br />

c<strong>as</strong>es. A CT sc<strong>an</strong> may be useful <strong>in</strong> the diagnosis of <strong>an</strong> <strong>in</strong>carcerated<br />

<strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong> ma<strong>in</strong>ly due to its ability to demonstrate<br />

the layers of the <strong>an</strong>terior abdom<strong>in</strong>al wall [11,12].<br />

Reduction of the content with excision of the sac <strong>an</strong>d closure<br />

of the f<strong>as</strong>cia defect with non-absorbable sutures is the<br />

most common <strong>an</strong>d widely accepted surgical approach,<br />

present<strong>in</strong>g the lowest recurrence rates [1,3,7]. Recent studies<br />

support the possible role of laparoscopy <strong>in</strong> the diagnosis<br />

<strong>an</strong>d treatment of <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong> <strong>in</strong> children,<br />

suggest<strong>in</strong>g that it may represent <strong>an</strong> acceptable therapeutic<br />

alternative [4,5].<br />

Conclusion<br />

<strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong> occurs through a congenital or, more<br />

often, <strong>an</strong> acquired defect <strong>in</strong> the <strong>Spigeli<strong>an</strong></strong> f<strong>as</strong>cia, lateral to<br />

the rectus muscle sheath. It is very rare <strong>in</strong> children. The<br />

c<strong>as</strong>e that we present is unique because it is probably the<br />

first report of a lower <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong> present<strong>in</strong>g <strong>as</strong> acute<br />

scrotum. No similar c<strong>as</strong>es have been reported <strong>in</strong> the literature.<br />

The aim of the present c<strong>as</strong>e report is to alert the physici<strong>an</strong><br />

of this rare entity because, although it is difficult to<br />

diagnose pre-operatively, accurate diagnosis <strong>an</strong>d appropriate<br />

treatment are essential to avoid future recurrences.<br />

Abbreviations<br />

SH: <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong>; CT: computed tomography.<br />

Compet<strong>in</strong>g <strong>in</strong>terests<br />

The authors declare that they have no compet<strong>in</strong>g <strong>in</strong>terests.<br />

Authors' contributions<br />

All authors contributed equally to the treatment of the<br />

patient (EC, NP, GF, DS, SR, DF). EC, NP <strong>an</strong>d GF wrote<br />

the draft. SR <strong>an</strong>d DF approved it. GF, DS <strong>an</strong>d DF carried<br />

out the revision.<br />

Consent<br />

Written <strong>in</strong>formed consent w<strong>as</strong> obta<strong>in</strong>ed from the patient's<br />

parents for publication of this c<strong>as</strong>e report <strong>an</strong>d <strong>an</strong>y accomp<strong>an</strong>y<strong>in</strong>g<br />

images. A copy of the written consent is available<br />

for review by the Editor-<strong>in</strong>-Chief of this journal.<br />

Acknowledgements<br />

The authors would like to th<strong>an</strong>k Dr Stolkidis Dimitris from Kor<strong>in</strong>thos General<br />

Hospital for his support <strong>in</strong> the treatment of this rare type of <strong>in</strong>gu<strong>in</strong>al<br />

<strong>hernia</strong>.<br />

References<br />

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reports of three c<strong>as</strong>es. Surg Today 1994, 24:1081-1084.<br />

2. Al-Salem AH: Congenital spigeli<strong>an</strong> <strong>hernia</strong> <strong>an</strong>d cryptorchidism:<br />

cause or co<strong>in</strong>cidence? Pediatr Surg Int 2000, 16:433-436.<br />

3. White JJ: Concomit<strong>an</strong>t spigeli<strong>an</strong> <strong>an</strong>d <strong>in</strong>gu<strong>in</strong>al <strong>hernia</strong>s <strong>in</strong> a<br />

neonate. J Pediatr Surg 2002, 37:659-660.<br />

4. Rodgers BM, McGahren ED, Burns R: Pediatric <strong>hernia</strong>s. In Abdom<strong>in</strong>al<br />

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al. New York: Spr<strong>in</strong>ger-Verlag; 2001:591-609.<br />

5. Sp<strong>an</strong>gen L: <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong>. In Hernia Edited by: Nyhus LM, Condon<br />

RE. Philadelphia, PA: Lipp<strong>in</strong>cott Co; 1995:381-392.<br />

6. Donnell<strong>an</strong> WL: Umbilical <strong>hernia</strong>s, <strong>Spigeli<strong>an</strong></strong> <strong>an</strong>d other unusual<br />

abdom<strong>in</strong>al <strong>hernia</strong>s. In Abdom<strong>in</strong>al Surgery of Inf<strong>an</strong>cy <strong>an</strong>d Childhood<br />

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Publishers; 1996:1-7.<br />

7. Iswariah H, Metcalfe M, Morrison CP, Maddern GJ: Facilitation of<br />

open spigeli<strong>an</strong> <strong>hernia</strong> repair by laparoscopic location of the<br />

<strong>hernia</strong> defect. Surg Endosc 2003, 17:832.<br />

8. Los<strong>an</strong>off J, Richm<strong>an</strong> B, Jones J: <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong> <strong>in</strong> a child: c<strong>as</strong>e<br />

report <strong>an</strong>d review of the literature. Hernia 2004, 6:191-193.<br />

9. Kirby RM: Str<strong>an</strong>gulated <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong>. Postgrad Med J 1987,<br />

63:51-52.<br />

10. Sp<strong>an</strong>gen L: <strong>Spigeli<strong>an</strong></strong> <strong>hernia</strong>. World J Surg 1989, 13:573-580.<br />

11. Levy G, Nagar H, Blachar A, Ben-Sira L, Kessler A: Preoperative<br />

sonographic diagnosis of <strong>in</strong>carcerated neonatal <strong>Spigeli<strong>an</strong></strong><br />

<strong>hernia</strong> conta<strong>in</strong><strong>in</strong>g the testis. Pediatr Radiol 2003, 33:307-409.<br />

12. Toms AP, Dixon AK, Murphy JMP, Jameson NV: Illustrated review<br />

of new imag<strong>in</strong>g techniques <strong>in</strong> the diagnosis of abdom<strong>in</strong>al wall<br />

<strong>hernia</strong>s. Br J Surg 1999, 86:1243-1249.<br />

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