Amyand’s hernia in adult and pediatric practice. Urological aspects

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Abstract

Amyand’s hernia, an inguinal hernia that contains the appendix within the hernia sac, is a rare condition in surgical, and even more so in urological practice. It is difficult to diagnose due to its low frequency, anatomical atypicality and urgency. There are no review publications on this issue, and those that are available are mainly descriptions of individual cases. In this article an attempt was made to highlight the largest possible number of clinical observations that reflect the urological aspects of the problem and present them in a systematic manner. Aim — to analyze the available publications covering the urological aspects of Amyand’s hernia.

The literature review was carried out on the basis of publications covering the urological aspects of Amyand’s hernia in adults and children, published in PubMed databases and Scientific Electronic Library eLibrary.ru. The search was carried out by the following keywords: “Amyand’s hernia”, “acute scrotum”, “inflammatory diseases of the scrotum”, “Fournier’s phlegmon”, “scrotal fistula” (in Russian and English). Taking into account the rarity of the analyzed problem, the analysis includes publications for the last 15 years. 189 publications related to the topic of the review were identified. 38 publications of the greatest scientific and practical interest were selected directly for citation in the review. The maximum number of available publications reflecting the “urological aspects” of Amyand’s hernia has been analyzed, and an attempt has been made to systematize them clinically. The variety of clinical manifestations caused by Amyand’s hernia is commonly referred to in the English literature as “Amyand’s hernia syndrome”. We have identified the following variants of the clinical course: scrotal fistula — a casuistic variant described in detail only by C. Amyand; picture of the “acute scrotum” syndrome — the most frequent variant of the clinical course of Amyand’s hernia with scrotal manifestations is noted mainly in childhood, however, it can also occur in adults in all age groups; phlegmon of the scrotum, phlegmon of Fournier — an extremely rare variant of the clinical course with scrotal manifestations, publications on this subject are rare.

Amyand’s hernia being a rare surgical problem in both children and adults, has a number of urological “masks”, causing diagnostic difficulties and is of some interest to the urologists.

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Introduction.

Amiand hernia is an infrequent condition in surgical, and especially in urological practice. If cases of detection of an unchanged vermiform process in the inguinal canal and scrotum are often detected during operations both on the abdominal cavity and on the inguinal canal, then the pathology of the appendix in the hernial sac is much less common. Amiand's hernia was first described in 1735, when the military surgeon C.Amiand at St. George's Hospital in London successfully operated on an 11-year-old boy with a right-sided inguinal-scrotal hernia containing a vermiform appendix [1]. Ryan W.J. (1937) back in the 30s of the XX century, he pointed out the possibility of the formation of an abscess in the scrotum with the infringement of the vermiform process in the inguinal canal with a very low frequency, citing the figure of 0.13% [2]. A. Creese in 1953, the term "Amiand hernia" was introduced into clinical practice [3].  Despite the popularity of the condition since the middle of the XVIII century, it remains difficult to diagnose due to the rarity of the problem, anatomical atypicity and urgency. The specialized literature contains mainly descriptions of individual cases or small series of observations, but there are no generalizing publications. Variants of the scrotal manifestations of this disease are described in isolated works and are not subject to any analysis, more often than not, simply indications of the possibility of such a variant of the clinical picture prevail [4; 5; 6]. The diagnosis of Amiand hernia is complex and is based mainly on computed tomography and laparoscopy data, which are not always performed routinely in acute diseases of the abdominal cavity, the scrotal localization of clinical manifestations even less encourages the use of these methods, making it as difficult as possible to establish a diagnosis [7]. It is generally believed that Amiand hernia, including those with scrotal clinical manifestations, is detected three times more often in childhood [8]. Ivanschuk G. (2014) noted such changes in adult patients, pointing to the complexity of differential diagnosis with orchitis and testicular torsion [9]. Taking into account the rarity of pathology, we believe that any description and publication on this issue is of some scientific value. In our publication, an attempt has been made to highlight the largest possible number of clinical observations reflecting the "urological" aspects of the problem and to attempt to systematize them.

Objective. To analyze the available publications covering the urological aspects of Amanda's hernia.

Materials and methods.

The literature review was carried out on the basis of publications covering the "urological" aspects of Amyand hernia in adults and children – variants of the clinical course of Amyand hernia with imitation of the "acute scrotum" syndrome, published in PubMed databases (https://pubmed.ncbi.nlm.nih.gov/) and Scientific Electronic Library Elibrary.ru (https://elibrary.ru /). The selection of publications cited in the review is limited to articles in peer-reviewed journals.

The search was carried out by the following keywords: "Amiand hernia", "acute scrotum", "inflammatory diseases of the scrotum", "Fournier's phlegmon", "scrotal fistula" (analogy in English).

The rarity of the sanctified aspect made it possible to include publications in the work over the past 15 years (2007-2022). At the first stage, 189 publications related to the review topic were identified. Publications that do not carry information available for analysis and duplicate publications were excluded from them. The second stage, based on the relevance of the material and the degree of reliability of sources, impact factors of journals and the logical presentation of data in the work, 38 works of the greatest scientific and practical interest were selected directly for citation in the review.

Results.

An attempt has been made to analyze the maximum possible number of publications reflecting the "urological aspects" of Amiand hernia and systematize them according to clinical variants.

The variety of clinical manifestations caused by Amiand hernia is commonly referred to in the English literature as "Amiand hernia syndrome". We have identified the following variants of the clinical course:

- scrotal fistula;

- picture of the "acute scrotum" syndrome;

- phlegmon of the scrotum, phlegmon of Fournier.

The primary description of the disease itself is somehow related to urology and is represented by a fecal fistula of the scrotum - on 6.12.1753, the surgeon C.Amiand operated on an 11-year-old patient whose inflamed vermiform process containing a steel pin in the inguinal-scrotal hernia was complicated by destructions with the formation of a fecal fistula of the scrotum [1].

Manifestations of the syndrome of "acute scrotum" in some cases may be manifested in Amyand hernia [10]. Khanal B. (2020) noted the possibility of enlargement of the scrotum and its soreness in the clinical picture of Amiand hernia [11]. Brainwood M. (2020), analyzing the variants of impaired obliteration of the vaginal process and their complications, also pointed to Amyand hernia as one of the rare causes of the formation of the "acute scrotum" symptom complex [12]. The need for differential diagnosis with various forms of acute scrotum syndrome – strained hydrocele, testicular torsion, epididymorochitis, was pointed out in his work by Mahajan A. [13]. The development of the same phenomena in Amiand hernia was indicated by Salvatore P. [14]. Diagnosing the true cause of a scrotal disaster can be difficult. Yodoshi T. (2018) described a newborn patient treated with a picture of acute scrotum syndrome. At the initial treatment (physical data, ultrasound, X-ray), the picture was regarded as acute epididymitis, conservative treatment was prescribed. Only upon repeated visit to the clinic, against the background of the progression of the destructive process in the scrotal cavity, when repeating instrumental methods, free gas was detected in the scrotal cavity on an X-ray and a tubular structure that did not exclude the location of the vermiform process in the scrotal cavity according to ultrasound, which required surgical intervention that confirmed inflammation of the vermiform process in the scrotal cavity [15]. Erginel B., (2017) published a similar observation of perforated appendicitis in an inguinal-scrotal hernia in a newborn with significant diagnostic difficulties [16]. Omran A. (2019) presented an observation of a patient of 14 days of life, in whom the clinical picture indicated exclusively scrotal pathology, ultrasound did not allow to exclude the diagnosis, the patient was operated on with suspicion of a torsion of the spermatic cord, an intraoperative pyocele was detected against the background of destructive appendicitis with the location of the process in the scrotum [17]. Mohamed A. (2019) described a case of Amyand hernia in a newborn 19 days of life; upon examination, a picture of acute epididymitis was found. During ultrasound examination of the scrotum and inguinal region, only an increase in the echogenicity of the testicle on the right and hypervascularization were detected. Only the development of the pattern of abdominal complications after 12 hours forced a repeated ultrasound examination and followed by laparotomy [18].

A similar pattern is possible at an older age. Khorramirouz R. (2015) cited the observation of a 5-year-old patient whose picture of an acute inflammatory process in the scrotum was formed against the background of an Amyand hernia complicated by an omentum infarction. The author noted the absence of characteristic abdominal symptoms with the development of exclusively scrotal changes. An interesting fact is that with the involvement of preoperative ultrasound, a true diagnosis was established before the operation, which is not characteristic of Amyand hernia, especially with its atypical course. They performed appendectomy, omentum resection and inguinal herniation with inguinal access [19]. A similar case was described by Sharma S.B. (2004) in a 6-year-old patient. He observed diffuse abdominal pain, vomiting, which may occur with testicular torsion and the development of local symptoms of acute scrotum syndrome; the diagnosis was made intraoperatively, appendectomy and ligation of the vaginal process of the peritoneum were performed [20]. In rare cases, the authors appreciate the possibilities of ultrasound and Dopplerography in the diagnosis of Amyand hernia more highly. So, Prando D. (2009) pointed out that UZDG can be very useful for excluding testicular torsion from differential diagnosis with sensitivity from 80 to 98% and accuracy of 97% [21].

There is an opinion in the literature that in the neonatal period, an Amiand hernia should be included in the differential diagnosis of testicular torsion and other forms of acute scrotum syndrome due to the similarity of the clinical picture [22; 16]. Milburn J.A. (2006) presented a description of a newborn patient with testicular ischemia due to compression of the spermatic cord against the background of inflammation of the appendix in the inguinal canal. [23].

Probert S. (2022) and Fernando J. (2002) pointed out the need to take these conditions into account in the differential diagnosis and in older children, the first of them represented their own clinical observation with diagnostic difficulties in an 11-year-old patient [24; 25].

Less often there is a picture of the left-sided syndrome of the "acute scrotum". Serrano A. (1979) described a case of appendicitis in an inguinal-scrotal hernia on the left with the formation of an inflammatory infiltrate affecting the testicle and the spermatic cord. The patient underwent an orchofuniculectomy with an appenectomy [26]. Khan R.A. (2011) observed a patient for 10 months with a left testicular torsion pattern with a long period of ischemia, which turned out to be an inflamed process in a left-sided hernia [27]. Cases of such atypical localization are even more difficult to diagnose. Okunev N.A. (2022) observing a patient of 2 years and 8 months with a left-sided inguinal-scrotal hernia and suspected scrotal phlegmon in the preoperative period during the preparation, he performed an ultrasound examination twice, however, the use of the method did not allow to make the correct diagnosis and determine the hernial contents correctly [28]. Singh K. (2011) observed a patient with an external picture of acute scrotum syndrome on the left in a patient with a left-sided Amyand hernia with its infringement in a 1.5-year-old child, which also created additional diagnostic difficulties [29].

It is possible to combine Amiand hernia with other diseases of the inguinal-scrotal region. Fasecetti-Leon F. (2017) described a patient of 23 days of life who had a picture of an "edematous and hyperemic scrotum" with palpable volume in the scrotum in the absence of a testicle in the scrotum. Intraoperatively, an inguinal-scrotal hernia with a pinched process and an abdominal form of cryptorchidism were diagnosed [30]. Kumar R. (2008) presented in his observation a combination of Amiand hernia with an inflamed appendix and inguinal cryptorchidism in a patient of 26 days of life, the patient underwent appendectomy and simultaneous orchopexy [31]. Dhanasekarapandian V. (2018) published a clinical observation of a combined pathology associated with Amyand hernia in a child of 35 days – a combination of an inflamed appendix in a sliding inguinal hernia with inguinal cryptorchidism. The inflamed process caused compression of the elements of the spermatic cord and testicle in the inguinal canal and led to irreversible testicular ischemia, which required orchectomy. The author designated the triple association described by him as the "Amiand triad" - a combination of components of Amiand hernia, appendicitis and non-omission of the testicle, however, this term is not used in other sources and is exclusively author's [32]. Otty O. (2021) described the combination of Amiand hernia with cryptorchidism in a variant of cross ectopia in a 17-year-old patient, in his case, the presence of a developed scrotum and the apparent picture of volume in the scrotum during inflammation also simulated the clinic of acute scrotum syndrome; the author performed appendectomy and orchopexy simultaneously [33]. As an exceptional case, a combination of an Amiand hernia with the development of an inflammatory process in the scrotum, simulating the clinic of an "acute scrotum" in combination with an omphalocele, is described; interestingly, in this case, a left-sided lesion was found [34].

In adult practice, differential diagnosis with the syndrome of "acute scrotum" is carried out much less often, however, such descriptions are also available. Laermans S. (2007) stated a case of Amyand hernia treatment in an adult male under the guise of acute epididymitis [35]. Malik K. (2019) described the observation of an Amiand hernia in a 50-year-old man whose clinical picture was dominated by swelling and hyperemia of the scrotum, orchoepididymitis and infected hydrocele were considered in the differential diagnosis, and only with the use of additional methods was an inguinal-scrotal hernia with infringement of the vermiform process was detected [36].

Conditions when an Amiand hernia "simulates" Fournier's disease are extremely rare in the literature. Rajaguru K (2016) presented a 47-year-old patient who, upon admission to the hospital on the 5th day of the disease, was diagnosed with Fournier's phlegmon with a lesion area of 15 * 12 cm, covering the groin area and scrotum to the root. The true diagnosis was established only in the process of performing a necrectomy in the depth of the wound. Changes in the testicle and spermatic cord also forced orchofuniculectomy [37]. He pointed out the possibility of the formation of scrotal phlegmon in hernia of Amiand and Morales-Cárdenas A. (2015) with a frequency of 0.1%, but without detailing this observation [38].

Conclusion.

From the analysis it follows that among all the variants of the "urological" forms of the clinical course of Amiand hernia, the simulation of the picture of "acute scrotum syndrome" prevails, while variants with the formation of a fecal fistula or scrotal phlegmons are much less common. This pathology is more common in children, but at any age it cannot be excluded.

Knowing the variants of the course of Amiand hernia and taking it into account in the differential diagnosis, in our opinion, will avoid a number of difficulties both in the diagnosis and in the surgical treatment of diseases of the inguinal-scrotal region in adult and pediatric patients.

 

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About the authors

Dmitry N. Shchedrov

Yaroslavl State Medical University; Regional Pediatric Clinical Hospital

Email: shedrov.dmitry@yandex.ru
ORCID iD: 0000-0002-0686-0445
SPIN-code: 7354-7379
ResearcherId: HHZ-7778-2022

Dr. Sci. (Med.), assistant professor of the Department of Urology and Nephrology, head of the Department of Pediatric Uroandrology

Russian Federation, 5 Revolutsionnaya st., Yaroslavl, 150000; Yaroslavl

Igor S. Shormanov

Yaroslavl State Medical University

Email: i-s-shormanov@yandex.ru
ORCID iD: 0000-0002-2062-0421
SPIN-code: 7772-8420
Scopus Author ID: 6507085029

Dr. Sci. (Med.), Professor, head of the Department of Urology with Nephrology

Russian Federation, 5 Revolutsionnaya st., Yaroslavl, 150000

Daria Yu. Garova

Yaroslavl State Medical University

Email: dar.garova@yandex.ru
ORCID iD: 0000-0003-4457-9694
SPIN-code: 5789-8889

clinical resident

Russian Federation, 5 Revolutsionnaya st., Yaroslavl, 150000

Natalia A. Sidorova

Yaroslavl State Medical University

Author for correspondence.
Email: nsidorova775@gmail.com
ORCID iD: 0009-0000-3624-8639

student

Russian Federation, 5 Revolutsionnaya st., Yaroslavl, 150000

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