1 SUBMITTED 26 JUL 22 1 REVISION REQ. 13 SEP 22; REVISION RECD. 7 OCT 22 2 ACCEPTED 24 OCT 22 3 ONLINE-FIRST: DECEMBER 2022 4 DOI: https://doi.org/10.18295/squmj.12.2022.72 5 6 A Rare Case of a Direct Incarcerated Inguinal Hernia Containing an 7 Epiploic Appendage and a Literature Review 8 *Stella Papamichail,1 Eleni Karlafti,2 Petra Malliou,1 9 Apostolos Zatagias,1 Aristeidis Ioannidis,1 Smaro Netta,1 10 Stavros Panidis,1 Daniel Paramythiotis1 11 12 11st Propaedeutic Surgical Department, University Hospital of Thessaloniki AHEPA, 13 Aristotle University of Thessaloniki, Thessaloniki, Greece; 2Emergency Department, 14 University Hospital of Thessaloniki AHEPA, Aristotle University of Thessaloniki, 15 Thessaloniki, Greece. 16 *Corresponding Author’s e-mail: stellapz@auth.gr 17 18 Abstract 19 Inguinal hernias are a widespread condition, responsible for a large number of acute 20 abdomen cases. Typically, indirect, rather than direct, hernias are the ones leading to 21 complications, as a consequence of their narrower hernial defect. Our case concerns a 22 rather rare incidence of a direct incarcerated hernia in a 71-year-old man who presented 23 with acute pain in the left inguinal area. Upon clinical examination, an irreducible 24 inguinal mass was palpated. Therefore, the existence of a complicated hernia was 25 suspected. The patient underwent an emergency repair, during which it was established 26 that the hernia was direct and incarcerated and that its sac contained an ischemic epiploic 27 appendage. The hernia was successfully repaired with mesh, the patient recovered 28 uneventfully and was discharged five days later. Despite the rarity of complicated direct 29 2 inguinal hernias, they should always be included in the differential diagnosis of 30 irreducible groin masses, because they can increase severe complications. 31 Keywords: Direct inguinal hernia; Appendix epiploica. 32 33 Introduction 34 Inguinal hernias are the most frequent type of hernia and their repair is among the most 35 common procedures general surgeons perform.1 Various risk factors can cause a 36 predisposition to the development of hernias, such as male sex, old age, a high body mass 37 index, connective tissue disorders and activity that increases intra-abdominal pressure, 38 like chronic coughing or weight-lifting.2 Inguinal hernias are divided into two categories 39 depending on the point of protrusion of the tissue. The hernia is indirect, when the 40 protrusion occurs through the internal inguinal ring, whereas direct hernias arise from the 41 posterior wall of inguinal canal, in the Hesselbach triangle.3 Due to their wider neck, 42 direct hernias are far less prone to complications.4 An uncomplicated or reducible 43 inguinal hernia typically presents as an inguinal bulge whose contents can return to the 44 abdomen, either spontaneously or by applying pressure.5 Complications arise when the 45 content becomes trapped or incarcerated, whereas strangulation involves reduced blood 46 supply and can cause obstruction, bowel necrosis and perforation.6 Treatment options 47 include ‘watchful waiting’ or elective repair for asymptomatic patients. Reinforcement of 48 the abdominal wall defect through a mesh repair is necessary when complications 49 emerge.7 50 51 In this case report we describe the case of a patient with a complicated direct inguinal 52 hernia who underwent emergency surgery, at the University General Hospital. 53 54 Case Report 55 In 2017, a 71-year-old male presented to the surgical emergency department due to pain, 56 located in the left inguinal region. The pain had started 72 hours before his admission, 57 after lifting weight. Clinical examination revealed a moderately distended abdomen, 58 diminished bowel sounds and mild diffuse tenderness, without signs of peritonitis. In the 59 left inguinal region there was a tender hernia. Reduction of the hernia was attempted but 60 https://pubmed.ncbi.nlm.nih.gov/31482863/ https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5614933/ https://www.aafp.org/afp/2020/1015/p487.html https://link.springer.com/article/10.1007/BF01207592 https://www.ncbi.nlm.nih.gov/books/NBK555972/ https://www.ncbi.nlm.nih.gov/books/NBK459309/ https://link.springer.com/article/10.1007/s10029-017-1668-x#citeas 3 proved impossible. Blood pressure, heart rate, oxygen saturation and body temperature 61 were within normal range. No chronic diseases, past surgeries, allergies of any sort or a 62 history of smoking were mentioned. The laboratory tests were normal except the elevated 63 levels of total white blood cell count (10,56K/μL). 64 65 Moreover, the X – ray of the abdomen was clear. Ultrasonography revealed a large hernia 66 in the left inguinal area [Figure 1]. Doppler ultrasonography showed a reduction of blood 67 flow to the hernial content, a finding on which the diagnosis of an incarcerated inguinal 68 hernia was based [Figure 2]. 69 70 The patient underwent surgery in order to reduce the hernia and repair the abdominal wall 71 defect. A left sided inguinal skin incision was performed to access the inguinal canal. 72 During surgery it was confirmed that the hernia was direct and its content was found to 73 be an ischemic, yet not necrotic epiploic appendage arising from the sigmoid colon 74 [Figure 3]. After the appendage was pushed back into the abdomen and blood flow was 75 restored, the abdominal wall weakening was reinforced using synthetic mesh. 76 Postoperative recovery was smooth, the patient was discharged after 5 days and presented 77 no complications during follow up. 78 79 Written informed consent has been obtained from the patient to publish this paper. 80 81 Discussion 82 Inguinal hernias constitute quite a common condition, affecting approximately 27% of 83 men and 3% of women across the world, and are typically classified as either direct or 84 indirect, based on differences in anatomy.2 Usually, inguinal hernias are asymptomatic 85 and do not alarm the patient until a straining event, such as lifting weight, raises the 86 intraabdominal pressure, causing soft tissue to protrude through an anatomical defect.6 87 The lifetime risk of dangerous complications following such an event has been found to 88 be rather low, estimated around 1-3%.5 Nevertheless, an increased risk has been strongly 89 associated with indirect hernias, whereas the less prevalent direct hernias, are about three 90 times less likely to become complicated which can be attributed to their wider neck.4,8,9 91 https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5614933/ https://www.ncbi.nlm.nih.gov/books/NBK459309/ https://www.ncbi.nlm.nih.gov/books/NBK555972/ https://link.springer.com/article/10.1007/BF01207592 https://pubmed.ncbi.nlm.nih.gov/30031240/ https://pubmed.ncbi.nlm.nih.gov/15008294/ 4 Specifically, indirect hernias herniate through the internal inguinal ring, which has 92 narrow diameter, while direct hernias protrude through Hesselbach’s triangle, medial to 93 the inferior epigastric vessels.10 However, despite the fact, that the neck of the fascial 94 defect in direct hernias is initially wide and soft, studies have shown that it can become 95 fibrotic and inelastic over time, and the above may multiply the risk of incarceration.4 96 97 Regarding diagnosis, the physical examination that involves inspection and palpation, 98 usually suffices to confirm the presence of the inguinal hernia.11 Further diagnostic 99 investigation using imaging methods such as ultrasonography, computed tomography 100 (CT), magnetic resonance imaging (MRI) or herniography is required only in cases of 101 pain and/ or swelling that suggest the presence of a complication. Differentiating between 102 direct and indirect hernias during preoperative care is meaningless and is in fact quite 103 challenging to achieve clinically or even through imaging.12 Concerning differential 104 diagnosis, if the initial clinical presentation includes edema, then lymph node 105 enlargement, aneurysm, saphena varix, soft-tissue tumor, abscess or genital anomalies 106 (such as ectopic testis) must be excluded. In case of the presence of pain, then adductor 107 tendonitis, pubic osteitis and hip arthritis should be considered likely.7 108 109 Regarding recommended treatment, options depend on the severity of the patient’s 110 symptoms. Asymptomatic or mild symptoms cases, can be managed with the ‘watchful 111 waiting’ approach or a scheduled repair, while complicated hernias require emergency 112 surgical repair.7 Moreover, the surgical techniques include tissue, open mesh and laparo-113 endoscopic mesh repair techniques, with a mesh-based repair being strongly 114 recommended for the majority of cases.7 115 116 Epiploic appendages are located in the large bowel and can be found in inguinal hernia 117 sacs, though this incident is quite rare and few cases have been reported.13 These 118 appendages are outpouchings of fatty tissue, covered by serosa that project into the 119 peritoneal cavity and that are supplied by one or two small arteries. Due to the limited 120 arterial blood supply, along with their pedunculated structure that allows increased 121 movement, epiploic appendages are prone to torsion and ischemia or bleeding, which can 122 https://pubmed.ncbi.nlm.nih.gov/34322367/ https://link.springer.com/article/10.1007/BF01207592 https://pubmed.ncbi.nlm.nih.gov/26987468/ https://link.springer.com/article/10.1007/s10029-009-0529-7#citeas https://link.springer.com/article/10.1007/s10029-017-1668-x#citeas https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5809582/ https://link.springer.com/article/10.1007/s10029-017-1668-x#citeas https://www.hindawi.com/journals/cris/2013/890234/ 5 also be caused by the thrombosis of the central vein.14 Epiploic appendagitis is also 123 related to diverticulitis because of the local spread of inflammation. CT scans are the 124 preferred imaging method of diagnosing epiploic appendagitis, which when primary does 125 not necessarily require surgical intervention and can be treated with non-steroidal anti-126 inflammatory drugs. However, in cases where the appendages become incarcerated in an 127 irreducible inguinal hernia, an emergency surgery can be called for.15 128 129 Despite the unlikelihood of direct hernia complications, there have been a few 130 documented cases of strangulated direct hernias arising in various ways. One such case 131 involved a life threatening bowel perforation, secondary to ischemic necrosis, which 132 required emergent resection of the necrotic bowel.16 In addition, incarcerated direct 133 hernias have also been reported as the cause of acute bowel obstruction.8 Moreover, a 134 complicated direct inguinal hernia containing the urinary bladder has led to obstructive 135 uropathy presenting with severe acute kidney failure, requiring emergency surgery and 136 dialysis.17 Finally, we describe two cases very similar to ours, one of which concerns an 137 irreducible direct inguinal hernia that was found to contain inflamed and hypertrophic 138 epiploic appendices which had to be resected before the hernia could be repaired.18 The 139 second one is a case of an incarcerated inguinal hernia which during emergent surgical 140 hernia reduction and herniorraphy was revealed to contain not only epiploic appendices, 141 but also part of the sigmoid colon.19 142 143 Eventually, after searching for similar cases on international literature, we found few 144 relevant case reports of direct strangulated or incarcerated hernias and even fewer of 145 hernias containing epiploic appendices. Our main findings are summarized in Table 1. It 146 is important to mention that the majority of reported cases of epiploic appendages being 147 found in inguinal hernias concerned indirect hernias.15 Hence this case report is unique in 148 that it describes a direct hernia. 149 150 Conclusion 151 Strangulation and incarceration occur scarcely among direct inguinal hernias. General 152 surgeons usually do not repair asymptomatic direct hernias and choose to follow the 153 https://insightsimaging.springeropen.com/articles/10.1186/s13244-019-0715-9 https://www.cureus.com/articles/77749-epiploic-appendagitis-in-an-incarcerated-inguinal-hernia-a-case-report-and-literature-review https://pubmed.ncbi.nlm.nih.gov/33489592/ https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6076353/ https://pubmed.ncbi.nlm.nih.gov/32300033/ https://pubmed.ncbi.nlm.nih.gov/19547689/ https://pubmed.ncbi.nlm.nih.gov/35636214/ https://www.cureus.com/articles/77749-epiploic-appendagitis-in-an-incarcerated-inguinal-hernia-a-case-report-and-literature-review 6 ‘watch and wait approach’. However, the risk of complications increases significantly 154 with age and in the presence of certain concomitant diseases. Consequently, being aware 155 of the fact that elective surgery for groin hernia is known to be a low-risk procedure, 156 patients suspected for groin hernia, should be considered for hernia repair depending on 157 their age, sex and clinical presentation, in order to avoid severe complications. 158 159 Authors’ Contributions 160 EK, PM, AZ, AI, Span and DP managed the patient. SPap and PM performed the 161 investigation. SPap provided the required resources. SN and DP curated the data. SN and 162 DP supervised the work. SPap and EK drafted the initial manuscript. EK, AZ and DP 163 reviewed and edited the manuscript. All authors approved the final version of the 164 manuscript. 165 166 References 167 1. Berndsen MR, Gudbjartsson T, Berndsen FH. Laeknabladid. 2019;105(9):385-391. 168 doi:10.17992/lbl.2019.09.247. 169 2. Öberg S, Andresen K, Rosenberg J. Etiology of Inguinal Hernias: A Comprehensive 170 Review. Front Surg. 2017;4:52. 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Int J Surg Case Rep. 2022;95:107237. doi:10.1016/j.ijscr.2022.107237 219 220 221 Figure 1: Ultrasonography of left inguinal region, revealing hernia (white arrow) 222 223 224 Figure 2: Doppler ultrasonography revealing reduced blood flow of the hernia content. 225 226 9 227 Figure 3: Direct inguinal hernia containing an ischemic epiploic appendage (white 228 arrow); image taken during surgery. 229 230 Table 1: Examples of cases of complicated direct inguinal hernias. 231 Author Content Sherif Monib et al. 2020 58-year-old man with a direct strangulated hernia, complicated with a small bowel perforation. Jacob Levi et al. 2020 72-year-old man presenting with hematuria, urinary retention and severe acute kidney failure who was diagnosed with a direct incarcerated hernia containing the urinary bladder. Manmohan Kamat et al. 2018 83-year-old male with a direct obstructed hernia of sliding type containing congested loops of ileum as well as part of the urinary bladder. Mayank Jain et al. 2008 52-year-old man whose irreducible direct hernia contained inflamed epiploic appendices. Ahmadullah Danish 2022 65-year old man with an incarcerated inguinal hernia containing sigmoid colon and epiploic appendices. 232