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Surgical Approach in a Patient with Intestinal Malrotation Associated with Adult Hypertrophic Pyloric Stenosis

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Surgical Approach in a Patient with Intestinal Malrotation Associated with Adult Hypertrophic Pyloric Stenosis Archives of the Balkan Medical Union vol. 55, no. 3, pp. 483-488 Copyright © 2020 Balkan Medical Union September 2020 CASE REPORT SURGICAL APPROACH IN A PATIENT WITH INTESTINAL MALROTATION ASSOCIATED WITH ADULT HYPERTROPHIC PYLORIC STENOSIS Veselin IVANOV1, Ventsislav DIMOV2 , Tsonka LUKANOVA1, Ivan POPADIIN1, Nikola VLADOV1 1˝Department of Gastroenterology, Hepatopancreatic Surgery and Transplantology, Military Medical Academy, Sofia, Bulgaria 2 Department of Anaesthesiology and Intensive Care, Military Medical Academy, Sofia, Bulgaria Received 19 May 2020, Corrections received 29 June 2020, Accepted 24 July 2020 https://doi.org/10.31688/ABMU.2020.55.3.14 ABSTRACT RÉSUMÉ Introduction. Intestinal malrotation and hypertroph- Approche chirurgicale chez un patient souffrant ic pyloric stenosis (HPS) are both rare diseases, usually d’une malrotation intestinale associée à une sténose diagnosed during infancy. In the medical literature pylorique hypertrophique de l’adulte there are only a few reports about infants who suffer from a combination of these diseases. The simulta- Introduction. La malrotation intestinale et la sténose neous occurrence of both diseases in adult patients pylorique hypertrophique (SPH) sont des maladies is extremely rare. However, in this case, the surgical rares, et toutes les deux sont normalement diagnosti- approach has to be a combination of different tech- quées, dans la plupart des cas, pendant la petite en- niques, aiming at treating each one of the diseases fance. On peut trouver dans la littérature un nombre separately. restreint des rapports d’enfants qui présentent une Case presentation. A 37-years-old female patient combinaison des signes de ces deux maladies. Leur who presented for upper gastrointestinal symptoms apparition simultanée chez un[e] patient[e] adulte est was diagnosed with adult hypertrophic pyloric stenosis extrêmement rare. Si tel est le cas quand même, l’ap- (AHPS), intestinal malrotation and consequent dilated proche chirurgicale devrait donc combiner des tech- stomach. The diagnosis was based on the history, clini- niques différentes qui visent un traitement différentiel cal findings, imaging examinations (X-ray, abdominal de chaque maladie. computed tomography), and gastroscopy. The surgi- Rapport du cas. Une patiente de 37 ans qui s’est cal approach included division of Ladd’s band and présentée pour des symptômes gastro-intestinaux su- Jaboulay pyloroplasty. périeurs, diagnostiquée avec une sténose pylorique Conclusions. The simultaneous occurrence of intesti- hypertrophique adulte (SPHA), une malrotation in- nal malrotation associated with AHPS is an extremely testinale, et une dilatation de l’estomac conséquente. Address for correspondence: Ventsislav DIMOV Department of Anaesthesiology and Intensive Care, Military Medical Academy, Sofia, Bulgaria Address: Sveti Georgi Sofiyski Str. 3, Sofia, Bulgaria Email: [email protected]; Phone: +359 88 777 3202 Surgical approach in a patient with intestinal malrotation associated with adult hypertrophic… – IVANOV et al rare condition. Applying a combined surgical approach Le diagnostic a été mis sur la base de l’anamnèse, des may have good postoperative results. résultats cliniques, des examens d’imagerie (radiogra- phie, tomodensitométrie abdominale) et de la gastros- Keywords: intestinal malrotation, adult pyloric ste- copie. L’approche chirurgicale a inclus la division de la nosis, dilated stomach, Ladd’s band, Jaboulay pyloro- bande de Ladd et la pyloroplastie Jaboulay. plasty. Conclusions. Le cas d’une malrotation intestinale associée à SPHA est une maladie extrêmement rare. Abbreviations list: L’application d’une approche chirurgicale combinée a AHPS – adult hypertrophic pyloric stenosis bien démontré de très bons résultats postopératoires. HPS – hypertrophic pyloric stenosis CT – computed tomography Mots-clés: malrotation intestinale, sténose pylorique adulte, estomac dilaté, bande de Ladd, pyloroplastie Jaboulay. INTRODUCTION or as an acute condition with small bowel obstruc- tion5. Intestinal malrotation is a rare congenital dis- Adult hypertrophic pyloric stenosis (AHPS) is ease, being diagnosed in 0.2% of all births1. Most also a rare disease. It may be divided into 3 types: cases are diagnosed in the first year of life (75-85%)2. idiopathic, concomitant and late stage of infantile hy- Other cases are discovered in a later stage of life, as pertrophic pyloric stenosis6,7. The symptoms of AHPS an accidental finding or a symptomatic disease and and the chronic type of intestinal malrotation over- the rest of the cases remain undiagnosed. Adult in- lap to some extent. testinal malrotation is a very rare condition; its inci- In this article, we present a rare clinical case of dence has been reported to be between 0.0001 and an adult patient with intestinal malrotation associ- 0.19%3,4. The diagnosis of intestinal malrotation in ated with AHPS and its surgical treatment. adults is difficult, because of the lack of specific symp- toms and the low frequency in the population. The CASE REPORT symptoms in adult patients may present as a chronic condition, with intermittent intestinal obstruction or A 27-years-old woman presented with symp- vague abdominal complaints, for months and years, toms of epigastric and back pain, nausea, vomiting, Fig. 1. Abdominal CT scan. Pyloric stenosis associated with dilated stomach. 484 / vol. 55, no. 3 Archives of the Balkan Medical Union Fig. 2. Abdominal X-ray. Left-sided large intestine. the symptoms. The patient was referred to a surgical examination and a computed tomography (CT) im- aging with intravenous contrast was performed. CT scan has shown a dilated stomach, pyloric stenosis and intestinal malrotation (Fig. 1). The patient was admitted to the Military Medical Academy, Sofia, Bulgaria, for elective surgery one month later, in January 2020. Abdominal X-ray and gastroscopy were performed, in order to confirm the diagnosis. The X-ray has shown a left-sided large intestine (Fig. 2) and the gastroscopy has shown a dilated stomach due to pyloric stenosis, with inability to pass the gastroscope distally through the obstruc- tion (Fig. 3). An upper and middle midline laparotomy Fig. 3. Gastroscopy. Pyloric stenosis. was performed under general anaesthesia. Intra- operatively, the examination of the abdominal cavity has found (Fig. 4,5): decreased appetite, abdominal bloating and 10 kg a) Pyloric stenosis with inflammation and oedema. b) Duodenal obstruction due to band. weight loss in one month. The gastroscopy showed c) Dilated stomach. a duodenal ulcer (size 1/1 cm) on the anterior wall d) Intestinal malrotation (type 2A): of duodenum 1, stenosis in the same part of the duo- – Duodenum – no duodenal rotation, intraperi- denum and an erythema-exudative pangastritis. The toneal location. histopathology of the duodenal ulceration has exclud- – Left-sided large intestine. ed a neoplastic infiltration. Helicobacter pylori test – Ladd’s band. was negative. Conservative treatment was prescribed, The surgical technique consisted in a division with esomeprazole 40 mg and itopride hydrochloride of the Ladd’s band and Jaboulay pyloroplasty with 50 mg three times per day. Hygienic-dietetic regimen truncal vagotomy (Fig. 6). has been recommended. The postoperative evolution was good, without The patient was followed up by a gastroenterolo- any complications. The normal passage through the gist. Five months later, there was no improvement in gastrointestinal tract was restored. The patient was September 2020 / 485 Surgical approach in a patient with intestinal malrotation associated with adult hypertrophic… – IVANOV et al Fig. 4. Intraoperative photo of the intraperitoneal location Fig. 5. Intraoperative photo of the intraperitoneal location of duodenum. of the duodenum and the Ladd’s band. DISCUSSION Intestinal malrotation is a rare congenital dis- ease (0.2% of the new-born children) and it appears as a consequence of incomplete gut rotation and fixa- tion. It was first described in animals by Aristotle and in humans by Fabricii8. Intestinal rotation pri- marily involves the midgut. The rotation of intes- tinal development has been divided into 3 stages. Stage I occurs in week 5 to 10. It includes extrusion of the midgut into the extra-embryonic cavity, a 90° anti-clockwise rotation, and return of the midgut into the foetal abdomen. Stage II occurs in week 11 and involves further anti-clockwise rotation within the abdominal cavity, completing a 270° rotation. This rotation brings the duodenal „C“ loop behind the superior mesenteric artery with the ascending colon to the right, the transverse colon above, and descend- ing colon to the left. Stage III involves fusion and anchoring of the mesentery. The caecum descends, and the ascending and descending colon attach to the 9 Fig. 6. Intraoperative photo of the Jaboulay pyloroplasty posterior abdominal wall . The type of intestinal mal- (side-to-side gastroduodenostomy). rotation depends on the stage at which the embryonic rotation of the bowel has stopped (Table 1). The type of intestinal malrotation in our case is 2A. discharged in the 7th postoperatory day. The follow-up Intestinal malrotations are most frequently di- examination obtained one month after the hospital agnosed in infancy, 75-85% of all cases in the first discharge showed increased quality of life, lack of up- year of life1. Adults with intestinal malrotation could per gastrointestinal symptoms and a weight gain of be asymptomatic or symptomatic (acute or chronic 5 Kg. type)12. The
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