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Gallstone ileus displaying the typical Rigler triad and an occult second ectopic stone

Gallstone ileus displaying the typical Rigler triad and an occult second ectopic stone Rationale: Gallstone ileus is a rare complication of cholecystolithiasis. It has a female predominance and can result in high mortality rates. Patient concerns: A 71-year-old woman complaining of recurrent vomiting and vague epigastralgia for > 2 weeks presented to our department. Diagnosis: Based on her physical examination, laboratory test results and radiographic findings (the typical Rigler’s triad), she was diagnosed with gallstone ileus caused by multiple ectopic stones. Interventions: After correction of electrolyte imbalances through parenteral nutrition and fluid management, the patient’s condition improved and she underwent enterolithotomy. A 5.3-cm stone located 40cm from the ligament of Treitz was extracted, and a second ectopic stone, not detected on any imaging modality, was found during laparotomy. Given her comorbidities and overall poor condition, cholecystectomy and fistula repair were not performed. Outcomes: The patient gradually recovered postoperatively. Lessons: Clinical symptoms including epigastralgia with nausea and vomiting, and abdominal CT findings of Rigler’s triad (ectopic gallstone, bowel obstruction, and pneumobilia) may lead to early diagnosis of gallstone ileus and enterolithotomy may be the preferred treatment for this condition in the elderly. Laparotomy should involve a systematic and meticulous search for the presence of additional ectopic enteric stones. Abbreviations: CRP = C-reactive protein, CT = computed tomography, ESR = erythrocyte sedimentation rate. Keywords: cholecystoenteric fistula, enterolithotomy, gallstone ileus, multiple stones, Rigler triad 1. Introduction Rigler triad, a pathognomonic radiographic finding is not routinely observed on plain x-ray or ultrasound but is identified Gallstone ileus is a rare complication caused by repeated episodes [4] on CT scan with much higher accuracy. Given the rare of cholecystitis, or rarely malignancy, with a predominance in occurrence of gallstone ileus, a clear consensus on the optimal [1,2] elderly patients with comorbidities. Poor appetite and [1,5] surgical intervention has not been reached. Despite the lack of recurrent vomiting caused by ileus can worsen existing poor consensus, careful and thorough examination of the entire small [2] health conditions in the elderly. In patients of advanced age bowel during laparotomy has been generally accepted as with associated comorbidities and a delayed presentation, the standard practice, as multiple stones can occur in 5% to 25% [1] mortality rate for gallstone ileus can be as high as 18%. Early [1,2] of cases of this disease. diagnosis followed by suitable and timely surgical intervention In this report, we present a case of surgically treated gallstone [1–3] are considered 2 predictors of improved prognosis. ileus with findings of the typical Rigler triad as seen on 3- dimensional reconstruction images. Moreover, after removal of the primary stone (5.3cm in diameter), an ectopic stone not Editor: Somchai Amornyotin. detected on any imaging examination was found during The authors report no conflicts of interest. laparotomy in the same patient. a b Department of Hepato-Bilio-Pancreatic Surgery, Department of Radiology and Nuclear Medicine, West China Hospital, Sichuan University, Chengdu, P.R. 2. Case report China. Correspondence: Bole Tian, Department of Hepato-Bilio-Pancreatic Surgery, A 71-year-old woman presented to the Hepato-Bilio-Pancreatic West China Hospital, Sichuan University, Chengdu, 610041, P.R. China Department complaining of recurrent attacks of vomiting bile- (e-mail: [email protected]). stained gastric contents with palpitations, weakness, difficulty in Copyright © 2017 the Author(s). Published by Wolters Kluwer Health, Inc. defecation, and loss of appetite. She had sought medical help at a This is an open access article distributed under the Creative Commons local hospital during the initial stage of this episode. Unfortu- Attribution-NoDerivatives License 4.0, which allows for redistribution, commercial nately, nonsurgical management did not relieve her symptoms and non-commercial, as long as it is passed along unchanged and in whole, with credit to the author. and her overall condition worsened. About 1 year before this episode, a gallbladder stone had been detected on ultrasound Medicine (2017) 96:45(e8541) following an episode of right upper quadrant pain. She had been Received: 25 June 2017 / Received in final form: 6 October 2017 / Accepted: 9 October 2017 diagnosed with type 2 diabetes mellitus for >20 years and had an oophorocystectomy 40 years prior. http://dx.doi.org/10.1097/MD.0000000000008541 1 Wang et al. Medicine (2017) 96:45 Medicine On physical examination she was afebrile (36.9°C), normo- we prescribed oral medical paraffin oil. However, after becoming tensive (92/60 mm Hg), and without signs of jaundice. aware of the size of the stone, conservative treatment was Abdominal examination revealed remarkable abdominal disten- abandoned and more aggressive treatments were considered. We tion with sensitivity in the periumbilical area. Murphy sign was did not attempt endoscopic management of the stone that was in negative. Laboratory test results were as follows: complete blood the intestinal lumen. Ultimately, after improvement in her general 12 1 count revealed anemia (red blood cell count: 2.9810 L , condition, the patient underwent enterolithotomy. During this 12 1 reference range: 4.3–5.8 10 L ; hemoglobin 87g/L, refer- procedure, we removed a 5.3-cm ectopic stone (Fig. 4) that was ence range 130–175g/L); blood biochemical studies revealed located 40cm from the ligament of Treitz in the proximal jejunal slightly decreased total bilirubin (4.6mmol/L, reference range lumen. Interestingly, while inspecting the entire length of the 5.0–28mmol/L), significantly decreased albumin (19.2g/L, refer- small and large intestines, we found and extracted another small ence range 40–55g/L), and decreased calcium (1.69mmol/L, stone measuring 2.0cm in diameter (Fig. 4) in the distal ileal reference range 2.1–2.7mmol/L). Inflammatory factors including lumen. This stone had not previously been detected on any leukocyte count, erythrocyte sedimentation rate (ESR), and C- imaging modalities. Given the severe adhesions in the abdominal reactive protein (CRP) were normal. cavity, the comorbidities, and the poor overall condition of this Abdominal sonography revealed a slightly thickened gallblad- patient, cholecystectomy and fistula repair were not performed. der wall with air in the gallbladder. The plain abdominal film Despite development of a severe infection in the surgical incision depicted air-fluid levels and dilatation of the loops of the small and persistently poor overall health, the patient gradually bowel (Fig. 1, black arrows). To identify the obstructive cause, recovered and was discharged 22 days postoperatively. abdominal computed tomography (CT) was performed with a This case report was approved by the Ethics Committee of helical unit (slice thickness 5 mm, reconstruction interval 2 mm, West China Hospital of Sichuan University, Chengdu, China, Somatom Definition Flash, Siemens Healthcare, Forcheim, and written informed consent was obtained. Germany). Transverse, non-enhanced CT images revealed a round intraluminal lesion measuring 3.4  3.8cm with central 3. Discussion hypoattenuation in the jejunum (Fig. 2A). The 3-dimensional reconstruction images revealed the typical Rigler triad of ectopic Gallstone ileus, a rare complication of cholecystolithiasis gallstone (Fig. 2), bowel obstruction (Fig. 2B, white arrow), and occurring in <1% of patients, is the cause of 1% to 4% of [1–3] pneumobilia (Fig. 3). Coronal abdominal CT images also cases of small bowel obstruction. As a pathogenesis of demonstrated a cholecystoduodenal fistula between the gallblad- gallstone ileus, cholecystoenteric fistula caused by recurrent der and the descending duodenum (Fig. 3B, white arrow). These cholecystitis is identified most commonly in elderly women with [1,2] radiographic findings, particularly the abdominal CT images comorbidities. Gallstones pass through fistulas into the revealing the typical Rigler triad, strongly supported the duodenum, stomach, colon, or intestine, and most of the them [2,3] diagnosis of gallstone ileus and cholecystoenteric fistula. are excreted. Ectopic stones obstructing the intestinal lumen The initial management for this elderly woman was to improve can lead to ileus. The atypical presentation, along with the low her poor general condition and correct her electrolyte imbalances incidence of the disease in elderly patients, decreased doctors’ [1,2] through parenteral nutrition and fluid management. In addition vigilance upon first presentation of these patients. Conse- to the treatment aimed at improving her overall condition, we quently, there was a delay in correctly diagnosing these patients’ illness, leading to a worsening general condition. Although expected that the stone could pass through the ileocecal valve if advances in anesthesia and perioperative management can improve patients’ prognoses dramatically, the mortality and [1–3] morbidity remain considerable. To the best of our knowl- edge, the presence of the typical Rigler triad visualized on ultrasound and plain x-ray is only obvious in a small number of cases of this disease (11.11% and 14.81%, respectively). Additionally, the incidence of a second, or multiple additional, ectopic stones was as low as 5% for this relatively rare disease. In this report, we presented the typical Rigler triad based on 3- dimensional reconstruction CT images. We also noted the presence of a second ectopic stone that was not detected on any imaging modality in the same patient. Abdominal pain and vomiting are constant but nonspecific features for gallstone ileus, and >50% of patients present with features of intestinal obstruction. Other nonspecific signs include dehydration, upper gastrointestinal hemorrhage, abdominal [1–3] tenderness, and pyrexia. In summary, the clinical manifesta- tion of gallstone ileus is variable and nonspecific and attention should be paid to any history of recurrent episodes of acute cholecystitis, which occurred in approximately half of these [1,2] cases. In this case, the patient’s main clinical manifestation was recurrent vomiting without obvious abdominal pain. Imaging studies play a vital part in the early diagnosis of gallstone ileus. Rigler triad, described as signs of partial or Figure 1. Plain abdominal film showing air-fluid levels and small bowel loops complete intestinal obstruction, air or contrast medium in the dilatation (black arrows). biliary tree, and ectopic gallstone is the specific imaging finding 2 Wang et al. Medicine (2017) 96:45 www.md-journal.com Figure 2. (A, B, and C) The 3-dimensional unenhanced abdominal reconstruction CT depicting a round intraluminal lesion with central hypoattenuation in proximal jejunum and distended duodenum and small bowel loops (B, white arrow). Figure 3. (A, B, and C) The 3-dimensional unenhanced abdominal reconstruction CT revealing pneumobilia and cholecystoduodenal fistula (B, white arrow). for this disease, and can be detected on x-ray, ultrasound, and [4,6] CT. Unfortunately, only 13.81% of gallstone ileus patients [4] present with the typical Rigler triad on plain x-ray. Ultrasound is useful in detecting cholecystitis and pneumobilia, but not valuable in diagnosing the intestinal situation or the location of [4] an ectopic gallstone. Presently, CT has been proven to be the most valuable diagnostic modality for gallstone ileus, having optimal sensitivity, specificity, and accuracy (93%, 100%, and [7] 99%, respectively). In addition to the detection of Rigler triad, CT can also provide vital information such as fistula location, or severe inflammation of the gallbladder and its surrounding tissues. This type of information is useful for early diagnosis and [4,7] optimal therapies. MRI could be used for confirmation of [4] findings before treatment. Unfortunately, some smaller and less [4,7] calcified stones may still be missed by this imaging format, as described in this case. The debate over which surgical procedure should be undertaken has lasted for years due to the low incidence of [1,8–10] this disease. The primary conflicts involve the decision whether to perform cholecystoenteric fistula repair during the [8,9] initial procedure. A review of 1001 cases concluded that simple enterolithotomy was both safe and effective in managing a [2] patient with gallstone ileus. A review of recent cases of patients Figure 4. Macroscopic view of these stones. with gallstone ileus who underwent surgical treatment concluded that even after adjusting for patient and hospital factors, 3 Wang et al. Medicine (2017) 96:45 Medicine compared with simple enterolithotomy, fistula repair performed test in early diagnosis, should be performed in any elderly patient as the initial procedure was associated not only with higher complaining of abdominal pain and recurrent vomiting with a mortality rates, but also with longer hospital stays and greater history of cholecystolithiasis. Actually, enterolithotomy was [1] total treatment expense. Despite being less efficient in detecting proven to be the most beneficial operation for patients in poor multiple ectopic stones, laparoscopic enterolithotomy was overall health, and laparotomy should involve a systematic and [8–10] considered as a safer option for high-risk patients. In our meticulous search for the presence of further enteric stones. case, although we solely performed enterolithotomy, the patient still recovered slowly due to a severe infection of the abdominal References incision and hospitalization for 22 days postoperatively. Indeed, [1] Halabi WJ, Kang CY, Ketana N, et al. Surgery for gallstone ileus: a a more aggressive surgical procedure might cause even greater nationwide comparison of trends and outcomes. Ann Surg 2014;259: difficulties with postoperative recovery. 329–35. The presence of second or multiple stones may be as low as 5% [2] Reisner RM, Cohen JR. Gallstone ileus: a review of 1001 reported cases. [2] in patients with gallstone ileus. However, the inability to detect Am Surg 1994;60:441–6. these stones might result in extremely poor outcomes. Indeed, [3] Kirchmayr W, Mühlmann G, Zitt M, et al. Gallstone ileus: rare and still controversial. ANZ J Surg 2005;75:234–8. even 1 stone remaining in the colon could lead to a lethal [11] [4] Lassandro F, Gagliardi N, Scuderi M, et al. Gallstone ileus analysis of outcome. CT has been proven to be the most useful radiological findings in 27 patients. Eur J Radiol 2004;50:23–9. radiographic modality for diagnosis and preoperative evalua- [5] Rodríguez-Sanjuán JC, Casado F, Fernández MJ, et al. Cholecystectomy [4,6,7] tion. However, in the present case, after meticulous and fistula closure versus enterolithotomy alone in gallstone ileus. Br J examination of the small and large bowel, we found a second Surg 1997;84:634–7. [6] Maglinte DD, Reyes BL, Harmon BH, et al. Reliability and role of plain ectopic stone in the distal ileum that was not detected by any of film radiography and CT in the diagnosis of small-bowel obstruction. the preoperative imaging modalities, including 3-dimensional AJR Am J Roentgenol 1996;167:1451–5. reconstruction CT images. [7] Yu CY, Lin CC, Shyu RY, et al. Value of CT in the diagnosis and management of gallstone ileus. World J Gastroenterol 2005;11: 4. Conclusions 2142–7. [8] Fitzgerald JE, Fitzgerald LA, Maxwell-Armstrong CA, et al. Gallstone ileus is a rare but severe complication of cholecystitis. Recurrent gallstone ileus: time to change our surgery? J Dig Dis 2009;10: Nonspecific symptoms and a predilection in the elderly caused a 149–51. [9] Ravikumar R, Williams JG. The operative management of gallstone delay in diagnosis. The presence of ileus, electrolyte disturbances, ileus. Ann R Coll Surg Engl 2010;92:279–81. hypoproteinemia, anemia, delay in diagnosis, and comorbidities, [10] Shiwani MH, Ullah Q. Laparoscopic enterolithotomy is a valid option to all contributed to the patient’s poor overall condition. Early treat gallstone ileus. JSLS 2010;14:282–5. surgical intervention following early diagnosis of gallstone ileus is [11] Vaughan-Shaw PG, Talwar A. Gallstone ileus and fatal gallstone coleus: the importance of the second stone. BMJ Case Rep 2013;2013. of vital importance. CT, which was demonstrated to be a valuable http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Medicine Wolters Kluwer Health

Gallstone ileus displaying the typical Rigler triad and an occult second ectopic stone

Wang, Li; Dong, Ping; Zhang, Yi; Tian, Bole; Amornyotin., Somchai
Medicine , Volume 96 (45) – Nov 1, 2017
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Wolters Kluwer Health
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Copyright © 2017 the Author(s). Published by Wolters Kluwer Health, Inc.
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0025-7974
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Abstract

Rationale: Gallstone ileus is a rare complication of cholecystolithiasis. It has a female predominance and can result in high mortality rates. Patient concerns: A 71-year-old woman complaining of recurrent vomiting and vague epigastralgia for > 2 weeks presented to our department. Diagnosis: Based on her physical examination, laboratory test results and radiographic findings (the typical Rigler’s triad), she was diagnosed with gallstone ileus caused by multiple ectopic stones. Interventions: After correction of electrolyte imbalances through parenteral nutrition and fluid management, the patient’s condition improved and she underwent enterolithotomy. A 5.3-cm stone located 40cm from the ligament of Treitz was extracted, and a second ectopic stone, not detected on any imaging modality, was found during laparotomy. Given her comorbidities and overall poor condition, cholecystectomy and fistula repair were not performed. Outcomes: The patient gradually recovered postoperatively. Lessons: Clinical symptoms including epigastralgia with nausea and vomiting, and abdominal CT findings of Rigler’s triad (ectopic gallstone, bowel obstruction, and pneumobilia) may lead to early diagnosis of gallstone ileus and enterolithotomy may be the preferred treatment for this condition in the elderly. Laparotomy should involve a systematic and meticulous search for the presence of additional ectopic enteric stones. Abbreviations: CRP = C-reactive protein, CT = computed tomography, ESR = erythrocyte sedimentation rate. Keywords: cholecystoenteric fistula, enterolithotomy, gallstone ileus, multiple stones, Rigler triad 1. Introduction Rigler triad, a pathognomonic radiographic finding is not routinely observed on plain x-ray or ultrasound but is identified Gallstone ileus is a rare complication caused by repeated episodes [4] on CT scan with much higher accuracy. Given the rare of cholecystitis, or rarely malignancy, with a predominance in occurrence of gallstone ileus, a clear consensus on the optimal [1,2] elderly patients with comorbidities. Poor appetite and [1,5] surgical intervention has not been reached. Despite the lack of recurrent vomiting caused by ileus can worsen existing poor consensus, careful and thorough examination of the entire small [2] health conditions in the elderly. In patients of advanced age bowel during laparotomy has been generally accepted as with associated comorbidities and a delayed presentation, the standard practice, as multiple stones can occur in 5% to 25% [1] mortality rate for gallstone ileus can be as high as 18%. Early [1,2] of cases of this disease. diagnosis followed by suitable and timely surgical intervention In this report, we present a case of surgically treated gallstone [1–3] are considered 2 predictors of improved prognosis. ileus with findings of the typical Rigler triad as seen on 3- dimensional reconstruction images. Moreover, after removal of the primary stone (5.3cm in diameter), an ectopic stone not Editor: Somchai Amornyotin. detected on any imaging examination was found during The authors report no conflicts of interest. laparotomy in the same patient. a b Department of Hepato-Bilio-Pancreatic Surgery, Department of Radiology and Nuclear Medicine, West China Hospital, Sichuan University, Chengdu, P.R. 2. Case report China. Correspondence: Bole Tian, Department of Hepato-Bilio-Pancreatic Surgery, A 71-year-old woman presented to the Hepato-Bilio-Pancreatic West China Hospital, Sichuan University, Chengdu, 610041, P.R. China Department complaining of recurrent attacks of vomiting bile- (e-mail: [email protected]). stained gastric contents with palpitations, weakness, difficulty in Copyright © 2017 the Author(s). Published by Wolters Kluwer Health, Inc. defecation, and loss of appetite. She had sought medical help at a This is an open access article distributed under the Creative Commons local hospital during the initial stage of this episode. Unfortu- Attribution-NoDerivatives License 4.0, which allows for redistribution, commercial nately, nonsurgical management did not relieve her symptoms and non-commercial, as long as it is passed along unchanged and in whole, with credit to the author. and her overall condition worsened. About 1 year before this episode, a gallbladder stone had been detected on ultrasound Medicine (2017) 96:45(e8541) following an episode of right upper quadrant pain. She had been Received: 25 June 2017 / Received in final form: 6 October 2017 / Accepted: 9 October 2017 diagnosed with type 2 diabetes mellitus for >20 years and had an oophorocystectomy 40 years prior. http://dx.doi.org/10.1097/MD.0000000000008541 1 Wang et al. Medicine (2017) 96:45 Medicine On physical examination she was afebrile (36.9°C), normo- we prescribed oral medical paraffin oil. However, after becoming tensive (92/60 mm Hg), and without signs of jaundice. aware of the size of the stone, conservative treatment was Abdominal examination revealed remarkable abdominal disten- abandoned and more aggressive treatments were considered. We tion with sensitivity in the periumbilical area. Murphy sign was did not attempt endoscopic management of the stone that was in negative. Laboratory test results were as follows: complete blood the intestinal lumen. Ultimately, after improvement in her general 12 1 count revealed anemia (red blood cell count: 2.9810 L , condition, the patient underwent enterolithotomy. During this 12 1 reference range: 4.3–5.8 10 L ; hemoglobin 87g/L, refer- procedure, we removed a 5.3-cm ectopic stone (Fig. 4) that was ence range 130–175g/L); blood biochemical studies revealed located 40cm from the ligament of Treitz in the proximal jejunal slightly decreased total bilirubin (4.6mmol/L, reference range lumen. Interestingly, while inspecting the entire length of the 5.0–28mmol/L), significantly decreased albumin (19.2g/L, refer- small and large intestines, we found and extracted another small ence range 40–55g/L), and decreased calcium (1.69mmol/L, stone measuring 2.0cm in diameter (Fig. 4) in the distal ileal reference range 2.1–2.7mmol/L). Inflammatory factors including lumen. This stone had not previously been detected on any leukocyte count, erythrocyte sedimentation rate (ESR), and C- imaging modalities. Given the severe adhesions in the abdominal reactive protein (CRP) were normal. cavity, the comorbidities, and the poor overall condition of this Abdominal sonography revealed a slightly thickened gallblad- patient, cholecystectomy and fistula repair were not performed. der wall with air in the gallbladder. The plain abdominal film Despite development of a severe infection in the surgical incision depicted air-fluid levels and dilatation of the loops of the small and persistently poor overall health, the patient gradually bowel (Fig. 1, black arrows). To identify the obstructive cause, recovered and was discharged 22 days postoperatively. abdominal computed tomography (CT) was performed with a This case report was approved by the Ethics Committee of helical unit (slice thickness 5 mm, reconstruction interval 2 mm, West China Hospital of Sichuan University, Chengdu, China, Somatom Definition Flash, Siemens Healthcare, Forcheim, and written informed consent was obtained. Germany). Transverse, non-enhanced CT images revealed a round intraluminal lesion measuring 3.4  3.8cm with central 3. Discussion hypoattenuation in the jejunum (Fig. 2A). The 3-dimensional reconstruction images revealed the typical Rigler triad of ectopic Gallstone ileus, a rare complication of cholecystolithiasis gallstone (Fig. 2), bowel obstruction (Fig. 2B, white arrow), and occurring in <1% of patients, is the cause of 1% to 4% of [1–3] pneumobilia (Fig. 3). Coronal abdominal CT images also cases of small bowel obstruction. As a pathogenesis of demonstrated a cholecystoduodenal fistula between the gallblad- gallstone ileus, cholecystoenteric fistula caused by recurrent der and the descending duodenum (Fig. 3B, white arrow). These cholecystitis is identified most commonly in elderly women with [1,2] radiographic findings, particularly the abdominal CT images comorbidities. Gallstones pass through fistulas into the revealing the typical Rigler triad, strongly supported the duodenum, stomach, colon, or intestine, and most of the them [2,3] diagnosis of gallstone ileus and cholecystoenteric fistula. are excreted. Ectopic stones obstructing the intestinal lumen The initial management for this elderly woman was to improve can lead to ileus. The atypical presentation, along with the low her poor general condition and correct her electrolyte imbalances incidence of the disease in elderly patients, decreased doctors’ [1,2] through parenteral nutrition and fluid management. In addition vigilance upon first presentation of these patients. Conse- to the treatment aimed at improving her overall condition, we quently, there was a delay in correctly diagnosing these patients’ illness, leading to a worsening general condition. Although expected that the stone could pass through the ileocecal valve if advances in anesthesia and perioperative management can improve patients’ prognoses dramatically, the mortality and [1–3] morbidity remain considerable. To the best of our knowl- edge, the presence of the typical Rigler triad visualized on ultrasound and plain x-ray is only obvious in a small number of cases of this disease (11.11% and 14.81%, respectively). Additionally, the incidence of a second, or multiple additional, ectopic stones was as low as 5% for this relatively rare disease. In this report, we presented the typical Rigler triad based on 3- dimensional reconstruction CT images. We also noted the presence of a second ectopic stone that was not detected on any imaging modality in the same patient. Abdominal pain and vomiting are constant but nonspecific features for gallstone ileus, and >50% of patients present with features of intestinal obstruction. Other nonspecific signs include dehydration, upper gastrointestinal hemorrhage, abdominal [1–3] tenderness, and pyrexia. In summary, the clinical manifesta- tion of gallstone ileus is variable and nonspecific and attention should be paid to any history of recurrent episodes of acute cholecystitis, which occurred in approximately half of these [1,2] cases. In this case, the patient’s main clinical manifestation was recurrent vomiting without obvious abdominal pain. Imaging studies play a vital part in the early diagnosis of gallstone ileus. Rigler triad, described as signs of partial or Figure 1. Plain abdominal film showing air-fluid levels and small bowel loops complete intestinal obstruction, air or contrast medium in the dilatation (black arrows). biliary tree, and ectopic gallstone is the specific imaging finding 2 Wang et al. Medicine (2017) 96:45 www.md-journal.com Figure 2. (A, B, and C) The 3-dimensional unenhanced abdominal reconstruction CT depicting a round intraluminal lesion with central hypoattenuation in proximal jejunum and distended duodenum and small bowel loops (B, white arrow). Figure 3. (A, B, and C) The 3-dimensional unenhanced abdominal reconstruction CT revealing pneumobilia and cholecystoduodenal fistula (B, white arrow). for this disease, and can be detected on x-ray, ultrasound, and [4,6] CT. Unfortunately, only 13.81% of gallstone ileus patients [4] present with the typical Rigler triad on plain x-ray. Ultrasound is useful in detecting cholecystitis and pneumobilia, but not valuable in diagnosing the intestinal situation or the location of [4] an ectopic gallstone. Presently, CT has been proven to be the most valuable diagnostic modality for gallstone ileus, having optimal sensitivity, specificity, and accuracy (93%, 100%, and [7] 99%, respectively). In addition to the detection of Rigler triad, CT can also provide vital information such as fistula location, or severe inflammation of the gallbladder and its surrounding tissues. This type of information is useful for early diagnosis and [4,7] optimal therapies. MRI could be used for confirmation of [4] findings before treatment. Unfortunately, some smaller and less [4,7] calcified stones may still be missed by this imaging format, as described in this case. The debate over which surgical procedure should be undertaken has lasted for years due to the low incidence of [1,8–10] this disease. The primary conflicts involve the decision whether to perform cholecystoenteric fistula repair during the [8,9] initial procedure. A review of 1001 cases concluded that simple enterolithotomy was both safe and effective in managing a [2] patient with gallstone ileus. A review of recent cases of patients Figure 4. Macroscopic view of these stones. with gallstone ileus who underwent surgical treatment concluded that even after adjusting for patient and hospital factors, 3 Wang et al. Medicine (2017) 96:45 Medicine compared with simple enterolithotomy, fistula repair performed test in early diagnosis, should be performed in any elderly patient as the initial procedure was associated not only with higher complaining of abdominal pain and recurrent vomiting with a mortality rates, but also with longer hospital stays and greater history of cholecystolithiasis. Actually, enterolithotomy was [1] total treatment expense. Despite being less efficient in detecting proven to be the most beneficial operation for patients in poor multiple ectopic stones, laparoscopic enterolithotomy was overall health, and laparotomy should involve a systematic and [8–10] considered as a safer option for high-risk patients. In our meticulous search for the presence of further enteric stones. case, although we solely performed enterolithotomy, the patient still recovered slowly due to a severe infection of the abdominal References incision and hospitalization for 22 days postoperatively. Indeed, [1] Halabi WJ, Kang CY, Ketana N, et al. Surgery for gallstone ileus: a a more aggressive surgical procedure might cause even greater nationwide comparison of trends and outcomes. Ann Surg 2014;259: difficulties with postoperative recovery. 329–35. The presence of second or multiple stones may be as low as 5% [2] Reisner RM, Cohen JR. Gallstone ileus: a review of 1001 reported cases. [2] in patients with gallstone ileus. However, the inability to detect Am Surg 1994;60:441–6. these stones might result in extremely poor outcomes. Indeed, [3] Kirchmayr W, Mühlmann G, Zitt M, et al. Gallstone ileus: rare and still controversial. ANZ J Surg 2005;75:234–8. even 1 stone remaining in the colon could lead to a lethal [11] [4] Lassandro F, Gagliardi N, Scuderi M, et al. Gallstone ileus analysis of outcome. CT has been proven to be the most useful radiological findings in 27 patients. Eur J Radiol 2004;50:23–9. radiographic modality for diagnosis and preoperative evalua- [5] Rodríguez-Sanjuán JC, Casado F, Fernández MJ, et al. Cholecystectomy [4,6,7] tion. However, in the present case, after meticulous and fistula closure versus enterolithotomy alone in gallstone ileus. Br J examination of the small and large bowel, we found a second Surg 1997;84:634–7. [6] Maglinte DD, Reyes BL, Harmon BH, et al. Reliability and role of plain ectopic stone in the distal ileum that was not detected by any of film radiography and CT in the diagnosis of small-bowel obstruction. the preoperative imaging modalities, including 3-dimensional AJR Am J Roentgenol 1996;167:1451–5. reconstruction CT images. [7] Yu CY, Lin CC, Shyu RY, et al. Value of CT in the diagnosis and management of gallstone ileus. World J Gastroenterol 2005;11: 4. Conclusions 2142–7. [8] Fitzgerald JE, Fitzgerald LA, Maxwell-Armstrong CA, et al. Gallstone ileus is a rare but severe complication of cholecystitis. Recurrent gallstone ileus: time to change our surgery? J Dig Dis 2009;10: Nonspecific symptoms and a predilection in the elderly caused a 149–51. [9] Ravikumar R, Williams JG. The operative management of gallstone delay in diagnosis. The presence of ileus, electrolyte disturbances, ileus. Ann R Coll Surg Engl 2010;92:279–81. hypoproteinemia, anemia, delay in diagnosis, and comorbidities, [10] Shiwani MH, Ullah Q. Laparoscopic enterolithotomy is a valid option to all contributed to the patient’s poor overall condition. Early treat gallstone ileus. JSLS 2010;14:282–5. surgical intervention following early diagnosis of gallstone ileus is [11] Vaughan-Shaw PG, Talwar A. Gallstone ileus and fatal gallstone coleus: the importance of the second stone. BMJ Case Rep 2013;2013. of vital importance. CT, which was demonstrated to be a valuable

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