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Tropeano G, Di Grezia M, Puccioni C, Bianchi V, Pepe G, Fico V, Altieri G, Brisinda G. The spectrum of pneumatosis intestinalis in the adult. A surgical dilemma. World J Gastrointest Surg 2023; 15:553-565. [PMID: 37206077 PMCID: PMC10190725 DOI: 10.4240/wjgs.v15.i4.553] [Citation(s) in RCA: 4] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/07/2022] [Revised: 02/10/2023] [Accepted: 03/21/2023] [Indexed: 04/22/2023] Open
Abstract
Pneumatosis intestinalis (PI) is a striking radiological diagnosis. Formerly a rare diagnostic finding, it is becoming more frequently diagnosed due to the wider availability and improvement of computed tomography scan imaging. Once associated only with poor outcome, its clinical and prognostic significance nowadays has to be cross-referenced to the nature of the underlying condition. Multiple mechanisms of pathogenesis have been debated and multiple causes have been detected during the years. All this contributes to creating a broad range of clinical and radiological presentations. The management of patients presenting PI is related to the determining cause if it is identified. Otherwise, in particular if an association with portal venous gas and/or pneumoperitoneum is present, the eventual decision between surgery and non-operative management is challenging, even for stable patients, since this clinical condition is traditionally associated to intestinal ischemia and consequently to pending clinical collapse if not treated. Considering the wide variety of origin and outcomes, PI still remains for surgeons a demanding clinical entity. The manuscript is an updated narrative review and gives some suggestions that may help make the decisional process easier, identifying patients who can benefit from surgical intervention and those who can benefit from non-operative management avoiding unnecessary procedures.
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Affiliation(s)
- Giuseppe Tropeano
- Emergency Surgery and Trauma Center, Fondazione Policlinico Universitario A Gemelli IRCCS, Rome 00168, Italy
| | - Marta Di Grezia
- Emergency Surgery and Trauma Center, Fondazione Policlinico Universitario A Gemelli IRCCS, Rome 00168, Italy
| | - Caterina Puccioni
- Emergency Surgery and Trauma Center, Fondazione Policlinico Universitario A Gemelli IRCCS, Rome 00168, Italy
| | - Valentina Bianchi
- Emergency Surgery and Trauma Center, Fondazione Policlinico Universitario A Gemelli IRCCS, Rome 00168, Italy
| | - Gilda Pepe
- Emergency Surgery and Trauma Center, Fondazione Policlinico Universitario A Gemelli IRCCS, Rome 00168, Italy
| | - Valeria Fico
- Emergency Surgery and Trauma Center, Fondazione Policlinico Universitario A Gemelli IRCCS, Rome 00168, Italy
| | - Gaia Altieri
- Emergency Surgery and Trauma Center, Fondazione Policlinico Universitario A Gemelli IRCCS, Rome 00168, Italy
| | - Giuseppe Brisinda
- Emergency Surgery and Trauma Center, Fondazione Policlinico Universitario A Gemelli IRCCS, Rome 00168, Italy
- Department of Surgery, Università Cattolica del Sacro Cuore, Rome 00168, Italy
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Martín-Soberón MC, Ruiz S, De Velasco G, Yarza R, Carretero A, Castellano D, Sepúlveda-Sánchez JM. Pneumatosis intestinalis in a radioactive iodine-refractory metastasic thyroid papillary carcinoma with BRAF V600E mutation treated with dabrafenib-trametinib: a case report. J Med Case Rep 2021; 15:109. [PMID: 33653337 PMCID: PMC7927265 DOI: 10.1186/s13256-020-02581-9] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/10/2019] [Accepted: 11/11/2020] [Indexed: 11/10/2022] Open
Abstract
BACKGROUND Pneumatosis intestinalis (PI) is a rare entity which refers to the presence of gas within the wall of the small bowel or colon which is a radiographic sign. The etiology and clinical presentation are variable. Patients with PI may present either with chronic mild non-specific symptoms or with acute abdominal pain with peritonitis. Some cases of intestinal pneumatosis have been reported as adverse events of new oncological treatments such as targeted therapies that are widely used in multiple tumors. CASE PRESENTATION A 59-year-old caucasian female with radioactive iodine-refractory metastatic thyroid papillary carcinoma with BRAFV600E mutation was treated with dabrafenib and trametinib as a compassionate use. After 4 months treatment, positron emission tomography-computed tomography (PET-CT) showed PI. At the time of diagnosis, the patient was asymptomatic without signs of peritonitis. The initial treatment was conservative and no specific treatment for PI was needed. Unfortunately, after dabrafenib-trametinib withdrawal, the patient developed tumor progression with significant clinical worsening. CONCLUSIONS This case report is, in our knowledge, the first description of PI in a patient treated with dabrafenib-trametinib. Conservative treatment is feasible if there are no abdominal symptoms.
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Affiliation(s)
- M C Martín-Soberón
- Medical Oncology Department, University Hospital 12 de Octubre, Madrid, Spain.
| | - S Ruiz
- Nuclear Medicine Department, University Hospital 12 de Octubre, Madrid, Spain
| | - G De Velasco
- Medical Oncology Department, University Hospital 12 de Octubre, Madrid, Spain
| | - R Yarza
- Medical Oncology Department, University Hospital 12 de Octubre, Madrid, Spain
| | - A Carretero
- Medical Oncology Department, University Hospital 12 de Octubre, Madrid, Spain
| | - D Castellano
- Medical Oncology Department, University Hospital 12 de Octubre, Madrid, Spain
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Dhadlie S, Mehanna D, McCourtney J. Pneumatosis intestinalis a trap for the unwary: Case series and literature review. Int J Surg Case Rep 2018; 53:214-217. [PMID: 30428434 PMCID: PMC6232619 DOI: 10.1016/j.ijscr.2018.10.079] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/14/2018] [Revised: 10/22/2018] [Accepted: 10/25/2018] [Indexed: 11/06/2022] Open
Abstract
Pneumatosis Intestinalis is a rare condition that may manifest with a wide range of symptoms from mild abdominal pain to acute peritonitis. In general, surgery is only indicated in symptomatic patients where medical therapy has failed and in patients presenting acutely unwell with a surgical abdomen. Treatment is generally medical and even with radiological evidence of perforation laparotomy may not be indicated if the patient is clinically well. Treatment with oxygen or hyperbaric oxygen has been shown to be effective. Introduction Pneumatosis Intestinalis (PI) can present with a broad range of presentations from chronic and non-specific to acute and life threatening. It is paradoxically one of the few conditions where a pneumoperitoneum found in the diagnostic workup is not necessarily an indication for laparotomy. Presentation of cases The first case is of a 75 year old gentlemen who attended the emergency department after several weeks of worsening abdominal pain and weight loss. At laparotomy, two segments of nodular, abnormal-looking small bowel were identified with bubbles of air seen in the small bowel mesentery. The second case is of an 86 year old man with of a 5-month history of abdominal pain, weight loss, nausea and diarrhoea. A subsequent endoscopy revealed diffuse gastritis with a small antral gastric ulcer with a small amount of blood. The CT scan demonstrated free air within the bowel wall, with a follow up scan performed 3 weeks later revealing an increase in the amount of free air. Discussion PI is a rare condition characterised by the presence of subserosal and submucosal gas filled cysts occurring anywhere in the gastrointestinal tract from the oesophagus to the anus. Numerous conditions have been associated with PI including bowel obstruction, infections, ischemia and there have been reported cases suggesting as association with Crohn’s disease. Conclusion PI is a condition that may manifest with a wide range of symptoms from mild abdominal pain to acute peritonitis. Its treatment is generally medical and even with radiological evidence of perforation laparotomy may not be indicated if the patient is clinically well.
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Affiliation(s)
- Sunny Dhadlie
- Caboolture Hospital, 120 McKean Street, 4510, Queenland, Australia.
| | - Daniel Mehanna
- Caboolture Hospital, 120 McKean Street, 4510, Queenland, Australia.
| | - James McCourtney
- Royal Alexandra Hospital, Corsebar Rd, Paisely, PA2 9PN, Scotland, UK.
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Kim I, Kim JI, Kwon EJ, Jang JH, Jung SM, Seong HJ, Cheung DY, Park SH. [Clinical Course of Three Cases of Pneumatosis Intestinalis]. THE KOREAN JOURNAL OF GASTROENTEROLOGY 2016; 67:262-266. [PMID: 27206438 DOI: 10.4166/kjg.2016.67.5.262] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/03/2022]
Abstract
Pneumatosis cystoides intestinalis (PCI) is a rare condition characterized by multiple gas-filled cysts of varying size in the wall of gastrointestinal tract. PCI may idiopathic or secondary to various disorders. The etiology and pathogenesis of PCI are unclear. Treatment is usually conservative, and includes oxygen and antibiotics therapy. Surgery is reserved for cases of suspected inconvertible intestinal obstruction or perforation. Eleven patients who were diagnosed with PI between 2005 and 2015 were reviewed. We report three cases of PCI and describe causes and complications. The most important point in the treatment of PCI is to determine whether the patient needs surgery. Conservative care should be considered first if the patient is stable. If any complication is observed, such as ischemia in the intestine, surgery is needed. It is important to choose the best treatment based on prognostic factors and CT findings.
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Affiliation(s)
- Il Kim
- Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea
| | - Jin Il Kim
- Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea
| | - Eui Jong Kwon
- Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea
| | - Jae Hyuk Jang
- Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea
| | - Sung Min Jung
- Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea
| | - Hyeon Jin Seong
- Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea
| | - Dae Young Cheung
- Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea
| | - Soo Hern Park
- Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea
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Neesse A, Nimphius W, Schoppet M, Gress TM. Abdominal pain following percutaneous mitral valve repair (MitraClip). Pneumatosis intestinalis (PI) of the ascending colon. Gut 2015; 64:458, 494. [PMID: 25360035 DOI: 10.1136/gutjnl-2014-308495] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/19/2022]
Affiliation(s)
- Albrecht Neesse
- Department of Gastroenterology II, University Medicine Goettingen, Goettingen, Germany
| | - Wilhelm Nimphius
- Department of Pathology, University Hospital Giessen und Marburg GmbH, Standort Marburg, Marburg, Germany
| | - Michael Schoppet
- Department of Cardiology, University Hospital Giessen und Marburg GmbH, Standort Marburg, Marburg, Germany
| | - Thomas M Gress
- Department of Gastroenterology, Endocrinology, Infectiology and Metabolism, University Hospital Giessen und Marburg GmbH, Standort Marburg, Marburg, Germany
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Wayne E, Ough M, Wu A, Liao J, Andresen KJ, Kuehn D, Wilkinson N. Management algorithm for pneumatosis intestinalis and portal venous gas: treatment and outcome of 88 consecutive cases. J Gastrointest Surg 2010; 14:437-48. [PMID: 20077158 DOI: 10.1007/s11605-009-1143-9] [Citation(s) in RCA: 106] [Impact Index Per Article: 7.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/23/2009] [Accepted: 12/14/2009] [Indexed: 01/31/2023]
Abstract
BACKGROUND Pneumatosis intestinalis (PI) and portal venous gas (PVG) historically mandated laparotomy due to the high mortality rate associated with mesenteric ischemia. Computed tomography (CT) can identify PI/PVG in patients with ischemic emergencies and benign idiopathic conditions. METHODS A consecutive series of patients with PI or PVG was reviewed from a single institution over 5 years. Eighty-eight cases of PI/PVG were studied: 74 initial patients (year 1-4) were used to generate a treatment algorithm and fourteen additional cases were used to test the algorithm. RESULTS PI and PVG were associated with three major clinical subgroups: mechanical causes (n=29), acute mesenteric ischemia (n=29), and benign idiopathic (n=26); four were unclassifiable. Patients with acute mesenteric ischemia were associated with abdominal pain (p=0.01), elevated lactate (>or=3.0 mg/dL; p=0.006), small bowel PI (p=0.04), and calculated vascular disease score (p<0.0005). The three subgroups could be distinguished using the generated algorithm with a sensitivity of 89%, specificity of 100%, and positive predictive value of 100%. CONCLUSIONS With greater sensitivity of modern CT scans, PI and PVG are being detected in patients with a wide range of surgical and non-surgical conditions. This clinical algorithm can identify subgroups to direct surgical intervention for acute ischemic insults and prevent non-therapeutic laparotomies for benign idiopathic PI and PVG.
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Affiliation(s)
- Erik Wayne
- Department of Surgery, University of Louisville, Louisville, KY 40202, USA
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Management algorithm for pneumatosis intestinalis and portal venous gas: treatment and outcome of 88 consecutive cases. J Gastrointest Surg 2010. [PMID: 20077158 DOI: 10.1007/s11605–009–1143–9] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 08/10/2023]
Abstract
BACKGROUND Pneumatosis intestinalis (PI) and portal venous gas (PVG) historically mandated laparotomy due to the high mortality rate associated with mesenteric ischemia. Computed tomography (CT) can identify PI/PVG in patients with ischemic emergencies and benign idiopathic conditions. METHODS A consecutive series of patients with PI or PVG was reviewed from a single institution over 5 years. Eighty-eight cases of PI/PVG were studied: 74 initial patients (year 1-4) were used to generate a treatment algorithm and fourteen additional cases were used to test the algorithm. RESULTS PI and PVG were associated with three major clinical subgroups: mechanical causes (n=29), acute mesenteric ischemia (n=29), and benign idiopathic (n=26); four were unclassifiable. Patients with acute mesenteric ischemia were associated with abdominal pain (p=0.01), elevated lactate (>or=3.0 mg/dL; p=0.006), small bowel PI (p=0.04), and calculated vascular disease score (p<0.0005). The three subgroups could be distinguished using the generated algorithm with a sensitivity of 89%, specificity of 100%, and positive predictive value of 100%. CONCLUSIONS With greater sensitivity of modern CT scans, PI and PVG are being detected in patients with a wide range of surgical and non-surgical conditions. This clinical algorithm can identify subgroups to direct surgical intervention for acute ischemic insults and prevent non-therapeutic laparotomies for benign idiopathic PI and PVG.
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Min SY, Kim JI, Choi PW, Park JH, Heo TG, Lee MS, Kim CN, Chang SH, Kim HS. Clinical Review of Pneumatosis Intestinalis. JOURNAL OF THE KOREAN SURGICAL SOCIETY 2010. [DOI: 10.4174/jkss.2010.79.6.467] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
Affiliation(s)
- Sun Young Min
- Department of Surgery, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Korea
| | - Jae Il Kim
- Department of Surgery, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Korea
| | - Pyong Wha Choi
- Department of Surgery, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Korea
| | - Je Hoon Park
- Department of Surgery, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Korea
| | - Tae Gil Heo
- Department of Surgery, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Korea
| | - Myung Soo Lee
- Department of Surgery, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Korea
| | - Chul Nam Kim
- Department of Surgery, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Korea
| | - Surk Hyo Chang
- Department of Surgery, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Korea
| | - Han Seong Kim
- Department of Pathology, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Korea
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Silvera M, Finn B, Reynolds KM, Taylor DJ. Clostridium tertium as a cause of enteritis in cattle. Vet Rec 2003; 153:60. [PMID: 12885216 DOI: 10.1136/vr.153.2.60] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022]
Affiliation(s)
- M Silvera
- Department of Veterinary Pathology, University of Glasgow Veterinary School, Bearsden Road, Bearsden, Glasgow G61 1QH
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Hwang J, Reddy VS, Sharp KW. Pneumatosis Cystoides Intestinalis with Free Intraperitoneal Air: A Case Report. Am Surg 2003. [DOI: 10.1177/000313480306900414] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/29/2022]
Abstract
Pneumatosis cystoides intestinalis (PCI) is a rare disorder of the medical management and clinical outcome of which largely depend on the severity of its clinical manifestation. A limited number of cases have been reported in the literature although it is suggested that the true incidence of this disorder is higher than observed in clinical practice. This is the case of a 76-year-old woman with a past medical history of Crohn's disease found to have PCI. The patient initially complained of abdominal pain, distention, and weight loss. Chest and abdominal radiographs demonstrated free intraperitoneal air. CT scans revealed characteristic air-filled cysts in the intestinal wall, which established the diagnosis of PCI. Because the patient did not have an acute abdomen or findings requiring emergency laparotomy she was treated nonoperatively with supportive care. Her symptoms resolved gradually over several days. The patient was discharged home in stable condition tolerating a regular diet and was doing well at follow-up. The sole finding of free air with PCI does not mandate exploratory laparotomy.
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Affiliation(s)
- Joanna Hwang
- From the Division of General Surgery, Vanderbilt University Medical Center, Nashville, Tennessee
| | - V. Sreenath Reddy
- From the Division of General Surgery, Vanderbilt University Medical Center, Nashville, Tennessee
| | - Kenneth W. Sharp
- From the Division of General Surgery, Vanderbilt University Medical Center, Nashville, Tennessee
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Abstract
Pneumatosis cystoides intestinalis (PCI) is a rare disease usually occurring in association with a large variety of gastrointestinal (GI) and non GI conditions in the majority of cases, although idiopathic PCI is also known to occur. There are two theories regarding the development of these intramural gas cysts--the mechanical and bacterial theories. PCI usually runs a benign course, although fulminant PCI can be present both in infants and adults. The importance of this condition for the surgeon lies in its early recognition, in order to prevent unnecessary surgical intervention, especially when pneumoperitoneum without clinical evidence of peritonitis is encountered. Oxygen therapy has been shown to lead to regression of PCI, although recurrences have been reported. Elemental diets and antimicrobial agents have provided symptomatic relief in a few reported cases. The association of PCI with a wide variety of conditions leads us to conclude that PCI may not be a disease in itself, but a sequel to these varied conditions.
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Affiliation(s)
- M A Rogy
- Department of Surgery I, University of Vienna Medical School, Austria
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Sequeira W. Pneumatosis cystoides intestinalis in systemic sclerosis and other diseases. Semin Arthritis Rheum 1990; 19:269-77. [PMID: 2192457 DOI: 10.1016/0049-0172(90)90049-l] [Citation(s) in RCA: 52] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Affiliation(s)
- W Sequeira
- Division of Rheumatology, Cook County Hospital, Chicago, IL 60612
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Abstract
Pneumatosis cystoides intestinalis is a condition in which submucosal or subserosal gas cysts are found in the wall of the small or large bowel. Both fulminant and benign forms exist. Fulminant pneumatosis cystoides intestinalis is associated with an acute bacterial process, sepsis, and necrosis of the bowel, while benign pneumatosis cystoides intestinalis can be totally asymptomatic and observed as an incidental finding at laparotomy. Many different causes of pneumatosis cystoides intestinalis have been proposed, including mechanical and bacterial causes. A review of the literature is presented with emphasis on the etiology of pneumatosis cystoides intestinalis, which probably can be attributed to bacterial and mechanical factors. Diagnosis and different forms of therapy and their efficacy are also discussed.
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Galandiuk S, Fazio VW, Petras RE. Pneumatosis cystoides intestinalis in Crohn's disease. Report of two cases. Dis Colon Rectum 1985; 28:951-6. [PMID: 4064857 DOI: 10.1007/bf02554315] [Citation(s) in RCA: 51] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/08/2023]
Abstract
Two cases of pneumatosis cystoides intestinalis in patients with Crohn's disease are reported. These cases illustrate both benign and fulminant forms of pneumatosis cystoides intestinalis, and highlight the diagnostic and therapeutic dilemma of patients in whom pneumatosis cystoides intestinalis and Crohn's disease coexist. While therapy is seldom indicated in benign forms of pneumatosis cystoides intestinalis, mortality rates with fulminant pneumatosis cystoides intestinalis are high, regardless of the type of therapy.
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Abstract
Pneumatosis coli is an uncommon disease which is frequently misdiagnosed and inappropriately treated. The condition can usually be managed conservatively but complications requiring surgical treatment are occasionally encountered. We describe 4 patients with pneumatosis coli and associated volvulus of the sigmoid colon. Some of the factors that may have contributed to pneumocyst formation in these 4 patients are discussed with reference to recent advances in our understanding of the pathogenesis of pneumatosis coli.
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Abstract
Pneumatosis cystoides intestinalis (PCI) is an uncommon condition of unknown aetiology. Bacterial gas production may be an important aetiological factor, but experimental evidence in humans has been lacking. We have studied breath hydrogen excretion as an index of bacterial gas production in 12 patients with PCI and have shown that four out of five with demonstrable cysts produced unusually high levels of hydrogen while fasting. This abnormality has not been previously reported. One patient showed resolution of PCI after antibiotic treatment. These findings confirm the importance of bacterial gas production in the pathogenesis of PCI.
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Abstract
5 infants with no growth of bacteria on cultures of blood and peritoneal fluid recovered from necrotising enterocolitis after medical treatment alone. 12 infants with positive cultures required surgery. 5 of these 12, who did not harbour clostridia, had a mild clinical course and all 5 survived segmental bowel resection. The 7 infants who harboured clostridia had a more severe clinical course and 4 died. In 3 of 4 infants with Clostridium perfringens, the necrotising enterocolitis was fulminant, characterised by severe pneumatosis intestinalis, extensive gangrene, early intestinal perforation, and a fatal outcome.
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