1086

Letters to the Editor

gluten sensitivity. On the other hand, some other authors have recently also shown the utility of detection of interferon-γ (IFN-γ) and IFN-γ-inducible protein-10 (IP-10) by ELISA or IFN-γ ELISpot for the detection of gluten-reactive T cells in HLA-DQ2.5 + associated CD (2). With this in mind, we aimed to study the importance of the detection of IP-10 levels in patients with a debut confirmed diagnosis of CD and to compare these with levels in children affected with food allergies. Patients with CD had a wide spectrum of gastrointestinal symptoms, isolated or not, including abdominal discomfort, nausea, vomiting, failure to thrive, constipation, or abdominal pain with iron deficiency. All patients of the CD group were positive for anti-transglutaminase antibodies (Tgt Abs), which were detected using antiIgA fluoroenzymoimmunoanalysis against human and recombinant Tgt (EliaTM Immunocap 250), and for anti-deaminated gliadine antibodies, which were detected using standard ELISA (Quanta LiteTM Gliadin IgA II). The allergy group consisted of subjects with food allergies and hypersensitivity I symptoms as described elsewhere (3). A healthy control group consisted of patients who had assisted in the study of drug allergies and who were negative for either anti-TgT or anti-Gliadin antibodies. Informed consent from patients and/or controls was obtained. The ethics committee approved the study (PI: 35/11 and PI: 12/14). IP-10 levels measured using Immunoassay (Procarta, Affymetrix) and read by flow cytometry (Luminex) were higher (statistically significant by the Kruskal– Wallis nonparametric method for three groups) in CD patients (n = 19; average of 212 pg/ml) compared with control patients (n = 10; 111 pg/ml). However, levels in CD patients were significantly lower than those in patients with a food allergy (n = 18; 627 pg/ml). As it has been reported that duodenal biopsies of pediatric CD patients have shown an inflammatory profile consisting of augmented IL8 and IL17 (4,5), we also measured these cytokines. In our sample, IL8 and IL17 were also clearly elevated, thus confirming the possibility of an immunotherapeutic intervention at this point. The American Journal of GASTROENTEROLOGY

It is known that IP-10 is secreted by several cell types such as monocytes, endothelial cells, and fibroblasts in response to IFN-γ. IP-10 has been attributed several roles, such as chemoattraction for monocytes/macrophages. It is possible that the IP-10 levels before intervention are predictive of a favorable response and have a supporting role in differential diagnosis of CD and food hypersensitivity. CONFLICT OF INTEREST Guarantor of the article: Victor Matheu, MD, PhD. Specific author contributions: Yvelise Barrios and Paloma Poza conceived the original idea and were mainly responsible for the work. Inmaculada Sanchez-Machín and Ruperto Gonzalez were responsible for control patients and responsible for the following up of patients with food allergy along with Paloma Poza. Andres Franco was responsible for the in vitro assays along with Y. Barrios. Honorio Armas was responsible for following up the patients with celiac disease. Victor Matheu was the senior researcher responsible for the work, had financial support, and wrote the final version of the manuscript. All authors approved the final version of the manuscript. Financial support: Supported by self-funding; Unidad de Alergología, Hospital Tórax. Potential competing interests: Yvelise Barrios, Paloma Poza, Andres Franco, Inmaculada Sanchez-Machín, Ruperto Gonzalez, Honorio Armas, and Victor Matheu declare that they do not have any conflict of interest. REFERENCES 1. Kabbani TA, Vanga RR, Leffler DA et al. Celiac disease or non-celiac gluten sensitivity? an approach to clinical differential diagnosis. Am J Gastroenterol 2014;109:741–6. 2. Ontiveros N, Tye-Din JA, Hardy MY et al. Ex vivo whole blood secretion of interferon-gamma (IFNgamma) and IFN-gamma-inducible protein-10 (IP-10) measured by ELISA are as sensitive as IFN-gamma ELISpot for the detection of gluten-reactive T cells in HLA-DQ2.5+ associated celiac disease. Clin Exp Immunol 2014;175: 305–15. 3. Glez PP, Franco YB, Matheu V. MIP-1alpha, MCP-1, and desensitization in anaphylaxis from cow’s milk. N Eng J Med 2012;367:282–4. 4. Eiro N, González-Reyes S, González L et al. Duodenal expression of Toll-like receptors and interleukins are increased in both children and adult celiac patients. Dig Dis Sci 2012;57:2278–85.

5. Bondar C, Araya RE, Guzman L et al. Role of CXCR3/CXCL10 axis in immune cell recruitment into the small intestine in celiac disease. PLoS One 2014;9:e89068. 1 Department of Immunology, Hospital Universitario de Canarias, Tenerife, Spain; 2Department of Allergy, Hospital del Tórax, Santa Cruz de Tenerife, Spain; 3Department of Pediatrics, Hospital Universitario de Canarias, San Cristóbal de La Laguna, Spain; 4Department of Medical Sciences, Lund University, Lund, Sweden; 5The first two authors are considered as shared first authors. Correspondence: Victor Matheu, MD, PhD, Department of Allergy, Hospital del Tórax, Ofra, Santa Cruz de Tenerife 38320, Spain. E-mail: [email protected]

A Fatal Case of Diffuse Enteritis After Colectomy for Ulcerative Colitis: A Case Report and Review of the Literature Joseph D. Feuerstein, MD1, Sveta Shah, MD1, Robert Najarian, MD2, Deborah Nagle, MD3 and Alan C. Moss, MD1 doi:10.1038/ajg.2014.118

To the Editor: Colectomy is considered curative in patients with refractory ulcerative colitis (UC). However, case reports exist of patients developing de novo jejunitis years after colectomy for UC. We present a case of a fatal severe enteritis that developed immediately following colectomy in a patient with UC, and a review of the literature. A 56-year-old woman was admitted to our center with hematemesis, high ileostomy output, and lethargy 11 days after a total proctocolectomy for refractory UC. She had a 12-year history of left-sided UC, but relapsed in the last 6 months despite mesalamine. During her preceding admission, enteric infections and cytomegalovirus (CMV) were excluded. She failed to respond to 5 days of IV steroids and salvage therapy with IV cyclosporine. She underwent a proctocolectomy with ileal j-pouch formation, and a diverting loop ileostomy, with a normal-appearing small VOLUME 109 | JULY 2014 www.amjgastro.com

Letters to the Editor

a

b

c

d

b

Figure 1. Endoscopic and pathology imaging. (a) Diverted ileal pouch with normal mucosa. (b) Jejunum with erythema, edema, congestion, friability, and ulceration. (c) Low power of representative section of small intestine demonstrating deep ulceration and associated inflammation involving the mucosa, submucosa, and superficial muscularis propria (hematoxylin and eosin (H&E) stain, ×40). (d) High-power view of the ‘‘ischemic pattern’’ of injury with regenerative basal crypts on the left and dilated atrophic basal crypts with abundant apoptotic debris forming crypt abscesses (H&E stain, ×400).

Table 1. Summary of cases of diffuse enteritis presenting within 1 month of colectomy Case 1 (current patient)

Case 2 (1)

Case 3a (2)

Case 4 (3)

Case 5 (3)

Case 6 (4)

Age

56

37

25

61

23

17

Time to readmission following colectomy admission

11 Days (26 days post-colectomy)

15 Days (28 days post-colectomy)

Unknown

1 Month

14 Days

Same admission (8 days post-colectomy)

Symptoms

Nausea, vomiting, lethargy, increased ostomy output and bright red blood in ileostomy bag and coffee ground emesis

Fevers, nausea, vomiting, epigastric pain, unable to tolerate oral intake, and melena

Fevers, bloody diarrhea, and abdominal pain

Severe abdominal pain and high output-ileostomy

Ileus

High ostomy output

Endoscopic/surgical findings

Diffuse mucosal friability, granularity, and deep linear ulcers of the mucosa

Diffuse mucosal friability, petechiae, and erosions of the mucosa

Diffuse mucosal edema, erosions and ulcerations

Diffuse erythema and small apthous ulcers

Diffuse inflammatory foci with multiple areas of perforation

Ulcerative colitis-like with active diffuse duodenitis and ileitis

Pathology

Severely active enteritis with ulceration and prominent epithelial cell apoptotic debris

Dense inflammatory infiltrate in lamina propria with extension into the submucosal layer, composed of small lymphocytes, plasma cells, histiocytes, monocytes, few neutrophils and occasional eosinophils.

Unknown

Marked apoptosis of epithelial cells, diffuse active chronic inflammatory infiltrate

Marked apoptosis of epithelial cells, lamina propria with lymphoplasmacellular infiltrate with eosinophilic and neutrophilic granulocytes

Diffuse mucosal inflammation with plasmacytosis of the lamina propria, neutrophilic cryptitis, crypt abscesses, and architectural distortion

Treatment at time of symptoms

Prednisone 10 mg and loperamide

Ceftriaxone, metronidazole, (treatment for e-coli positive culture) and total parenteral nutrition.

Unknown

None

None

High-dose steroids

Time to treatment after symptoms

18 Days

20 Days

Unknown

0 Day

0 Day surgery, 16 days for medical therapy

0 Day

© 2014 by the American College of Gastroenterology

The American Journal of GASTROENTEROLOGY

1087

1088

Letters to the Editor

Table 1. Continued Case 1 (current patient)

Case 2 (1)

Case 3a (2)

Case 4 (3)

Case 5 (3)

Case 6 (4)

Treatment after diagnosis

High-dose steroids methylprednisolone 20 mg three times daily

High-dose steroids 1 mg/kg

High-dose dexamethasone

Oral steroids

Surgery, calcineurin inhibitor, oral steroids, and parenteral nutrition

High-dose steroids continued

Outcome

Death 24 days after admission from multiorgan failure

Death 30 days after admission of multiorgan failure

Survives

Survives (multiple complications including recurrent bleeding and pancreatitis)

Survives (3-year follow-up)

Survives

a

Article in Japanese. Only abstract translated into English.

bowel. The removed colon demonstrated severe chronic active colitis, and the terminal ileum was normal. She was discharged on 20 mg of prednisone daily with a taper by 5 mg weekly. On readmission, she reported increasing ileostomy output, lethargy, and hematemesis over the prior week. She denied any use of nonsteroidal anti-inflammatory drugs, alcohol, or illicit drugs. Stool studies were negative for Clostridium difficile by polymerase chain reaction testing. Endoscopic examination of the duodenum, proximal ileum, and ileal pouch revealed diffuse granularity, deep linear ulcers, and friability of the mucosa (Figure 1). Only the stomach and diverted ileal limb were normal. Pathology was notable for acute inflammation, fibrinopurulent exudate, and granulation tissue. She was initially treated empirically with gancyclovir, without clinical improvement. Stains for CMV and herpes simplex virus were negative. She was commenced on IV steroids for presumed immune-mediated enteritis. Her bloody ileostomy output continued, because of early disseminated intravascular coagulation, and she became hypotensive. A computed tomography-angiogram showed findings consistent with diffuse small intestinal ischemia, with a patent superior mesenteric artery. Emergency surgery revealed diffuse small bowel necrosis. The majority of her small intestine was resected. Unfortunately, her clinical status rapidly deteriorated, and it was subsequently decided to provide comfort measures only. A postmortem examination noted signifiThe American Journal of GASTROENTEROLOGY

cant mucosal and submucosal ulceration with marked epithelial apoptosis and crypt dropout but no transmural inflammation (Figure 1). Additional findings were septic emboli in both lungs, septic necrosis of the spleen, acutely inflamed intrahepatic bile ducts, and a left-shifted myeloid dominant marrow. The presumptive diagnosis was diffuse immune-mediated enteritis leading to a systemic inflammatory response and multiorgan failure. A review of the literature revealed five other case reports with similar presentation within 1 month of colectomy for UC (1–4). In these five cases, the patients developed varying symptoms including fever, nausea, vomiting, diarrhea, epigastric pain, and gastrointestinal bleeding. In all cases an endoscopy and pouchoscopy showed diffuse enteritis (Table 1). The pathology in all cases lacked any features consistent with Crohn’s disease. In addition, infectious and viral etiologies were negative. In our case and one other case report (1), the patients were restarted on intravenous steroid only 2 weeks after readmission. In both cases the patients succumbed to multiorgan failure. In the other cases (2–4), three patients were already on steroids or were restarted earlier on with intravenous steroids or a calcineurin inhibitor. In the two fatalities we report, restart of immunosuppressive therapy occurred 2 weeks after the initial presentation (1). We postulate that, in the setting of severe UC, the massive T-cell and cytokine-mediated inflammatory response may continue post colectomy, with infiltration of the small bowel once the colon

is removed. Reinstitution of immunosuppression early in the successfully treated cases may have been the critical factor in determining the outcome. CONFLICT OF INTEREST

Guarantor of the article: Joseph D. Feuerstein, MD. Specific author contributions: Each author participated in the acquisition of data in this case and in the manuscript. Study design and manuscript preparation: Feuerstein, Shah, Moss, Nagle, and Najarian. Critical revisions: Feuerstein and Moss. Financial support: None. Potential competing interests: None. REFERENCES 1. Annese V, Caruso N, Bisceglia M et al. Fatal ulcerative panenteritis following colectomy in a patient with ulcerative colitis. Dig Dis Sci 1999;44:1189–95. 2. Nakajima M, Nakashima H, Kiyohara K et al. [Case with diffuse duodenitis and enteritis following total colectomy for ulcerative colitis]. Nihon Shokakibyo Gakkai Zasshi 2008;105:382–90. 3. Corporaal S, Karrenbeld A, van der Linde K et al. Diffuse enteritis after colectomy for ulcerative colitis: two case reports and review of the literature. Eur J Gastroenterol Hepatol 2009;21:710–5. 4. Valdez R, Appelman HD, Bronner MP et al. Diffuse duodenitis associated with ulcerative colitis. Am J Surg Pathol 2000;24:1407–13. 1

Department of Medicine and Division of Gastroenterology, Beth Israel Deaconess Medical Center, Harvard Medical School, Boston, Massachusetts, USA; 2Department of Pathology, Beth Israel Deaconess Medical Center, Harvard Medical School, Boston, Massachusetts, USA; 3 Department of Surgery and Division of Colo-Rectal Surgery, Beth Israel Deaconess Medical Center,

VOLUME 109 | JULY 2014 www.amjgastro.com

Letters to the Editor

Harvard Medical School, Boston, Massachusetts, USA. Correspondence: Joseph Feuerstein, MD, Department of Medicine and Division of Gastroenterology, Beth Israel Deaconess Medical Center, Harvard Medical School, 330 Brookline Avenue E/Dana 501, Boston, Massachusetts 02215, USA. E-mail: [email protected]

Terminal Ileitis Found Upon Imaging: Is It Always Crohn’s Disease? Teresa Pinto Pais, MD1, Sónia Fernandes, MD1, Carlos Fernandes, MD1, Iolanda Ribeiro, MD1 and João Carvalho, MD1 doi:10.1038/ajg.2014.114

To the Editor: Terminal ileitis (TI), defined as inflammation of the terminal ileum, is a common condition in clinical practice and is classically associated with Crohn’s disease (CD). However, a wide variety of diseases may be associated with ileitis (1,2). Diagnosis of the specific cause of ileitis is of paramount importance because misdiagnosis may result in critical delays or errors in patient management (3–5). Nonetheless, distinguishing between the various forms of ileitis remains a test of clinical acumen. Accurate diagnosis is suggested by a detailed history and physical examination, by laboratory testing, and by ileocolonoscopy and/or radiological evaluation. To study the different entities associated with TI in clinical practice, we reviewed all patients with confirmed imaging of TI on ultrasound admitted to our department over four consecutive years. An observational, descriptive, longitudinal study was performed, with a retrospective review of demographics, clinical presentation, laboratory data, treatment, and follow-up. Abdominal ultrasonography was performed in all patients and was complemented with computed tomography when necessary. Sonographic features compatible with TI included hypoechogenic mural thickening of the terminal ileum and hypoechoic enlarged mesenteric lymph nodes (6). The diagnosis of ileal CD was based on clinical, endoscopic, and histopathological data. Acid-fast bacilli smear and culture © 2014 by the American College of Gastroenterology

were performed in ileal biopsy specimens. An infectious etiology was presumed when there was clinical, analytical, and imaging remission after empirical antibiotic therapy, or when confirmed by isolation of pathogenic microorganisms in stool cultures. This study assessed 62 patients with a median age of 38.2 years (18–82), with a slight predominance of females (56.4%). The main form of presentation was abdominal pain (93.5%) and diarrhea (67.7%). All patients presented with ultrasonographic features of TI, which were corroborated by computed tomography in 62.9%. In this study, infectious etiology was confirmed in 62.9% (n = 39) of patients, CD was diagnosed in 32.2% (n = 20), and ileum cancer in 4.9% (ileal lymphoma—2, adenocarcinoma—1). Fecal pathogens were identified in 7 patients: Campylobacter—3, Yersinia—2, and Salmonella—2. No parasitic or tuberculosis infection was identified. On comparing the groups with confirmed CD and infectious ileitis, we found in the first group higher median values of serum inflammatory parameters (C-reactive protein: 9.8 vs. 7.4 mg/dl; leukocyte count: 16.650 vs. 11.170/μl), lower hemoglobin levels (10.45 vs. 13.5 g/dl, t-test, P=0.03), and a longer inpatient period (10 vs. 5 days). Patients diagnosed with CD maintain longterm follow-up in our outpatient clinic. In the current study, the diagnosis of CD of the ileum was confirmed histopathologically in 32.2% of patients with imaging of TI. We highlight that inflammation of the terminal ileum is not always CD, and infectious etiology is a common cause (62.9% in our study). The differential diagnosis of TI found on imaging, although sometimes difficult, is of critical importance to avoid further unnecessary diagnostic workup and inappropriate treatment. CONFLICT OF INTEREST Guarantor of the article: Teresa Pinto Pais, MD. Specific author contributions: Teresa Pinto Pais was involved in planning and conducting the study, collecting and interpreting data, and writing the paper. She has approved the final draft submitted. Sónia Fernandes was involved in planning the study, interpreting data, and drafting the

manuscript. She has approved the final draft submitted. Carlos Fernandes was involved in collecting and interpreting data. He has approved the final draft submitted. Iolanda Ribeiro was involved in collecting and interpreting data. She has approved the final draft submitted. João Carvalho was involved in planning the study, interpreting data, and drafting the manuscript. He has approved the final draft submitted. Financial support: None. Potential competing interests: None.

REFERENCES 1. Dilauro S, Crum-Cianflone NF. Ileitis: when it is not Crohn’s disease. Curr Gastroenterol Rep 2010;12:249–58. 2. Greaves ML, Pochapin M. Asymptomatic ileitis: past, present, and future. J Clin Gastroenterol 2006;40:281–5. 3. Lee YJ, Yang SK, Byeon JS et al. Analysis of colonoscopic findings in the differential diagnosis between intestinal tuberculosis and Crohn’s disease. Endoscopy 2006;38:592–7. 4. Jeong SH, Lee KJ, Kim YB et al. Diagnostic value of terminal ileum intubation during colonoscopy. J Gastroenterol Hepatol 2008;23:51–5. 5. Chang HS, Lee D, Kim JC et al. Isolated terminal ileal ulcerations in asymptomatic individuals: natural course and clinical significance. Gastrointest Endosc 2010;72:1226–32. 6. Ledermann HP, Börner N, Strunk H et al. Bowel wall thickening on transabdominal sonography. Am J Roentgenol 2000;174:107–17. 1

Department of Gastroenterology and Hepatology, Centro Hospitalar de Gaia/Espinho, Vila Nova de Gaia, Portugal. Correspondence: Teresa Pinto Pais, MD, Department of Gastroenterology and Hepatology, Centro Hospitalar de Gaia/Espinho, Rua Conceição Fernandes, 4434-502 Vila Nova de Gaia, Portugal. E-mail: [email protected]

Patient Satisfaction Does Not Correlate With Established Colonoscopy Quality Metrics Rena Yadlapati, MD1, Andrew Gawron, MD1,2 and Rajesh N. Keswani, MD1 doi:10.1038/ajg.2014.115

To the Editor: Quality metrics for colonoscopy are increasingly being measured and reported, as procedure quality correlates The American Journal of GASTROENTEROLOGY

1089

A fatal case of diffuse enteritis after colectomy for ulcerative colitis: a case report and review of the literature.

A fatal case of diffuse enteritis after colectomy for ulcerative colitis: a case report and review of the literature. - PDF Download Free
466KB Sizes 2 Downloads 4 Views