CASE HISTORY

The patient was a 13-year-old girl presenting with abdominal pain, weight loss, and bloody diarrhea. One month before diagnosis, she was treated with a course of azithromycin for pneumonia. Two weeks later, she developed crampy abdominal pain and bloody diarrhea, occurring 3 to 4 times per day. She developed fevers, increased fatigue, nausea, and vomiting. Initial evaluation at an outside emergency department (ED) revealed normal complete blood count and urinalysis. Clostridium difficile antigen/toxin as well as stool cultures were negative. Erythrocyte sedimentation rate (ESR) was elevated at 54 mm/hour. The patient was transferred to Seattle Children's Hospital. On examination, she was afebrile with normal vitals. She had diffuse abdominal tenderness on examination, but no peritoneal signs. Given a history of bloody diarrhea with negative stool studies, she underwent endoscopic evaluation, which revealed erythema, edema, and friability in the stomach, and duodenal bulb. Colonoscopy revealed moderate-to-severe erythema, edema in the entire colon, and ulceration in the proximal colon. The terminal ileum was also erythematous and edematous. Biopsies revealed moderate-to-severe chronic and mild-to-moderate active colitis with chronic active ileitis (Fig. 2). Given endoscopic findings in the ileum, an initial diagnosis of Crohn disease was given and patient was initiated on exclusive enteral nutritional (EEN) therapy. Patient was reclassified later as UC after review of biopsies, imaging, serology, and conferencing with proceduralist. The patient responded well with gradual resolution of abdominal pain and decreased stooling to 2 to 3 semi-formed nonbloody stools per day. C-reactive protein decreased from a maximum of 4.2 to 1.6 mg/dL before discharge from hospital. At follow-up after discharge, CRP completely normalized (Fig. 1).

Initial laboratory studies at Seattle Children's also revealed elevation of alkaline phosphatase (1378 IU/L; normal 130–560 IU/L) and GGT (851 IU/L; normal 5–55 IU/L) (Fig. 1). Magnetic Resonance Cholangiopancreatography (MRCP) during hospitalization showed common bile duct dilatation with regions demonstrating a beaded appearance as well as an abrupt transition at the distal common bile duct suggestive of possible stricture. There was evidence of mild dilatation and beading appearance of the intrahepatic bile duct as well. Further evaluation revealed an elevated immunoglobulin G titer with a normal IgG subclass, antimitochondrial antibody and anti-liver kidney microsomal antibody negativity, and serum carbohydrate antigen (SCA) 19-9 elevation of 906 U/mL (normal <55 U/mL). She was ASCA IgA- and IgG negative and ANCA positive (Table 1). She underwent percutaneous liver biopsy as well as endoscopic retrograde cholangiopancreatography with sphincterotomy and stricture dilatation. Biopsy results were consistent with biliary ductal obstruction and PSC (Fig. 2).

She was maintained on EEN for 10 weeks and then transitioned to the SCD. She remained asymptomatic and clinically well for over a year. She has 2 bowel movements per day, formed, nonbloody, nonmucousy. She has had good weight gain, good energy, no oral ulcerations, no joint pains, and no rashes. She has had complete normalization of her labs with normal ESR, C-reactive protein, haemoglobin, and albumin as well as her liver enzymes including GGT and alkaline phosphatase (Fig. 1). Initial calprotectin done 6 weeks after initial diagnosis was 1127 mg/kg (normal <163 mg/kg). This normalized at 14 (108 mg/kg) and 34 weeks (78 mg/kg). In addition, repeat SCA 19-9 as well as immunoglobulins normalized (Table 1). Repeat endoscopy/colonoscopy 1 year after diagnosis, revealed normal appearing mucosa in the stomach, duodenum, terminal ileum, and colon. Biopsies revealed mild chronic colitis with focal activity (Fig. 2). MRE results showed focal regions of minimal intrahepatic biliary duct prominence within hepatic segment IV.

DISCUSSION

PSC is generally a progressive disease, which ultimately leads to severe complications including cholestasis and hepatic failure. Hepatic transplant is the only curative therapy. Approximately 50% of symptomatic patients do not survive beyond 15 years from diagnosis unless transplanted. Currently, there is no effective medical therapy that alters PSC disease progression, although ursodeoxycholic acid and vancomycin have been associated with biochemical improvements in PSC (8).

The intestinal microbiota of patients with PSC is characterized by decreased microbial diversity, and a significant overrepresentation of Enterococcus, Fusobacterium, and Lactobacillus genera. This dysbiosis is present in patients with PSC with and without concomitant IBD and is distinct from IBD without PSC (9). As the relationship between PSC, IBD, and the intestinal microbiome is becoming better elucidated, therapeutic options aimed at manipulating the intestinal microbiome will likely play an important role in treatment. A prime example is vancomycin, which has been shown to have a beneficial effect in PSC (10). Although dietary therapy has not been rigorously studied in PSC, a disease without known therapy to prevent progression, we suggest that diet may play a critical role. It is known that immunosuppressive therapy can help control symptoms, but the effect on the intestinal microbiome from immunosuppression differs from the changes resulting from dietary therapies. Whereas immunosuppression may help control IBD-associated enteral inflammation, it may not address underlying triggers of pathogenesis such as the microbiota, dietary exposures, and the resultant metabolites produced. This case is the first to report clinical remission and laboratory normalization of PSC with dietary therapy. Though IBD activity may be a confounder, we suggest that both control of IBD activity and alterations in the intestinal microbiota may be within the pathway of effective therapy for PSC. Further prospective studies are merited and will help elucidate the role of diet in either primary or adjunctive therapy for PSC.

A 20-year-old female was diagnosed with ulcerative colitis (UC) at age 14 and primary sclerosing cholangitis (PSC) at age 16. The PSC was successfully treated with high doses of oral vancomycin; however, the UC was more difficult to manage. After many drug treatments failed to treat the UC, the patient began following the specific carbohydrate diet (SCD). This report documents fecal microbiome changes resulting from following the SCD for two weeks. The DNA extracted from fecal samples was subjected to 16S rRNA gene sequencing to quantify bacterial species abundance. Not only were substantial changes in the fecal bacterial composition detectable within two weeks, but all UC symptoms were also controlled as early as one week following the start of the diet. The patient's fecal microbiota was dramatically different from those of three healthy control subjects and showed remarkable loss of bacterial diversity in terms of species richness, evenness, and overall diversity measures. Other specific changes in bacterial composition included an increase in Enterobacteriaceae, including Escherichia and Enterobacter species. A two- to three-fold decrease was observed in the prevalence of the most dominant fecal bacterial species, Fusobacterium ulcerans, after two weeks on the SCD. Overall species diversity and evenness increased to levels near the controls, although species richness remained low. These findings provide information on the fecal bacteria from a patient with PSC and UC, following prolonged oral vancomycin treatment, and identifies a potentially specific microbial effect for the SCD.

GOAL:

To determine the effect of the specific carbohydrate diet (SCD) on active inflammatory bowel disease (IBD).

BACKGROUND:

IBD is a chronic idiopathic inflammatory intestinal disorder associated with fecal dysbiosis. Diet is a potential therapeutic option for IBD based on the hypothesis that changing the fecal dysbiosis could decrease intestinal inflammation.

STUDY:

Pediatric patients with mild to moderate IBD defined by pediatric Crohn's disease activity index (PCDAI 10-45) or pediatric ulcerative colitis activity index (PUCAI 10-65) were enrolled into a prospective study of the SCD. Patients started SCD with follow-up evaluations at 2, 4, 8, and 12 weeks. PCDAI/PUCAI, laboratory studies were assessed.

RESULTS:

Twelve patients, ages 10 to 17 years, were enrolled. Mean PCDAI decreased from 28.1±8.8 to 4.6±10.3 at 12 weeks. Mean PUCAI decreased from 28.3±23.1 to 6.7±11.6 at 12 weeks. Dietary therapy was ineffective for 2 patients while 2 individuals were unable to maintain the diet. Mean C-reactive protein decreased from 24.1±22.3 to 7.1±0.4 mg/L at 12 weeks in Seattle Cohort (nL<8.0 mg/L) and decreased from 20.7±10.9 to 4.8±4.5 mg/L at 12 weeks in Atlanta Cohort (nL<4.9 mg/L). Stool microbiome analysis showed a distinctive dysbiosis for each individual in most prediet microbiomes with significant changes in microbial composition after dietary change.

CONCLUSIONS:

SCD therapy in IBD is associated with clinical and laboratory improvements as well as concomitant changes in the fecal microbiome. Further prospective studies are required to fully assess the safety and efficacy of dietary therapy in patients with IBD.

Abstract

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Abstract

Here we report the treatment of a pediatric Crohn's disease patient with the Specific Carbohydrate Diet (SCD) and without medications. The SCD excludes most dairy products and complex carbohydrates to reduce bacterial growth. This is thought to decrease local inflammation and allow the gastrointestinal tract to heal. Some studies suggest that this diet can help control and improve symptoms of Crohn’s disease.

Purpose

The purpose of this paper was to investigate the impact of specific carbohydrate restriction (polysaccharides and disaccharides) in the form of the specific carbohydrate diet (SCD) in treating irritable bowel syndrome (IBS).

Design/methodology/approach

A female patient diagnosed with diarrhea predominant IBS was assigned to the SCD for six months. The diet occurred in phases and was advanced based on the individual’s tolerance level under the guidance of a registered dietitian. Quality of life was measured by a pre- and post-IBS severity score questionnaire. Gastrointestinal symptoms were measured by self-assessment of IBS symptoms using a seven-point Likert-like scale, with −3 = substantially worse to +3 = substantially better. Probiotics were consumed throughout the duration of the study.

Findings

The quality of life severity score significantly improved from a severity of 315 (with 500 being the most severe) to 15. The initial symptoms from the first day on the diet compared to the total period for bloating, abdominal pain/discomfort, flatulence/wind, diarrhea, bowel urgency, stool consistency, stool frequency, energy levels, incomplete evacuation and abdominal rumbling were improved significantly (p < 0.0005). The SCD diet significantly improved the quality of life and IBS symptoms in a female patient with IBS-diarrhea.

Originality/value

This study is the first of its kind to evaluate the efficacy of the SCD to treat IBS. The SCD should be considered a therapeutic option to treating IBS after fermentable carbohydrate restriction.

Abstract

Neither the characteristics of patients who are following the SCD nor the benefits of this diet have been well described in the medical literature. Herein, we report on the largest series of patients with IBD following the SCD to date and describe their clinical characteristics.

Abstract

Objective: The human intestine harbors trillions of commensal microbes that live in homeostasis with the host immune system. Changes in the composition and complexity of gut microbial communities are seenininflammatoryboweldisease (IBD),indicating disruption in host-microbe interactions. Multiple factors including diet and inflammatory conditions alter the microbial complexity. The goal of this study was to develop an optimized methodology for fecal sample processing and to detect changes in the gut microbiota of patients with Crohn’s disease receiving specialized diets.

Design: Fecal samples were obtained from patients with Crohn’s disease in a pilot diet crossover trial comparing the effects of a specific carbohydrate diet (SCD) versus a low residue diet (LRD) on the composition and complexity of the gut microbiota and resolution of IBD symptoms. The gut microbiota composition was assessed using a high-density DNA microarray PhyloChip.

Results: DNA extraction from fecal samples using a column based method provided consistent results. The complexity of the gut microbiome was lower in IBD patients compared to healthy controls. An increased abundance of Bacteroides fragilis (B. fragilis) was observed in fecal samples from IBD positive patients. The temporal response of gut microbiome to the SCD resulted in an increased microbial diversity while the LRD diet was associated with reduced diversity of the microbial communities.

Conclusion: Changes in the composition and complexity of the gut microbiome were identified in response to specialized carbohydrate diet. The SCD was associated with restructuring of the gut microbial communities.

Keywords: IBD; Crohn’s Disease; Fecal microbiome; Diet Modification; PhyloChip

Abstract

Abstract

Due to both perceived and real risks of current medical therapies for Crohn's disease (CD), other safe and effective approaches, particularly those utilizing nutrition and enteral therapy, have been sought. The specific carbohydrate diet (SCD) has become one alternative for CD considered by parents and patients, yet no prospective pediatric trials which employ mucosal healing as an outcome exist. Methods: Pts with active CD (PCDAI ≥ 15) interested in the SCD as adjunctive therapy and able to swallow a video capsule (VC), were eligible for this study. Subjects underwent a patency capsule, and if passed intact, VC was administered. They were maintained on their prescribed medications and reviewed the SCD with a dietician who then monitored their intake. VC was then repeated at 12 weeks(wk). Demographic, dietary and clinical information were collected at both time points. VC at wks 0 and12 were evaluated by a reader blinded to patient results and timing. PCDAI, Harvey Bradshaw (HB) and Lewis score (LS) were calculated at study visits as well. Means for outcome variables are reported here because of the few pts enrolled as yet. Results: The SCD has been offered to 10 pts to date. Two declined because of the stringency of the SCD; 2 were unable to ingest the VC; with 6 enrolled. Four (2 M, 2 F; average age 13.5 y; disease duration 4.5 y) have completed the trial to date; 1 (20 yo F) ceased at 8 wk because of difficulty with the SCD. The 4 completers received an average of 72.4 % of their estimated caloric needs, respectively, prior to the SCD, and 82.6 % on the SCD. Weight, Hgb, WBC, ESR, and albumin were essentially unchanged. Mean HB decreased from 3 to 1 and PCDAI from 20 to 6.2. Small bowel (SB) ulcers seen on initial VC in 3 were not seen on the 12 wk VC, with LS decreasing in all pts. In1 pt not rigidly adherent to the SCD, the number of stenotic areas decreased and the LS declined, but additional aptha developed in a new location. Impressions: Mucosal and clinical improvement were seen in the first 4 patients completing this pilot study (with SB mucosal healing in 2). VC appears to offer an important means to monitor mucosal improvement even over a relatively short interval. Completion of this trial and additional studies are needed to understand the changes described here and the mechanisms contributing to this improvement.

This case series indicates the potential for the IBD-AID to be used as an adjunctive or alternative therapy for the treatment of IBD. Notably, 9 out of 11 patients were able to be managed without anti-TNF therapy, and 100% of the patients had their symptoms reduced. To make clear recommendations for its use in clinical practice, randomized trials are needed alongside strategies to improve acceptability and compliance with the IBD-AID.

Over the years, there have been numerous studies examining diet and Inflammatory Bowel Disease (IBD). Six decades ago, prior to the identification of gluten as the principal offending agent, S.V. Haas successfully developed the Specific Carbohydrate Diet (SCD) for the treatment of celiac disease. The SCD has as its basis a strict grain-free, sugar-free, and complex carbohydrate-free dietary regimen. In theory, it is similar to an elemental diet, the thought being that foods easily absorbed provide bowel rest. The SCD, however, strives to use readily available foods such as fruits, meats, nuts, eggs, and vegetables. In addition, it is thought the SCD may alter gut flora and thus remove bacterial antigens thought to be responsible for the immune hypersensitivity seen in IBD.

After reviewing two cases in which individuals adhering to a strict SCD showed a positive outcome, it was decided to conduct an internet survey to ascertain whether there were other cases to support such findings.

Journal of Pediatric Gastroenterology and Nutrition

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Volume 39 Supplement 1 June 2004 pp S299-S300
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P0637 THE SPECIFIC CARBOHYDRATE DIET - A TREATMENT FOR CROHN'S DISEASE?

Fridge, J. L.¹; Kerner, J.¹; Cox, K.¹

¹Pediatric Gastroenterology, Hepatology and Nutrition, Lucile Packard Children's Hospital, Stanford Medical Center, Palo Alto, United States

Submitted by: jacqueline.fridge@medcenter.stanford.edu

Introduction:

Many diet therapies for Crohn's disease are known to be effective. It is not known which components of diets give the benefit, or which component of a regular diet is perpetuating the disease. We report two children with Crohn's disease who made a complete recovery on the Specific Carbohydrate Diet (SC Diet). One child received no other therapy, the other was steroid dependent prior to the diet. The diet eliminates all complex carbohydrates and refined sugars. In theory the diet deprives intestinal bacteria of the substrates they need to survive, reducing bacterial growth and the harmful products of fermentation.

Methods:

This is a case series to describe a novel diet therapy.

Results:

Case 1: An 11 year old girl presented with abdominal pain, weight loss and diarrhea. Laboratory examination revealed an albumin of 3.0 g/dL, hemoglobin (Hb) 11.6 g/dL and erythrocyte sedimentation rate (ESR) 17 mm/Hr. Serologies were positive with ASCA IgA 27.7 EU/ml and IgG 53.9 EU/ml, but pANCA was negative. Upper GI series (UGIS) showed multiple areas of stricture and mucosal cobblestoning in the jejunum and ileum, and a 5-6 cm stricture in the terminal ileum. Pathology was non-specific. The family refused standard care and following their own research elected to start the SC Diet. After 6 months on the diet her UGIS is normal, labs including ASCA antibodies have normalized and the patient is growing and symptom free.

Case 2:

A 9 year old boy presented with a history of diarrhea, abdominal pain, poor appetite and no weight gain for 2 years. Laboratory examination revealed albumin 3.4 g/dL, Hb 12.0 g/dL and ESR 43 mm/Hr. ASCA IgA, IgG and pANCA were negative. UGIS showed narrowing of the distal ileum and proximal cecum. Pathology demonstrated focal acute colitis with crypt abcesses and granuloma formation. The patient responded well to treatment with sulfasalazine, prednisone and 6 mercaptopurine. Ten weeks after starting medications, during a steroid taper, his symptoms flared and his anemia returned. His prednisone was increased, but at this time the family elected to start the SC Diet. After 3 months on the diet the patient is off steroids, symptom free and all blood tests have normalized.

Conclusion:

The apparent effectiveness of the Specific Carbohydrate Diet in Crohn's Disease warrants more study. There is current interest in the manipulation of intestinal flora using probiotics and prebiotics. If this diet works by changing the bacterial flora of the bowel, then it adds weight to the role of bacteria in the pathogenesis of Crohn's Disease.

Reference(S):

Gottschall, Elaine: Breaking the Vicious Cycle - Intestinal Health Through Diet. Baltimore, Ontario, The Kirkton Press 2002.

We present in this paper the results of our experience with 603 cases of celiac disease from which has emerged a useful method of diagnosis of this confusing condition and, more important, an effective cure by diet which we have found successful in cases of all types and degrees of severity.

Since the etiology of celiac disease is unknown and its symptoms occur in many other conditions, it may be helpful to review briefly the history of attempts to find a cure, especially by dietary methods, and to describe the clinical picture of the disease. We shall then outline our diet in detail, report statistically on our 603 cases, and suggest further lines of study.

It is not generally realized that celiac disease occurs at all ages, although Gee, who first described it, called attention to this fact. It usually occurs before the fifth year, beginning most frequently in the latter half of the first year. The onset is usually so gradual that the date is difficult to set; occasionally it is abrupt. The course is chronic, and there is a marked tendency to relapse. The symptoms are usually characteristic. There is a marked hypotonia. The face is edematous, distressed, pale and emaciated, but is less emaciated than the extremities, which the patient may be unable or disinclined to move. There is great irritability, and usually complete anorexia. The abdomen is very large, protuberant and soft, except when distended by gas. The stools are frequent, large, pale and foul-smelling, rarely watery, and appear to be greater in bulk than can be accounted for by the intake. There is a marked retardation in growth.

Some years ago I treated a child, aged 3 years, who suffered from a severe case of anorexia nervosa. She had reached a serious state of depletion and weakness from her self imposed starvation, refusing all food and regurgitating that fed to her by gavage. She finally accepted a banana, with the result that other food was taken in a more or less normal amount within forty-eight hours. There was a complete relapse when the banana was withheld, and food was taken normally only with bananas.

This experiment was repeated to test the validity of the observation, always with the same result, until a time came when her appetite was normal whether bananas were included in the diet or not. The action was such as is attributed to a hormone. It was natural, therefore, to test bananas in a case of celiac disease where anorexia was a prominent symptom.