A nationwide survey of non-IgE-mediated gastrointestinal food allergies in neonates and infants.

BACKGROUND: Non-IgE-mediated gastrointestinal food allergies (non-IgE-GIFAs) seem to be increasing rapidly worldwide. However, nationwide studies have been limited to food-protein-induced enterocolitis (FPIES) and food-protein-induced allergic proctocolitis (FPIAP), with little attention to other non-IgE-GIFA subgroups. The aim of this study was to elucidate the clinical features of all patients with non-IgE-GIFAs, not just certain subgroups. METHODS: We conducted a nationwide cross-sectional survey of non-IgE-GIFAs in Japan from April 2015 through March 2016. A questionnaire was sent to hospitals and clinics throughout Japan. The questionnaire asked about the number of physician-diagnosed non-IgE-GIFA patients, the status of fulfillment of the diagnostic criteria, tentative classification into 4 clusters based on the initial symptoms, the day of onset after birth, complications, and the suspected offending food(s). RESULTS: The response rate to that questionnaire was 67.6% from hospitals and 47.4% from clinics. Analyses were conducted about "diagnosis-probable" patient cohort (n = 402) and the "diagnosis-confirmed" patients (n = 80). In half of the reported non-IgE-GIFA patients, onset occurred in the neonatal period. The patients were evenly distributed among 4 non-IgE-GIFA clusters. In Cluster 1, with symptoms of vomiting and bloody stool, the onset showed a median of 7 days after birth, which was the earliest among the clusters. Cow's milk was the most common causative food. CONCLUSIONS: In half of the patients, the onset of non-IgE-GIFAs was in the neonatal period. This highlights the importance of studying the pathogenesis in the fetal and neonatal periods.

A different starting line for allergic march: food protein-induced allergic proctocolitis.

OBJECTIVE: The aim of this study is to investigate the long-term prognosis of food protein--induced allergic proctocolitis (FPIAP) patients, the risk of developing both allergic and gastrointestinal diseases, and to evaluate whether it leads to allergic march. METHODS: A total of 149 children who were diagnosed with FPIAP and developed tolerance at least 5 years prior to the study and 41 children (with no history of food allergy) as a control group were enrolled. Both groups were re-evaluated for allergic diseases as well as gastrointestinal disorders. RESULTS: The mean age of diagnosis for the FPIAP group was 4.2 ± 3.0 months, while the mean age of tolerance was 13.9 ± 7.7 months. The mean age of both FPIAP and control groups at the last visit was 101.6 ± 24.4 and 96.3 ± 24.1 months, respectively (P = 0.213). At the final evaluation of both groups, the comorbid allergic disease was significantly higher in the FPIAP group (P < 0.001). There was no significant difference between the two groups in terms of functional gastrointestinal disorders (FGIDs), eosinophilic gastrointestinal diseases, and inflammatory bowel disease (P = 0.198, 0.579, and 0.579, respectively).In the FPIAP group, the allergic disease was significantly higher at the final visit in patients with comorbid allergic disease at diagnosis (P < 0.001). In the FPIAP group, FGID was significantly higher in the group that developed allergic diseases in the future, compared to the group that did not develop allergic diseases in the future (P = 0.034). The proportion of both FGID and allergic diseases was significantly higher in subjects that developed tolerance at >18 months, compared to subjects that developed tolerance at >18 months (P < 0.001 and <0.001, respectively). CONCLUSIONS: Patients with FPIAP may develop allergic diseases as well as FGID in the long term.

Dietary Factors May Delay Tolerance Acquisition in Food Protein-Induced Allergic Proctocolitis.

BACKGROUND: Dietary and environmental factors may influence tolerance acquisition in food protein-induced allergic proctocolitis (FPIAP). This retrospective observational study explored the role of maternal diet during pregnancy and breastfeeding in tolerance acquisition in infantile FPIAP. METHODS: Breastfed infants with FPIAP from six diverse regions in Greece were divided into two groups, based on development of tolerance to the trigger food: Group A (n = 43), before, and Group B (n = 53), after, the 6th month of age. Maternal diet during pregnancy and breastfeeding was elicited using the Mediterranean Diet Score Questionnaire and the Mediterranean Oriented Culture Specific Semi-Quantitative Food Frequency Questionnaire. RESULTS: Mean age at diagnosis of FPIAP (1.5 months) and weaning (5.5 months) were the same in both groups. The main trigger was cow's milk. Group A received infant milk formula earlier than Group B. Group B had a higher incidence of asthma/wheeze, siblings with milk allergy, maternal smoking and rural residence. On multivariate analysis, earlier resolution of FPIAP was associated with higher maternal education and with salt intake and consumption of goat/sheep cheese during pregnancy and olive oil during breastfeeding. Consumption of multivitamins during pregnancy and meat, winter fruits, green vegetables, butter, salt, "ready-to-eat" meals and pastries during breastfeeding were correlated with longer duration of symptoms. CONCLUSIONS: Mothers of children with FPIAP to cow's milk protein can be advised to eat more yogurt, cheese and olive oil during subsequent pregnancies, and avoid multivitamins, grilled food, "ready-to-eat" meals, pastries, meat and alcohol during breastfeeding, to reduce the duration of FPIAP presenting in future infants.

Assessment of intracellular zinc levels in infants with food protein-induced allergic proctocolitis.

BACKGROUND: Food protein-induced allergic proctocolitis (FPIAP) is characterized by bloody stools in well-appearing infants. Zinc is a micronutrient that plays a crucial role in immune modulation and is essential for cellular function during immune response. Although there are studies on the assessment of intracellular zinc levels in allergic diseases, no data is available on erythrocyte zinc levels of patients with FPIAP. OBJECTIVE: This study aimed to assess the erythrocyte zinc levels of children with allergic proctocolitis and compare zinc levels with clinical and demographic characteristics. METHODS: This was a case-control study that prospectively compared 50 patients with FPIAP and 50 healthy children without malnutrition. The erythrocyte zinc levels of children were determined using atomic absorption spectrophotometry. RESULTS: Fifty patients with FPIAP, including 28 (51%) girls, with median age of 7.1 ± 2.9 (3-14) months and 50 healthy children, including 26 (53.1%) girls, with median age of 7.7 ± 2.8 (3-13) months were included in the study. Seventy percent (n = 35) of the patients with FPIAP started to have symptoms while they were exclusively breastfeeding. Offending allergen foods were cow's milk (78%), egg (40%), sesame (10%), hazelnut (8%), almond (6%), beef (6%), and peanuts (6%, n = 3). Intracellular (erythrocyte) zinc levels in patients with FPIAP were lower than in the healthy control group (495.5 ± 134 µg/dL, 567.3 ± 154.4 µg/dL, respectively, P = 0.01). Patients with FPIAP aged younger than 6 months had lower intracellular zinc levels compared with those aged above 6 months (457 ± 137 µg/dL; 548 ± 112 µg/dL, respectively, P = 0.01). There was no relationship between zinc levels and time of symptom onset, presence of concomitant disease, being allergic to multiple foods, and family history of atopy (P > 0.05). CONCLUSIONS: FPIAP is a food allergy with limited information on its pathogenesis. Considering the beneficial effects on gastrointestinal system epithelia, zinc may be involved in the pathogenesis of FPIAP. Future comprehensive prospective research on this subject is of importance.

Longitudinal disease-associated gut microbiome differences in infants with food protein-induced allergic proctocolitis.

BACKGROUND: Complex interactions between the gut microbiome and immune cells in infancy are thought to be part of the pathogenesis for the marked rise in pediatric allergic diseases, particularly food allergies. Food protein-induced allergic proctocolitis (FPIAP) is commonly the earliest recognized non-immunoglobulin E (IgE)-mediated food allergy in infancy and is associated with atopic dermatitis and subsequent IgE-mediated food allergy later in childhood. Yet, a large prospective longitudinal study of the microbiome of infants with FPIAP, including samples prior to symptom onset, has not been done. RESULTS: Here, we analyzed 954 longitudinal samples from 160 infants in a nested case-control study (81 who developed FPIAP and 79 matched controls) from 1 week to 1 year of age by 16S rRNA ribosomal gene sequencing as part of the Gastrointestinal Microbiome and Allergic Proctocolitis (GMAP) study. We found key differences in the microbiome of infants with FPIAP, most strongly a higher abundance of a genus of Enterobacteriaceae and a lower abundance of a family of Clostridiales during the symptomatic period. We saw some of these significant taxonomic differences even prior to symptom onset. There were no consistent longitudinal differences in richness or stability diversity metrics between infants with FPIAP and healthy controls. CONCLUSIONS: This study is the first to identify differences in the infant gut microbiome in children who develop FPIAP, some even before they develop symptoms, and provides a foundation for more mechanistic investigation into the pathogenesis of FPIAP and subsequent food allergic diseases in childhood. Video abstract.

Chronic Lymphogranuloma Venereum Proctocolitis Masquerading as Inflammatory Bowel Disease.

Rectal infection with the L1, L2, and L3 serovars of Chlamydia trachomatis can cause lymphogranuloma venereum (LGV) proctocolitis, particularly among men who have sex with men (MSM). Symptoms of this sexually transmitted infection include anal pain, rectal bleeding and discharge, tenesmus, constipation, and fever. Clinicians should consider LGV when there is a history of receptive anal intercourse and symptoms of proctocolitis. A positive nucleic acid amplification test (NAAT) on a rectal sample is diagnostic. This report describes a man with HIV and chronic proctocolitis in whom the diagnosis of LGV was delayed because the clinical picture mimicked inflammatory bowel disease.

First reported lymphogranuloma venereum cases in Russia discovered in men who have sex with men attending proctologists.

BACKGROUND: Several lymphogranuloma venereum (LGV) outbreaks among men who have sex with men (MSM) have been reported throughout the world since 2003. Nevertheless, no LGV cases have been internationally reported from Russia. We evaluated the prevalence of LGV among MSM attending proctologists in Moscow, Russia, and compared the LGV and non-LGV rectal Chlamydia trachomatis (CT) infections. METHODS: MSM (n = 534) attending for proctologic care were included. Rectal specimens were sampled for CT and Neisseria gonorrhoeae (NG) by nucleic acid amplification tests (NAATs). All CT-positive patients were tested with an LGV-specific NAAT. RESULTS: In total, 37.3% (95% CI 33.3-41.5; 199/534) of MSM were CT positive. Of these, 68.8% (95% CI 62.1-74.9; 137/199) had LGV and 31.2% (95% CI 25.1-37.9; 62/199) a non-LGV rectal CT infection. Older age (34 years vs. 31 years, p = 0.035) and group-sex practices (67.2% (92/137) vs. 33.9% (21/62), p < 0.0001) were associated with LGV. The LGV-positive MSM were also more likely to be HIV-positive (67.2% (92/137) vs. 41.9% (26/62), p = 0.001). Proctoscopy revealed ulcerative proctitis/proctocolitis in 99.3% (136/137) of LGV-positive MSM. No ulcerative or erosive proctitis was found in the MSM with non-LGV CT infection, but 58.1% (36/62) of them had anorectal disorders. Finally, mild catarrhal or hemorrhagic proctitis was diagnosed in only 21.6% (8/37) of MSM with non-LGV CT infection lacking concomitant NG or syphilis (p < 0.0001). CONCLUSIONS: LGV is widely spread among MSM attending proctologists in Moscow. Clinically, acute LGV proctitis/proctocolitis can be difficult to distinguish from inflammatory bowel disease that leads to mismanaged LGV infections. LGV diagnostic laboratory testing is essential, however, currently mainly lacking for MSM in Russia. All MSM with CT-positive rectal specimens should be subsequently tested for LGV.

Clinical Features and Clinical Course of Food Protein-Induced Allergic Proctocolitis: 10-Year Experience of a Tertiary Medical Center.

BACKGROUND: Food protein-induced allergic proctocolitis (FPIAP) is a food allergy characterized by bloody stools in well-appearing breast-fed infants. OBJECTIVE: To determine the clinical course of FPIAP and the factors affecting the development of tolerance. METHODS: Over a 10-year period, patients with a diagnosis of FPIAP who were followed at the outpatient Allergy-Immunology clinic in a tertiary care children's hospital in Turkey were retrospectively analyzed. RESULTS: The frequency of FPIAP was 0.18% among 64,549 patients. The median age of symptom onset was 2 months (interquartile range, 1.0-3.5 months), and the median age of tolerance development was 12 months (interquartile range, 8.0-17.21 months). The occurrence of symptoms in the neonatal period was associated with a history of premature birth (odds ratio, 3.75; 95% CI, 1.33-10.59; P = .031) and neonatal intensive care unit hospitalization (odds ratio, 4.72; 95% CI, 1.78-12.53; P = .002). Use of a cow's milk-based formula was associated with a higher risk of the onset of symptoms after 1 month (odds ratio, 2.69; 95% CI, 1.19-6.07; P = .016). The use of an amino acid-based formula and the presence of diarrhea at admission were associated with later development of tolerance (P = .023 and P < .001, respectively). An IgE-mediated reaction was observed during oral food challenge testing in 6% of the patients. CONCLUSIONS: The manifestations of FPIAP appeared earlier in premature infants and later in infants using formula. The use of amino acid-based formula and having had diarrhea were associated with delayed tolerance.

A Prospective Assessment of Clinical Characteristics and Responses to Dietary Elimination in Food Protein-Induced Allergic Proctocolitis.

BACKGROUND: Some aspects of diagnostic elimination/challenge diets in food protein-induced allergic proctocolitis (FPIAP) are still poorly defined. OBJECTIVE: This study investigated the symptom spectrum, time required for resolution of each symptom, triggering foods, and risk factors for multiple food allergies (MFA) in FPIAP. METHODS: Infants referred with visible blood in stool were enrolled after etiologies other than FPIAP had been excluded. Laboratory evaluation, clinical features, and elimination/challenge steps were performed prospectively during diagnostic management. RESULTS: Ninety-one of 102 infants (53 boys) were diagnosed with FPIAP. Eleven children did not bleed during challenges. Visible blood in stool began before 2 months of age in 63.6% of the infants not diagnosed with FPIAP, compared with 18.9% of the patients with FPIAP (P = .003). Offending foods were identified as cow's milk (94.5%), egg (37.4%), beef (10.9%), wheat (5.5%), and nuts (3.3%). MFA was determined in 42.9% of patients. Multivariate logistic regression analysis identified atopic dermatitis (AD) (odds ratio [OR]: 2.98, 95% confidence interval [CI]: 1.18-7.55, P = .021) and an eosinophil count ≥300 cells/µL (OR: 2.72, 95% CI: 1.09-6.80, P = .032) as independent risk factors for MFA. Blood and mucus in stool disappeared in a median 3 days (interquartile range [IQR]: 1-14.5 days) and 30 days (IQR: 8-75 days), respectively. CONCLUSIONS: A tendency to transient bleeding occurs in infants who present with bloody stool before 2 months of age. A 2-week duration of elimination for blood in stool is sufficient to reach a judgment of suspected foods for FPIAP. Mucus in stool is the last symptom to disappear. Concurrent AD suggests a high probability of MFA in FPIAP.

Non-allergic benign infantile proctocolitis: a neglected nosographic entity.

The presence of rectal bleeding in the first months of life is very often diagnosed as food protein-induced allergic proctocolitis (FPIAP). The symptoms typically start in infancy, and most cases resolve by age 12 months. Infants with FPIAP usually present bloody stools mixed with mucus, with or without reduced stool consistency. Most affected infants are generally healthy-appearing. We used the story of an infant with rectal bleeding as a reminder that there is also the possibility of a form of benign non-allergic proctocolitis that is not usually included in the differential diagnosis. In the absence of warning signs and in case of infant well-being, it should be the first clinical entity to suspect. Therefore, we suggest we should wait at least 2 months before starting to eliminate cow milk or other foods from the diet.

Atopy Patch Test in the Diagnosis of Food Allergens in Infants with Allergic Proctocolitis Compared with Elimination/Introduction C.

Allergic proctocolitis is a cell-dependent food allergy that is present in both breast and formula-fed infants. The presence of blood with different amounts in the stool is the main manifestation of the disease. Different results have been published on the accuracy and specificity of the atopic patch test (APT). The purpose of this study was to evaluate the results of the APT and compare them with those obtained in the food elimination/introduction (E/I) challenge, as the gold standard of confirming the allergy. Twenty-eight patients (18 boys, 10 girls, <1 year) with allergic proctocolitis were recruited in this study. The mean age of the disease onset and enrolling the study were 2.23±1.7 and 5.25±2.19 months, respectively. After performing APT with fresh foods, an E/I challenge was done in a patient with positive tests, and results were analyzed. APT was positive in 14/28 (50%) individuals. The most common foods detected by APT in all of the individuals were: milk (10/28), rice (5/28), soy (4/28), and egg white (4/28), while in E/I challenge in the APT-positive individuals were: milk (8/10), rice (3/5), egg white (1/4), and soy (0/4). APT was positive in half of the infants<1 year with allergic proctocolitis and there was no significant correlation between the APT results and the E/I challenge test for all foods. Comparing the results of APT and E/I challenge methods showed a convergence between the milk and rice sensitivity, thus we suppose APT to be a useful tool in identifying these two allergens in cell-mediated food allergies like allergic proctocolitis.

2021 European Guideline on the management of proctitis, proctocolitis and enteritis caused by sexually transmissible pathogens.

This guideline intents to offer guidance on the diagnosis and management of patients with gastrointestinal symptoms and a suspected sexually transmitted cause. Proctitis is defined as an inflammatory syndrome of the anal canal and/or the rectum. Infectious proctitis can be sexually transmitted via genital-anal mucosal contact, but some also via digital contact and toys. Neisseria gonorrhoeae, Chlamydia trachomatis (including lymphogranuloma venereum), Treponema pallidum and herpes simplex virus are the most common sexually transmitted anorectal pathogens. Shigellosis can be transferred via oral-anal contact and may lead to proctocolitis or enteritis. Although most studies on these infections have concentrated on men who have sex with men (MSM), women having anal intercourse may also be at risk. A presumptive clinical diagnosis of proctitis can be made when there are symptoms and signs, and a definitive diagnosis when the results of laboratory tests are available. The symptoms of proctitis include anorectal itching, pain, tenesmus, bleeding, constipation and discharge in and around the anal canal. The majority of rectal chlamydia and gonococcal infections are asymptomatic and can only be detected by laboratory tests. Therefore, especially when there is a history of receptive anal contact, exclusion of anorectal infections is generally indicated as part of standard screening for sexually transmitted infections (STIs). Condom use does not guarantee protection from STIs, which are often spread without penile penetration. New in this updated guideline is: (i) lymphogranuloma venereum proctitis is increasingly found in HIV-negative MSM, (ii) anorectal Mycoplasma genitalium infection should be considered in patients with symptomatic proctitis after exclusion of other common causations such N. gonorrhoeae, C. trachomatis, syphilis and herpes, (iii) intestinal spirochetosis incidentally found in colonic biopsies should not be confused with syphilis, and (iv) traumatic causes of proctitis should be considered in sexually active patients.

Portal vein thrombosis and food protein-induced allergic proctocolitis in a premature newborn with hypereosinophilia: a case report.

BACKGROUND: Peripheral blood eosinophilia is identified in numerous medical conditions associated with allergic, infectious, and inflammatory processes mostly as reactive eosinophilia with or without tissue eosinophilia. In hospitalized neonates, eosinophilia is common with an inverse relationship with gestational age and occurs solely as mild eosinophilia in the majority of cases. In the literature, eosinophilia has been proposed as a possible risk factor for venous thromboembolism. However, few reports are found on thromboembolic events including portal vein thrombosis (PVT) associated with eosinophilia in the newborn period. Neonates, particularly preterm infants, are vulnerable to thrombosis due to the immature and developing hemostatic system with little reserve capacity, which occurs as catheter-related thrombosis in most cases. CASE PRESENTATION: A male newborn at 34+ 5 weeks' gestation presented with a left portal venous thrombus and hematochezia after initial cow's milk feeding in the setting of blood hypereosinophilia for a prolonged period of time without central venous catheterization. The infant was diagnosed with PVT and food protein-induced allergic proctocolitis (FPIAP) and showed complete resolution of the conditions with expectant management with food avoidance, including the normalized eosinophil count. CONCLUSIONS: Our experience suggests that in the setting of hypereosinophilia with a prolonged duration in premature neonates, FPIAP should be suspected in case of hematochezia in otherwise healthy infants, and considering the increased thrombotic risk by the hypereosinophilia and premature newborn status, evaluation for neonatal thrombosis may be needed, including PVT with the potential risk for the more serious, but uncommon, late complications encompassing portal hypertension.

Low Incidence of Inflammatory Bowel Disease Adverse Events in Adalimumab Clinical Trials Across Nine Different Diseases.

OBJECTIVE: Adalimumab is approved for treatment of Crohn's disease and ulcerative colitis. Thus, we postulated that exacerbation or new-onset of inflammatory bowel disease (IBD) would be rare events in patients treated with adalimumab for non-IBD indications. The objective was to evaluate the incidence of IBD adverse events (AEs) across adalimumab trials. METHODS: IBD AE rates in 75 adalimumab clinical trials in rheumatoid arthritis, polyarticular juvenile idiopathic arthritis, pediatric enthesitis-related arthritis, uveitis, hidradenitis suppurativa, adult and pediatric psoriasis, psoriatic arthritis, nonpsoriatic arthritis peripheral spondyloarthritis (SpA), axial SpA, including nonradiographic axial SpA, and ankylosing spondylitis, were analyzed. Search terms for IBD AEs (new onset or worsening/flare) included IBD, ulcerative colitis, Crohn's disease, and ulcerative proctitis. RESULTS: This analysis included 24,114 patients, representing 36,508 patient-years of adalimumab exposure. The overall rate of IBD AEs in adalimumab-treated patients was 0.1 (95% confidence interval [95% CI] 0.1-0.2)/100 patient-years (41 events), ranging from no events (psoriatic arthritis, uveitis, and pediatric trials) to 0.8 (95% CI 0.2-2.2)/100 patient-years in peripheral SpA. The rate of IBD in axial SpA was 0.6 (95% CI 0.4-1.0)/100 patient-years. During placebo-controlled trials, the overall IBD rate was 0.1 (95% CI 0.0-0.3)/100 patient-years for adalimumab groups (3 events in 6,781 patients; 2,752 patient-years of exposure) and 0.1 (95% CI 0.0-0.4)/100 patient-years for placebo groups (1 event in 3,493 patients; 1,246 patient-years of exposure). IBD rates in axial SpA were 0.5 (95% CI 0.1-1.4)/100 patient-years for adalimumab and 0.6 (95% CI 0.0-3.1)/100 patient-years for placebo. CONCLUSION: The rates of IBD AEs in adalimumab clinical trials were generally low across the evaluated diseases, including axial SpA; all events occurred in adult patients.

Food protein-induced allergic proctocolitis may have distinct phenotypes.

BACKGROUND: Food protein-induced allergic proctocolitis (FPIAP) is a non-immunoglobulin E (IgE)-mediated food allergy, which presents with bloody mucoid stool in infants. Although IgE-mediated allergy and sensitizations to offending foods have been described in other non-IgE-mediated food allergies, it has not been investigated in FPIAP. OBJECTIVE: To investigate IgE-mediated allergy and sensitization to offending foods in FPIAP. METHODS: Patients (n = 204) were retrospectively recruited and grouped as FPIAP (n = 180; FPIAP with or without the symptoms of IgE-mediated food hypersensitivity to offending and nonoffending foods at initial consultation), FPIAP-IgE sensitization to offending foods (n = 17), and FPIAP-transition to IgE-mediated allergy to offending foods (n = 7). The study was performed in accordance with the protocol approved by the local ethical committee of the Hacettepe University. RESULTS: The median age of onset of symptoms and the development of tolerance was 2 months (interquartile range [IQR], 1.0-3.0) and 12 months (IQR, 10.0-14.0), respectively, and of the patients with skin prick test or serum specific IgE tests (n = 196), 38 (19.4%) had evidence of IgE sensitization to offending foods at the initial consultation or during follow-up; 17 (8.6%) had IgE sensitization, 7 (3.6%) indicated a transition to IgE-mediated allergy to FPIAP-induced foods. The median age of tolerance development of the FPIAP-transition group (19 months, IQR, 18.0-29.0) was significantly later than that of the FPIAP group (11 months, IQR, 10.0-14.0; P < .001) and the FPIAP-IgE sensitization group (11.0 months, IQR, 9.5-12.0; P < .001). Tolerance was observed within the study period in almost all the patients. CONCLUSION: Children with FPIAP may have sensitization or develop IgE-mediated allergy over time to offending foods. In addition, IgE sensitization in FPIAP does not have an unfavorable effect on tolerance development; however, the transition to an IgE-mediated phenotype may delay tolerance for a brief time.

Anal skin tag - An unusual presenting feature of food protein-induced allergic proctocolitis in a neonate.

BACKGROUND: Anal skin tags are commonly seen with anal fissures, haemorrhoids, inflammatory bowel disease and their association have been extensively studied. However the presence of anal skin tag in food protein-induced allergic proctocolitis has rarely been reported in literature. We report a neonate with food protein-induced allergic proctocolitis who presented with blood in stool and anal skin tag. CASE DESCRIPTION: A 26-day-old baby presented with history of passing intermittent blood in stools for two days. The baby was exclusively breast-fed and was well-appearing with no failure to thrive. Two anal skin tags were present but there was no evidence of anal fissures or haemorrhoids. The biopsy of anal skin tag showed fibroepithelial polyp. Colonoscopy was suggestive of food protein-induced allergic proctocolitis. In view of poor response to elimination diet in the mother and extensively hydrolysed formula, the baby was started on amino acid formula with complete recovery. CONCLUSION: Through this case we wish to highlight that clinicians should consider food protein-induced allergic proctocolitis in their differential diagnosis in a neonate presenting with blood in stools and anal skin tag.

Using Eosinophil Biomarkers From Rectal Epithelial Samples to Diagnose Food Protein-induced Proctocolitis: A Pilot Study.

OBJECTIVES: The gold standard diagnostic procedure for food protein-induced proctocolitis (FPIP) requires flexible sigmoidoscopy (FS). To date there is no validated, noninvasive test to confirm FPIP diagnosis. Eosinophil-derived neurotoxin (EDN), a product of eosinophil (EOS) degranulation, has been shown to correlate with eosinophil infiltration in other tissues. Our objective was to compare EDN concentrations in rectal epithelial samples from infants with FPIP with those from a control population. METHODS: Children who underwent routine FS at Arnold Palmer Hospital for Children were enrolled in an IRB-approved, prospective, open-label pilot study between July 2017 and May 2019. We obtained rectal epithelial samples via: rectal swab, cytology brushing through FS, and rectal biopsy through FS. We then measured EDN levels in the samples and compared levels found in infants with FPIP against levels found in the control group. FPIP was defined as more than 60 EOS per 10 high-power fields (HPF) in rectal epithelial tissue obtained via rectosigmoid biopsy. RESULTS: Twenty-four patients were enrolled. The control group (n = 13) included patients with normal histopathology (84% boys, mean age 19 months, SD 6 months) and the FPIP group (n = 11) included patients with FPIP confirmed via biopsy (45% boys, mean age 6.9 months, SD 9 months). EDN concentration was significantly higher in the FPIP group than in the control group, for 2 sampling methods: rectal biopsy (183.6 ±â€Š114.6 vs 76.6 ±â€Š71.0 µg/mL; P = 0.010) and rectal swab (66.2 ±â€Š64.8 vs 20.4 ±â€Š22.2 µg/mL; P = 0.025). CONCLUSIONS: EDN concentrations measured from rectal swab and rectal biopsy samples is elevated and may be a useful tool to screen for FPIP in children.