6. (ihazanfar MN, Thomsen SF. OrnaLzumab for Urticarial Vasculitis: Case Report and Review of the Literature. Case Rep Dermatol Med. 2015;2015:576893. 7. Sussman G, Hebert J, Barron C. Bian J, Ca'on-Guay RM, Laflarnme S at al. Real-life experiences with omalizumab for the treatment of chronic urticaria. Ann Allergy r'lsthrna Irnrnunol. 2014; 112:170-4. 8. Diez LS, 1amayo LM, Cardona R. [Omalizumab: therapeutic option inchronic spontaneous urticaria difficult to control With associated vasculitis, report of three cases). Biomedica. 2013;33:503-12. 9. Enriquez 'I, Sirvenl rnernbr anoproiiferative hypocomplementemic 200~i;18:318- n. O. Kai AC, Hohr C. Grattan CE. Improvement in quality of life impairment followed by relapse with 6-monthly periodic administration of ornalizumab for severe treatment· refractory chronic urtiCiJlia and urticarial vasculitis. Clin Exp Dermatoi. 2014;39:651-2. AE, Amoros F. glom er lJ Ion eph r iti s urticarial vasculitis. J Crescentic and Nephrol. M Manuscript received June 9, 2017; accepted for publication July 26.2017. Fabio Basta Unit of Allergology, Immunology and Rheumatology Department of Medicine Campus Bio-Medico di Roma Via Alvaro del Portillo, 200 00128 Roma, Italy E-mail fbasta@unicampus.it Alves C'. Afonso 1'. Leiria-Pinto 1"; i Dona Estefdnia Hospital. Immlll10allelgulugy department, Centro llmpitalar de Lisboa Central,Lisbon, Portugal Dona Estefdnia !Iospiral, Pediatric Gastroenterolug)) Department. Cenlro Huspitalar de Lisboa Central, Lisbon, Portugal 'CEDOC. Respiratory Research Group. ,yova Medical School. Lisbon, Portugal .J lnvestig Allergol Clin 11111T1unol 2017, Vo!. 27(6) 384-386 doi 10.18176!jinci.OI94 Key words: Saccharomyces boulardii. Hypersensitivity reactions. Food protein-inouced enterocolitissyndrome-like. Palabras clave: Saccharomycesboulardii. Reaccionesde hipersensibilidad. SindrolTIedeenterocolitis inducida par prateinas alrillentarias. Saccharomyces boulardii is a nonpathogenic yeast isolated fi'om theskin of lychees grown in Indochina that has been widely prescribed in the lust 30 years for prophylaxis and treatment of bacterial diarrhea11,2]. Hypersensitivity reactions to S bOlllaldii are rare, and.to the authors' knowledge, only 2 cases have been reported in the literature [3,4]. We present a case of an exclusively breastfed 2-month- old girl, who presented with blood and mucus instool. Enteritis (viral and bacterial) was excluded, and the mother was advised to begin a dairy-tree diet. However. the blood andmueus persisted despite the ehunge to the mother's diel. Rectosigmoidoscopy with biopsy revealed nodular lymphoid hyperplasia and histopathologic evidence ofcolitis characterized byedematous and hemorrhagic focal areas with eosinophil infiltration (more than 6 per high-power-field) and occasional lymphoid nodes. suggesting a diagnosis ofallergic colitis. We therefore reinforced the importance ora dairy-fi'ee diet while breast feeding. The patient was a healthy, full-term infantdelivered by cesarean birth. with normal growth for age. Blood and mucus persisted in stool, ulbeit at a lower intensity. At 3 months. the infant began to produce watery stool. S bOlllaldii (liL 250 sachets) was prescribed. The child began to vomitprohJsely approximately 2 hours after ingestion, ,,,,ith spontaneous resolution within a few hours. Three weeks later. as gastrointestinal symptoms persisted (intermittent vvatery stools with blood and mucus). she was again treated ",ith S bOlllal'dii, 2hours after which she experienced ,ill episode of uncontrollable vomiting, with severe prostration. She was seen at theemergency department. where she received replacement fluid therapy. The vom iting resolved on the same day. and no other signs of inJection were observed. At that time. she was referred to our lmmunoallergology Department. Skin prick tests and specific IgE for 111 ilk, casein, n-laetalbumin, and [.I-lactoglobulin were negative. We diagnosed non-·IgE-mediated cow's milk allergy and
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6. (ihazanfar MN, Thomsen SF. OrnaLzumab for UrticarialVasculitis: Case Report and Review of the Literature. Case RepDermatol Med. 2015;2015:576893.
7. Sussman G, Hebert J, Barron C. Bian J, Ca'on-Guay RM,Laflarnme S at al. Real-life experiences with omalizumabfor the treatment of chronic urticaria. Ann Allergy r'lsthrnaIrnrnunol. 2014; 112:170-4.
8. Diez LS, 1amayo LM, Cardona R. [Omalizumab: therapeuticoption in chronic spontaneous urticaria difficult to controlWith associated vasculitis, report of three cases). Biomedica.2013;33:503-12.
9. Enriquez 'I, Sirvenlrnernbr anoproiiferativehypocomplementemic200~i;18:318- n.
O. Kai AC, Hohr C. Grattan CE. Improvement in quality of lifeimpairment followed by relapse with 6-monthly periodicadministration of ornalizumab for severe treatment· refractorychronic urtiCiJlia and urticarial vasculitis. Clin Exp Dermatoi.2014;39:651-2.
AE, Amoros F.glom er lJ Ion eph riti s
urticarial vasculitis. J
Crescenticand
Nephrol.
M Manuscript received June 9, 2017; accepted for publication July26.2017.
Fabio BastaUnit of Allergology, Immunology and Rheumatology
Department of MedicineCampus Bio-Medico di Roma Via Alvaro del Portillo, 200
00128 Roma, ItalyE-mail [email protected]
Alves C'. Afonso 1'. Leiria-Pinto 1";iDona Estefdnia Hospital. Immlll10allelgulugy department, Centrollmpitalar de Lisboa Central, Lisbon, PortugalDona Estefdnia !Iospiral, Pediatric Gastroenterolug)) Department.Cenlro Huspitalar de Lisboa Central, Lisbon, Portugal'CEDOC. Respiratory Research Group. ,yova Medical School.Lisbon, Portugal
.J lnvestig Allergol Clin 11111T1unol2017, Vo!. 27(6) 384-386doi 10.18176!jinci.OI94
Key words: Saccharomyces boulardii. Hypersensitivity reactions. Foodprotein-inouced enterocolitis syndrome-like.
Palabras clave: Saccharomycesboulardii. Reaccionesde hipersensibilidad.SindrolTIede enterocolitis inducida par prateinas alrillentarias.
Saccharomyces boulardii is a nonpathogenic yeast isolatedfi'om the skin of lychees grown in Indochina that has beenwidely prescribed in the lust 30 years for prophylaxis andtreatment of bacterial diarrhea 11,2].
Hypersensitivity reactions to S bOlllaldii are rare, and. tothe authors' knowledge, only 2 cases have been reported inthe literature [3,4].
We present a case of an exclusively breastfed 2-month-old girl, who presented with blood and mucus in stool.Enteritis (viral and bacterial) was excluded, and the motherwas advised to begin a dairy-tree diet. However. the bloodand mueus persisted despite the ehunge to the mother'sdiel. Rectosigmoidoscopy with biopsy revealed nodularlymphoid hyperplasia and histopathologic evidence of colitischaracterized by edematous and hemorrhagic focal areas witheosinophil infiltration (more than 6 per high-power-field) andoccasional lymphoid nodes. suggesting a diagnosis of allergiccolitis. We therefore reinforced the importance ora dairy-fi'eediet while breast feeding.
The patient was a healthy, full-term infant delivered bycesarean birth. with normal growth for age.
Blood and mucus persisted in stool, ulbeit at a lowerintensity. At 3 months. the infant began to produce waterystool. S bOlllaldii (liL 250 sachets) was prescribed. Thechild began to vomit prohJsely approximately 2 hours afteringestion, ,,,,ith spontaneous resolution within a few hours.Three weeks later. as gastrointestinal symptoms persisted(intermittent vvatery stools with blood and mucus). shewas again treated ",ith S bOlllal'dii, 2 hours after which sheexperienced ,ill episode of uncontrollable vomiting, with severeprostration. She was seen at the emergency department. whereshe received replacement fluid therapy. The vom iting resolvedon the same day. and no other signs of inJection were observed.
At that time. she was referred to our lmmunoallergologyDepartment. Skin prick tests and specific IgE for 111 ilk,casein, n-laetalbumin, and [.I-lactoglobulin were negative.We diagnosed non-·IgE-mediated cow's milk allergy and
reinforced the importance for both mother and infant of adairy-l1-ee diet. Cow's milk was successfully introduced tothe patient's diet at the age of 12 months.
Despite the suspicion of S houlardii hypersensitivityreactions, the parents refused an oral challenge test with thisprobiotic because their child had tolerated another probiotic(Lactohacillus reuteri).
S houlardii is considered a safe probiotic, althoughhypcrsensitivity reactions-while ran;--·-ean occur [3,4].
The patient we report presented symptoms indicative ofgastrointestinal allcrgy. such as that occurring in food protein-induced enterocolitis syndrome (FPfES). as in a previouslypublished case of hypersensitivity to S houlardii [3]. Weexcluded a possible IgE-mediated reaction owing to theabsence of associated classic allergic skin or respiratorymanifestations and the late onset of the reaction [4,5.1-
FPl ES is a non-lgE-mediated food allergy characterizedby profuse vomiting accompanied by pallor and lethargywithin I to 4 hours (usually 2 hours) after ingestion of theoffending food. It also can be followed by diarrhea in 5 to 8hours in some patients and may last J(lr up to 24 hours afterexposure [5,6].
The diagnosis is based on the clinical history, recognitionof clinical symptoms, exclusion of other etiologies, and asupervised oral challenge test. Although the challenge testis considered the gold standard, a history of severe, repeatedreactions in a patient who becomes asymptomatic afterelimination of the suspected culprit is sufficient to make adiagnosis 15,6].
We assessed causality in this suspected adverse drugreaction using the probability scale of Naranjo [7], in \vhicha score of 10 was obtaincd, indicating a definitive diagnosis.Even though the patient"s parents rcftlsed an oral challengetest. the Naranjo score supports the link between exposure toS' houlardii and FPIES-like reaction [7].
According to international consensus guidelines [51, themajor criterion and at least 3 minor criteria must be met forthe diagnosis of FPIES. Our patient experienced profusevomiting 2 hours after ingestion of S boulardii and. ,vhenIe-exposed. experienced a second episode of repetiti vevomiting with extreme lethargy and need for replacementfluid, therapy in the emergency department. thus fulfilling therequirements for the diagnosis ofFPIES-like reaction. Basedon the convincing clinical history with a repeated reaction tothe same drug. complete resolution over a 111atterof hours,and the absence of infectious gastroenteritis, the reaction wedescribe can be considered an FPIES-like allergic reactioncaused by S bOlllardii.
We hope that our findings raise awareness of possiblehypersensitivity reaction to S boulardii.
Funding
The authors declare that no funding was received for thepresent study.
Conflicts of Interest
The authors declare that they have no conflicts ofinterest.
1. Kelesidisl, Pothoulakis C. Efficacy and safety of the plObioticSaccharomyces boulardii for the prevention and therapyof gastrointestinal disorders. Ther Adv Ciastroenterol.20'12;5(2):11125.
2. Vandenplas Y. Brunser 0, SzaJewska H. Saccharomycesboulardii in childnood. Eur J Pediatr 2009; 168: 2';3-65.
3. Hwang JB, Kang KJ, Kang YN, Kim AS. Probiotic gastrointestinalallerqic reaction caused by Saccharomyces boulardii. AnnAllergy i\sthma Immunol. 2009 Jul; 103(1 )87-8.
4, Kartal 0, Demirel F, Baysan !-\, Gulec M, Yesillik S, Uyanyk M,et al. !-\f1 unexpected allergic reaction with Saccharomycesboulardii: a case report. Clin Transl Allergy. 2014;4(Suppl3):['100.
5. Nowak-Wegrzyn A, Chehade \11, Groetd, M, Spergel J,Wood R, Allen K, et al. International consr:nsus guidelinesfor the diagnosis and management of food protein·induced enterocolitis syndrome, J Allergy Clin Irnmunol.2017;139:1111-26.
6. Nowak-Wegrzyn A, Jarocka-Cyrta E, Moschione Ca~tlo APB.Food protein-Induced enterocolitis syndrome. J InvestigAllergol (In Immunol. 2017; vol.27 (1): 1-18,
7. Doherty MJ. i\lgorithrns for assessing the probability ofan Adverse Drug Reaction. Respiratory Medicine CME 2,2009;63-67.
ill Manuscript received June 5, 2017; accepted for publication July31,2017
CMia AlvesSelvi<;o de Imunoalergologia
Hospital D. EstefaniaRua Jacinta Marto, 1169-045 Lisboa, Portugal
E-mail: [email protected]
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