ASTARITA C

AYALA, FABIO

D'ARIENZO A.

GARGANO D

MANGUSO F

PELUSO, ROSARIO

SCARPA, RAFFAELE

STAIANO T

Archivio della ricerca - Università degli studi di Napoli Federico II

Int J Colorectal Dis (2005) 20:79–80DOI 10.1007/s00384-004-0626-0 L E T T ER TO THE ED I TORFrancesco MangusoTeresa StaianoCorrado AstaritaRaffaele ScarpaRosario PelusoDomenico GarganoFabio AyalaAgesilao D’ArienzoConsecutive occurrence of rhinoconjunctivitis,seronegative spondyloarthritisand pyoderma gangrenosum in a patientwith ulcerative colitisAccepted: 26 May 2004Published online: 14 August 2004 Springer-Verlag 2004F. Manguso ()) · T. Staiano ·A. D’ArienzoSection of Gastroenterology,Department of Clinicaland Experimental Medicine,Faculty of Medicine,Federico II University of Naples,Naples, Italye-mail: manguso@unina.itTel.: +39-081-7462708Fax: +39-081-7462708C. Astarita · D. GarganoSection of Allergologyand Clinical Immunology,Department of Clinicaland Experimental Medicine,Second University of Naples,Naples, ItalyR. Scarpa · R. PelusoSection of Rheumatology,Department of Clinicaland Experimental Medicine,Faculty of Medicine,Federico II University of Naples,Naples, ItalyF. AyalaSection of Dermatology,Department of Systematic Pathology,Faculty of Medicine,Federico II University of Naples,Naples, ItalyDear Editor:Ulcerative colitis (UC) is a chronicinflammatory disorder of the colonwith a clinical course characterizedby periods of exacerbation and re-mission. Its aetiology is unknown,although genetic, environmental, in-fectious and immunological factorsare thought to be involved. In UC,data seem insufficient to characterizethe T-helper (Th) cell cytokine phe-notype as either Th1 or Th2, eventhough there is more evidence ofantibody-mediated hypersensitivityin tissue damaging, and less evidenceof Th1 cell responses. UC is associ-ated with a variety of extraintestinalmanifestations (EIMs) and up to one-third of patients experience at leastone in their lifetime. The most fre-quent EIM in UC is an arthritis in-cluded among the seronegativespondyloarthropathies (SpA), whichis characterized by inflammatory ax-ial spine disease, asymmetric periph-eral arthritis and enthesopathy. Fewstudies have been performed to es-tablish the profile of pro-inflamma-tory cytokines in SpA, and a Thpredominance has not yet been dem-onstrated, even though the efficacy ofanti-TNF-alpha treatment and otherindirect evidence suggest a Th1 im-plication. Pyoderma gangrenosum(PG) is a dermatologic manifestationseen in about 0.5–5% of UC patients.There are four prototypic forms ofPG (ulcerative, pustular, bullous andvegetative), but even in PG the Thprofile has not yet been clarified. Inthis report, we describe the case of aUC patient who showed the consec-utive occurrence of allergicrhinoconjunctivitis, SpA and PG overa period of 10 years.A 14-year-old boy presented to theGastroenterology Unit in November1991 with a 1-year history of UC.He had been subjected to tonsillec-tomy and adenoidectomy at the ageof 6 years. The pan-colonoscopyshowed an active pancolitis and bi-opsy confirmed the diagnosis. Overthe next 4 years he was admitted tothe hospital on four occasions formoderate to severe activity of theintestinal disease. The therapy wasestablished according to the clinicalcourse of UC and was based onchronic administration of mesalazine.Corticosteroids were added toaminosalicylates when needed. At theage of 16, he started to smoke regu-larly 10 cigarettes per day. In 1993 heunderwent an allergologic evaluationfor the presence of an increasing pe-rennial rhinoconjunctivitis that hadstarted at the age of 12 with a mild,intermittent clinical profile. He wassubjected to skin prick testing (SPT)with a panel of common, localallergens. The patient showed a moreintense positive SPT response tomites than to histamine (positivecontrol). Total serum IgE levels were452 mg/l (normal range 12–240 mg/l),and the circulating eosinophil countwas 220 cells/ml (normal count80<500 cells/ml). Allergic rhinocon-junctivitis was confirmed after achallenge test and treated with anti-histamine drugs, topical corticoste-roid and ophthalmic preparations.No injection immunotherapy wasplanned. Over the next 6 years wenoted an improvement in the allergicsymptoms, which gradually disap-peared, together with a slow im-provement in the intestinal disease inboth severity and number of flare-ups. An allergologic evaluation madein June 1998 showed negative skinreaction to the same SPT panel in thepresence of normal reactivity to his-tamine. Total serum IgE levels fellwithin the normal range (194 mg/l).Eosinophil count was 140 cells/ml.The allergologic status remained un-modified during subsequent follow-ups, and the pan-colonoscopy withbiopsy confirmed the remission ofUC. In October 1999, the patient wasobserved at the Rheumatology Unitwith severe inflammatory polyarthri-tis. A bone scintiscan examinationwas performed after i.v. administra-tion of 99mTc methylene diphospho-nate (555 MBq), using a large-fieldgamma camera (Starport 400 AT,General Electric, Milwaukee, WI,USA), to determine the distributionand activity of the disease. The sac-roiliac involvement was defined onthe basis of the sacroiliac/sacrumuptake ratio as suggested by Percyand Lentle [1]. The search for serumrheumatoid factor was negative. Ar-thritis was classified as seronegativespondyloarthropathy, based on theEuropean Spondyloarthropathy StudyGroup criteria, and required treatmentwith methotrexate (10 mg/week). Thehuman leukocyte antigen (HLA)haplotypes, tested with a microlym-phocytotoxicity technique, showedno particular rheumatologic suscep-tibilities (A24, A28, B62, BW4,BW6, CW3, CW7, DR11, DR13,DR52, DQ7). In February 2000, thepatient was admitted to the Derma-tologic Unit with a large ulcerativepyoderma gangrenosum in the leftleg, which required a systemic treat-ment with prednisone (40 mg/day)and a local therapeutic option withCastellani’s paint. At the same time,there appeared in the right leg a smallfollicular-based pustule (initial lesionof PG) that did not break down andrapidly disappeared.The patient’s history showed theoccurrence of UC together with IgE-mediated allergic disease. During thecourse of UC, symptoms of atopydisappeared at the same time as animprovement in the intestinal disease,while SpA and PG emerged. T-helperlymphocytes have been shown toplay an important role in immuneresponses, and at least three func-tional subsets of Th cells have beendistinguished according to their cy-tokine secretion profiles. Each subsetregulates the others in a dynamicprocess. A prominence of one orother of the Th1 and Th2 phenotypesis described in several diseases, andcross-regulation of the Th1 immuneresponse to suppress Th2 disease, orvice versa, has been established inhuman and animal models. Recently,we have shown that UC atopic dis-eases and SpA, as well as atopicdiseases and allergic contact derma-titis (ACD), are strongly polarizedimmunological conditions tendingto mutual exclusion, while SpA andACD may coexist. To explain themutual exclusion of atopic diseasesand SpA or ACD, and the coexistenceof SpA and ACD, we have postulatedthat the diseases might lie at oppositeor equal ends of the immunologicalspectrum according to T-helper cellresponses. It should be noted thatatopy is a well-known Th2 condition,while in ACD the Th1 subtype pre-dominates.The clinical course shown by ourpatient seems to indicate high Th2activity at the beginning of his med-ical history. In contrast, the late onsetof SpA and PG, together with a bettercourse of intestinal disease and thedisappearance of atopy symptoms,suggests that immunological changes,promoting Th1 manifestations, oc-curred later in the course of UC.These results clearly indicate dy-namic changes in the immune re-sponse in our patient. From a dy-namic point of view, the immunesystem could express over time, atthe individual level, different re-sponses according to the prevalent Thprofile, as found in a patient withrheumatoid arthritis. In particular, ourcase possibly represents the clinicalexpression of the linear differentia-tion model postulated for lymphocytedevelopment. In this model, Th2represents an immature pattern that,through a Th0 stadium, terminallydifferentiates along type-1 effectorcells. In fact, our patient, after de-veloping UC together with rhino-conjunctivitis, and after a period ofremission of both diseases, showedarthritis and PG. Unfortunately, thestimuli promoting the Th1/Th2 bal-ancing or alternation of different Th1/Th2 polarized diseases in vivo remainfar from clear. Recently, we havedemonstrated that cigarette smokingand appendectomy are risk factors forthe development of EIMs in patientswith UC. In our case, the patient wasexposed to both risk factors, whichmay have contributed to a T-helperprofile modification over time.The development of these con-cepts will not only allow a betterunderstanding of the mechanismsinvolved in the pathogenesis andcourse of UC, but will also providethe basis for the development ofnovel strategies for its treatment.Acknowledgements Francesco Mangusois the recipient of a fellowship from Ex-traintestinal Manifestations in Inflammato-ry Bowel Disease, Study of the ImmuneResponses and the Cytokine Profiles,Federico II University of Naples, whichwas funded in part by a grant from GiulianiS.p.A., Milan, Italy.Reference1. Percy FS, Lentle B (1980) Radiologicaland scintigraphic features. In: Moll JM(ed) Ankylosing spondylitis. ChurchillLivingstone, Edinburgh, p 76

Consecutive occurrence of rhinoconjunctivitis, seronegative spondyloarthritis and pyoderma gangrenosum in a patient with ulcerative colitis.

https://www.iris.unina.it/retrieve/handle/11588/203250/298113/Int%20J%20colorectal%20Dis%202005.pdf

Consecutive occurrence of rhinoconjunctivitis, seronegative spondyloarthritis and pyoderma gangrenosum in a patient with ulcerative colitis.

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