Systemic involvement of acute generalized exanthematous pustulosis: a retrospective study on 58 patients
Identifieur interne : 000198 ( France/Analysis ); précédent : 000197; suivant : 000199Systemic involvement of acute generalized exanthematous pustulosis: a retrospective study on 58 patients
Auteurs : C. Hotz [France] ; L. Valeyrie-Allanore [France] ; C. Haddad [France] ; S. Bouvresse [France] ; N. Ortonne [France] ; T. A. Duong [France] ; S. Ingen-Housz-Oro [France] ; J. C. Roujeau [France] ; P. Wolkenstein [France] ; O. Chosidow [France]Source :
- British Journal of Dermatology [ 0007-0963 ] ; 2013-12.
English descriptors
- Teeft :
- Acute, Agep, Alanine aminotransferase, Amoxicillin, Amoxicillin rechallenge, Arch dermatol, Aspartate aminotransferase, Blood gases, Body surface area, British association, British journal, Bronchoalveolar lavage, Cholestasis, Creatinine, Cutaneous, Dermatol, Dermatologist, Dermatology, Diffuse, Distress syndrome, Drug reactions, Euroscar, Euroscar score, Exanthematous, Exanthematous pustulosis, Generalized pustular psoriasis, Hepatic, Hepatic dysfunction, Hepatic function tests, Hepatocellular, High fever, Hotz, Involvement, Liver cholestasis, Neutrophil, Neutrophil count, Normal range, Normal ranges, Past infections, Pristinamycin, Protein level, Psoriasis, Pulmonary involvement, Pustular, Pustule, Pustulosis, Putative culprit drugs, Rechallenge, Renal, Renal function tests, Retrospective study, Severe cutaneous, Severe cutaneous involvement, Syndrome, Systemic, Systemic involvement, Systemic symptoms, Upper limit.
Abstract
Background: Acute generalized exanthematous pustulosis (AGEP) is a severe cutaneous adverse reaction characterized by rash with sterile pustules, high fever and elevated circulating neutrophil counts. Objectives: To investigate the frequency and clinical features of AGEP systemic involvement. Methods: This retrospective study included all patients hospitalized in our department between 2000 and 2010 with a discharge diagnosis of AGEP. Patients had to fulfil the following criteria: (i) a specific EuroSCAR score > 4 and (ii) biological and radiological work‐up available. Results: Among the 58 patients enrolled, 10 had at least one systemic involvement: hepatic function test results were abnormal for seven; six had renal insufficiency; two developed acute respiratory distress, with one patient's bronchoalveolar lavage fluid containing many neutrophils but no microorganisms; one was agranulocytotic. Mean peripheral neutrophil counts and mean C‐reactive protein levels were elevated significantly in patients with systemic involvement. Amoxicillin rechallenge and hospitalization duration were associated with systemic involvement. AGEP systemic involvement was observed in 17% of cases studied, including liver, kidney, bone‐marrow and lung involvement. Outcomes were favourable after drug withdrawal, and symptomatic and topical steroid treatments. Conclusions: The neutrophil count–systemic involvement association may suggest a role for neutrophils in AGEP systemic involvement. Physicians should be aware of the possibility of systemic involvement in AGEP and should actively look for signs of extracutaneous reactions.
What's already known about this topic? Internal organ dysfunction is not usually expected in acute generalized exanthematous pustulosis (AGEP). Systemic involvement in AGEP has rarely been reported. What does this study add? AGEP systemic involvement does exist. Increased physician awareness and better knowledge of the condition should improve its management.
Url:
DOI: 10.1111/bjd.12502
Affiliations:
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Hotz</name><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>Department of Dermatology, Referral Center for Toxic and Autoimmune Blistering Diseases, Université–Paris‐Est Créteil Val de Marne (UPEC), Henri‐Mondor Hospital, 51, avenue du Maréchal de Tassigny, 94010, Créteil CEDEX</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation></author><author><name sortKey="Valeyrie Llanore, L" sort="Valeyrie Llanore, L" uniqKey="Valeyrie Llanore L" first="L." last="Valeyrie-Allanore">L. Valeyrie-Allanore</name><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>Department of Dermatology, Referral Center for Toxic and Autoimmune Blistering Diseases, Université–Paris‐Est Créteil Val de Marne (UPEC), Henri‐Mondor Hospital, 51, avenue du Maréchal de Tassigny, 94010, Créteil CEDEX</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>Assistance Publique–Hôpitaux de Paris (APHP), LIC EA 4393, Université–Paris‐Est Créteil Val de Marne (UPEC), Henri‐Mondor Hospital, 51, avenue du Maréchal de Tassigny, 94010, Créteil CEDEX</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation><affiliation></affiliation><affiliation wicri:level="1"><country wicri:rule="url">France</country></affiliation></author><author><name sortKey="Haddad, C" sort="Haddad, C" uniqKey="Haddad C" first="C." last="Haddad">C. Haddad</name><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>Department of Dermatology, Referral Center for Toxic and Autoimmune Blistering Diseases, Université–Paris‐Est Créteil Val de Marne (UPEC), Henri‐Mondor Hospital, 51, avenue du Maréchal de Tassigny, 94010, Créteil CEDEX</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation></author><author><name sortKey="Bouvresse, S" sort="Bouvresse, S" uniqKey="Bouvresse S" first="S." last="Bouvresse">S. Bouvresse</name><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>Department of Dermatology, Referral Center for Toxic and Autoimmune Blistering Diseases, Université–Paris‐Est Créteil Val de Marne (UPEC), Henri‐Mondor Hospital, 51, avenue du Maréchal de Tassigny, 94010, Créteil CEDEX</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation></author><author><name sortKey="Ortonne, N" sort="Ortonne, N" uniqKey="Ortonne N" first="N." last="Ortonne">N. Ortonne</name><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>Department of Pathology, Assistance Publique – Hôpitaux de Paris (APHP), Université – Paris‐Est Créteil Val de Marne (UPEC), Henri‐Mondor Hospital, 51, avenue du Maréchal de Tassigny, 94010, Créteil CEDEX</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation></author><author><name sortKey="Duong, T A" sort="Duong, T A" uniqKey="Duong T" first="T. A." last="Duong">T. A. Duong</name><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>Department of Dermatology, Referral Center for Toxic and Autoimmune Blistering Diseases, Université–Paris‐Est Créteil Val de Marne (UPEC), Henri‐Mondor Hospital, 51, avenue du Maréchal de Tassigny, 94010, Créteil CEDEX</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation></author><author><name sortKey="Ingen Ousz Ro, S" sort="Ingen Ousz Ro, S" uniqKey="Ingen Ousz Ro S" first="S." last="Ingen-Housz-Oro">S. Ingen-Housz-Oro</name><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>Department of Dermatology, Referral Center for Toxic and Autoimmune Blistering Diseases, Université–Paris‐Est Créteil Val de Marne (UPEC), Henri‐Mondor Hospital, 51, avenue du Maréchal de Tassigny, 94010, Créteil CEDEX</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation></author><author><name sortKey="Roujeau, J C" sort="Roujeau, J C" uniqKey="Roujeau J" first="J. C." last="Roujeau">J. C. Roujeau</name><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>Department of Dermatology, Referral Center for Toxic and Autoimmune Blistering Diseases, Université–Paris‐Est Créteil Val de Marne (UPEC), Henri‐Mondor Hospital, 51, avenue du Maréchal de Tassigny, 94010, Créteil CEDEX</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation></author><author><name sortKey="Wolkenstein, P" sort="Wolkenstein, P" uniqKey="Wolkenstein P" first="P." last="Wolkenstein">P. Wolkenstein</name><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>Department of Dermatology, Referral Center for Toxic and Autoimmune Blistering Diseases, Université–Paris‐Est Créteil Val de Marne (UPEC), Henri‐Mondor Hospital, 51, avenue du Maréchal de Tassigny, 94010, Créteil CEDEX</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>Assistance Publique–Hôpitaux de Paris (APHP), LIC EA 4393, Université–Paris‐Est Créteil Val de Marne (UPEC), Henri‐Mondor Hospital, 51, avenue du Maréchal de Tassigny, 94010, Créteil CEDEX</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation></author><author><name sortKey="Chosidow, O" sort="Chosidow, O" uniqKey="Chosidow O" first="O." last="Chosidow">O. Chosidow</name><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>Department of Dermatology, Referral Center for Toxic and Autoimmune Blistering Diseases, Université–Paris‐Est Créteil Val de Marne (UPEC), Henri‐Mondor Hospital, 51, avenue du Maréchal de Tassigny, 94010, Créteil CEDEX</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation><affiliation wicri:level="3"><country xml:lang="fr">France</country><wicri:regionArea>INSERM, Centre d'Investigation Clinique 006, APHP, F‐94000, Créteil</wicri:regionArea><placeName><region type="region" nuts="2">Île-de-France</region><settlement type="city">Créteil</settlement></placeName></affiliation></author></analytic><monogr></monogr><series><title level="j" type="main">British Journal of Dermatology</title><title level="j" type="alt">BRITISH JOURNAL OF DERMATOLOGY</title><idno type="ISSN">0007-0963</idno><idno type="eISSN">1365-2133</idno><imprint><biblScope unit="vol">169</biblScope><biblScope unit="issue">6</biblScope><biblScope unit="page" from="1223">1223</biblScope><biblScope unit="page" to="1232">1232</biblScope><biblScope unit="page-count">10</biblScope><date type="published" when="2013-12">2013-12</date></imprint><idno type="ISSN">0007-0963</idno></series></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0007-0963</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="Teeft" xml:lang="en"><term>Acute</term><term>Agep</term><term>Alanine aminotransferase</term><term>Amoxicillin</term><term>Amoxicillin rechallenge</term><term>Arch dermatol</term><term>Aspartate aminotransferase</term><term>Blood gases</term><term>Body surface area</term><term>British association</term><term>British journal</term><term>Bronchoalveolar lavage</term><term>Cholestasis</term><term>Creatinine</term><term>Cutaneous</term><term>Dermatol</term><term>Dermatologist</term><term>Dermatology</term><term>Diffuse</term><term>Distress syndrome</term><term>Drug reactions</term><term>Euroscar</term><term>Euroscar score</term><term>Exanthematous</term><term>Exanthematous pustulosis</term><term>Generalized pustular psoriasis</term><term>Hepatic</term><term>Hepatic dysfunction</term><term>Hepatic function tests</term><term>Hepatocellular</term><term>High fever</term><term>Hotz</term><term>Involvement</term><term>Liver cholestasis</term><term>Neutrophil</term><term>Neutrophil count</term><term>Normal range</term><term>Normal ranges</term><term>Past infections</term><term>Pristinamycin</term><term>Protein level</term><term>Psoriasis</term><term>Pulmonary involvement</term><term>Pustular</term><term>Pustule</term><term>Pustulosis</term><term>Putative culprit drugs</term><term>Rechallenge</term><term>Renal</term><term>Renal function tests</term><term>Retrospective study</term><term>Severe cutaneous</term><term>Severe cutaneous involvement</term><term>Syndrome</term><term>Systemic</term><term>Systemic involvement</term><term>Systemic symptoms</term><term>Upper limit</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract">Background: Acute generalized exanthematous pustulosis (AGEP) is a severe cutaneous adverse reaction characterized by rash with sterile pustules, high fever and elevated circulating neutrophil counts. Objectives: To investigate the frequency and clinical features of AGEP systemic involvement. Methods: This retrospective study included all patients hospitalized in our department between 2000 and 2010 with a discharge diagnosis of AGEP. Patients had to fulfil the following criteria: (i) a specific EuroSCAR score > 4 and (ii) biological and radiological work‐up available. Results: Among the 58 patients enrolled, 10 had at least one systemic involvement: hepatic function test results were abnormal for seven; six had renal insufficiency; two developed acute respiratory distress, with one patient's bronchoalveolar lavage fluid containing many neutrophils but no microorganisms; one was agranulocytotic. Mean peripheral neutrophil counts and mean C‐reactive protein levels were elevated significantly in patients with systemic involvement. Amoxicillin rechallenge and hospitalization duration were associated with systemic involvement. AGEP systemic involvement was observed in 17% of cases studied, including liver, kidney, bone‐marrow and lung involvement. Outcomes were favourable after drug withdrawal, and symptomatic and topical steroid treatments. Conclusions: The neutrophil count–systemic involvement association may suggest a role for neutrophils in AGEP systemic involvement. Physicians should be aware of the possibility of systemic involvement in AGEP and should actively look for signs of extracutaneous reactions.</div><div type="abstract" xml:lang="en">What's already known about this topic? Internal organ dysfunction is not usually expected in acute generalized exanthematous pustulosis (AGEP). Systemic involvement in AGEP has rarely been reported. What does this study add? AGEP systemic involvement does exist. Increased physician awareness and better knowledge of the condition should improve its management.</div></front></TEI><affiliations><list><country><li>France</li></country><region><li>Île-de-France</li></region><settlement><li>Créteil</li></settlement></list><tree><country name="France"><region name="Île-de-France"><name sortKey="Hotz, C" sort="Hotz, C" uniqKey="Hotz C" first="C." last="Hotz">C. Hotz</name></region><name sortKey="Bouvresse, S" sort="Bouvresse, S" uniqKey="Bouvresse S" first="S." last="Bouvresse">S. Bouvresse</name><name sortKey="Chosidow, O" sort="Chosidow, O" uniqKey="Chosidow O" first="O." last="Chosidow">O. Chosidow</name><name sortKey="Chosidow, O" sort="Chosidow, O" uniqKey="Chosidow O" first="O." last="Chosidow">O. Chosidow</name><name sortKey="Duong, T A" sort="Duong, T A" uniqKey="Duong T" first="T. A." last="Duong">T. A. Duong</name><name sortKey="Haddad, C" sort="Haddad, C" uniqKey="Haddad C" first="C." last="Haddad">C. Haddad</name><name sortKey="Ingen Ousz Ro, S" sort="Ingen Ousz Ro, S" uniqKey="Ingen Ousz Ro S" first="S." last="Ingen-Housz-Oro">S. Ingen-Housz-Oro</name><name sortKey="Ortonne, N" sort="Ortonne, N" uniqKey="Ortonne N" first="N." last="Ortonne">N. Ortonne</name><name sortKey="Roujeau, J C" sort="Roujeau, J C" uniqKey="Roujeau J" first="J. C." last="Roujeau">J. C. Roujeau</name><name sortKey="Valeyrie Llanore, L" sort="Valeyrie Llanore, L" uniqKey="Valeyrie Llanore L" first="L." last="Valeyrie-Allanore">L. Valeyrie-Allanore</name><name sortKey="Valeyrie Llanore, L" sort="Valeyrie Llanore, L" uniqKey="Valeyrie Llanore L" first="L." last="Valeyrie-Allanore">L. Valeyrie-Allanore</name><name sortKey="Valeyrie Llanore, L" sort="Valeyrie Llanore, L" uniqKey="Valeyrie Llanore L" first="L." last="Valeyrie-Allanore">L. Valeyrie-Allanore</name><name sortKey="Wolkenstein, P" sort="Wolkenstein, P" uniqKey="Wolkenstein P" first="P." last="Wolkenstein">P. Wolkenstein</name><name sortKey="Wolkenstein, P" sort="Wolkenstein, P" uniqKey="Wolkenstein P" first="P." last="Wolkenstein">P. Wolkenstein</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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