Letter to the Editor Two Cases of Extensive Limb Swelling After Influenza Vaccination To The Editors:
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wo children were referred to our clinic on separate occasions, both presenting with extensive swelling of the left upper arm in which they had received an influenza vaccination 2 days earlier. We diagnosed both children with extensive limb swelling (ELS) and no management was required. As not many clinicians in our department were familiar with ELS, we found it important to present these cases and to stress the self-limiting character of the condition to prevent unnecessary diagnostic testing and treatment. A boy of 5 years complained about his vaccinated arm the day following vaccination (Vaxigrip, intramuscular triceps, Sanofi Pasteur, Lyon, France). The second day after injection the parents noticed extreme swelling and redness of his vaccinated limb. We saw impressive circumferential swelling of the left arm, extending from just below the shoulder to halfway down the forearm (see Figure, Supplemental Digital Content 1, http://links.lww.com/ INF/B956). The swelling was erythematous and was warmer than the surrounding skin. Movements of the elbow and shoulder were not compromised. There was no tenderness upon palpation. Blood tests showed a normal blood count and low C-reactive protein. A girl of 4 years developed a localized swelling around the injection site a few hours after vaccination (Vaxigrip, intramuscular deltoid). Over the following days the swelling extended further along the arm and the arm became slightly painful. She did not complain of itch. Because an allergic reaction was suspected, desloratadine was prescribed, which had no effect on the symptoms. Our physical examination showed swelling and erythema of the left upper arm extending from the shoulder to just above the elbow (see Figure, Supplemental Digital Content 2, http://links.lww.com/INF/B957). There were some small blisters located centrally around the injection site. Movement of the arm was not restricted. Palpation was
The authors have no funding or conflicts of interest to disclose. Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal’s website (www.pidj.com).
mildly painful. Both children appeared well and did not have a fever. Their medical history showed atopic constitution. We diagnosed both children with ELS. After a few days the condition spontaneously resolved without sequelae. According to The Netherlands Pharmacovigilance Centre, ELS can be described as a swelling occurring in the same limb to which a vaccination has been administered, extending from the joint proximal, to the joint distal of the injection site, or circumferencing entirely around the limb.1 In the majority of patients, symptoms present within 1 day after vaccination— sometimes as early as only hours after the injection—and in all cases swelling becomes apparent within 48 hours.1,2 Swelling is usually accompanied by other signs of inflammation, such as erythema, warmth or pain.1,2 The pain is usually mild and there is no functional impairment. Other symptoms have also been reported, such as blistering, which was also seen with one of our patients.1,2 The children appear well and do not experience a high-grade fever.1,2 The exact incidence of ELS is unclear, which is partly because no uniform definition exists and cases may also be misdiagnosed or underreported. In the literature ELS is mainly known for its occurrence in children after a booster vaccination with diphtheria tetanus acellular pertussis-containing vaccine; however, it can occur with any vaccine and it is not unusual for it to develop after influenza vaccination.2 The pathophysiology of ELS has not been elucidated. Several hypotheses exist, including it being a continuum of a local inflammatory reaction1 and being a contribution of a type III3 or IV hypersensitivity reaction.4 Although the pathophysiology remains unclear and recurrence rates are high (reported up to 20%), it is thought that revaccination is safe as ELS is generally well tolerated and the condition is self-limiting.5 It is important to note that all authors investigated ELS only with regard to diphtheria tetanus acellular pertussis-containing vaccines. Due to its striking appearance, ELS might be mistaken for bacterial cellulitis or a type I allergic reaction. As in our cases, this might lead to unnecessary diagnostic testing and treatment. It is important for the clinician to recognize ELS as a benign and self-limiting condition that does not require intervention.
Emily Cohen, MD Sabrin Tahri, BSc Ingrid Lukkassen, MD
REFERENCES 1. Netherlands Pharmacovigilance Centre LAREB. Season influenza vaccines and extensive limb swelling (ELS). Lareb Q Rep. 2012;2:28–34. 2. Woo EJ, Burwen DR, Gatumu SN, et al; Vaccine Adverse Event Reporting System Working Group. Extensive limb swelling after immunization: reports to the vaccine adverse event reporting system. Clin Infect Dis. 2003;37:351–358. 3. Liese JG, Stojanov S, Zink TH, et al. Safety and immunogenicity of Biken acellular pertussis vaccine in combination with diphtheria and tetanus toxoid as a fifth dose at four to six years of age. Munich Vaccine Study Group. Pediatr Infect Dis J. 2001;20:981–988. 4. Scheifele DW, Halperin SA, Ferguson AC. Assessment of injection site reactions to an acellular pertussis-based combination vaccine, including novel use of skin tests with vaccine antigens. Vaccine. 2001;19:4720–4726. 5. Rennels MB, Black S, Woo EJ, et al. Safety of a fifth dose of diphtheria and tetanus toxoid and acellular pertussis vaccine in children experiencing extensive, local reactions to the fourth dose. Pediatr Infect Dis J. 2008;27:464–465.
Risk of Rotavirus Vaccination for Children with SCID To the Editors: uinn et al.1 demonstrated reduced morbidity and mortality from acute gastroenteritis by rotavirus vaccination in young children in Australia. We agree with the consensus statement highlighting these benefits in both developed and developing settings, which clearly outweigh the small increased risk of intussusception.2 Nevertheless, we think that the additional risk rotavirus vaccination poses for children with severe combined immunodeficiency (SCID) needs comment. Recently, we saw a 21-month-old boy from a country in the Middle-East who was in reduced general condition for further diagnosis and management at our center. Although his older brother died at 20 months of age suffering from postvaccinational BCGitis and severe cytomegalovirus infection, our patient had received rotavirus vaccine (Rotarix, GlaxoSmithKline Biologicals, Rixensart, Belgium) twice at 3 and 4 months of age. Ever since, he suffered from chronic gastroenteritis, which resulted in severe stunting and wasting. The patient had a x-linked hemizygous mutation
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The authors have no conflicts of interest or funding to disclose. Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal’s website (www.pidj.com).
Copyright © 2014 by Lippincott Williams & Wilkins ISSN: 0891-3668/15/3401-0114 DOI: 10.1097/INF.0000000000000475
Department of Pediatrics St. Antonius Hospital Utrecht/Nieuwegein, The Netherlands
114 | www.pidj.com
The Pediatric Infectious Disease Journal • Volume 34, Number 1, January 2015
Copyright © 2014 by Lippincott Williams & Wilkins DOI: 10.1097/INF.0000000000000507
T
wo children were referred to our clinic on separate occasions, both presenting with extensive swelling of the left upper arm in which they had received an influenza vaccination 2 days earlier. We diagnosed both children with extensive limb swelling (ELS) and no management was required. As not many clinicians in our department were familiar with ELS, we found it important to present these cases and to stress the self-limiting character of the condition to prevent unnecessary diagnostic testing and treatment. A boy of 5 years complained about his vaccinated arm the day following vaccination (Vaxigrip, intramuscular triceps, Sanofi Pasteur, Lyon, France). The second day after injection the parents noticed extreme swelling and redness of his vaccinated limb. We saw impressive circumferential swelling of the left arm, extending from just below the shoulder to halfway down the forearm (see Figure, Supplemental Digital Content 1, http://links.lww.com/ INF/B956). The swelling was erythematous and was warmer than the surrounding skin. Movements of the elbow and shoulder were not compromised. There was no tenderness upon palpation. Blood tests showed a normal blood count and low C-reactive protein. A girl of 4 years developed a localized swelling around the injection site a few hours after vaccination (Vaxigrip, intramuscular deltoid). Over the following days the swelling extended further along the arm and the arm became slightly painful. She did not complain of itch. Because an allergic reaction was suspected, desloratadine was prescribed, which had no effect on the symptoms. Our physical examination showed swelling and erythema of the left upper arm extending from the shoulder to just above the elbow (see Figure, Supplemental Digital Content 2, http://links.lww.com/INF/B957). There were some small blisters located centrally around the injection site. Movement of the arm was not restricted. Palpation was
The authors have no funding or conflicts of interest to disclose. Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal’s website (www.pidj.com).
mildly painful. Both children appeared well and did not have a fever. Their medical history showed atopic constitution. We diagnosed both children with ELS. After a few days the condition spontaneously resolved without sequelae. According to The Netherlands Pharmacovigilance Centre, ELS can be described as a swelling occurring in the same limb to which a vaccination has been administered, extending from the joint proximal, to the joint distal of the injection site, or circumferencing entirely around the limb.1 In the majority of patients, symptoms present within 1 day after vaccination— sometimes as early as only hours after the injection—and in all cases swelling becomes apparent within 48 hours.1,2 Swelling is usually accompanied by other signs of inflammation, such as erythema, warmth or pain.1,2 The pain is usually mild and there is no functional impairment. Other symptoms have also been reported, such as blistering, which was also seen with one of our patients.1,2 The children appear well and do not experience a high-grade fever.1,2 The exact incidence of ELS is unclear, which is partly because no uniform definition exists and cases may also be misdiagnosed or underreported. In the literature ELS is mainly known for its occurrence in children after a booster vaccination with diphtheria tetanus acellular pertussis-containing vaccine; however, it can occur with any vaccine and it is not unusual for it to develop after influenza vaccination.2 The pathophysiology of ELS has not been elucidated. Several hypotheses exist, including it being a continuum of a local inflammatory reaction1 and being a contribution of a type III3 or IV hypersensitivity reaction.4 Although the pathophysiology remains unclear and recurrence rates are high (reported up to 20%), it is thought that revaccination is safe as ELS is generally well tolerated and the condition is self-limiting.5 It is important to note that all authors investigated ELS only with regard to diphtheria tetanus acellular pertussis-containing vaccines. Due to its striking appearance, ELS might be mistaken for bacterial cellulitis or a type I allergic reaction. As in our cases, this might lead to unnecessary diagnostic testing and treatment. It is important for the clinician to recognize ELS as a benign and self-limiting condition that does not require intervention.
Emily Cohen, MD Sabrin Tahri, BSc Ingrid Lukkassen, MD
REFERENCES 1. Netherlands Pharmacovigilance Centre LAREB. Season influenza vaccines and extensive limb swelling (ELS). Lareb Q Rep. 2012;2:28–34. 2. Woo EJ, Burwen DR, Gatumu SN, et al; Vaccine Adverse Event Reporting System Working Group. Extensive limb swelling after immunization: reports to the vaccine adverse event reporting system. Clin Infect Dis. 2003;37:351–358. 3. Liese JG, Stojanov S, Zink TH, et al. Safety and immunogenicity of Biken acellular pertussis vaccine in combination with diphtheria and tetanus toxoid as a fifth dose at four to six years of age. Munich Vaccine Study Group. Pediatr Infect Dis J. 2001;20:981–988. 4. Scheifele DW, Halperin SA, Ferguson AC. Assessment of injection site reactions to an acellular pertussis-based combination vaccine, including novel use of skin tests with vaccine antigens. Vaccine. 2001;19:4720–4726. 5. Rennels MB, Black S, Woo EJ, et al. Safety of a fifth dose of diphtheria and tetanus toxoid and acellular pertussis vaccine in children experiencing extensive, local reactions to the fourth dose. Pediatr Infect Dis J. 2008;27:464–465.
Risk of Rotavirus Vaccination for Children with SCID To the Editors: uinn et al.1 demonstrated reduced morbidity and mortality from acute gastroenteritis by rotavirus vaccination in young children in Australia. We agree with the consensus statement highlighting these benefits in both developed and developing settings, which clearly outweigh the small increased risk of intussusception.2 Nevertheless, we think that the additional risk rotavirus vaccination poses for children with severe combined immunodeficiency (SCID) needs comment. Recently, we saw a 21-month-old boy from a country in the Middle-East who was in reduced general condition for further diagnosis and management at our center. Although his older brother died at 20 months of age suffering from postvaccinational BCGitis and severe cytomegalovirus infection, our patient had received rotavirus vaccine (Rotarix, GlaxoSmithKline Biologicals, Rixensart, Belgium) twice at 3 and 4 months of age. Ever since, he suffered from chronic gastroenteritis, which resulted in severe stunting and wasting. The patient had a x-linked hemizygous mutation
Q
The authors have no conflicts of interest or funding to disclose. Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal’s website (www.pidj.com).
Copyright © 2014 by Lippincott Williams & Wilkins ISSN: 0891-3668/15/3401-0114 DOI: 10.1097/INF.0000000000000475
Department of Pediatrics St. Antonius Hospital Utrecht/Nieuwegein, The Netherlands
114 | www.pidj.com
The Pediatric Infectious Disease Journal • Volume 34, Number 1, January 2015
Copyright © 2014 by Lippincott Williams & Wilkins DOI: 10.1097/INF.0000000000000507
