LETTER TO THE EDITOR

Fibromyalgia-Like Illness in 2 Girls After Human Papillomavirus Vaccination To the Editor: hronic arthralgias are recognized human papillomavirus (HPV) vaccine side effects. The United Kingdom National Health Service Web page contains the following statement: “More than 1 in 100 people who have the Gardasil HPV vaccine experience fever, nausea (feeling sick), and painful arms, hands, legs, or feet.”1 In 2013, the Japanese Health Ministry stopped actively recommending HPV vaccination in young girls. The ministry was investigating 24 cases of an illness similar to complex regional pain syndrome that emerged after HPV vaccination.2 The name complex regional pain syndrome is a relatively new term to describe a condition formerly known as reflex sympathetic dystrophy. Fibromyalgia is characterized by chronic widespread pain and widespread allodynia. Other fibromyalgia distinctive features are paresthesias, chronic fatigue, and sleeping difficulties.3 Fibromyalgia and complex regional pain syndrome share clinical and possibly pathogenetic features4. A consistent line of investigation suggests that fibromyalgia is a sympathetically maintained neuropathic pain syndrome.5 The objective of this communication was to describe 2 previously healthy girls who developed a chronic incapacitating fibromyalgia-like illness after HPV vaccination.

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CASE 1 An 11-year-old girl had a single quadrivalent recombinant HPV vaccine (Gardasil) injection. She did not receive the recommended 3 injections regimen. Eleven months later, her physician prescribed a new complete 3-dosing program. Right after the second jab, she developed severe pain in the injected arm that lasted for a week. Two days after the third vaccine shot, she developed again severe pain in the injected arm, now accompanied by a swollen red hand. Two weeks later, her symptoms spread to the opposite arm and then all over her body. Her pain was accompanied by severe paresthesias. She was not able to attend school anymore. She developed insomnia and profound fatigue. Before the onset of her chronic illness, there

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was no history of trauma or psychiatric or family problems. Extensive diagnostic workup was normal. Blood test was negative for infectious, inflammatory, or autoimmune markers. Cerebrospinal fluid analysis, head and neck magnetic resonance imaging, and electromyography were normal. A bone scan was not done. Different analgesics, anti-inflammatory drugs, corticosteroids, or antineuropathic medications gave her no sustained relief. Seven months after the onset of her illness, widespread pain and paresthesias continued. At this point, physical examination showed a distressed girl. Neurological examination was normal except for severe generalized allodynia. Eighteen of 18 fibromyalgia tender points turned out to be positive. There was no joint swelling. The remainder of the physical examination was unrevealing.

CASE 2 A healthy 14-year-old girl developed severe neck pain 4 weeks after her second HPV vaccine shot (Gardasil). For the ensuing weeks, pain spread to her arms and then to her legs. She developed paresthesias in the 4 extremities. Because of her symptoms, she could not sleep well or attend school. She lived in a harmonic household, and there was no personal history of trauma, anxiety, or depression. She has a family history of spondyloarthropathy. Physical examination showed an uncomfortable-looking girl crying in pain. She had exquisite tenderness at palpations all over her body including all 18 fibromyalgia tender point sites. The remainder of the physical examination was normal. There was no arthritis or enthesopathy. Extensive diagnostic workup was negative. No infectious, inflammatory, or auto immune illness could be detected. HLAB27 was negative. Several analgesic, corticosteroid, or antineuropathic medication provided only transient mild relief. Five months after the onset of her illness, she still had widespread pain and paresthesias.

DISCUSSION Both patients fulfilled the fibromyalgia criteria endorsed by the American College of Rheumatology in 1990 and also in 2010. Both girls have chronic widespread pain and widespread allodynia with tender points evident in 12 or more specific anatomical sites. Both cases also had other fibromyalgia typical features such as paresthesias, insomnia, and chronic fatigue.2 In case 1, there was immediate temporal

cause-effect relationship between vaccine injection and severe pain. The illness in case 1 began as a complex regional pain syndrome located in the injected arm. She satisfied the International Association for the Study of Pain diagnostic criteria for complex regional pain syndrome6 that requires (1) preceding noxious event; (2) spontaneous pain or hyperalgesia/ hyperesthesia not limited to a single nerve territory and disproportionate to the inciting event; and (3) edema, temperature, or sudomotor abnormalities present in the affected limb, in particular distal sites. This girl’s condition evolved to a widespread fibromyalgia-like illness. Spreading of complex regional pain syndrome to other extremities is a well-recognized phenomenon.7 In case 2, vaccination-pain relationship is less clear; her intriguing illness developed 4 weeks after her second vaccine injection. In both instances, multiple specialist consultations and diagnostic procedures found no other plausible explanation for a severe persistent fibromyalgia-like illness developing at their young age. Both complex regional pain syndrome and fibromyalgia are controversial entities. We propose that fibromyalgia is a generalized complex regional pain syndrome based on the following arguments: In both conditions, there is female predominance, frequent onset after trauma, chronic nonnociceptive pain, paresthesias, sympathetic instability, and allodynia/ hyperesthesia.4 Our group has proposed that dorsal root ganglia may play a key role in fibromyalgia pain. Trauma or viral infection can induce dorsal root ganglia sympathetic fiber sprouting establishing abnormal sympathetic-nociceptive short circuits.5,8 It is tempting to speculate that in genetically susceptible individual an intramuscular-injected vaccine containing noninfectious virus plus the aluminum adjuvant substance could elicit similar changes. Literature review disclosed few case reports of fibromyalgia developing after other types of vaccination. The US National Vaccine Injury Compensation Program reported 11 fibromyalgia cases developing after rubella immunization.9 There is also single case of fibromyalgia starting after the third hepatitis B vaccination.10 Macrophagic myofasciitis is a well-established complication of aluminumcontaining vaccines. This condition may present as a fibromyalgia-like illness.11 Cases of macrophagic fasciitis may have raised

JCR: Journal of Clinical Rheumatology • Volume 20, Number 7, October 2014

Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.

JCR: Journal of Clinical Rheumatology • Volume 20, Number 7, October 2014

Letter to the Editor

REFERENCES

8. Vargas-Alarcon G, Alvarez-Leon E, Fragoso JM, et al. A SCN9A gene-encoded dorsal root ganglia sodium channel polymorphism associated with severe fibromyalgia. BMC Musculoskeletal Dis. 2012;13:23.

creatine kinase concentrations, increased erythrocyte sedimentation rate, myopathic electromyography, and a positive response to corticosteroid therapy.12 These features were not present in these 2 girls. The 2 cases herein described have prominent paresthesias and allodynia suggestive of a neuropathic rather than a myopathic pain etiology. Understandably, after their prolonged illness ordeal, these 2 girls and their families were reluctant to undergo an additional invasive procedure such as muscle biopsy. We are not aware of previous description of fibromyalgia-like illness after HPV immunization. We suggest that the medical community and regulatory agencies be aware of these possible adverse effects in order to define their real magnitude and, if indicated, to take corrective actions. Manuel Martínez-Lavín, MD Rheumatology Department Instituto Nacional de Cardiología Ignacio Chávez Mexico City Mexico [email protected]

© 2014 Lippincott Williams & Wilkins

1. HPV vaccines side effects. Available at: http:// www.nhs.uk/Conditions/vaccinations/Pages/ hpv-vaccine-cervarix-gardasil-side-effects. aspx. Accessed June 18, 2014. 2. World Health Organization. Weekly Epidemiological Record 2013;88: 301–312. 3. Wolfe F, Smythe HA, Yunus MB, et al. The American College of Rheumatology 1990 criteria for the classification of fibromyalgia: report of the multicenter criteria committee. Arthritis Rheum. 1990;33:160–172. 4. Martínez-Lavin M. Is fibromyalgia a generalized reflex sympathetic dystrophy? Clin Exp Rheumatol. 2001;19:1–3. 5. Martinez-Lavin M. Biology and therapy of fibromyalgia. Stress, the stress response system, and fibromyalgia. Arthritis Res Ther. 2007;9:216–219. 6. Birklein F. Complex regional pain syndrome. J Neurol. 2005;252:131–138. 7. Maleki J, LeBel AA, Bennett GJ, et al. Patterns of spread in complex regional pain syndrome, type I (reflex sympathetic dystrophy). Pain. 2000;88:259–266.

9. Weibel RE, Benor DE. Chronic arthropathy and musculoskeletal symptoms associated with rubella vaccines. A review of 124 claims submitted to the National Vaccine Injury Compensation Program. Arthritis Rheum. 1996;39:1529–1534. 10. Nancy AL, Shoenfeld Y. Chronic fatigue syndrome with autoantibodies—the result of an augmented adjuvant effect of hepatitis-B vaccine and silicone implant. Autoimmun Rev. 2008;8:52–55. 11. Authier FJ, Sauvat S, Champey J, et al. Chronic fatigue syndrome in patients with macrophagic myofasciitis. Arthritis Rheum. 2003;48:569–570. 12. Gherardi RK, Coquet M, Chérin P, et al. Macrophagic myofasciitis: an emerging entity. Groupe d'Etudes et Recherche sur les Maladies Musculaires Acquises et Dysimmunitaires (GERMMAD) de l’Association Française contre les Myopathies (AFM). Lancet. 1998; 352:347–352.

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Fibromyalgia-like illness in 2 girls after human papillomavirus vaccination.

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