Abstract

A 19-year-old Japanese woman was referred to us with the complaints of arthralgia and meralgia following human papillomavirus (HPV) vaccination. She received HPV vaccination at the age of 15 years and three years later, she developed intermittent arthralgia, meralgia, and numbness in limbs. There were no orthostatic dysregulation symptoms. She had hypertelorism, and brachydactyly in both the hands, and revealed mild cubitus varus deformity with lateral instability. X-ray examination disclosed hypoplasia of the humeral capitellum and trochlea in elbow joints. G-banded chromosomes were shown to be composed of 48, XXXX. She was, therefore, diagnosed with XXXX syndrome, which explained the reason for her limb symptoms. Although some girls with HPV vaccination complain of various symptoms including limb pain and numbness, exact underlying cause of these symptoms needs to be ascertained carefully for reaching a final diagnosis.

Highlights

  • Human papillomavirus (HPV) vaccine was introduced for the prevention of uterine and cervical cancer [1]

  • Cases with polysomy X accompanied with systematic lupus erythematosus (SLE) were reported and it is likely that X-chromosome polysomy plays a role in higher incidence of autoimmune diseases [14]

  • We previously reported patients with arthritis after human papillomavirus (HPV) vaccination [15]

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Summary

Introduction

Human papillomavirus (HPV) vaccine was introduced for the prevention of uterine and cervical cancer [1]. This post-HPV vaccination syndrome is explainable by a combination of orthostatic dysregulation, chronic regional pain syndrome (CRPS), and brain cognitive dysfunction [10]. We report a case wherein the patient was diagnosed with XXXX syndrome during examination for suspected post-HPV vaccination syndrome. We obtained the patient’s consent for publishing this case report

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