Recovery of Resistant Alopecia Areata Treated with Tofacitinib: An 8-Year-Old Child's Case Report.

Tofacitinib has been reported to activity in the recovery of alopecia areata (AA) in several reports, mainly from Western countries. We report the case of a young Indian boy with resistant AA who was treated with tofacitinib-based therapy. Improvements in terms of hair regrowth were observed within 6-8 weeks of treatment with oral tofacitinib 2.5 mg BID tablets, and the hair regrowth was nearly complete by 5 months. There were no associated side effects and the treatment with tofacitinib was well-tolerated. Copyright:

INTRODUCTION

Alopecia areata (AA) is an autoimmune hair loss disorder that affects people of all ages, but the severe disease forms usually start during childhood.[12] The pediatric-onset AA has a worse prognosis as compared to the adult-onset AA.[34] The successful use of Janus kinase (JAK) inhibitors such as ruxolitinib, tofacitinib, and baricitinib, for the treatment of AA, has been reported in several reports.[456] Tofacitinib, a JAK 1/3 inhibitor, has shown a response in alopecia patients.[78] In this report, we present the case of a child with AA who was treated with tofacitinib.

CASE REPORT

An 8-year-old boy was presented to the clinical facility with complaints of patchy hair loss for the past 3–4 years. He had received oral minipulse therapy, azathioprine, and topical treatments including calcineurin inhibitors, steroids, and immunomodulatory drugs, which failed to show results, and the patient was resistant to these treatments.

The patient had extensive hair loss on the scalp. Based on the symptoms and history, the patient was diagnosed with nonresponsive AA. His vital signs and laboratory values including complete blood counts, liver function tests, thyroid function tests, etc., were reported normal. His initial Severity of Alopecia Tool score was around 50.

The patient's treatment was started with tofacitinib oral tablet 2.5 mg once daily along with mometasone cream once daily. After 1 month, tofacitinib dose was increased to 2.5 mg twice daily. He was followed up regularly to access hair growth.

After treatment initiation, appreciable hair growth was reported within 6–8 weeks. After 5 months, there was a nearly complete hair growth observed as compared to the baseline [Figure 1]. To avoid relapse, nearly after 5 months when the complete hair growth was seen, tofacitinib dose of 2.5 mg twice daily was continued for one additional month, then reduced to 2.5 mg once daily for the next 1 month, and further reduced to 2.5 mg every alternate day for another 1½ month. The laboratory investigation reports were normal during and posttreatment. Overall, the patient was on therapy without any side effects and highly satisfied with the treatment response.

Figure 1

Before and after pic

DISCUSSION

The JAK inhibitors, especially tofacitinib, have shown effectiveness in the AA through the diminution of the inflammatory cascade,[67] but the reports are mainly from the westernized world. Furthermore, the reports on JAK use in the pediatric population with AA include topical formulations majorly, considering the potential toxicity issues of oral formulations including increased risk for infection and myelosuppression.[29] The evidence on the use of oral tofacitinib in Indian children with AA is scarce. Our case report provides information on the use of oral tofacitinib tablet 2.5 mg twice daily to a young boy with AA for 5 months.

The long-term safety of JAK inhibitors has not been still established in children, a few clinical trials (NCT02592434 and NCT01500551) are underway to evaluate tofacitinib for arthritis in children (2–18 years). Tofacitinib was used at a dose of 2.5 mg twice daily is lower than the standard dose (5 mg twice daily) being used in previous reports,[10] but the treatment response was satisfactory in our patient. In line with previous reports, our experience suggests that tofacitinib was effective and well-tolerated in the treatment of AA in a young Indian boy and may be a therapeutic option for children with AA resistant to conventional therapy.

Consent

The patient's written informed consent was obtained.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understand that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.