An Excellent Response to Tofacitinib in a Pediatric Alopecia Patient: A Case Report and Review
August 2018 | Volume 17 | Issue 8 | Features | 914 | Copyright © August 2018
Liza Brown DO and Stanley Skopit DO MSE FAOCD FAAD
Larkin Community Hospital/NSU-COM, New York, NY
KD is an 8 year-old male patient who presented to our clinic in December 2016 with a history of patchy hair loss for many months duration that was worsening. KD’s past medical history was notable for atopic dermatitis, and a positive family history of autoimmune thyroid disease. Upon examination he had well circumscribed areas of hair loss throughout his scalp, with exclamation mark hairs seen on dermoscopy. Eyebrows and eyelashes were intact, no epidermal changes of scale or erythema were noted on the scalp and no palpable lymph nodes were present. He was diagnosed with alopecia areata at this time and was treated with Clobetasol 0.05% solution QHS as well as Kenalog 2.5 mg/ml injections to the areas of hair loss. Patient followed up two months later with worsening of his alopecia at a rapid pace, presenting now with hair loss of the entire scalp and loss of the eyebrows. He was diagnosed with progression to alopecia universalis at that time, with a corresponding SALT (Severity of Alopecia Tool) score of 100.
Both KD and his mother stated the hair loss was causing much distress in the patient’s life both at school and at home. After a thorough discussion of treatment alternatives to include continued topical high dose steroids, intralesional injections, high dose oral methylprednisolone, topical irritation with anthralin, topical immunotherapy with diphenylcyclopropenone (DPCP) or squaric acid dibutylester (SADBE) and systemic immunosuppressives, both the mother, patient and clinician agreed to try tofacitinib 5 mg twice daily with continued usage of topical steroids. Patient and his family was counseled about support groups, and local meetings to ease the mental distress associated with this condition.
After baseline labs were obtained and reported within normal limits, to include CBC, CMP, thyroid studies, lipids and Quanitferon gold, KD was started on tofacitinib 5 mg BID. Labs were repeated one month later, and 3 months ongoing thereafter.
At KD’s 3 month follow up, after starting tofacitinib 5 mg twice daily, KD showed complete regrowth of the eyebrows with minimal hair growth of the posterior occiput (Figure 1 a-d). At KD’s 6 month follow up he had 100% regrowth of eyebrows and complete scalp regrowth, resulting in a SALT score of 0 (Figure 2).
KD reported no side effects until month 6, after full hair regrowth, when patient started to report mild headaches. Drug holiday was offered but the patients family chose to discontinue treatment at this time as they were concerned side effects were secondary to medication usage. Unfortunately, patient was lost to follow-up after the discontinuation of treatment. From previous case reports we can postulate that his alopecia returned to baseline after discontinuation of tofacitinib.
KD had an incredible response to treatment, as has been reported previously in literature of adolescents using these novel therapies. This is the youngest patient ever reported to be successfully treated with oral tofacitinib 5 mg twice daily for alopecia and its variants.
J Drugs Dermatol. 2018;17(8):914-917.
Alopecia areata (AA) is an autoimmune disorder characterized by the sudden onset of non-scarring hair loss in a patch distribution.1 The hair loss can occur in any hair bearing area, with the scalp being the most common location in 90% of cases.2 Alopecia is defined by the extent of hair loss and distribution.2 Variations of alopecia occur and include reticular alopecia, ophiasis (band like area of hairloss on occiput), sisapho or ophiasis inversus (pattern opposite to ophiasis) and diffuse alopecias with complete hair loss on the scalp (alopecia totalis, AT), and complete scalp and body hair loss (alopecia universalis, AU).2 Exclamation point hairs, which are broken hairs that taper at the proximal end, are pathognomonic for this disease.1 The incidence of alopecia areata is between 0.1%- 0.2% of the population, occurring equally in males and females.1,2 Pediatric alopecia accounts for roughly 20% of overall alopecia cases and it is believed roughly 60% of AA patients will develop alopecia before the age of 20.2 Of note, AT occurring in a child less than one year of age is rare, and atrichia congenita should be considered.1 In the U.S,. an estimated 1.7% of the population will experience at least one episode of alopecia areata during their lifetime.1,2While the etiology and pathogenesis of alopecia remain uncertain, many factors have been described in its pathogenesis and include genetic factors, history of atopy, stress, non-specific immune response and personal and family history of autoimmune diseases.1 Autoimmune thyroid disease has the strongest association with AA with an incidence reported between 8-28%.2 A higher