Remission of Alopecia Universalis Following Successful Clinical Islet Transplantation

To the Editor: Previously, we reported three cases of alopecia after clinical islet transplantation in type 1 diabetic patients (1Zuk DM Koh A Imes S Shapiro AMJ Senior PA Three cases of alopecia following clinical islet transplantation.Am J Transplant. 2011; 11: 163-168Abstract Full Text Full Text PDF Scopus (10) Google Scholar). Like type 1 diabetes, alopecia is an autoimmune condition, but autoreactive T cells are directed against hair follicles. Alopecia is an uncommon complication of solid organ transplantation and has been attributed to immunosuppressant drugs (2Tricot L Lebbé C Pillebout E Martinez F Legendre C Thervet E Tacrolimus-induced alopecia in female kidney-pancreas transplant recipients.Transplantation. 2005; 80: 1546-1549Crossref PubMed Scopus (47) Google Scholar). Remission of alopecia following clinical islet transplantation is, therefore, unexpected. A 48-year-old Caucasian female with type 1 diabetes since age 22, developed alopecia universalis (including loss of eyebrows, eyelashes and pubic hair) 9 years later. Because of frequent, severe episodes of hypoglycemia with unawareness (Clarke score 7, HYPO score 3464) she underwent two islet infusions in May and June 2010, receiving 6358 and 6272 IE/kg, and achieved insulin independence by September 2010. For the initial islet infusion, thymoglobulin (6 mg/kg) was used for induction with etanercept (50 mg, day 0 and 25 mg, days 3, 7, 10). Before the second infusion, basiliximab (20 mg, days 0 and 4) was administered and etanercept dosing repeated. Maintenance immunosuppression was with tacrolimus (10–12 ng/mL for 3 months, then 8–10 ng/mL) and mycophenolate mofetil (1 g twice daily). Profound lymphopenia (0.1 × 109/l) resulted from the administration of thymoglobulin and relative lymphopenia (0.6 × 109/l) persists 6 months later. Eight weeks after the initial infusion, the patient reported growth of eyelashes, eyebrows, scalp and body hair which was easily seen on clinical examination. Hair growth has continued to date, now 12 months since transplant, although hair growth on the scalp is relatively sparse. She remains insulin-independent. Alopecia universalis is the rarest (estimated 1 per 100 000) and most severe form of alopecia areata. The course of alopecia areata is variable. Hair regrowth can occur in 50% by 1 year (3Tobin DJ Characterization of hair follicle antigens targeted by the anti-hair follicle immune response.J Investig Dermatol Symp Proc. 2003; 8: 176-181Abstract Full Text Full Text PDF PubMed Scopus (63) Google Scholar), but new areas of hair loss develop in most, and remission is uncommon in cases progressing to alopecia universalis. Experimental data supports a causative role for both cytotoxic T cells and humoral factors. Putative autoantigens expressed by growing hair follicles include hair-specific keratin and trichohyalin (3Tobin DJ Characterization of hair follicle antigens targeted by the anti-hair follicle immune response.J Investig Dermatol Symp Proc. 2003; 8: 176-181Abstract Full Text Full Text PDF PubMed Scopus (63) Google Scholar). This remission of alopecia universalis following successful clinical islet transplantation supports the autoimmune basis of this condition and further indicates that this autoimmune attack causes the hair follicles to remain in telogen rather than being completely destroyed. Although this remission could simply be due to the effects of maintenance immunosuppression to prevent ongoing autoimmune attack on hair follicles, the use of lymphodepleting antibodies for induction may involve the deletion of auto-reactive T-lymphocytes or the emergence of regulatory T cells. Remission due to alterations in T-cell behavior seems more likely because remission of alopecia has been described after allogeneic hematopoietic stem cell transplantation (4Seifert B Passweg JR Heim D et al.Complete remission of alopecia universalis after allogeneic hematopoietic stem cell transplantation.Blood. 2005; 105: 426-427Crossref PubMed Scopus (18) Google Scholar). Although manipulation of autoreactive T cells has formed the basis for clinical trials to reverse new onset type 1 diabetes (5Keymeulen B Vandemeulebroucke E Ziegler A et al.Insulin needs after CD3-antibody therapy in new-onset type 1 diabetes.N Engl J Med. 2005; 352: 2598-2608Crossref PubMed Scopus (947) Google Scholar), the regulation of autoimmunity is complex. In general, lymphodepletion is a potent stimulus for the development of autoimmunity by inducing homeostatic proliferation (6Baccala R Theofilopoulos A The new paradigm of T-cell homeostatic proliferation-induced autoimmunity.Trends Immunol. 2005; 26: 5-8Abstract Full Text Full Text PDF PubMed Scopus (94) Google Scholar) whereas in this case, remission of alopecia may have been due to depletion of autoreactive T cells. The authors of this manuscript have no conflicts of interest to disclose as described by the American Journal of Transplantation.

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