ABSTRACT
Vitiligo is an acquired idiopathic pigmentary skin disorder characterized by the development of white macules and patches due to the loss of functioning melanocytes. In this report, we describe a case of a patient with a longstanding history of dermatitis herpetiformis (DH) and celiac disease that developed rapidly progressing, biopsy-confirmed generalized vitiligo after 11 months of treatment with anti-inflammatory medication sulfasalazine, prescribed for the patient's DH. To the best of our knowledge, this is the first case report which has demonstrated the possible biochemical pathways, triggered by sulfasalazine, in the development of vitiligo.
Subject(s)
Celiac Disease/drug therapy , Dermatitis Herpetiformis/drug therapy , Skin/pathology , Sulfasalazine/adverse effects , Vitiligo/chemically induced , Anti-Inflammatory Agents, Non-Steroidal/adverse effects , Biopsy , Celiac Disease/complications , Celiac Disease/diagnosis , Dermatitis Herpetiformis/complications , Dermatitis Herpetiformis/diagnosis , Humans , Male , Middle Aged , Sulfasalazine/therapeutic use , Vitiligo/diagnosisABSTRACT
Pyoderma gangrenosum (PG)-like ulcerations are a rare clinical manifestation of methylenetetrahydrofolate reductase (MTHFR) mutation. We describe a patient considered to have PG who was treated with long-term high doses of systemic corticosteroids and multiple immunosuppressive agents for several years. In spite of this continuous aggressive therapy, the lesions did not improve but continued to get worse. She developed many significant and catastrophic side effects to them. When referred to our dermatology centre, on investigation, it was discovered that she has an MTHFR mutation. It seemed reasonable to presume that PG-like lesions were related to it. Treatment with a biologically active form of folate-[6S]-5-MTHF-with vitamins B6 and B12 was initiated. It was considered to be beneficial and capable of reducing hyperhomocysteinaemia and endothelial damage consequent from it. Since the institution of this treatment, the patient has begun to show very gradual but slow and incremental improvement.
Subject(s)
Methylenetetrahydrofolate Reductase (NADPH2)/genetics , Pyoderma Gangrenosum/pathology , Skin Ulcer/pathology , Tetrahydrofolates/therapeutic use , Diagnosis, Differential , Female , Humans , Hyperhomocysteinemia/blood , Middle Aged , Mutation , Pyoderma Gangrenosum/diagnosis , Pyoderma Gangrenosum/drug therapy , Rare Diseases , Skin Ulcer/drug therapy , Skin Ulcer/genetics , Tetrahydrofolates/administration & dosage , Treatment Outcome , Vitamin B 12/administration & dosage , Vitamin B 12/therapeutic use , Vitamin B 6/administration & dosage , Vitamin B 6/therapeutic use , Vitamin B Complex/therapeutic useABSTRACT
In this report, we describe a case of a patient with a clinical history of systemic sarcoidosis and psoriasis who developed biopsy-confirmed perforating and necrotizing cutaneous granulomas after 12 months of treatment with adalimumab, a tumor necrosis factor-alpha-inhibiting, anti-inflammatory, biologic medication, prescribed for the patient's psoriasis. Although rare reports of a "sarcoidosis-like" reaction associated with select tumor necrosis factor-alpha agents exist, to the best of our knowledge, perforating and necrotizing cutaneous granulomas after treatment with adalimumab has not been previously reported. Given the patient's history of systemic sarcoidosis, the differential diagnosis includes reactivation of latent sarcoidosis with adalimumab as a trigger.
Subject(s)
Adalimumab/adverse effects , Granuloma/chemically induced , Granuloma/therapy , Psoriasis/drug therapy , Sarcoidosis/drug therapy , Wound Healing/physiology , Adalimumab/therapeutic use , Aged , Anti-Bacterial Agents/therapeutic use , Antibodies, Monoclonal, Humanized/adverse effects , Antibodies, Monoclonal, Humanized/therapeutic use , Biopsy, Needle , Combined Modality Therapy , Drug-Related Side Effects and Adverse Reactions/etiology , Drug-Related Side Effects and Adverse Reactions/pathology , Drug-Related Side Effects and Adverse Reactions/therapy , Follow-Up Studies , Granuloma/pathology , Humans , Immunohistochemistry , Leg , Male , Necrosis/chemically induced , Necrosis/surgery , Prednisone/therapeutic use , Psoriasis/complications , Psoriasis/pathology , Recurrence , Sarcoidosis/complications , Sarcoidosis/pathology , Severity of Illness Index , Skin Transplantation/methods , Skin Ulcer/chemically induced , Skin Ulcer/pathology , Skin Ulcer/surgery , Time FactorsABSTRACT
BACKGROUND: Existing therapies for vitiligo are limited in efficacy and can be associated with undesirable side effects. Topical Janus kinase inhibitors may offer a new therapeutic option for vitiligo. OBJECTIVE: We sought to assess the role of topical ruxolitinib 1.5% cream, a Janus kinase inhibitor, in vitiligo treatment. METHODS: This 20-week, open-label, proof-of-concept trial of twice-daily topical ruxolitinib 1.5% cream was conducted in 12 patients with a minimum of 1% affected body surface area of vitiligo. The primary outcome was percent improvement in Vitiligo Area Scoring Index from baseline to week 20. RESULTS: Of 12 patients screened, 11 were enrolled and 9 completed the study (54.5% men; mean age, 52 years). Four patients with significant facial involvement at baseline had a 76% improvement in facial Vitiligo Area Scoring Index scores at week 20 (95% confidence interval, 53-99%; P = .001). A 23% improvement in overall Vitiligo Area Scoring Index scores was observed in all enrolled patients at week 20 (95% confidence interval, 4-43%; P = .02). Three of 8 patients responded on body surfaces and 1 of 8 patients responded on acral surfaces. Adverse events were minor, including erythema, hyperpigmentation, and transient acne. LIMITATIONS: Limitations of the study include the small sample size and open-label study design. CONCLUSIONS: Topical ruxolitinib 1.5% cream provided significant repigmentation in facial vitiligo and may offer a valuable new treatment for vitiligo.
