A Case of Bilateral Acute Depigmentation of the Iris in One of Two Identical Twins

Abstract

Background: Bilateral Acute Depigmentation of the Iris (BADI) is a condition which was first described in a case series from Turkey by Tugal-Tutkin and Urgancioglu in 2006.1 The condition is characterized by bilateral acute depigmentation and discoloration of the iris stroma, pigment dispersion, and deposition of pigment in the angle. In our case we report a patient who developed BADI after receiving pitcher plant extract injections for chronic migraine, while her identical twin sister has normal iris architecture and pigmentation and never received any pitcher plant injections. Case Presentation: Patient is a 41-year-old female with history of pitcher plant extract injections to her face for chronic migraine, who later developed bilateral depigmentation of the iris. She did not have any signs of anterior segment uveitis or iridocyclitis. She has an identical twin sister who maintained normal iris pigmentation during the entire course. Discussions and Conclusion: Bilateral Acute depigmentation of the is a recently discovered condition described in the literature in Turkish patients1,2. This condition affects mainly young females and is characterized by acute bilateral stromal depigmentation, without other pathologic ocular findings. These patients usually maintain normal vision and do not develop significant glaucoma from pigment collecting in the anterior chamber angle. This condition can be mistaken for Fuchs’ heterochromic iridocyclitis, pigment dispersion syndrome, pseudoexfoliation syndrome, and viral iridocyclitis. This is the first reported case in North America and is important for differentiation from the above pathologies. Our patient had a history of pitcher plant extract injections to the face but it is unclear if this is associated with our patient’s development of BADI. As awareness of this condition progresses, a possible etiology may be elucidated.

Bilateral Acute Depigmentation of Iris (BADI) and Bilateral Acute Iris Transillumination (BAIT)-An Update

“…By contrast, BADI has a more benign course with a shorter duration of pigment discharge from the iris stroma, a lower incidence of IOP rise that is only transient, and iris changes that can be reversible. 3,4,5,7,12,18…”

“…6 Other triggers have been reported as well, including accidental exposure to insecticide spray, fumigation therapy, and pitcher plant extract injections to the face for chronic migraine. 5,9,33 Viral serology for varicella-zoster virus, cytomegalovirus, Epstein-Barr virus, and herpes simplex virus has proven unhelpful 3,4,6,7,12 and polymerase chain reaction analysis of aqueous humor for these pathogens has yielded negative results. 3,10,13,19,34 During the recent COVID-19 pandemic, a total of 19 cases of BADI and BAIT were reported, 18 cases from Turkey and 1 from India.…”

“…4,9 On the other hand, persistent depigmentation was seen at 4 and 5 years in two other cases reported in the literature. 5,8 There are no fundus changes throughout the disease course and visual acuity is not affected in BADI patients. 3,7 Clinical Features of BAIT In 2004, Bringas Calvo and Iglesias Cortiñas 2 reported bilateral acute anterior uveitis with pigment dispersion in an older woman treated with moxifloxacin for pneumonia.…”

“…Following these early publications, many cases have been reported from other countries. 4,5,6,7,8,9 BADI affects predominantly young females. It presents as a sudden-onset redness, ocular pain, tearing, and photophobia in both eyes simultaneously or within a few days of each other.…”

“…The patients usually maintain normal visual acuity. 1,3,4,5 Patients with BADI present with severe diffuse episcleral injection, more pronounced than ciliary injection. They do not have signs of intraocular inflammation, but they present with circulating pigment in the anterior chamber, which may…”