Autopsy and Case Reports
https://autopsyandcasereports.org/article/doi/10.4322/acr.2020.197
Autopsy and Case Reports
Clinical Case Report

Exogenous Ochronosis (EO): Skin lightening cream causing rare caviar-like lesion with banana-like pigments; review of literature and histological comparison with endogenous counterpart

Amir Qorbani; Adnan Mubasher; George Peter Sarantopoulos; Scott Nelson; Maxwell Alexander Fung

Downloads: 4
Views: 2179

Abstract

Ochronosis is a cutaneous disorder caused by the accumulation of phenols, either endogenously as homogentisic acid in patients with alkaptonuria (autosomal recessive disorder with deficiency of the enzyme homogentisic acid oxidase), or exogenously in patients using phenol products such as topical creams containing hydroquinone or the intramuscular application of antimalarial drugs. Exogenous ochronosis (EO) typically affects the face and was reported in patients with dark skin such as Black South Africans or Hispanics who use skin-lightening products containing hydroquinone for extended periods. Recently more cases have been reported worldwide even in patients with lighter skin tones, to include Eastern Indians, Asians, and Europeans. However, just 39 cases of EO have been reported in the US literature from 1983 to 2020. Here we present two cases; a 69 and a 45-year-old female who were seen for melasma, given hydroquinone 4% cream daily and tretinoin 0.05%. Both patients noticed brown spots on their cheeks, which progressively enlarged and darkened in color. The diagnosis of ochronosis was confirmed by characteristic histopathological features on the punch biopsy. Unfortunately, neither patient responded to multiple treatments (to include, tazarotene 0.1% gel and pimecrolimus ointment, topical corticosteroids, and avoidance of hydroquinone containing products). We also present a case of classic (endogenous) ochronosis in a patient with alkaptonuria to picture the histological similarities of these two entities. EO is an important clinical consideration because early diagnosis and treatment may offer the best outcome for this notoriously refractory clinical diagnosis.

Keywords

Ochronosis, Ochronosis, Skin Care, Skin Cream, Skin Diseases, Skin Pigmentation

References

Findlay GH. Ochronosis.

 

Clin Dermatol. 1989;7(2):28-35. [https://doi.org/10.1016/0738-081X(89)90054-0]. [PMID:2667739]

 

Laymon CW. Ochronosis.

 

 

AMA Arch Derm Syphilol. 1953;67(6):553-60. [https://doi.org/10.1001/archderm.1953.01540060015003]. [PMID:13050178]

 

Phillips JI, Isaacson C, Carman H. Ochronosis in black South Africans who used skin lighteners.

 

 

Am J Dermatopathol. 1986;8(1):14-21. [https://doi.org/10.1097/00000372-198602000-00003]. [PMID:3706666]

 

Kramer KE, Lopez A, Stefanato CM, Phillips TJ. Exogenous ochronosis.

 

 

J Am Acad Dermatol. 2000;42(5 Pt 2):869-71. [https://doi.org/10.1016/S0190-9622(00)90257-3]. [PMID:10767690]

 

Findlay GH, Morrison JG, Simson IW. Exogenous ochronosis and pigmented colloid milium from hydroquinone bleaching creams.

 

 

Br J Dermatol. 1975;93(6):613-22. [https://doi.org/10.1111/j.1365-2133.1975.tb05110.x]. [PMID:1220808]

 

Beddard AP, Plumtre CM. A further note on ochronosis associated with carboluria.

 

 

Q J Med. 1912(4);5:505-8.

 

Jordaan HF, Van Niekerk DJ. Transepidermal elimination in exogenous ochronosis.

 

 

Am J Dermatopathol. 1991;13(4):418-24. [https://doi.org/10.1097/00000372-199108000-00015]. [PMID:1928626]

 

Penneys NS. Ochronosislike pigmentation from hydroquinone bleaching creams.

 

 

Arch Dermatol. 1985;121(10):1239-40. [https://doi.org/10.1001/archderm.1985.01660100019003]. [PMID:4037813]

 

Bruce S, Tschen JA, Chow D. Exogenous ochronosis resulting from quinine injections.

 

 

J Am Acad Dermatol. 1986;15(2 Pt 2):357-61. [https://doi.org/10.1016/S0190-9622(86)70178-3]. [PMID:3734183]

 

Jacyk W. Annular granulomatous lesions in exogenous ochronosis are manifestation of sarcoidosis.

 

 

Am J Dermatopathol. 1995;17(1):18-22. [https://doi.org/10.1097/00000372-199502000-00004]. [PMID:7695007]

 

Levin CY, Maibach H. Exogenous ochronosis: an update on clinical features, causative agents and treatment options.

 

 

Am J Clin Dermatol. 2001;2(4):213-7. [https://doi.org/10.2165/00128071-200102040-00002]. [PMID:11705248]

 

Hull P, Procter P. The melanocyte: an essential link in hydroquinone-induced ochronosis.

 

 

J Am Acad Dermatol. 1990;22(3):529-31. [https://doi.org/10.1016/S0190-9622(08)80401-X]. [PMID:2312837]

 

Touart D, Sau P. Cutaneous deposition diseases. Part II.

 

 

J Am Acad Dermatol. 1998;39(4 Pt 1):527-44. [https://doi.org/10.1016/S0190-9622(98)70001-5]. [PMID:9777759]

 

Ho JD, Vashi N, Goldberg LJ. Exogenous ochronosis as an elastotic disease: a light-microscopic approach.

 

 

Am J Dermatopathol. 2019;Publish Ahead of Print. [https://doi.org/10.1097/DAD.0000000000001571]. [PMID:31725487]

 

Andersen FA, Bergfeld WF, Belsito DV, et al. Final amended safety assessment of hydroquinone as used in cosmetics.

 

 

Int J Toxicol. 2010;29(6, Suppl):274S-87. [https://doi.org/10.1177/1091581810385957]. [PMID:21164074]

 

Simmons BJ, Griffith RD, Bray FN, Falto-Aizpurua LA, Nouri K. Exogenous ochronosis: a comprehensive review of the diagnosis, epidemiology, causes, and treatments.

 

 

Am J Clin Dermatol. 2015;16(3):205-12. [https://doi.org/10.1007/s40257-015-0126-8]. [PMID:25837718]

 

Levitt J. The safety of hydroquinone: a dermatologist’s response to the 2006 Federal Register.

 

 

J Am Acad Dermatol. 2007;57(5):854-72. [https://doi.org/10.1016/j.jaad.2007.02.020]. [PMID:17467115]

 

Dogliotti M, Leibowitz M. Granulomatous ochronosis -- a cosmetic-induced skin disorder in Blacks.

 

 

S Afr Med J. 1979;56(19):757-60. [PMID:505210]

 

Gil I, Segura S, Martínez-Escala E, et al. Dermoscopic and reflectance confocal microscopic features of exogenous ochronosis.

 

 

Arch Dermatol. 2010;146(9):1021-5. [https://doi.org/10.1001/archdermatol.2010.205]. [PMID:20855704]

 

Qorbani A, Fereidouni F, Levenson R, et al. Microscopy with ultraviolet surface excitation (MUSE): A novel approach to real-time inexpensive slide-free dermatopathology.

 

 

J Cutan Pathol. 2018;45(7):498-503. [https://doi.org/10.1111/cup.13255]. [PMID:29660167]

 

Hardwick N, Van Gelder LW, Van der Merwe CA, Van der Merwe MP. Exogenous ochronosis: an epidemiological study.

 

 

Br J Dermatol. 1989;120(2):229-38. [https://doi.org/10.1111/j.1365-2133.1989.tb07787.x]. [PMID:2923796]

 

Bellew SG, Alster TS. Treatment of exogenous ochronosis with a Q-switched alexandrite (755 nm) laser.

 

 

Dermatol Surg. 2004;30(4 Pt 1):555-8. [PMID:15056151]

 

Kramer K, Lopez A, Stefanato C, Phillips TJ. Exogenous ochronosis.

 

 

J Am Acad Dermatol. 2000;42(5 Pt 2):869-71. [https://doi.org/10.1016/S0190-9622(00)90257-3]. [PMID:10767690]

 

Kanechorn-Na-Ayuthaya P, Niumphradit N, Aunhachoke K, Nakakes A, Sittiwangkul R, Srisuttiyakorn C. Effect of combination of 1064 nm Q-switched Nd:YAG and fractional carbon dioxide lasers for treating exogenous ochronosis.

 

 

J Cosmet Laser Ther. 2013;15(1):42-5. [https://doi.org/10.3109/14764172.2012.748198]. [PMID:23368689]

 

Martins VM, Sousa AR, Portela NC, Tigre CA, Gonçalves LM, Castro RJ Fo. Exogenous ochronosis: case report and literature review.

 

 

An Bras Dermatol. 2012;87(4):633-6. [https://doi.org/10.1590/S0365-05962012000400021]. [PMID:22892783]

 

Lawrence N, Bligard CA, Reed R, Perret WJ. Exogenous ochronosis in the United States.

 

 

J Am Acad Dermatol. 1988;18(5 Pt 2):1207-11. [https://doi.org/10.1016/S0190-9622(88)70126-7]. [PMID:3372785]

 

Gandhi V, Verma P, Naik G. Exogenous ochronosis after prolonged use of topical hydroquinone (2%) in a 50-year-old Indian female.

 

 

Indian J Dermatol. 2012;57(5):394-5. [https://doi.org/10.4103/0019-5154.100498]. [PMID:23112363]

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 

 


Submitted date:
02/21/2020

Accepted date:
06/17/2020

Publication date:
09/02/2020

5f4fee3d0e8825ee227dc2e3 autopsy Articles
Links & Downloads

Autops Case Rep

Share this page
Page Sections